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International Journal of
Case Reports and Images
Contents Vol. 5, No. 9 (September 2014)
Cover Image 642 A huge retroperitoneal lymphatic cyst

presenting as a mesenteric cyst, managed
Figure 3: X-ray features of both lower limbs laparoscopically
showing fusion of distal part of left tibia with the Manash Ranjan Sahoo, Leesa Misra,
right with lateral bending of left leg. Raghavendra Mohan Kaladagi, Manoj
Srinivas Gowda, Abinash Panda, Syam
Sundar Behera
Review Article 646 Appendicular abscess in left inguinal hernia

mimicking strangulation
Manash Ranjan Sahoo, Basavaraja C.,
604 Radioisotopes: An overview Kumar A.T., Sunil Jaiswal
Cover Figure: JKotya Naik Maloth, Nagalaxmi Velpula, Sridevi
Ugrappa, Srikanth Kodangal 650 Mixed connective tissue disorder: A case
report
Rishi Katewa, Jakhar R.S., Barala G.L.
Case Series
656 Primary ciliary dyskinesia (Kartagener
syndrome) in a 38-year-old Egyptian male:
610 Laparoscopic plication and mesh repair for A rare case
diastasis recti: A case series Motaz Badr Abdellatif Ibrahim
Manash Ranjan Sahoo, Kumar A.T.
Clinical Images
Case Report
661 Differentiating a simple cyst or metastatic
breast cancer: A medical dilemma
614 Cutaneous metastatic breast adenocarcinoma Waqas Jehangir, Zorawar Singh, Abdul I
twenty years post-mastectomy: A lesson to Mahmad, Teena Mathew
learn
Armand Asarian, Olubunmi Esan, Philip Xiao, 665 Multidrug resistant pyogenic liver
Segun Adeoye abscesses: A rare but fatal complication of
619 Aortoduodenal fistula after transperitoneal a life-saving procedure
repair of an inflammatory abdominal aortic Waqas Jehangir, Shilpi Singh, Andrea A
aneurysm: A case report Lewis, Shuvendu Sen
Denise MD Özdemir-van Brunschot, Giel G
Koning, J Adam van der Vliet
Letter to Editors
625 Unusually large sialolith of submandibular gland
Haci Taner Bulut
668 Trapezo-metacarpal dislocation diagnosed
629 Malignant mucosal melanoma of the nasal cavity: as sprain
A case report Monsef Boufettal, Rida-Allah Bassir,
Devika Gupta, Niti Goyal, Vandana Rana, Rajat Mohamed S. Berrada, Moradh El Yaacoubi
Jagani, Davendra Swarup
634 Erythropoietin induced miliaria crystallina: A
All Articles: possible new adverse effect of erythropoietin
Sumir Kumar, B.B. Mahajan, Sandeep Kaur,
Amarbir Singh
638 Sirenomelia: A case report
Swagat Mahapatra, Suruchi Ambasta
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Int J Case Rep Images 2014;5(9):604–609. Maloth et al. 604
www.ijcasereportsandimages.com
Review Article OPEN ACCESS
Radioisotopes: An overview
Kotya Naik Maloth, Nagalaxmi Velpula, Sridevi Ugrappa,
Srikanth Kodangal
Abstract to diagnose or treat a disease based on the

cellular function and physiology rather than
Many elements which found on earth exist relying on the anatomy.
in different atomic configurations and are
termed isotopes which have same atomic Keywords: Radioisotope, Nuclear medicine,
number but differ in their atomic mass. These Radiopharmaceuticals, Radionuclide imaging
unstable element decay by emission of energy
such isotopes, which emit radiation, are *********
called radioisotopes. Using of these isotopes
in various sectors like industries, agriculture,
healthcare and research centres has got a great How to cite this article
importance at present. In health care sector,
these isotopes are used in nuclear medicine Maloth KN, Velpula N, Ugrappa S, Kodangal S.
as diagnostic and therapeutic modalities. Radioisotopes: An overview. Int J Case Rep Images
Radionuclide imaging (or functional imaging) 2014;5(9):604–609.
is a branch of medicine which provides the
only means of assessing physiologic changes
doi:10.5348/ijcri-201457-RA-10012
that is a direct result of biochemical alterations
and is based on the radiotracer method. In
nuclear medicine procedures, radionuclides are
combined with other chemical compounds or
pharmaceuticals to form radiopharmaceuticals. Introduction
These radiopharmaceuticals, once administered
to the patient, can localize to specific organs Radionuclide imaging (or functional imaging) is a
or cellular receptors. This unique ability of branch of medicine which provides the only means of
radiopharmaceuticals allows nuclear medicine assessing physiologic changes that is a direct result of
biochemical alterations. In most cases, the information
is used by physicians to make a quick, accurate diagnosis
Kotya Naik Maloth1, Nagalaxmi Velpula2, Sridevi Ugrappa3,
Srikanth Kodangal4 of the patient’s illness [1]. This imaging is based on the
radiotracer method which assumes that radioactive
Affiliations: 1MDS, Assistant professor, Mamata Dental
College and Hospital, Khammam, Telangana, India; 2MDS,
atoms or molecules in an organism behave in a manner
Professor & Head, Sri Sai College of Dental Surgery, identical to that of their stable counterparts because they
Vikarabad, Telangana, India; 3MDS, Postgraduate, Sri Sai are chemically indistinguishable. Radiotracers allow
College of Dental Surgery, Vikarabad, Telangana, India; measurement of tissue function in vivo and provide
4
MDS, Associate professor, Sri Sai College of Dental an early marker of disease through measurement of
Surgery, Vikarabad, Telangana, India. biochemical change. As, other X-rays imaging methods
Corresponding Author: Dr. Kotya Naik. Maloth, H.No.5-5- assess changes by their differential absorption in tissues
47/1, Mustafa Nagar, Khammam-507001, Telangana, India; (tissue electron density); only anatomical or structural
Ph: +919885617131; Email: dr.kotyanaik.maloth@gmail.com changes —which are the later effects of some biochemical
process— can be assessed by these methods. Nuclear
medicine has applications in neurology, cardiology,
Received: 24 March 2014
oncology, endocrinology, lymphatic’s, urinary functions,
Accepted: 28 April 2014
Published: 01 September 2014
gastroenterology and pulmonology. Some forms of
International Journal of Case Reports and Images, Vol. 5 No. 9, September 2014. ISSN – [0976-3198]
radiation therapy are administered by nuclear specialists TYPES OF ISOTOPES USED IN MEDI-
and these include radioisotope administration. CAL FIELD
Isotopes have the same number of protons but
different number of neutrons and these elements have Various isotopes used in medical field as in diagnostic
same atomic number but differ in atomic mass. These and therapeutic aspects (Tables 1 and 2) [9, 10].
unstable element decay by emission of energy in the form In diagnostic application depending upon the type of
of alpha, beta (electron)/beta plus (positron) and gamma production these isotopes can listed as follows reactor
rays. Such isotopes, which emit radiation, are called isotopes and cyclotron isotopes (Table 3).
radioisotopes [2, 3]. These radioisotopes are also known
as radionuclides. These isotopes are used in various Mode of administration
sectors like industries, agriculture, healthcare and These isotopes either during diagnostic or therapeutic
research centres because of their characteristic nature of can be administered by inhalation (xenon, argon,
emitting radiation and their energies. nitrogen), oral (iodine) or intravenous (thallium, gallium).
Radioactive products which are used in medicine The most commonly used liquid radionuclides are
are referred to as radiopharmaceuticals [4]. technetium-99m, iodine-123, iodine-131, thallium-201,
Radiopharmaceuticals differ from other medically and gallium-67.
employed drugs since they generally elicit no The most commonly used gaseous/aerosol/
pharmacological response (owing to the minute radionuclides are xenon-133, krypton-81m, Technetium-
quantities administered) and they contain radionuclide. 99M and DTPA (diethylene-triamine-pentaacetate).
They are prepared by tagging the chosen carrier
component with an appropriate radioactive isotope. APPLICATIONS OF RADIOISOTOPES
The carrier component of the radiopharmaceutical is a
biologically active molecule used to localize the drug in In diagnostic aspects
a specific organ or group of organs to provide diagnostic
In nuclear medicine with advances included as
information about those tissues such as pyrophosphate
positron emission tomography (PET), imaging has
and methylene diphosphonate (MDP) compounds in
value in cardiovascular, neurological, psychiatric, and
skeleton bone tissues.
oncological diagnosis. Positron emission tomography is a
functional imaging modality that allows the measurement
EVOLUTION OF RADIOISOTOPES of metabolic reactions within the whole body. F-18 in
In 1898, discovery of polonium by Pierre and Marie FDG (fluorodeoxyglucose) which is an analog of glucose
Curie introduced the term “radioactive” [5]. Radium was has become very important in detection of cancers and
discovered by the Curie six months after the discovery of the monitoring of progress in their treatment, using PET.
polonium with the collaboration of the chemist G. Bemont The combination of PET scan and computed tomography
[6]. Radium played by far a more important role than (CT) scan in a single device provides simultaneous
polonium. Its separation in significant amount opened structural and biochemical information (fused images)
the way to its medical and industrial application and also under almost identical conditions, minimizing the
its use in laboratories. Later ‘uranic rays’ was discovered temporal and spatial differences between the two imaging
by Henri Becquerel in 1900 [5]. modalities and is called Fusion imaging [11, 12].
Overall 1800 isotopes are present, but at present only Single-photon emission computed tomography
up to 200 radioisotopes are used on a regular basis, and (SPECT) imaging technique was developed as an
most of them are produced artificially. Radioisotopes can enhancement of planar imaging enables the exact
be manufactured in several ways. anatomical site of the source of the emission to be
The most common is by neutron activation in a determined. This technique involves the detection
nuclear reactor. This involves the capture of a neutron by of gamma rays emitted singly (single photon) from
the nucleus of an atom resulting in an excess of neutrons radionuclides such as technetium-99m and thallium-201.
(neutron rich) which leads to the production of desired Radioimmunodetection/radioimmunoassay is an
radioisotope [2, 3]. in vitro nuclear medicine, is a very sensitive technique
Some radioisotopes are manufactured in a cyclotron, used to measure concentrations of antigens by use of
devised by Lawrence and Livingston in 1932 [7, 8] in antibodies.
which charged particles such as protons, deuterons and
alpha particles are introduced to the nucleus resulting In therapeutics aspects
in a deficiency of neutrons (proton rich). These particles The radiations given out by some radioisotopes
are accelerated to high energy levels and are allowed are very effective in curing certain diseases such as
to impinge on the target material. 11C, 13N, 18F, 123I, radiocobalt (60Co) is used in the treatment of brain tumor,
etc. are some of the isotopes that can be produced in a radiophosphorus (32P) in bone diseases and radioiodine
cyclotron. (131I) in thyroid cancer [4].
Table 1: Reactor isotopes used for diagnostic purpose [9, 10]
Isotope Half -life Uses

Used as the 'parent' in a generator to produce technetium-99m. Most widely used
Molybdenum-99 66 hours
isotope in nuclear medicine.
Used in to image the skeleton and heart muscle in particular, but also used for brain,
thyroid, lungs (perfusion and ventilation), liver, spleen, kidney (structure and filtration
Technetium-99m 6 hours
rate), gallbladder, bone marrow, salivary and lacrimal glands, heart blood pool, infection
and numerous specialised medical studies.
Chromium-51 27.7 days Used to label red blood cells and quantify gastrointestinal protein loss.
Used to study genetic diseases affecting copper metabolism, such as Wilson's and
Copper-64 13 hours
Menke diseases.
Holmium-166 26 hours Being developed for diagnosis and treatment of liver tumors.
Used in diagnostically to evaluate the filtration rate of kidneys and to diagnose deep
Iodine-125 60 days vein thrombosis in the leg. It is also widely used in radioimmunoassays to show the
presence of hormones in tiny quantities.
Used in diagnosis of abnormal liver function, renal (kidney) blood flow and urinary
Iodine-131 8 days
tract obstruction.
Iron-59 46 days Used in studies of iron metabolism in the spleen.
Potassium-42 12 hours Used for the determination of exchangeable potassium in coronary blood flow.
Rhenium-188 17 hours Used to beta irradiate coronary arteries from an angioplasty balloon.
Selenium-75 120 days Used in the form of selenomethionine to study the production of digestive enzymes.
Table 2: Reactor isotopes used in therapeutics [9, 10]

Isotopes Half -life Uses
Cobalt-60 10.5 months Formerly used for external beam therapy.
Dysprosium-165 2 hours Used as an aggregated hydroxide for synvectomy treatment of arthritis.
Erbium-169 9.4 days Used for relieving arthritis pain in synovial joints.
Iodine-125 60 days Used in cancer brachytherapy (prostrate and brain)
Iodine-131 8 days Widely used in treating thyroid cancer.
Iridium-192 74 days Supplied in wire form for use as an internal radiotherapy source for cancer treatment.
Palladium-103 17 days Used to make brachytherapy permanent implant seeds for early stage prostate cancer.
Phosphorus-32 14 days Used in the treatment of polycythemia vera.

Used for cancer brachytherapy and as silicate colloid for the relieving the pain of
Yttrium-90 64 hours
arthritis in larger synovial joints.
Radioactive sources are also available for carrier such as a monoclonal antibody tagged with the
brachytherapy with many nuclides and in various shapes alpha-emitting radionuclide. Lead-212 is being used
and size depending upon the type of their radiation in TAT for treating pancreatic, ovarian and melanoma
energy and emission [13]. cancers. An experimental development of this is boron
A new field is targeted alpha therapy (TAT) or neutron capture therapy (BNCT) using boron-10 which
alpha radioimmunotherapy, especially for the control concentrates in malignant brain tumors. Radionuclide
of dispersed cancers. The short range of very energetic therapy has progressively become successful in treating
alpha emissions is targeted into cancer cells, with persistent disease and doing so with low toxic side-effects.
Table 3: Cyclotron Radioisotopes

Isotope Half -life Uses
Carbon-11 20 minutes
Nitrogen-13 10 minutes
Positron emitters used in PET scan.
Oxygen-15 2 minutes
Fluorine-18 110 minutes
Cobalt-57 272 days as a marker to estimate organ size and for invitro diagnostic kits.
Gallium-67 78 hours Used for tumor imaging and localization of inflammatory lesions (infections).
Used for specialist diagnostic studies, e.g., brain studies, infection and colon transit
Indium-111 2.8 days
studies.
Increasingly used for diagnosis of thyroid function, it is a gamma emitter without the
Iodine-123 13 hours
beta radiation of I-131.
Can yield functional images of pulmonary ventilation, e.g., in asthmatic patients, and for
Krypton-81m 13 seconds
the early diagnosis of lung diseases and function.
Rubidium-82 65 hours Convenient PET agent in myocardial perfusion imaging.
With any therapeutic procedure the aim is to confine the • It can display blood flow.
radiation to well-defined target volumes of the patient. • Assessment of physiologic or functional change in
The doses per therapeutic procedure are typically 20–60 tissues, because of disease process.
Gy [14]. • Computer analysis and enhancement of results are
Radioimmunotherapy is dependent on three principal available.
interdependent factors: the antibody, the radionuclide
and the target tumor, and host and is most commonly Disadvantages radioisotope imaging
employed in the management of hematopoietic • Poor image resolution—often only minimal
neoplasms, especially non-Hodgkin lymphoma (NHL) information is obtained on target tissue anatomy.
[15]. • The radiation dose to the whole body can be
relatively high.
Indications for radioisotope imaging in head and • Images are not usually disease-specific.
neck region • Difficult to localize exact anatomical site of source
The following are the indications for radioisotope of emissions.
imaging in head and neck region [16]:
• In assessment of sites and extent of bone metastases
as in tumor staging. CONCLUSION
• Bone scan scintigraphy as it is sensitive and non-
invasive technique for demonstrating osteoblastic Imaging technologies have become increasingly
lesions of the skeletal system. sophisticated in recent years. Nuclear medicine and
• Investigation of salivary gland function especially molecular imaging, which provides the only means of
in Sjögren’s syndrome (technetium-99m assessing physiologic changes that is a direct result
pertechnetate)-salivary gland scintigraphy. of biochemical alterations at cellular and molecular
• Investigation of thyroid gland. levels, and in combination with traditional anatomic
• Brain scans and assessment of breakdown of blood imaging such as computed tomography scan and
brain barrier. magnetic resonance imaging (MRI) scan, provide precise
• Growth assessment-evaluation of bone grafts localization of functional abnormalities. These imaging
• In growth pattern-assessment of continued growth techniques are based on the radiotracer method, and
in condylar hyperplasia allow the measurement of tissue function in vivo and
• Blood flow and blood pool examination into the provide an early marker of disease through measurement
specific tissue/organ. of biochemical change. Many elements which found on
earth exists in different atomic configurations used in
Advantages of radioisotope imaging medicine are referred to as radiopharmaceuticals which
• Target tissue function is investigated. are useful to get diagnostic and therapeutic information
• All similar target tissues can be imaged during one about those tissues.
investigation. For example, whole skeleton can be
imaged in one bone scan.
********* 8. Agarwal BS and Kedar Nath Ram Nath. Meerut (1996);

A report on Atomic Energy in India: A Perspective,
Author Contributions Government of India, DAE, September (2002); and
Kaplan I. Nuclear Physics, Narosa Publishing House,
Kotya Naik Maloth – Substantial contributions to
New Delhi (1992).
conception and design, Acquisition of data, Analysis 9. New reactor needed for medical imaging - why
and interpretation of data, Drafting the article, Revising cyclotrons cannot do the job. Australasian Science
it critically for important intellectual content, Final Magazine, May 1999 edition.
approval of the version to be published 10. Baum S and Bramlet R. Basic Nuclear Medicine
Nagalaxmi Velpula – Analysis and interpretation of data, (Appleton-Century-Crofts, New York, 1975).
Revising it critically for important intellectual content, 11. Griffeth LK. Use of PET/CT scanning in cancer
Final approval of the version to be published patients: technical and practical considerations Baylor
Sridevi Ugrappa – Analysis and interpretation of data, University Medical Center Proceedings 2005;18:321–
330
Revising it critically for important intellectual content,
12. Zaidi H and Montandon ML. The Clinical Role of
Final approval of the version to be published Fusion Imaging Using PET, CT, and MR Imaging.
Srikanth Kodangal – Analysis and interpretation of data, Magn Reson Imaging Clin N Am 2010;18(1):133-49.
Revising it critically for important intellectual content, 13. Flynn A et al.”Isotopes and delivery systems for
Final approval of the version to be published brachytherapy”. In Hoskin P, Coyle C. Radiotherapy
in practice: brachytherapy. New York: Oxford
Guarantor University Press 2005.
The corresponding author is the guarantor of submission. 14. Radioisotopes in Medicine. Nuclear Issues Briefing
Paper 26, May 2004. http://www.uic.com.au/nip26.
htm.
Conflict of Interest 15. Goldenberg DM. Targeted Therapy of Cancer with
Authors declare no conflict of interest. Radiolabeled Antibodies, The Journal Of Nuclear
Medicine 2002; 43(5):693-713.
Copyright 16. Matteson SR, Deahl ST et al. Advanced Imaging
© 2014 Kotya Naik Maloth et al. This article is distributed Methods. Crit Rev Oral Biol Med 1996; 7(4):346-295..
under the terms of Creative Commons Attribution
License which permits unrestricted use, distribution
and reproduction in any medium provided the original
author(s) and original publisher are properly credited.
Please see the copyright policy on the journal website for
more information.
REFERENCES
1. Hayter CJ. Radioactive Isotopes in Medicine: A
Review J. Clin. Path. 1960; 13:369-90.
2. Lilley J.S. Nuclear Physics- Principles and
Applications. John Wiley and Sons, Ltd, Singapore
(2002); Ananthakrishnan M, Seminar talk on
Applications of Radioisotopes and Radiation
Technology.
3. Bhubaneswar and Verma HC. Concepts of Physics –
Part 2, Bharati Bhawan Printers, Patna (1996).
4. Sahoo S. Production and Applications of Radioisotopes
Physics Education 2006; 3:1-11.
5. Becquerel H. Emission des radiations nouvelles
par l’uranium metallique. C R Acad Sci Paris. 1896;
122:1086.
6. Curie P, Curie M, Bémont G. Sur une nouvelle
substance fortement radio-active contenue dans la
pechblende. C R Acad Sci Gen. 1898; 127:1215–18.
7. Roy RR and Nigam BP. Nuclear Physics- Theory and
Experiment. John Wiley and Sons, Inc., New York
(1967); Manchanda VK, Technical talk on Radiation
Technology Applications and Indian Nuclear
Programme: Societal Benefits, in Aarohan 2K5 at NIT,
Durgapur; Beiser A, Concepts of Modern Physics,
McGraw-Hill Book Company, Singapore (1987).
About the Authors
Article citation: Maloth KN, Velpula N, Ugrappa S, Kodangal S. Radioisotopes: An overview. Int J Case Rep Images
2014;5(9):604–609.
Kotya Naik Maloth is Senior Lecturer in the Department of Oral Medicine and Radiology at Mamata
Dental College and Hospital, Khammam, Dr. NTR University of Health Sciences, Telangana, India.
He has published eight research papers in national and international academic journals. His research
interests include advance treatment modalities in oral cancer patients such as radiotherapy and radio
immunotherapy.
Nagalaxmi Velpula is Professor and Head of the Department of Oral Medicine and Radiology at
Sri Sai College of Dental Surgery, Vikarabad. Dr.NTR University of Health Sciences, Telangana, India.
She has published 30 research papers in national and international academic journals. Her research
interests include preventive measures and advance treatment modalities in oral cancer patients.
Sridevi Ugrappa is postgraduate student in the Department of Oral Medicine and Radiology at Sri Sai
College of Dental Surgery, Vikarabad, Dr. NTR University of Health Sciences, Telangana, India. She has
published six research papers in national and international academic journals. Her research interests
include recent diagnostic advances in maxilla-facial radiology.
Srikanth Kodangal is Associate Professor in the Department of Oral Medicine and Radiology at Sri
Sai College of Dental Surgery, Vikarabad, Dr. NTR University of Health Sciences, Telangana, India.
He has published 10 research papers in national and international academic journals. His research
interests include management and prevention of oral cancer.
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other devices other devices
Int J Case Rep Images 2014;5(9):610–613. Sahoo et al. 610
CASE series OPEN ACCESS
Laparoscopic plication and mesh repair for diastasis recti:

A case series
Manash Ranjan Sahoo, Kumar A. T.
Abstract Keywords: Diastasis recti, Laparoscopic plication,

Linea alba, Tissue separating mesh
Introduction: Diastasis recti is a disorder
defined as a separation of the rectus abdominis How to cite this article
muscle into right and left halves which
occurs principally in newborns and pregnant Sahoo MR, Kumar AT. Laparoscopic plication and
women. Reports on laparoscopic repair are mesh repair for diastasis recti: A case series. Int J
rare. Usually, the patients themselves request Case Rep Images 2014;5(9):610–613.
treatment. We here present a series of three
patients who underwent laparoscopic repair
doi:10.5348/ijcri-201460-CS-10046
for diastasis recti. Case Series: Three patients,
all female, presented with something bulging
out of the abdomen with no other symptoms.
All were multiparous. Laparoscopically, linea
alba was plicated in the midline after taking Introduction
simple horizontal sutures all along the defect
from xiphisternum to just below umbilical Diastasis recti is a disorder defined as a separation
region creating a neo-linea alba. Then a tissue of the rectus abdominis muscle into right and left halves
separating mesh was used to reinforce the which occurs principally in newborns and pregnant
plication either using tackers or transfascial women. Normally, the two sides of the muscle are joined
sutures. Postoperatively, there was significant at the linea alba at the body midline. There is no associated
decrease in abdominal girth and patients morbidity or mortality with this condition except for
complained of pain which decreased in 3–4 days cosmetic reasons. There are no current guidelines on the
and tightness in the abdomen which gradually treatment of diastasis recti. Divarication repair is not very
reduced with time after discharge. Conclusion: popular because of associated morbidity and cosmetically
Laparoscopic plication of linea alba and unacceptable results. More recently there are various
placement of prosthetic mesh is very promising, attempts by different surgeons to reduce the morbidity
safe operation for diastasis recti and could be and length of scar associated with conventional open
the future for treatment of the same. procedures [1]. Laparoscopic repair of diastasis recti has
seldom been described in literature [2].
Manash Ranjan Sahoo1, Kumar A.T.2
Affiliations: 1MS, Associate Professor, Department of
Surgery, S.C.B. Medical College, Cuttack, Odisha, India;
2
Post Graduate, Department of Surgery, S.C.B. Medical
CASE SERIES
College, Cuttack, Odisha, India.
Three patients, all female, presented with a bulging of
Corresponding Author: Manash Ranjan Sahoo, Mailing
Address: Orissa Nursing Home, Medical road, Ranihat,
the abdomen in the midline in two patients, and cosmetic
Cuttack, Odisha, India. Postal Code: 753007; Ph: disfigurement in another one, with uncomfortability and
+919937025779; Fax: 0671-2414034; E-mail: manash67@ no other symptoms. All were multiparous in the range of
gmail.com 35–45 years. There was no history of previous operation.
There was no history of chronic cough and ascites in any
patient. On examination in standing position midline
Received: 20 November 2012 bulge was seen. On supine position there was defect
Accepted: 23 February 2013 between both recti muscles. There were no surgical scar
Published: 01 September 2014 marks over the abdomen except for striae indicative of
previous pregnancies. Two females had three children and

another one had two children. Ultrasound of abdomen
did not reveal any hernias. Routine biochemical reports
were normal. Under general anesthesia through a three
port approach (Figure 1) laparoscopically, camera port
(11 mm) in epigastrium right to the falciform ligament,
two working port (6 mm) in right and left hypochondrium
on anterior axillary line, linea alba was plicated in the
midline after taking intracorporeal horizontal continuous
sutures using ethilon double loop sutures (Figure 2) 2–3 cm
on either side of midline through the separated rectus
sheath all along the defect from suprapubic area till 5–6 cm
above umbilicus tightened by reducing the intraperitoneal
pressure to 8 mmHg and with manual compression over
abdominal wall creating a neo-linea alba (Figure 3). Then
a tissue separating mesh (physiomesh, Ethicon make)
was used to reinforce the plication by placing over the
plicated length and fixing with both tackers in a double
crown fashion and transfascial sutures (Figure 4). After
desufflation, the reduced girth of abdomen created loose
skin folds (Figure 5). Adhesive compression bandage Figure 3: Completion of continuous suturing forming neo-linea
alba.
was given over the abdomen for about 72 hours and
abdominal binder for eight weeks. Postoperatively, there
Figure 4: Reinforcing the plication using tissue separating mesh

(physiomesh).
Figure 1: Three port approach for repair of diastasis recti.
Figure 2: Continuous suturing of the two separated rectus

Figure 5: Immediate postoperative photograph following
muscle with their sheath using ethilon double loop suture
surgery.
intracorporeally.
was decrease in abdominal girth of 12.4 ± 2.64 cm (range: diastasis recti and could be the future for treatment of the
10–15 cm) after six months when compared with baseline same.
measurement preoperatively, using measuring tape at
the level of umbilicus which was considered statistically *********
significant (p value <0.05). Patients complained of pain
in the immediate postoperative period which decreased in Author Contributions
3–4 days and tightness in the abdomen which gradually Manash Ranjan Sahoo – Conception and design,
reduced with time after discharge on fifth day. Follow- Acquisition of data, Analysis and interpretation of data,
up at first month, third month, sixth month, and one Drafting the article, Final approval of the version to be
year showed decreased in tightness in the abdomen and published
further decrease in girth of the abdomen. No patients have Kumar A. T. – Conception and design, Acquisition of
recurrence of abdominal bulging or bowel obstruction in data, Analysis and interpretation of data, Drafting the
the follow-up period. article, Critical revision of the article, Final approval of
the version to be published
DISCUSSION Guarantor
The corresponding author is the guarantor of submission.
Diastasis recti may appear as a ridge running down
the midline of the abdomen, anywhere from the xiphoid Conflict of Interest
process to the umbilicus. It becomes more prominent with Authors declare no conflict of interest.
straining and may disappear when the abdominal muscles
are relaxed. It is more common in multiparous women due Copyright
to repeated episodes of stretching. The condition must © 2014 Manash Ranjan Sahoo et al. This article is
be differentiated from an epigastric hernia or incisional distributed under the terms of Creative Commons
hernia, if the patient has had abdominal surgery. Hernias Attribution License which permits unrestricted use,
may be ruled out using ultrasound. distribution and reproduction in any medium provided
In some cases of adults, diastasis recti can be the original author(s) and original publisher are properly
corrected and/or mitigated by physiotherapy. A study credited. Please see the copyright policy on the journal
conducted at Columbia University Program in Physical website for more information.
Therapy established that the women utilizing the Tupler
Technique exercises had a smaller diastasis than the
control group who did not do these exercises. Controversy REFERENCES
still exists regarding operative repair for diastasis recti
[3, 4] and there are few studies which have assessed 1. Zukowski ML, Ash K, Spencer D, Malanoski M,
the effectiveness of surgical intervention. In extreme Moore G. Endoscopic intracorporeal abdominoplasty:
cases, diastasis recti is corrected during the cosmetic A review of 85 cases. Plast Reconstr Surg
surgery procedure known as a tummy tuck by creating a 1998;102(2):516–27.
2. Palanivelu C, Rangarajan M, Jategaonkar PA, Amar V,
plication or folding of the linea alba and suturing together.
Gokul KS, Srikanth B. Laparoscopic repair of diastasis
However, it is usually the patients themselves who recti using the ‘Venetian blinds’ technique of plication
request treatment. Many novel procedures have invaded with prosthetic reinforcement: A retrospective study.
all the specialties of surgery with the advent of endoscopy. Hernia 2009;13(3):287–92.
Reports on laparoscopic repair are still very rare. Open 3. Nahas FX, Augusto SM, Ghelfond C. Should
procedures for diastasis recti have many complications diastasis recti be corrected? Aesthetic Plast Surg
[5, 6] such as hematoma, seroma formation, flap 1997;21(4):285–9.
necrosis, hypertrophic scars, increased infection rate, 4. Lockwood T. Rectus muscle diastasis in males: Primary
contour abnormalities that may be permanent. However, indication for endoscopically assisted abdominoplasty.
Plast Reconstr Surg 1998;101(6):1685–91.
laparoscopic repair is cosmetically more acceptable
5. Chaouat M, Levan P, Lalanne B, Buisson T, Nicolau
without significant associated morbidities and are P, Mimoun M. Abdominal dermolipectomies:
promising future technique for repair of diastasis recti Early postoperative complications and long
[2]. We too were able to give cosmetically excellent repair term unfavourable results. Plast Reconstr Surg
with disappearance of diastasis and reduction in girth of 2000;106(7):1614–8.
the abdomen. 6. Vastine VL, Morgan RF, Williams GS, et al. Wound
complications of abdominoplasty in obese patients.
Ann Plast Surg 1999;42(1):34–9.
CONCLUSION
Laparoscopic plication of linea alba and placement
of prosthetic mesh is very promising, safe operation for

Int J Case Rep Images 2014;5(9):614–618. Asarian et al. 614
CASE REPORT OPEN ACCESS
Cutaneous metastatic breast adenocarcinoma twenty years

post-mastectomy: A lesson to learn
Armand Asarian, Olubunmi Esan, Philip Xiao, Segun Adeoye
Abstract Keywords: Cutaneous metastasis, Metastatic

breast cancer, Breast cancer with cutaneous
Introduction: Cutaneous metastasis complicating metastasis, Cutaneous metastatic breast ad-
solid organ malignancy is a common clinical entity, enocarcinoma
and the primary is often metastatic breast cancer.
Skin metastasis from breast cancer occurs in about How to cite this article
25% of breast cancer patients. Case Report: We
present a case report of a patient with a history Asarian A, Esan O, Xiao P, Adeoye S. Cutaneous
of modified left radical mastectomy and chemo- metastatic breast adenocarcinoma twenty years
therapy for breast cancer twenty years earlier, post-mastectomy: A lesson to learn. Int J Case Rep
who presented for surgical evaluation of multiple Images 2014;5(9):614–618.
painful skin lesions. Initial punch biopsy of skin
lesions reported negative for malignancy. Surgical
biopsy provided tissue which demonstrated a doi:10.5348/ijcri-2014108-CR-10419
rare histopathological morphology for cutaneous
metastatic breast adenocarcinoma. Conclusion:
This report makes the case for considering
surgical biopsy when conventional biopsy reports
Introduction
negative for malignancy, especially in the setting
of remote or recent cancer history. It also advises Cutaneous metastasis complicating solid organ
prolonged relapse surveillance, well beyond malignancy is a common clinical entity, and the primary is
the current protocol, as well as development of often metastatic breast cancer. Skin metastasis (local and
prognostic markers for identifying patients at risk distant) from breast cancer occurs in about 25% of breast
of long disease latency after primary treatment. cancer patients [1, 2]. There is an array of histopathological
morphologies of metastatic cutaneous cancer reflective
Armand Asarian1, Olubunmi Esan2, Philip Xiao3, Segun Adeoye4
of a wide range of potential primaries. The diversity
Affiliations: 1MD, Consultant Surgeon, Program Director,
is further compounded by the myriad of typologies of
General Surgery Residency Program, Department of
Surgery, The Brooklyn Hospital Center (TBHC), Brooklyn, the primary malignancy. A complete description of
NY, USA; 2MD, General Surgery Resident, Department of metastatic cutaneous lesions involves the identification
Surgery, The Brooklyn Hospital Center, Brooklyn, NY, USA; of the primary cancer as well as the categorization of
3
MD, Consultant Pathologist, Department of Pathology, The the histological subtype. An initial negative report from
Brooklyn Hospital Center, Brooklyn, NY, USA.; 4MD, MS, a conventional skin biopsy does not definitively exclude
Attending Physician, Hospital Unit, University of Pittsburgh the possibility of cutaneous metastasis, especially in the
Medical Center (UPMC), Horizon, Greenville, PA, USA. setting of a positive personal history of malignancy. The
Former Resident, The Brooklyn Hospital Center. explanations for false negative result include: inadequate
Corresponding Author: Segun Adeoye, MD, MS, Attending sampling, inappropriate handling, sub-optimal
Physician, Hospital Unit, University of Pittsburgh Medical
processing and erroneous reporting. Arguably, one of the
Center (UPMC), Horizon, Greenville, PA, USA, Former
most striking variables in metastatic disease is the length
Resident, The Brooklyn Hospital Center; Ph: +19174995846;
Email: adeoyesp@upmc.edu, deoye.segun@yahoo.com of clinical latency. Though most metastatic diseases are
identified within a relatively short-period (within 3 years)
of the primary cancer diagnosis, many are discovered
Received: 26 April 2014 more remotely (many years to decades).
Accepted: 03 June 2014
Int J Case Rep Images 2014;4(5(9):614–618 Asarian et al. 615
CASE REPORT prominent nucleoli in a background of desmoplastic

stroma (Figure 2). Immunohistochemical stain results
A 79-year-old female presented to the surgical showed that the tumor cells were positive for AE1/AE3,
outpatient clinic for evaluation of five distinctly painful CK7 and mammaglobin (Figure 3), focally positive for
skin lesions. The lesions appeared nine months earlier GCDFP-15, and negative for CK20, napsin A, thyroid
and had progressively increased in size. Initial punch transcription factor-1 (TTF1), p63, CA19.9 and CDX2.
biopsy done at the dermatology clinic did not show Negative staining for napsin A and TTF1 make lung
any evidence of malignancy. The patient’s history was primary less likely. Negative staining for CK20, CDX2
significant for recent development of cough, and dyspnea and positive staining for CK7 essentially exclude a
on exertion, associated with malaise and 20 lbs weight loss colorectal primary. Combined with the morphological
over a one-year period. Significant negatives included, features, the immunoprofile supported the diagnosis of
the absence of: fever, chest pain, gastrointestinal or metastatic adenocarcinoma with breast origin (Table 1).
neurological symptoms. Her medical history included Further study revealed that the tumor cells were positive
hypertension, dyslipidemia, diabetes mellitus (II), for estrogen and progesterone receptors, and negative
coronary artery disease and myocardial infarction for HER-2/Neu (Figure 4). The patient was discharged
status post-percutaneous coronary intervention. Family home postoperatively for outpatient management of
history was unremarkable. She had a modified left metastatic disease and is currently receiving treatment
radical mastectomy, and post-surgical chemotherapy as for metastatic disease.
well as tamoxifen therapy for left breast cancer twenty
years ago. She reported adherence with annual clinical
breast examination and mammographic surveillance DISCUSSION
schedules. The last mammogram of her right breast
lesion was reported as consistent with fibroadenoma The incidence of cutaneous metastasis from solid
with a BiRADs 2 rating, and demonstrated clinical and organ malignancy has been reported to range from 0.7–
radiological stability. The radiologist recommended 10% [1]. Though cutaneous metastasis occurs in only a
against further evaluation. An intravenous contrast-aided minority of patients diagnosed with breast cancer, it
chest tomography done earlier to evaluate respiratory
symptoms revealed multiple pulmonary lesions suspicious
for metastatic disease. On physical examination, she was
found to have five raised skin lesions: 2 cm lesions in the
right upper back and subclavicular area, 1 cm lesions over
the left shoulder and left chest wall; and 0.5 cm lesion
in the right supraclavicular area. The lesions were firm,
fixed and well circumscribed. Clinical breast examination
revealed a nodular lesion in the outer quadrant of the
right breast without associated skin changes or nipple
discharge. This lesion had been shown to be benign and
stable by mammogram evaluation. A scar noted on the
left side of the chest was consistent with her modified
radical mastectomy history.
The records on TNM classification of the primary
breast tumor, initial investigations and initial and
subsequent treatments of the breast malignancy were
not available as the hospital only kept records for up to
seven years after the medical encounter. More so, at that
time electronic medical record (EMR) system was non-
existent. Differentials diagnoses included: cutaneous
metastasis of breast carcinoma, cutaneous lymphoma
or sarcoidosis, cutaneous manifestation of mycobacteria
or fungi infection, allergic, hyperimmune or systemic
inflammatory disease. The multiplicity of lesions made
primary apocrine tumor very unlikely.
The patient underwent a successful excisional biopsy
of all the skin lesions. Microscopic examination revealed
sheets of poorly differentiated malignant cells under the
skin surface (Figure 1). Higher magnification revealed Figure 1: (A, B) Microscopic examination reveal there are
that the tumor was composed of sheets, nests and clusters of sheets of tumor cells infiltrating under the skin
cords or individual cells with pleomorphic nuclei and surface (H&E stain, x200).
Int J Case Rep Images 2014;5(9):614–618 Asarian et al. 616
Table 1: Summary of immunoprofile of biopsied sample

Results
Antibody Clone Description Results
AE1/AE3 AE1, AE3 Pancreatin cocktail, low or high molecular weight +ve
CK7 SP52 Carcinoma subset (non-gastrointestinal); 54kD Keratin +ve
CK20 SP33 Carcinoma subset (most-gastrointestinal, others); 46kD Keratin -ve
CEA polyclonal Polyclonal Carcinoembryonic antigen, adenocarcinoma +ve
p63 BCA4A4 Nuclear transcription factor, basal and myoepithelial -ve
GCDFP-15 EP1582Y Gross cystic fluid disease protein, breast tumor, others +ve (focal)
Mammaglobin 31A5 Breast terminal duct and lobular epithelium +ve
ER SP-1 Estrogen receptor +ve
PR 1E2 Progesterone receptor +ve
CDX2 EPR27G4Y Carcinoma subset; intestinal-type tumors -ve
TTF-1 8G7G3/1 Thyroid transcription factor-1; Thyroid and lung tumors -ve
Napsin A Polyclonal Type II pneumocytes, Pulmonary adenocarcinoma -ve
CA19.9 121SLE Gastrointestinal, ovarian and lung tumors +ve (focal)
occurs at a greater rate than with any other solid organ

malignancy. In women, the incidence of cutaneous
metastasis from breast cancer is second only to malignant
melanoma. 23% of patients diagnosed with breast cancer
have cutaneous manifestation, often in the form of skin
nodules [3]. The relatively high incidence of breast cancer
in the general population extrapolates to potentially
high metastatic events in dermatology practices. This
is even more of a truism in patients with a recent or
remote diagnosis of breast cancer [2]. The presentation Figure 2: Higher magnification revealed that tumor is composed
of metastatic cutaneous disease from metastatic breast of sheets, cords or individual cells with pleomorphic nuclei and
cancer has been reported as nodular (80%), telangiectatic prominent nucleoli in a background of desmoplastic stroma
(11%), en cuirasse (3%), alopecia neoplastica (2%), or (H&E stain, x200).
zosteriform type (0.8%) [1].
Accurate assessment of the lesions as metastatic can
be difficult because the lesions are often ambiguous, and
indistinguishable from the more common benign process,
such as cellulitis and lymphedema [4]. As exemplified
in our case, there may be long latency between the
diagnosis and treatment of the primary malignancy and
the identification of cutaneous metastasis [5]. Rarely,
the metastatic cutaneous lesions may be the only sinister
sign indicative of an underlying primary malignancy or
its recurrence [6]. Unfortunately, it often portends a poor Figure 3: Immunohistochemical stain results showing that
prognosis [4]. Though death usually occurs within six tumor cells are positive for mammaglobin (Immunoperoxidase,
months of diagnosis of the metastatic cutaneous disease, x200).
long-term survival (in years) with appropriate and timely
treatment has been reported. In this case, though tissue
interventions. The causes of false negative punch biopsy
biopsy was not pursued for chest tomography-identified
are enumerated above. We cannot with certainty pinpoint
metastatic pulmonary disease, and absent overt evidence
the reason for the false negative report in this case,
of another primary cancer, and an immunoprofile
though we lean towards inadequate tissue sampling. The
supportive of a breast origin, we found no reason to
experience leads us to suggest lowering the threshold
question our impression of a breast primary.
for recommending surgical biopsies when initial punch
The case we present is unique in two ways. Firstly,
biopsies report negative for malignant disease in patients
it emphasizes the potential fallibility of conventional
with recent or remote history of cancer. Furthermore,
dermatological biopsies (often punch biopsies), with
a recent history of constitutional symptoms (weight
the inherent risks of false reassurance and delayed
loss, as reported by our patient) should be sinister for
Int J Case Rep Images 2014;4(5(9):614–618 Asarian et al. 617
to prognosis should justify a confirmatory excisional

biopsy in the setting of recent or remote history of
cancer. Beyond circumventing the problem of a falsely
negative, falsely reassuring punch biopsy, surgical
(excisional) biopsy provides a more representative
sample for the complete elucidation of the morphology
of metastatic cutaneous disease; thus informing
the staging process, and directing management and
prognostic considerations.
Figure 4: Immunohistochemical stain results showed that tumor *********

cells are positive for estrogen receptor (Immunoperoxidase,
x200). Author Contributions
Armand Asarian – Substantial contributions to
conception and design, Acquisition of data, Analysis
the non-benign nature of such cutaneous lesions. The and interpretation of data, Drafting the article, Revising
patient’s chest computed tomography scan findings were it critically for important intellectual content, Final
suggestive of metastatic lung disease. It should have approval of the version to be published
necessitated complete elucidation of the nature of skin Olubunmi Esan – Analysis and interpretation of data,
lesions by excisional biopsy. Secondly, and perhaps more Revising it critically for important intellectual content,
importantly, the presentation of cutaneous metastasis Final approval of the version to be published
may be remote in temporality from the initial diagnosis Philip Xiao – Analysis and interpretation of data,
of the primary neoplasm. Latent metastases resulting in Revising it critically for important intellectual content,
late-onset relapses occur frequently in breast and bone Final approval of the version to be published
cancer [3]. The majority (two-thirds) occurring within Segun Adeoye – Analysis and interpretation of data,
three years of the diagnosis of breast cancer, but cases Revising it critically for important intellectual content,
presenting decades later have been reported [7–9]. In our Final approval of the version to be published
case, there was a 20-year latency.
The disseminated cells presenting as secondaries Guarantor
often progress and evolve independently of the primary The corresponding author is the guarantor of submission.
tumor, they may develop mutations over time, making
them more genetically and immunologically different Conflict of Interest
from the primary breast cancer. This observation informs Authors declare no conflict of interest.
the need for comprehensive genetic testing and cellular
targeting when selecting adjuvant chemotherapy, Copyright
radiotherapy or immunotherapy for latent metastatic © 2014 Armand Asarian et al. This article is distributed
disease [9]. Furthermore, it makes the case for extending under the terms of Creative Commons Attribution
the duration of surveillance for relapses well beyond the License which permits unrestricted use, distribution
current protocol. For example, the use of anastrozole and reproduction in any medium provided the original
(arimidex) and other adjuvant hormonal therapies are author(s) and original publisher are properly credited.
promoted for up to five years after definitive treatment Please see the copyright policy on the journal website for
of the breast cancer, a recommendation partially more information.
informed by the fact that most latent relapses occurs
within five years of the primary treatment. Perhaps,
REFERENCES
the development of prognostic markers associated with
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About the Authors
Article citation: Asarian A, Esan O, Xiao P, Adeoye S. Cutaneous metastatic breast adenocarcinoma twenty years
post-mastectomy: A lesson to learn. Int J Case Rep Images 2014;5(9):614–618.
Armand Asarian is a Colorectal Surgeon at Brooklyn Hospital Center. He is the Vice Chair of the
Department of Surgery, and doubles as Director of its General Surgery Residency Program. Dr. Asarian
is credited with a number of academic publications. Email: apa9001@nyp.org
Olubunmi Esan is the Chief Resident in the Department of General Surgery at Brooklyn Hospitalist
Center. He earned MB,BS degrees from the University of Lagos, Nigeria. He has interests in general and
vascular surgery and intends to complete Fellowship training in vascular surgery. Dr. Esan is credited with
a number of academic publications. Email: ooe9001@nyp.org
Philip Xiao is the Chair of the Department of Pathology at Brooklyn Hospital Center. Dr. Xiao is credited
with a number of academic publications. Email: pqx9001@nyp.org
Segun Adeoye is a Hospitalist at University of Pittsburgh Medical Centers in Greenville and Shenango Valley,
Pennsylvania, USA. Heearned MBBS degrees from the College of Medicine of University of Lagos, Nigeria. He
has published 11 research papers and peer-reviewed articles in national and international academic journals
and authored a book titled “Synopsis of Medical Biochemistry- A MediLag Experience”. His research interests
include primary care, preventive medicine, hospital medicine, complementary and integrative medicine. He is
currently a Fellow at the University of Arizona Complimentary and Integrative Medicine Program. He intends to
pursue a PhD in bio-informatics. Email: Adeoye.segun@yahoo.com; adeoyesp@upmc.edu

Int J Case Rep Images 2014;5(9):619–624. Brunschot et al. 619
Aortoduodenal fistula after transperitoneal repair of an

inflammatory abdominal aortic aneurysm: A case report
Denise MD Özdemir-van Brunschot, Giel G Koning, J Adam van der Vliet
Abstract How to cite this article

Introduction: An aortoduodenal fistula Özdemir- van Brunschot DD, Koning GG, vd Vliet JA.
is a potentially lethal complication after Aortoduodenal fistula after transperitoneal repair of
transperitoneal open repair of an abdominal an inflammatory abdominal aortic aneurysm: A case
aortic aneurysm. Case Report: A 77-year-old report. Int J Case Rep Images 2014;5(9):619–624.
Caucasian male who underwent a conventional
repair of an inflammatory infrarenal aortic
aneurysm, was readmitted with hematemesis doi:10.5348/ijcri-2014109-CR-10420
only six weeks after surgery. Gastroscopy
and computed tomography angiography
indicated an aortoduodenal fistula and urgent
aortic reconstruction was performed. An
aortoduodenal fistula is a potentially lethal Introduction
complication after transperitoneal open repair
of an abdominal aortic aneurysm. Conclusion: An aortoduodenal fistula was first described by Sir
An aortoduodenal fistula seldom occurs as Astley Cooper in 1829 and is defined as an abnormal
early after conventional transperitoneal open communication between the aorta and duodenum [1].
aneurysm repair as in our case. The early Communications of the aorta with other sites of the
occurrence of the aortoduodenal fistula can be gastrointestinal tract, such as jejunum, stomach and
explained by the inflammatory character of the sigmoid are possible, but are seen less often [2].
aneurysm.
Keywords: Aortoenteric, Aortoduodenal fistu- CASE REPORT

la, Conventional abdominal aortic aneurysm
repair, Abdominal, Aneurysm, Inflammatory, A 77-year-old Caucasian male patient was admitted to
Nevelsteen procedure the emergency department because of general discomfort,
nausea and dark stained emesis. Medical history of the
patient included diabetes mellitus type 2, a peritonitis
after appendicitis and peripheral artery occlusive disease
Denise MD Özdemir-van Brunschot1, Giel G Koning1, J (PAOD) stage 2a. Six weeks earlier, he underwent a
Adam van der Vliet1 conventional transperitoneal repair of an inflammatory
Affiliations: 1Department of Surgery, Division of Vascular and infrarenal aortic aneurysm measuring 7.6 cm in diameter.
Transplant Surgery, Radboud University Nijmegen Medical It was a difficult procedure because of dense adhesions
Centre, Nijmegen, The Netherlands. between the duodenum and the aortic aneurysm wall,
Corresponding Author: Denise MD Özdemir- van Brunschot, leading to a small serosal injury which was sutured
Department of Surgery, Division of Vascular- and Transplant peroperatively. Six days after surgery the patient was
Surgery, Radboudumc Geert Grooteplein-Zuid 10, 6525 discharged after an uneventful postoperative recovery.
GA Nijmegen, The Netherlands; Email: denise.ozdemir-
At readmission, the vital signs were stable and
vanbrunschot@radboudumc.nl
hemoglobin level was 6.1 g/dL. Shortly thereafter, the
patient produced hematemesis. A gastroscopy was
Received: 25 April 2014 performed subsequently. This examination revealed a
Accepted: 24 May 2014 vulnerable, edematous mucous membrane with a trace
Published: 01 September 2014 of blood in the duodenum. A computed tomography
angiography (CTA) was performed (Figure 1) because of

the high suspicion of an aortoduodenal fistula. Although
no clear contrast extravasation in the duodenum was
seen on this scan, an aortoduodenal fistula was presumed
because of the very close proximity of the duodenum to
the aorta and the air bubbles in and around the aneurysm
sac and the signs of local bowel wall thickening.
An urgent aortic reconstruction was performed with a
graft constructed from the right superficial femoral vein
according to Nevelsteen (Figure 2) [3]. Intraoperatively,
no signs of a duodenal perforation were detected.
An additional femoro-femoral crossover bypass was
constructed using a Dacron prosthesis (8 mm Gelsoft)
because of absent pulsations in the left leg.
Perioperative cultures of the removed aortic graft
turned out positive for Streptococcus anginosus and
Haemolytic streptococcus group B.
The fourth day after surgery, a sudden resuscitation
setting occurred and hemoglobin level declined to 3.5 g/ Figure 1: Computed tomography angiography (arterial phase)
showing gas bubbles (arrow) in the aneurysm sac and a very
dL. An emergency CTA was done, which showed no active
close (adhesive) relation with duodenum.
blush. Despite all measures to resuscitate his condition
worsened. Aortic stump blow-out was considered, but
since no active blush was seen and it was considered
unlikely. After careful discussion and consideration
among all treating physicians and his family, it was agreed
not to initiate any further surgical nor endovascular
intervention and the patient deceased shortly afterwards.
Permission for a postmortem examination was declined
by the relatives.
DISCUSSION
Aortoduodenal fistulas are difficult to diagnose
because patients often have non-specific complaints
such as general discomfort, weakness and weight loss
and imaging is seldom clear [2, 4]. Classical signs are
gastrointestinal bleeding, a pulsatile abdominal mass and
abdominal pain. Gastrointestinal bleeding can present
as melena (up to 50%) and/or hematemesis (up to two-
thirds). Typically, this bleeding is extensive but may be
preceded by intermittent bleeding or herald bleeding. The
sensitivity and specificity of computed tomography ranges
from 40–90% and from 33–100%, respectively [4]. Signs
include perigraft gas or fluid, soft tissue inflammation
with edema, loss of continuous wrap of tissue around
the graft and bowel wall thickening. Characteristics of
other modalities, such as magnetic resonance imaging/
angiography (MRI/MRA), have not yet been sufficiently
Figure 2: Intraoperative view autologous venous aortic
evaluated [4]. reconstruction (Nevelsteen procedure).
A secondary aortoduodenal fistula is fatal without
surgical intervention [3]. Treatment should consist of
resection of the aortoduodenal fistula and preferably also
the aortic graft. Revascularization can be undertaken
in a variety of ways, including primary aortic repair, significant complications including lower extremity
aortic replacement with a new prosthetic or venous amputations, aortic stump blow out and mortality [3, 5].
graft or an extra-anatomical bypass (for example, axillo- Like conventional aortic repair, recently more reports are
bifemoral bypass) [4]. Each procedure is associated with published of patients treated with various percutaneous
endovascular techniques [6, 7]. Also, injection of embolic or enteric fistula is given [9–25]. Chang et al. described a
material in the fistula followed by endovascular stent patient in 2002 who presented 20 days after emergency
graft has been described [8]. surgery because of a ruptured abdominal aneurysm
Despite the advancements in treatment of with an aortoduodenal fistula [16]. The perioperative
aortoduodenal fistulas and state-of-the-art intensive cultures were positive for Klebsiella pneumoniae. Tromp
care, the prognosis remains poor, with an overall 30-days et al. also described a patient presenting with an early
survival of 30–44% [4, 5]. aortoduodenal fistula six weeks after an endovascular
Due to the meticulous coverage of the aortic prosthesis aneurysm repair was performed [23].
after implantation by closing the aneurysm sac and closing In this patient, the early occurrence of an
the retroperitoneum, an aortoduodenal fistula usually aortoduodenal fistula may be explained by the
presents as a late complication after transperitoneal open perioperative duodenal serosal injury or by the
aneurysm repair with an incidence of 0.4–2.4%, mostly inflammatory character of the aneurysm. On the
3–5 years after surgery [4]. Mechanisms of secondary preoperative computed tomography scan, it was not
fistulas include direct mechanical erosion of the suture expected that an inflammatory aortic aneurysm was
line into the bowel, proximal suture line disruption with present in the patient. Best on based available evidence,
pseudoaneurysm formation and fistulization, transient if an inflammatory aneurysm had been expected, an open
bacteremia and graft infection from perioperative retroperitoneal approach may have been considered
contamination [4]. [26, 27]. A search of literature according to the Patient
The patient in this case was diagnosed with an Intervention Comparison Outcome (PICO) strategy and
aortoduodenal fistula only six weeks after surgery. An critical appraisal method in line with Guyatt [28] and the
aortoduodenal fistula seldom occurs this early after Cochrane Handbook for systematic reviews [29], showed
transperitoneal open aneurysm repair. In Table 1, the that repair of an inflammatory aneurysm may have a
postoperative time of presentation of an aortoduodenal higher risk of developing an aortoduodenal fistula [16, 30].
Table 1: Reports of secondary aortoduodenal fistulas since 1998 and the time of presentation after abdominal aortic aneurysm repair
Author Year of Number of Time of Fistula after Initial Type of Procedure

Publication Patients Procedure
Yabu [9] 1998 1 10 years Conventional repair
Constans [10] 1999 7 4-7 years Conventional repair
Hauseggers [11] 1999 1 20 months Endovascular repair
Makar [12] 2000 1 4 months Endovascular repair
Lau, [13] 2001 1 5 years Conventional repair
Ohki, [14] 2001 1 9 months Endovascular repair
Parry, [15] 2001 1 12 months Endovascular repair
Chang, [16] 2002 1 20 days Conventional repair
Kar, [17] 2002 1 22 months Endovascular repair
Berges, [18] 2003 1 2 years Endovascular repair
Elkouri, [19] 2003 1 17 months Endovascular repair
Probst, [20] 2006 1 8 months Conventional repair
Geraci [21] 2008 1 5 years Conventional repair
Tanaka [22] 2009 1 13 years Conventional repair
Tromp [23] 2009 1 6 weeks Endovascular repair
Billi [24] 2012 1 3 years Conventional repair
Perencevich [25] 2013 1 3 years Conventional repair
CONCLUSION Infection after Reconstructive Surgery for Aortoiliac

Disease. J Vasc Surg 1995;22(2):129–34.
In summary, an aortoduodenal fistula is a devastating 4. Mathias J, Mathias E, Jausset F, et al. Aorto-
enteric fistulas: A physiopathological approach
condition, associated with high morbidity and mortality,
and computed tomography diagnosis. Diagn Interv
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only six weeks after conventional aneurysm repair. The Lastdrager WB. Primary aortoenteric fistula: Report
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explained by the inflammatory character of the abdominal 6. Milnerowicz A, Milnerowicz A, Pawlowskii S, Skora
aortic aneurysm. J, Pupka A. The Treatment of Aortoduodenal Fistula
with the Use of the Endovascular Prosthesis. Polim
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7. Palma JH, Buffolo E, Gaia D. Endovascular
treatment for aorta diseases: Overview. Rev Bras
Author Contributions Cir Cardiovasc 2009;24(2 Suppl):40s–4s. [Article in
Denise MD Özdemir-van Brunschot – Substantial Portuguese].
contributions to conception and design, Acquisition 8. Busuttil RW, Rees W, Baker JD, Wilson SE.
of data, Drafting the article, Revising it critically for Pathogenesis of aortoduodenal fistula: Experimental
important intellectual content, Final approval of the and clinical correlates. Surgery 1979;85(1):1–13.
version to be published 9. Yabu M, Himeno S, Kanayama Y, et al. Secondary
Giel G Koning – Substantial contributions to conception Aortouodenal Fistula Complicating Aortic Grafting
and design, Drafting the article, Revising it critically as a Cause of Intermittent Chronic Intestinal
Bleeding. Intern Med 1998;37(1):47–50.
for important intellectual content, Final approval of the
10. Constans J, Midy D, Baste JC, Demortière F, Conri
version to be published C. Secondary Aortoduodenal Fistulas: Report of 7
J Adam van der Vliet – Substantial contributions to cases. Rev Med Interne 1999;20(2):121–7. [Article in
conception and design, Revising it critically for important French].
intellectual content, Final approval of the version to be 11. Hausegger KA, Tiesenhausen K, Karaic R, Tauss J,
published Koch G. Aortoduodenal Fistula: A Late Complication
of Intraluminal Exclusion of an Infrarenal Aortic
Guarantor Aneurysm. J Vasc Interv Radiol 1999;10(6):747–50.
The corresponding author is the guarantor of submission. 12. Makar R, Reid J, Pherwani AD, et al. Aorto-
enteric Fistula Following Endovascular Repair of
Abdominal Aortic Aneurysm. Eur J Vasc Endovasc
Conflict of Interest Surg 2000;20(6):588–90.
Authors declare no conflict of interest. 13. Lau H, Chew DK, Gembarowicz RM, Makrauer FL,
Conte MS. Secondary aortoduodenal fistula. Surgery
Copyright 2001;130(3):526–7.
© 2014 Denise MD Özdemir-van Brunschot et al. This 14. Ohki T, Veith FJ, Shaw P, et al. The Increasing
article is distributed under the terms of Creative Commons Incidence of Mid-term and Long-term Complications
Attribution License which permits unrestricted use, after Endovascular Graft Repair of Abdominal Aortic
Aneurysms: A Note of Caution based on 9 Years’
distribution and reproduction in any medium provided
Experience. Ann Surg 2001;234(3):323–34.
the original author(s) and original publisher are properly 15. Parry DJ, Waterworth A, Kessel D, Robertson I,
credited. Please see the copyright policy on the journal Berridge DC, Scott DJ. Endovascular Repair of
website for more information. an Inflammatory Abdominal Aortic Aneurysm
complicated by AortoduodenalFistulation with an
Unusual Presentation. J Vasc Surg 2001;33(4):874–
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About the Authors
Article citation: Özdemir- van Brunschot DD, Koning GG, vd Vliet JA. Aortoduodenal fistula after transperitoneal
repair of an inflammatory abdominal aortic aneurysm: A case report. Int J Case Rep Images 2014;5(9):619–624.
D.M.D. Özdemir-van Brunschot is PhD Student at the Department of Surgery, Radboudumc,

Nijmegen, The Netherlands.
G.G. Koning is Vascular Surgeon at the Department of Surgery, Radboudumc, Nijmegen, The
Netherlands.
J. Adam van der Vliet is Vascular Surgeon at the Department of Surgery, Radboudumc, Nijmegen,
The Netherlands.

Int J Case Rep Images 2014;5(9):625–628. Bulut 625
Unusually large sialolith of submandibular gland

Haci Taner Bulut
Abstract diagnostic strategy for treatment and follow-up

studies in sialolithiasis.
Introduction: Pathologically sialolithiasis is
a disease which results in the obstruction of a Keywords: Sialolithiasis, Large sialolith,
salivary gland by a sialolith. Sialolith is generally Submandibular gland, Imaging
seen in small size and their sizes range from 1
mm to 1 cm. Large salivary gland calculi are How to cite this article
infrequent and defined as the size of 1.5 cm or
larger. Only a few cases of large sialolith of the Bulut HT. Unusually large sialolith of submandibular
submandibular glands have been reported in gland. Int J Case Rep Images 2014;5(9):625–628.
literature. Imaging methods have an important
role in making a diagnosis and in planning further
management, operative or otherwise. Case doi:10.5348/ijcri-2014110-CR-10421
Report: A 52-year-old male patient with multiple
stones in the submandibular gland admitted to
our hospital with swelling at submandibular
region. Plain films show large stones at the
region of submandibular gland. The maximum
Introduction
stone size was 1.8x0.8 mm. Submandibular
Sialolithiasis accounts for the most common cause
gland size increased due to the sialoadenitis
of diseases of salivary glands [1–7]. Pathologically,
caused by stones. After surgery, the patient had
sialolithiasis is a disease results in the obstruction of a
a nearly normal function of the glands for three
salivary gland by a sialolith [8, 9]. The clinical symptoms
months. Conclusion: The swelling which is seen
of sialolithiasis are pain and swelling due to enlargement
in the submandibular region most commonly
of involved gland [1–3, 7, 9]. Thoese symptoms help
originates from sialolithiasis of submandibular
clinicians to diagnose easily. Nonetheless, pain is not
gland, so it should be carefully evaluated by
seen in all the cases [2, 10]. About 80% of all reported
clinicians. Diagnostic imaging methods may
cases of sialolith occur in the submandibular gland [1–
complement each other in examining glands
3, 6, 9]. The sublingual and minor salivary glands are
with sialolithiasis and may offer a promising
seldom involved [1–5, 7, 9]. Sialolithiasis is generally
found between 30 and 60 years of age and it has a
frequent prevalence in male patients [9, 11]. Sialoliths
Haci Taner Bulut are generally seen in small size and their sizes range
Affiliations: Department of Radiology, Medical Faculty of from 1 mm to 1 cm. Large salivary gland sialolith which
Adıyaman University, Adıyaman, Turkey. is large 15 mm are considered rare [7, 9]. Only a few cases
Corresponding Author: Haci Taner Bulut, MD, Department of large sialolith of the submandibular or parotid glands
of Radiology, Medical Faculty of Adıyaman University, have been reported in literature. Imaging methods have
Adıyaman, Turkey, Tel: +90 530 207 15 17, Fax: +90 416 an important role in making a diagnosis and in planning
227 08 63; Email: taner.bulut02@gmail.com management. Plain radiography and sialography,
magnetic resonance imaging (MRI) scan, computed
tomography (CT) scan, and ultrasound all have a role
Received: 05 June 2014
[12]. The aim of this study is to evaluate the clinical and
radiographical findings of the patient with large sialolith
of submandibular gland.
CASE REPORT
A 52-year-old male patient admitted to our hospital
with painless swelling at submandibular region. Extra-
oral examination revealed swelling and palpable mass.
In intraoral examination, bimanual palpation revealed
a hard elongated mass and multiple stones in a large
size. Plain film, axial CT, and MRI scans were obtained
for radiological examination. Stone location, shape, and
size were estimated on plain film, MRI and CT scans.
Posterior-anterior and sagittal plain films show large
calculi at the region of submandibular gland (Figure 1). Figure 3: Coronal short tau inversion recovery (STIR) (A)
Non-enhanced axial CT-scan showed large hyperdense Enhanced T1-weighted, (B) Magnetic resonance images of
masses (sialoliths), localized within the left Wharton duct affected gland showing higher signal intensity compared with
and enlargement of the affected submandibular gland. normal gland on right side.
The maximum stone size was 1.8x0.8 mm, as measured
directly on the axial CT scan (Figure 2). Submandibular
gland size increased due to the sialadenitis caused by
stones. Due to sialadenitis, coronal Short tau inversion DISCUSSION
recovery (STIR) and enhanced axial T1-weighted
magnetic resonance images of affected gland showed The most widespread illnesses of the salivary gland
higher signal intensity compared with normal gland on are sialoliths [1, 3, 9, 13]. Sialoliths are generally seen
right side (Figure 3). Sialoliths removed with surgery. in small size and their sizes range from 1 mm to 1 cm
After surgery, the patient had a nearly normal function of [1–3, 5, 9]. The mean size of sialoliths is reported as 6
the glands for three months. to 9 mm. They infrequently measure more than 1.5 cm.
Large salivary gland calculi are infrequent and defined as
the size of 1.5 cm or larger [2, 9–11]. Most of the studies
have conducted that the common symptoms of sialoliths
are recurrent pain and swelling of the associated gland,
because sialoliths generally does not block the flow of
saliva fully [1–3]. Nonetheless, large sialoliths have been
frequently reported in the body of salivary glands, they
have infrequently been described in the salivary ducts,
particularly without any complaints from the patients [2,
4, 14]. In this study, clinical and radiological features of
one case which have large sialoliths in the size of 1.8 cm
were presented. The sialoliths were located into Wharton
ducts and the patient complained painless swelling. Some
Figure 1: A 52-year-old male with left submandibular uncommon large salivary stones may be noticed unless
sialolithiasis. Posterior-anterior (A) Sagittal, (B) Plain films the patient has a long history, due to the fact that lesions
showing large stones at the region of submandibular gland. are usually asymptomatic. It is conducted that the stones
may expand in the proportion of about 1 to 1.5 mm each
year [2]. Hence, it is possible to presume that sialoliths of
our case began to develop many years ago.
In the diagnosis of sialoliths, history and careful
examination come to the fore. Pain and swelling of
involved gland at the time of meal are of great importance.
Bimanual examination in the floor of the mouth may
show a palpable stone in a great number of cases of
submandibular sialoliths. Bimanual palpation of the
gland is very useful because a uniformly solid and hard
gland indicates a hypo-functional or non-functional gland
[1, 2, 8]. A case in the this study has a history of painless
swelling in the floor of the mouth at mealtimes. Extra-
oral examination revealed swelling and palpable mass.
Figure 2: (A, B) Non-enhanced axial computed tomography
scan showing sialoliths and enlargement of the affected In intraoral examination, bimanual palpation revealed a
submandibular gland. hard elongated mass and multiple stones in a large size.
In the diagnoses of sialolithiasis, imaging methods presence of gland lithias. Large submandibular sialoliths
are very useful. Plain radiographs are useful in showing should be treated by appropriate approach to avoid
radiopaque stones. It is very uncommon for patients possible severe postoperative complications.
to have a combination of radiopaque and radiolucent
stones and 40% of parotid stones may be radiolucent
[15]. Sialography is thus useful in patients showing CONCLUSION
signs of sialadenitis related to radiolucent stones or
deep submandibular stones. Sialography is, however, The swelling which is seen in the submandibular
contraindicated in acute infection or in significant region most commonly originates from sialolithiasis of
patient contrast allergy. Nowadays, magnetic resonance submandibular gland, so it should be carefully evaluated by
sialography (MR sialography) imaging is recommended clinicians. Diagnostic imaging methods may complement
in diagnosis of sialoliths, but this method is not each other in examining glands with sialolithiasis and
appropriate to see the inner duct of the salivary glands. may offer a promising diagnostic strategy for treatment
Developed in the 1990’s as an endoscopic method, and follow-up studies in sialolithiasis.
sialoendoscopy technique enables clinicians to examine
the ductal system completely and it can be used not only *********
for diagnosis but also for treatment [16]. The CT scan
is useful in identifying small calculi within the salivary Author Contributions
gland or duct. It can also show localization and number of Haci Taner Bulut – Substantial contributions to
stones in the gland and measure size of stones. Contrast- conception and design, Acquisition of data, Analysis
enhanced CT scan has a potential to show enlargement of and interpretation of data, Drafting the article, Revising
gland due to sialadenitis. Features of the submandibular it critically for important intellectual content, Final
glands affected by sialolithiasis can well evaluate with approval of the version to be published
MRI scan [12]. Hence, it can possible to differentiate,
acute or chronic stage of sialadenitis with MRI scan. It Guarantor
can also show the location of stones and shapes of ducts. The corresponding author is the guarantor of submission.
In this context, MRI scan using T1-weighted and STIR
sequences, can provide effective information about the Conflict of Interest
pathologic status of the gland parenchyma affected Authors declare no conflict of interest.
by sialolithiasis [12]. Moreover, the extent, acute and
chronic nature of this obstruction may reflect by MRI Copyright
findings of the gland parenchyma [12]. In this study, the © 2014 Haci Taner Bulut. This article is distributed
sialoliths were observed clearly in plain radiographs, under the terms of Creative Commons Attribution
but estimation of size and location of stones is limited. License which permits unrestricted use, distribution
The MRI scan and CT scan were suitable in precise and reproduction in any medium provided the original
preoperative estimation of stone’s size and location. author(s) and original publisher are properly credited.
These results suggest that MRI features may reflect Please see the copyright policy on the journal website for
acute obstruction, and a combination of CT and MRI more information.
scans in examining glands with sialolithiasis may offer a
promising diagnostic strategy for treatment and follow-
up studies in sialolithiasis. REFERENCES
Different treatment options may be selected according
to the size and location of the sialolith. The treatment 1. Siddiqui SJ. Sialolithiasis: An unusually large
of choice of small sialolith should be medical instead of submandibular salivary stone. Br Dent J
surgical. However, if the stone is too large or located in 2002;193(2):89–1.
2. Alkurt MT, Peker I. Unusually large submandibular
the proximal of duct, piezoelectric extracorporal shock
sialoliths: Report of two cases. Eur J Dent
wave lithotripsy or surgical removal of the stone or gland 2009;3(2):135–9.
may be required [1–3]. Sialoendoscopy is a new way and 3. Iqbal A, Gupta AK, Natu SS, Gupta AK. Unusually
minimally invasive technique for treating obstructions large sialolith of Wharton’s duct. Ann Maxillofac
of the ductal system and can be used with operation Surg 2012;2(1):70–3.
in large salivary stones [17]. Recurrent or continuous 4. Leung AK, Choi MC, Wagner GA. Multiple sialoliths
obstruction of the salivary duct may lead to acute or and a sialolith of unusual size in the submandibular
chronic sialadenitis or even to the perforation of the oral duct: A case report. Oral Surg Oral Med Oral Pathol
mucosa [18]. In this case, the sialoliths were removed by Oral Radiol Endod 1999;87(3):331–3.
5. Parkar MI, Vora MM, Bhanushali DH. A Large
surgical excision. The clinicians should evaluate carefully
Sialolith Perforating the Wharton’s Duct: Review of
the painful or painless swellings in submandibular area. Literature and a Case Report. J Maxillofac Oral Surg
This condition seems to be the most common disease 2012;11(4):477–82.
in submandibular gland and Wharton duct due to the
6. Singhal A, Singhal P, Ram R, Gupta R. Self-exfoliation 13. Akin I, Esmer N. A submandibular sialolith of unusual
of large submandibular stone-report of two cases. size: A case report. J Otolaryngol 1991;20(2):123–5.
Contemp Clin Dent 2012;3(Suppl 2):S185–7. 14. Graziani F, Vano M, Cei S, Tartaro G, Mario
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submandibular gland duct: Report of two cases with submandibular gland: A clinical report. J Craniofac
unusual shape. Contemp Clin Dent 2013;4(1):78– Surg 2006;17(3):549–2.
80. 15. Isacsson G, Isberg A, Haverling M, Lundquist
8. Ledesma-Montes C, Garcés-Ortíz M, Salcido-García PG. Salivary calculi and chronic sialoadenitis of
JF, Hernández-Flores F, Hernández-Guerrero the submandibular gland: A radiographic and
JC. Giant sialolith: Case report and review of the histologic study. Oral Surg Oral Med Oral Pathol
literature. J Oral Maxillofac Surg 2007;65(1):128– 1984;58(5):622–7.
30. 16. Becker M, Marchal F, Becker CD, et al. Sialolithiasis
9. Oteri G, Procopio RM, Cicciu M. Giant Salivary and salivary ductal stenosis: Diagnostic accuracy of
Gland Calculi (GSGC): Report Of Two Cases. Open MR sialography with a three-dimensional extended-
Dent J 2011;5:90–5. phase conjugate-symmetry rapid spin-echo
10. Lustmann J, Regev E, Melamed Y. Sialolithiasis. A sequence. Radiology 2000;217(2):347–58.
survey on 245 patients and a review of the literature. 17. Yu CQ, Yang C, Zheng LY, Wu DM, Zhang J,
Int J Oral Maxillofac Surg 1990;19(3):135–8. Yun B. Selective management of obstructive
11. Bodner L. Giant salivary gland calculi: Diagnostic submandibular sialadenitis. Br J Oral Maxillofac
imaging and surgical management. Oral Surg Oral Surg 2008;46(1):46–9.
Med Oral Pathol Oral Radiol Endod 2002;94(3):320– 18. Sutay S, Erdag TK, Ikiz AO, Guneri EA. Large
3. submandibular gland calculus with perforation of
12. Sumi M, Izumi M, Yonetsu K, Nakamura T. The the floor of the mouth. Otolaryngol Head Neck Surg
MR imaging assessment of submandibular gland 2003;128(4):587–8.
sialoadenitis secondary to sialolithiasis: Correlation
with CT and histopathologic findings. AJNR Am J
Neuroradiol 1999;20(9):1737–43.
About the Author
Article citation: Bulut HT. Unusually large sialolith of submandibular gland. Int J Case Rep Images 2014;5(9):625–
628.
Haci Taner Bulut is Medical Faculty in Department of Radiology, Adiyaman University, Adiyaman,
Turkey. He has published many research papers in national and international academic journals. His
research interests include neuroradiology, MR imaging. He intends to pursue Postdoocs in future.
Email: taner.bulut02@gmail.com

Int J Case Rep Images 2014;5(9):629–633. Gupta et al. 629
Malignant mucosal melanoma of the nasal cavity:

A case report
Devika Gupta, Niti Goyal, Vandana Rana, Rajat Jagani, Davendra Swarup
Abstract wide local excision of nasal mass confirmed the

diagnosis. Conclusion: Malignant melanomas
Introduction: Mucosal melanoma of the can mimic a large number of malignant diseases
nasal cavity is a rare tumor. It is seen more and early diagnosis by astute pathologist can help
commonly in the elderly and is known to achieve attain long time remission.
have a male preponderance. Patients often
present with non-specific symptoms of nasal Keywords: Malignant diseases, Melanoma,
obstruction or epistaxis. These tumors carry Nasal cavity, Nasal mass, Sinonasal tract,
a poor prognosis because of higher rates of HMG-45
locoregional recurrence and distant metastasis.
Case Report: We report a 54-year-old male How to cite this article
who presented with submandibular swelling
and history of episodes of occasional epistaxis. Gupta D, Goyal N, Rana V, Jagani R, Swarup D.
Microscopic examination of the excision biopsy Malignant mucosal melanoma of the nasal cavity: A
of the submandibular lymph node supported with case report. Int J Case Rep Images 2014;5(9):629–
immunohistochemistry (IHC) was suggestive 633.
of metastatic deposit of malignant melanoma.
Clinical examination and radiological imaging
doi:10.5348/ijcri-2014111-CR-10422
for the primary tumor lead to detection of a
mass in the right nasal cavity. Histopathology of
Devika Gupta1, Niti Goyal2, Vandana Rana3, Rajat Jagani4,

Davendra Swarup5
Affiliations: 1MD, DNB Pathology, Assistant Professor,
Introduction
Armed Forces Medical College, Department of Pathology
and Laboratory Science, Command Hospital, Pune, India; Melanomas are tumors arising from melanocytes
2
Resident, MD, Pathology, Armed Forces Medical College, which are neural crest derived cells present in the basal
Department of Pathology and Laboratory Science, Command layer of skin, hair follicles, most squamous-covered
Hospital, Pune, India; 3MD, Pathology, Assistant Professor, mucosal membranes, leptomeninges and several other
Armed Forces Medical College, Department of Pathology sites [1].
and Laboratory Science, Command Hospital, Pune, India; Mucosal melanomas of the nasal cavity, paranasal
4
MD, Pathology, Associate Professor, Armed Forces Medical sinuses and nasopharynx (collectively called sinonasal
College, Department of Pathology and Laboratory Science, tract) are extremely rare. They constitute about 1% of all
Command Hospital, Pune, India; 5Professor, Armed Forces
melanomas. Patients have non-specific symptoms which
Medical College, Department of Pathology and Laboratory
Science, Command Hospital, Pune, India. is often the cause of delay in diagnosis. The cases are
Corresponding Author: Devika Gupta, MD, DNB, Pathology,
usually detected when there is extensive local invasion
Assistant Professor, Armed Forces Medical College, or distant metastasis. Majority of the tumors are melanin
Department of Pathology and Laboratory Science Command rich, however, some have scarcity of pigment and hence
Hospital, Pune - 411040, India; Ph: 9158984335; Email: called non-pigmented amelanotic tumors. It is often
devikalives5h@gmail.com difficult to diagnose pigment deficient melanomas as
these can morphologically resemble variety of other
malignant neoplasms. Immunohistochemistry is useful
Received: 23 April 2014 in confirming such cases. Treatment is wide local excision
Accepted: 24 May 2014 with radiotherapy and/or chemotherapy. Prognosis is
generally poor and unpredictable.
We are discussing this aggressive tumor because of its The patient was subsequently referred to the
rarity and to emphasize the importance of early detection radiotherapy department for further management.
of this lesion and high index of suspicion required when Presently, one year after the diagnosis the patient is
it presents with epistaxis and on histological examination disease free and is on regular follow-up at our hospital.
is found to be amelanotic.
DISCUSSION
CASE REPORT
Malignant melanomas originate from neural crest
A 48-year-old male, tobacco chewer, presented derived melanocytes present in the basal layer of skin,
with swelling in right angle of mandible of six months hair follicles and most squamous covered mucosal
duration. The patient related it to dental caries for membranes, leptomeminges and several other sites. Most
which he took dental consultation. The patient, however, of the melanomas arise in the sun exposed areas, i.e.,
noticed a gradual increase in the size of the swelling head and neck area and on lower extremities. Twenty to
without overlying skin changes. He also complained of twenty-five percent of melanoma cases occur in head and
occasional episodes of bleeding from right nostril. He neck region out of which approximately 6–8% originates
underwent fine-needle aspiration cytology (FNAC) of in mucous membrane of upper aerodigestive tract [2].
the right submandibular swelling in a local peripheral The most common site for mucosal melanomas in the
hospital which was reported as suggestive of non- head and neck region is oral cavity followed by sinonasal
Hodgkin lymphoma. He was referred to our tertiary care region and lastly pharynx. Sinonasal mucosal melanomas
centre for further treatment and management. On general are uncommon and comprise less than 1% of all
physical examination a single submandibular lymph melanomas and less than 5% of all sinonasal neoplasms
node, measuring approximately 2x3 cm, was palpated [3]. The most common site of origin for melanomas
on the right side. Swelling was firm, non-tender and within the nose is nasal septum followed by inferior and
non-mobile with no overlying skin changes. The patient middle turbinate [4]. The exact site of origin of the larger
underwent excision biopsy of the submandibular swelling lesions often cannot be determined. These tumors are
and histopathological examination showed effacement of aggressive with high incidence of locoregional recurrence
entire lymph node architecture by a tumor composed of and distant metastasis to lymph nodes and viscera. The
sheets of polygonal cells having high N:C ratio, variable incidence of regional lymph node metastasis is 5–15%
amount of cytoplasm, coarse vesicular chromatin and [5]. The submandibular lymph nodes are most commonly
prominent eosinophilic nucleoli. A few cells focally involved. Involvement of regional lymph nodes strongly
were found to contain intracytoplasmic pigment. suggests distant spread as seen in our case [6].
Immunohistochemistry confirmed the diagnosis of The diagnosis of mucosal melanomas is based on
metastatic deposit from malignant melanoma. Meanwhile histological finding and IHC because their microscopic
the patient was evaluated in ENT outpatient department features overlap with high grade lymphoma, poorly
for epistaxis. Nasal examination revealed a small, friable,
fleshy growth in the right nasal cavity. Nasal endoscopy
of the left side was normal. Computed tomography
(CT) scan of the head and neck area was inconclusive.
Computed tomography scan of chest and abdomen was
within normal limits.
Positron emission tomography (PET) scan was done
which showed an FDG-avid soft tissue density lesion in
the right nasal cavity measuring 36x12 mm (Figure 1).
Wide excision biopsy of nasal mass was done and sent
for histopathological confirmation. Hematoxylin &
Eosin (H&E) section of the nasal mass revealed round
to polygonal tumor cells disposed in sheets. The cells
had high N:C ratio, hyperchromatic pleomorphic nuclei
with prominent eosinophilic nucleoli. These cells had
variable amount of cytoplasm with only a few cells having
intracytoplasmic melanin pigment focally. On IHC the
tumor cells were positive for HMG-45 and S-100 and were
negative for LCA, Pan-CK and EMA. Based on HPE and
IHC the diagnosis of malignant melanoma was offered
(Figure 2A–C). This tumor was considered as primary
melanoma of nasal cavity due to the absence of previous Figure 1: Positron emission tomography scan showing FDG-
or concurrent pigmented lesions elsewhere. avid soft tissue density lesion within the right nasal cavity.
differentiated carcinomas, rhabdomyosarcomas,

plasmacytomas [7]. They are positive for S-100 protein
and specific melanocytic markers such as melan A
and HMB-45 antigens [8]. The diagnosis is further
complicated by the absence or presence of scanty melanin
pigment as seen in our case.
The management of nasal mucosal melanomas
has not been uniform. Standard treatment is surgical
resection associated with adjuvant radiotherapy
and chemotherapy. Surgery along with adjuvant
chemotherapy and radiotherapy should be used for
patients with either regional metastasis or large bulky
primary disease [9]. In our patient wide local excision
was performed followed by postoperative radiotherapy.
The purpose of presenting the case is rarity of the lesion
along with high index of suspicion which will help in early
diagnosis and treatment.
CONCLUSION
Malignant melanomas are the greatest mimickers
in pathology. They can be mistaken for a variety of
tumors especially in the nasal cavity where these tumors
usually do not show any junctional activity and also are
amelanotic. Hence a high degree of suspicion on part
of the pathologist is required to clinch the diagnosis in
early stages. Treatment of choice in mucosal melanomas
is combination of surgery with radiotherapy. Genesis
of targeted immunotherapy and chemotherapy against
melanomas based on clear understanding of biology of
these tumors will help achieve a higher response rate.
*********
Author Contributions
Devika Gupta – Conception and design, Acquisition of
data, Analysis and interpretation of data, Drafting the
article, Critical revision of the article, Final approval of
the version to be published
Niti Goyal – Conception and design, Acquisition of data,
Analysis and interpretation of data, Drafting the article,
Critical revision of the article, Final approval of the
version to be published
Vandana Rana – Acquisition of data, Analysis and
interpretation of data, Critical revision of the article,
Final approval of the version to be published
Rajat Jagani – Analysis and interpretation of data,
Figure 2: (A) Section from mass right nasal cavity showing
Drafting the article, Critical revision of the article, Final
an attenuated respiratory epithelial lining. The subepithelial approval of the version to be published
region is infiltrated by sheets of tumor cells (H&E stain, Davendra Swarup – Analysis and interpretation of data,
x100). (B) Section showing round to polygonal tumor cells Critical revision of the article, Final approval of the
having high nuclear-to-cytoplasmic ratio, coarse vesicular version to be published
nuclei with conspicuous, eosinophilic nucleoli (H&E stain,
x400). (C) Positive staining of tumor cells for (HMG-45, Guarantor
x200). The corresponding author is the guarantor of submission.
Conflict of Interest of tumors. Pathology and Genetics, Head and Neck

Authors declare no conflict of interest. Tumors. Lyon: IARC Press 2005. p. 65–75.
4. Díaz Molina JP, Rodrigo Tapia JP, Llorente Pendas
JL, Suárez Nieto C. Sinonasal mucosal melanomas:
Copyright Review of 17 cases. Acta Otorrinolaringol Esp
© 2014 Devika Gupta et al. This article is distributed 2008;59(10):489–3. [Article in Spanish].
under the terms of Creative Commons Attribution 5. Patrick RJ, Fenske NA, Messina JL. Primary mucosal
License which permits unrestricted use, distribution melanoma. J Am Acad Dermatol 2007;56(5):828–
and reproduction in any medium provided the original 34.
author(s) and original publisher are properly credited. 6. Batsakis JG, Suarez P, El-Naggar AK. Mucosal
Please see the copyright policy on the journal website for melanomas of the head and neck. Ann Otol Rhinol
more information. Laryngol 1998;107(7):626–30.
7. Iezzoni JC, Mills SE. “Undifferentiated” small
round cell tumors of the sinonasal tract: Differential
diagnosis update. Am J Clin Pathol 2005;124
REFERENCES Suppl:S110–21.
8. Hofbauer GF, Böni R, Simmen D, et al. Histological,
1. Gilchrest BA, Eller MS, Geller AC, Yaar M. The immunological and molecular features of a nasal
pathogenesis of melanoma induced by ultraviolet mucosa primary melanoma associated with nasal
radiation. N Engl J Med 1999;340(17):1341–8. melanosis. Melanoma Res 2002;12(1):77–82.
2. Bothale KA, Maimoon SA, Patrikar AD, Mahore SD. 9. Owens JM, Roberts DB, Myers JN. The role of
Mucosal malignant melanoma of the nasal cavity. postoperative adjuvant radiation therapy in the
Indian J Cancer 2009;46(1):67–70. treatment of mucosal melanomas of the head and
3. Wenig BM, Prasad ML, Dulguerov P, et al. neck region. Arch Otolaryngol Head Neck Surg
Neuroectodermal tumors. In: Barnes L, Eveson JW, 2003;129(8):864–8.
Reichart P, Sidransky D editors. WHO classification
About the Authors
Article citation: Gupta D, Goyal N, Rana V, Jagani R, Swarup D. Malignant mucosal melanoma of the nasal
cavity: A case report. Int J Case Rep Images 2014;5(9):629–633.
Devika Gupta is Assistant Professor in Department of Pathology, Armed Forces Medical College,
Pune, India. She has earned the undergraduate degree MBBS from Armed Forces Medical College
(Pune University), India and postgraduate degree of MD Pathology and DNB Pathology from Delhi
University, India. Her research interests include hematolymphoid malignancies and blood coagulation
disorders. She intends to pursue fellowship in transplant immunology in future.
Niti Goyal is final year Resident Pathology in the Department of Pathology at Armed Forces Medical
College, Pune, India. She earned the undergraduate degree (MBBS) from, India. She has keen interest
in oncopathology and wants to pursue her fellowship in same.
Vandana Rana is Associate Professor in Department of Pathology, Command Hospital, Pune, India.
She has earned her MBBS and MD (Pathology) from PGIMS Rohtak, Haryana, India. She has been
working in the field of oncopathology for last seven years. Her research interests include breast and
lymphoid malignancies. She has active interest in teaching and intends to keep on upgrading and
expanding her horizons.
Rajat Jagani is Associate Professor in Department of Pathology, Command Hospital, Pune, India.
He earned the undergraduate degree MBBS from University of Pune, Pune Maharashtra, India and
postgraduate degree form University of Pune, Pune Maharashtra, India. (Long-term Training in
oncopathology at Post-graduate Institute of Medical Education and Research, Chandigarh India). He
has published seven research papers in national and international academic journals. His research
interests include prostate pathology, breast pathology and effusion cytology. He intends to pursue PhD
in oncopathology in future.
Davendra Swarup is HOD Pathology in Department of Pathology, Command Hospital, Pune, India.
He passed his MBBS and MD (Pathology) from Agra University, India. He has teaching experience of 14
years for both undergraduate and postgraduate students. His research interests include histopathology
and transfusion medicine.

Int J Case Rep Images 2014;5(9):634–637. Kumar et al. 634
Erythropoietin induced miliaria crystallina: A possible new

adverse effect of erythropoietin
Sumir Kumar, B.B. Mahajan, Sandeep Kaur, Amarbir Singh
ABSTRACT no change in the treatment plan for the above

mentioned complaints except introduction of
Introduction: Erythropoietin, also known as erythropoietin recently for the management
EPO, is used for the treatment of anemia of of severe anemia secondary to chronic kidney
chronic kidney disease. Usually, the drug is well disease. The diagnosis of miliaria crystallina
tolerated with only a few side effects. Adverse was made clinically and spontaneous resolution
effects are mostly systemic with hypertension occurred in about seven days. Conclusion:
being seen frequently. Various cutaneous This case adds erythropoietin among the list of
side effects include pruritus, rash, urticaria drugs precipitating miliaria crystallina which
and erythema. However, miliaria induced by is being reported for the first time in literature.
erythropoietin has not been reported so far. Recognizing this otherwise benign clinical
Case Report: A 74-year-old female was referred entity is important as it can be confused with
from medicine department with possibility severe drug reactions such as toxic epidermal
of toxic epidermal necrolysis. She developed necrolysis. The absence of inflammatory signs in
crops of non-itchy tiny vesicles filled with clear skin, lack of mucosal involvement, no systemic
fluid on body for the last three days. Injection manifestations and spontaneous resolution can
erythropoietin was given six hours prior to help to distinguish it from other drug reactions.
onset of rash. Similar episode occurred one
week back after the use of erythropoietin. There Keywords: Erythropoietin, EPO, Miliaria,
was no history of any acute febrile illness. She Eccrine sweat glands
was a known case of hypertension, diabetes
mellitus with chronic kidney disease. There was How to cite this article
Sumir Kumar1, B.B. Mahajan2, Sandeep Kaur3, Amarbir Singh4 Kumar S, Mahajan BB, Kaur S, Singh A.
Erythropoietin induced miliaria crystallina: A
Affiliations: 1MD, Assistant Professor, Department of
Dermatology, Venereology & Leprology, GGS Medical
possible new adverse effect of erythropoietin. Int J
College & Hospital, Faridkot, Punjab, India; 2MD, Professor Case Rep Images 2014;5(9):634–637.
& Head, Department of Dermatology, Venereology &
Leprology, GGS Medical College & Hospital, Faridkot,
doi:10.5348/ijcri-2014112-CR-10423
Punjab, India; 3MBBS, Postgraduate Resident, Department
of Dermatology, Venereology & Leprology, GGS Medical
College & Hospital, Faridkot, Punjab, India; 4MD, Senior
Resident, Department of Dermatology, Venereology &
Leprology, GGS Medical College & Hospital, Faridkot,
Punjab, India. INTRODUCTION
Corresponding Author: Sandeep Kaur C/O Mr. B.D. Sharma,
House No. 49/A, Street No. 3, Guru Nanak Colony, Opposite Erythropoietin (EPO) is a glycoprotein that controls
GGS Medical College & Hospital, Sadiq Road, Faridkot, erythropoiesis. It is used frequently for the treatment of
Punjab, India. 151203; Mob: 91-9779845246; Email: anemia of chronic kidney disease. Its adverse effects are
docsandeep_2005@yahoo.com mostly systemic with hypertension being seen frequently.
We report a case of a 74-year-old female who got referred
to us with possibility of toxic epidermal necrolysis.
Subsequently, diagnosis of miliaria induced by EPO was
established and patient recovered spontaneously. This
side effect has not been reported so far. This case report,
thus, highlights this unrecognized adverse effect and Thus, both adrenergic and cholinergic stimulation results
importance of differentiating it from other serious drug in increased sweating. There is also role of intracellular
reactions. calcium in acetylcholine mediated stimulation of eccrine
sweating (Figure 2).
It is the obstruction or disruption of these eccrine
CASE REPORT glands that results in miliaria [1]. The three forms of
miliaria, miliaria crystallina, miliaria rubra (prickly
A 74-year-old female was referred to us from medicine heat) and miliaria profunda, differ in clinical form due
department with possibility of toxic epidermal necrolysis. to the different levels at which obliteration occurs, with
She complained of development of crops of non-itchy tiny pathology being present at the level of stratum corneum,
vesicles filled with clear fluid on body for last three days. intraepidermal and at or below the dermal-epidermal
Injection EPO was given six hours prior to onset of rash. junction, respectively.
Similar episode occurred one week back after the use of Miliaria crystallina is seen in conditions associated
EPO. There was no history of any acute febrile illness. with profuse sweating and high humidity. It occurs
She was a known case of hypertension, diabetes mellitus commonly in infants due to a delay in patency developing
with chronic kidney disease. There was no change in
the treatment plan for the above mentioned complaints
except introduction of EPO recently for management of
severe anemia secondary to chronic kidney disease.
Physical examination revealed afebrile patient with
blood pressure 160/90 mmHg. On local examination,
multiple, discrete, fragile vesicles filled with clear fluid on
normal appearing, non-tender skin were evident in left
inframammary area along with branny desquamation on
the back, abdomen and thighs, buttocks (Figure 1A–B).
Rest of the body including palms, soles and mucosae
was normal. Laboratory results of the patent revealed
hemoglobin 5 g/dL, with microcytic and normocytic
anemia, serum creatinine 3.5 mg/dL, blood urea 50
mg/dL, clotting time 8 minutes (normal 2–6 minutes), Figure 1: (A) Desquamation on trunk with background skin
being normal at the time of presentation, (B) Close-up view
prothrombin time 20 seconds (normal 11–15 seconds).
of vesicles: fragile vesicles filled with clear fluid without any
Liver function tests and electrolytes were within normal signs of inflammation, (C) Normal looking skin on anterior
limits. Viral markers were non-reactive. Biopsy was not trunk after resolution of miliaria, (D) Resolution of miliaria on
done in view of severe anemia, deranged coagulation posterior trunk.
profile, immunosuppressed state of CRF and clear
distinct clinical picture.
The diagnosis of miliaria crystallina was made
clinically. She was managed conservatively tab
hydroxyzine 10 mg HS, tab ranitidine 150 mg BD,
calamine lotion for local application BD. She improved
in about seven days without any sequelae (Figure 1C–D).
DISCUSSION
Eccrine sweat glands are a type of sweat glands that
are distributed widely over the body surface and produce
hypotonic sweat. The evaporation of sweat helps in
thermoregulation through the loss of extra heat. Activity
of these glands is controlled by the thermoregulatory
centre located in preoptic region of the hypothalamus.
Innervation occurs through the sympathetic post
ganglionic fibres arising at the spinal cord thoracic
and lumbar regions T1-L2. In contrast to ordinary
sympathetic innervation, acetylcholine is the principle
neurotransmitter. Although it also has an adrenergic
Figure 2: The role of intracellular calcium in the production of
component. Sweat cells exhibit cholinergic and alpha and
eccrine sweat.
beta adrenergic receptors on their basolateral membrane.
in the sweat ducts. It can also uncommonly occur in *********

adults during episodes of febrile illnesses associated
with profuse sweating. Profuse sweating results in Author Contributions
overhydration of stratum corneum which results in Sumir Kumar – Substantial contributions to conception
obstruction of acrosyringium. and design, Acquisition of data, Analysis and
Recognizing this otherwise benign clinical entity interpretation of data, Drafting the article, Revising
is important as it can be confused with drug reactions it critically for important intellectual content, Final
such as toxic epidermal necrolysis. The absence of approval of the version to be published
inflammatory changes in skin, and absence of systemic B.B. Mahajan – Analysis and interpretation of data,
manifestations and spontaneous resolution can help to Revising it critically for important intellectual content,
distinguish it from other drug reactions. Final approval of the version to be published
Erythropoietin is produced normally by interstitial Sandeep Kaur – Analysis and interpretation of data,
fibroblasts in the kidney in close association with Revising it critically for important intellectual content,
peritubular capillary and tubular epithelial tubule. Final approval of the version to be published
Erythropoietin available for use as therapeutic agents is Amarbir Singh – Analysis and interpretation of data,
produced by recombinant DNA technology in cell culture. Revising it critically for important intellectual content,
Its use is associated with some adverse effects with Final approval of the version to be published
hypertension being reported in 20–30% patients treated
for anemia of chronic kidney disease [2]. This side effect Guarantor
is less common with subcutaneous route as compared The corresponding author is the guarantor of submission.
with intravenous administration [3, 4]. Various factors
are thought to contribute to hypertension as exemplified Conflict of Interest
in various studies [5–7]. Of these mechanisms like alpha Authors declare no conflict of interest.
adrenergic hyper-responsiveness and marked increase
in intracellular calcium levels could possibly have Copyright
contributed to the pathogenesis of miliaria in our patient. © 2014 Sumir Kumar et al. This article is distributed
Other cutaneous side effects reported include pruritus, under the terms of Creative Commons Attribution
rash, urticaria (3%), erythema (1%). License which permits unrestricted use, distribution
When exogenous EPO is used as a performance- and reproduction in any medium provided the original
enhancing drug through the enhancement of oxygen author(s) and original publisher are properly credited.
delivery to muscles, it is classified as an erythropoiesis- Please see the copyright policy on the journal website for
stimulating agent (ESA) and has been used in various more information.
sports. Hence, it is included under category of doping
agents.
There are several case reports of drug precipitated REFERENCES
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so far. So this may be the first report of this unique side 1. Shuster S. Duct disruption, a new explanation of
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of follicular differentiation [8]. Bethanechol, a drug that 2. Maschio G. Erythropoietin and systemic hypertension.
promotes sweating due to its parasympathomimetic effect, Nephrol Dial Transplant 1995;10 Suppl 2:74–9.
3. Smith KJ, Bleyer AJ, Little WC, Sane DC. The
has been reported to cause miliaria, as have clonidine
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following doxorubicin administration has been reported, 4. Strippoli GF, Craig JC, Manno C, Schena FP.
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CONCLUSION 5. Krapf R, Hulter HN. Arterial hypertension induced by
erythropoietin and erythropoiesis-stimulating agents
This case adds erythropoietin (EPO) among the list (ESA). Clin J Am Soc Nephrol 2009;4(2):470–80.
of drugs precipitating miliaria crystallina which is being 6. Marrero MB, Venema RC, Ma H, Ling BN, Eaton DC.
Erythropoietin receptor-operated Ca2+ channels:
reported for the first time in literature. Recognizing this
Activation by phospholipase C-gamma 1. Kidney Int
otherwise benign clinical entity is important as it can 1998;53(5):1259–68.
be confused with severe drug reactions such as toxic 7. Abiose AK, Aronow WS, Moreno H Jr, Nair CK,
epidermal necrolysis. The absence of inflammatory Blaschke TF, Hoffman BB. Increased vascular alpha1-
signs in skin, lack of mucosal involvement, no systemic adrenergic sensitivity in patients with renal failure:
manifestations and spontaneous resolution can help to Receiving recombinant erythropoeitin. Am J Ther
distinguish it from other drug reactions. 2007;14(5):427–34.
8. Gupta AK, Ellis CN, Madison KC, Voorhees JJ.

Miliaria crystallina occurring in a patient treated with
isotretinoin. Cutis 1986;38(4):275–6.
9. Haas N, Martens F, Henz BM. Miliaria crystallina
in an intensive care setting. Clin Exp Dermatol
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10. Godkar D, Razaq M, Fernandez G. Rare skin
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Int J Case Rep Images 2014;5(9):638–641. Mahapatra et al. 638
Sirenomelia: A case report

Swagat Mahapatra, Suruchi Ambasta
Abstract Keywords: Sirenomelia, Caudal regression

syndrome, Deformity, Congenital structural
Introduction: Sirenomelia is a congenital anomaly, Fusion
structural anomaly characterized by abnormal
development of the caudal region of the body with How to cite this article
varying degrees of fusion of lower limbs. Most
of the times, the condition is fatal for the baby. Mahapatra S, Ambasta S. Sirenomelia: A case report.
Most babies do not survive even after surgery. Int J Case Rep Images 2014;5(9):638–641.
Fifty percent of cases are seen as stillbirths,
and it is much more common in identical twins.
This abnormality was initially confused with doi:10.5348/ijcri-2014113-CR-10424
caudal regression syndrome, but later was
given a new name, i.e., sirenomelia mermaid
syndrome. Case Report: We present a case of a
15-year-old boy with partially fused lower limbs.
Introduction
A provisional diagnosis of sirenomelia is made
and detailed review and surgery was planned. Sirenomelia is a congenital structural anomaly
The boy did not report for further treatment. characterized by abnormal development of the caudal
Conclusion: Very rare disorder with prevalence region of the body with varying degrees of fusion of lower
of 1 in 100,000 live births with a total of 300 cases limbs. Almost always the condition proves fatal for the
reported till today in which nine are from India. baby. Most babies do not survive even after surgery. Fifty
The precise etiology of sirenomelia was not well percent of cases are seen as stillbirths, and it is much
understood. Many theories have been proposed more common in identical twins. After initial confusion
but none of these is considered definitive. It with caudal regression syndrome, it was later given a new
is very important to diagnose this condition name—sirenomelia mermaid syndrome. Sirenomelia is
by ultrasonography so that termination of characterized by complete fusion of bilateral lower limbs,
pregnancy can be carried out. along with renal agenesis, gastrointestinal defects and
absent external genitalia. An important finding which
differentiates these two entities is the presence of single
Swagat Mahapatra1, Suruchi Ambasta2 umbilical and persistent vitelline artery.
Affiliations: 1
MS, Orthopedics, Senior Resident,
Department of Orthopedics, JIPMER, Pondicherry, India;
2
MD Anesthesiology, Senior Resident, Department of CASE REPORT
Anesthesiology and Critical Care, JIPMER, Pondicherry,
India. A 15-year-old boy presented to our outpatient
Corresponding Author: Swagat Mahapatra, Old MSR department with deformity of both lower limbs
Quarters No-21, Dhanawantary Nagar, JIPMER with partial fusion of lower legs since birth. He was
Campus, Puducherry, Pondicherry, India - 605006; Ph: accompanied by his parents. On taking detailed history, it
+918940482218; Email: drswagat@gmail.com was found that parents had not ever consulted any doctor.
Patient was able to ambulate independently with a single
stick. He was not having any other complaint as such and
was mentally normal. He was having no other physical
Accepted: 07 July 2014
anomalies in the skull, facies, chest and lumbar region.
On examination of both the lower limbs, it was seen that

pelvis and both femur were normal. The left popliteal
fossa was at a lower level than the right. Fusion of the
lower limbs was noted at the level of distal one-third of
the leg about 8 cm above bilateral ankle joint. Below the
level of fusion the right distal leg was straight but the left
distal leg and ankle foot complex was deviated laterally
with acute angulation. Both feet were maldeveloped with
rockerbottom deformity with the absence of great toe on
the left foot and few other phalanges in both feet (Figure
1 and 2). On palpation, it was felt that the proximal part
of the left tibia was fused with the right tibia. X-rays
confirmed our findings of fusion of bilateral tibia with
acute angulation of the left tibia with partial absence of
metatarsals and phalanges (Figure 3). The development
of feet above the level of fusion was adequate but below Figure 1: Clinical photograph of both lower limbs showing
this level both feet were maldeveloped. A provisional fusion of lower leg with both ill formed feet.
diagnosis of sirenomelia was made and further workup
and plan for surgery was made. The parents agreed for
workup and left to come prepared for admission but they
did not come for admission and also did not respond to
repeated calls.
DISCUSSION
A mermaid is a legendary aquatic creature with the
upper body of a female human and the tail of a fish.
Mermaids are associated with the biological order
sirenia comprising dugongs and manatees. Hence
this syndrome is named sirenomelia. Sirenomelia is a
congenital structural anomaly characterized by abnormal
development of the caudal region of the body with
varying degrees of fusion of lower limbs [1]. It bears the
resemblance of Mermaid of Greek mythology and hence
the synonym of Mermaid syndrome [2]. The prevalence
of this anomaly is 1:100.000 live births with a male to
female ratio of 3:1. About 300 cases are reported in which
nine are from India [3].
There are clinically mild and severe varieties. In mild
variety, the sirenomelia baby has two limbs fused into Figure 2: Clinical photograph of both feet with legs showing
one, only to the extent of the skin. The feet may be fully standing attitude with left popliteal fossa at a lower level than
formed and many are only attached at the ankles. All the right.
three main bones of the leg are fully and correctly formed.
In this situation, a small surgery can easily correct the
deformity whereas the severe variety is very difficult to
manage. Externally both limbs are completely joined and
appear ill-formed. There is a complete absence of foot
structures and out of the three long bones, only two are
present in the entire limb. Other internal abnormalities
can only be accessed with imaging studies. Distinction was
made between these due to the fact that the sirenomelia
had a specific pathogenic factor namely arterial steal
phenomenon. On the other hand, the caudal regression
syndrome was probably due to diabetic embryopathy. An
important finding which differentiates these two entities
is presence of single umbilical and persistent vitelline Figure 3: X-ray features of both lower limbs showing fusion of
distal part of left tibia with the right with lateral bending of left
artery. Sirenomelia has also been classified into three
leg.
types simpus apus (no feet, one tibia, one femur), simpus Copyright
unipus (one foot, two femur, two tibia, two fibula), © 2014 Swagat Mahapatra et al. This article is distributed
simpus dipus (two feet and two fused legs (flipper like)- under the terms of Creative Commons Attribution
this is called a mermaid). The first case of sirenomelia License which permits unrestricted use, distribution
was seen in 1542. Duhamel gave the term caudal and reproduction in any medium provided the original
regression syndrome in 1961 in which he described that author(s) and original publisher are properly credited.
sirenomelia was associated with anorectal, genitourinary Please see the copyright policy on the journal website for
and vertebral anomalies. more information.
The precise etiology of sirenomelia is not well
understood. Many theories have been proposed but none
of these is considered conclusive. Hibelink et al. [4] told IV REFERENCES
cadmium and lead can produce sirenomelia in the golden
hamster. Von Lennep et al. [5] stated teratogenic effects 1. Jones KL. Sirenomelia sequence in smith’s
of Vitamin A, Duhemmel et al. [6] told manifestation of recognizable patterns of humal malformation. WB
the caudal regression syndrome. Quan et al. [7] coined saunders company, Philadelphia 1998:634.
2. De Silva MV, Lakshman WD. Sirenomelia sequence
the term VATER, Stocker et al. [8] proposed defect in the
(Mermaid syndrome). Ceylon Med J 1999;44(1):34–
primitive streak, Stevenson [9] explained vitelline artery 5.
steal theory. 3. Parthasarathy S. Shivanarutselvan - Sirenomelia
Ultrasonography of a fetus with sirenomelia – A case report-Interesting cases in orthopaedics.
demonstrates the fused femur, decreased distance IJCP;2:23–5.
between two femurs and decreased or absent mobility 4. Hilbelink DR, Kaplan S. Sirenomelia: Analysis in the
of the two lower limbs with respect to each other. When cadmium and lead treated golden hamster. Teratog
infant is clinically examined, there may be only simple Carcinog Mutagen 1986;6(5):431–40.
fusion of skin of the limbs or there may be fusion of 5. Von Lennep E, El Khazen N, De Pierreux G, Amy
JJ, Rodesch F, Van Regemorter N. A case of partial
all long bones except fused femur. So imaging studies
Sirenomelia and possible vitamin A teratogenesis.
such as X-rays which show the bony abnormalities Prenat Diagn 1985;5(1):35–40.
and ultrasonography are advised to see for solid organ 6. Duhamel B. From the mermaid to anal imperforation:
abnormality. This disorder is universally lethal and hence The syndrome of caudal regression. Arch Dis Child
prenatal diagnosis with imaging studies is very helpful to 1961;36(186):152–5.
plan termination of pregnancy [10, 11]. 7. Quan L, Smith DW. The VATER association.
Vertebral defects, anal atresia, tracheo-esophageal
fistula with esophageal atresia, renal and radial
CONCLUSION dysplasia: A spectrum of associated defects. J Pediatr
1973;82(1):104–7.
8. Stocker JT, Heifetz SA. Sirenomelia. A morphological
In our case, we could not find many of the features as
study of 33 cases and review of the literature. Perspect
described for this rare condition. Also we could not further Pediatr Pathol 1987;10:7–50.
investigate to clinch the etiology due to non-cooperation 9. Stevenson RE, Jones KL, Phelan MC, et al. Vascular
of the patient. This rare abnormality is usually universally steal: The pathogenetic mechanism producing
fatal but this boy was found to be healthy at fifteen years sirenomelia and associated defects of the viscera and
of age. This case was reported due its rarity. soft tissues. Pediatrics 1986;78(3):451–7.
10. Duncan PA, Shapiro LR, Klien RM. Sacrococcygeal
********* dysgenesis association. Am J Med Genet
1991;41(2):153–61.
11. Schuler L, Salzano FM. Patterns in multimalformed
Author Contributions babies and the question of relationship between
Swagat Mahapatra – Substantial contributions to sirenomelia and VACTERL. Am J Med Genet
conception and design, Acquisition of data, Analysis 1994;49(1):29–35.
and interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Suruchi Ambasta – Analysis and interpretation of data,
Guarantor
Conflict of Interest
Authors declare no conflict of interest.

A huge retroperitoneal lymphatic cyst presenting as a

mesenteric cyst, managed laparoscopically
Manash Ranjan Sahoo, Leesa Misra, Raghavendra Mohan Kaladagi,
Manoj Srinivas Gowda, Abinash Panda, Syam Sundar Behera
Abstract such selected cases laparoscopy is helpful both

as a diagnostic and therapeutic modality.
Introduction: Retroperitoneal lymphatic cysts
are uncommon in occurrence. Due to presence Keywords: Erythropoietin, EPO, Miliaria,
of large potential space in the retroperitoneum, Eccrine sweat glands
the cyst grows to a considerable size in abdomen
before it presents clinically and the presentation How to cite this article
noted in most of the cases are non-specific,
confusing to the surgeons leading to delay in Sahoo MR, Misra L, Kaladagi RM, Gowda MS,
diagnosis. The treatment of choice is complete Panda A, Behera SS. A huge retroperitoneal
excision of cyst. Nowadays minimal access lymphatic cyst presenting as a mesenteric cyst,
procedures are tried. Case Report: We report managed laparoscopically. Int J Case Rep Images
a case of a huge retroperitoneal lymphatic cyst 2014;5(9):642–645.
which had complex presentation mimicking as
mesenteric cyst in a 22-year-old female, which doi:10.5348/ijcri-2014114-CR-10425
was managed laparoscopically. Conclusion:
Retroperitoneal cysts are uncommon, with a
very low estimated incidence. Approximately,
one-third of patients with retroperitoneal
cysts are asymptomatic and the cyst is found Introduction
incidentally. Retroperitoneal lymphatic cysts are
an uncommon lesion in adults. With complete Retroperitoneal cysts are uncommon with an
surgical excision these tumors have an excellent estimated incidence of 1/5,750 to 1/250,000 [1].
prognosis, with great symptomatic relief. In Approximately, one-third of patients with retroperitoneal
cysts is asymptomatic and found incidentally. Sometimes
they pose challenge for proper preoperative diagnosis
Manash Ranjan Sahoo1, Leesa Misra2, Raghavendra Mohan because of their location. With the recent development
Kaladagi3, Manoj Srinivas Gowda3, Abinash Panda3, Syam of surgical equipment and advance surgical techniques, a
Sundar Behera3
number of minimally invasive procedures are employed
Affiliations: 1MBBS, MS, FAIS, FAMS, Associate Professor, for treatment of such tumors. This case report describes
Surgery, S.C.B. Medical College, Cuttack, Odisha, India; a patient with lymphatic cyst mimicking as a mesenteric
2
MBBS, MD, Associate Professor, Obstetrics & Gynecology,
cyst who underwent a successful laparoscopic resection.
S.C.B. Medical College, Cuttack, Odisha, India; 3MBBS,
(MS), Postgraduate Student, Surgery, S.C.B. Medical
Corresponding Author: Manash Ranjan Sahoo, Associate CASE REPORT
Professor, Department of Surgery, SCB Medical College
and Hospital, Cuttack, Odisha, India. 753 001; Ph: A 22-year-old female presented with mass per
+919937025779, Fax: +916712414034; E-mail: manash67@ abdomen in right lower region since four months
gmail.com which was gradually increasing in size and associated
with vague abdominal discomfort which was diffuse,
constant, dull aching, nothing alleviated the pain. There
Received: 20 May 2014 were irregularities in her monthly cycles from last one
Accepted: 06 July 2014
year, no history of loss of appetite or weight, no history
of tuberculosis or any surgeries (appendicectomy) and
no significant changes in the bowel and bladder habits.

Past history was insignificant except the menstrual
changes. There was also no history of any gynecological
malignancies in family members.
On examination, general condition was satisfactory,
moderately built and nourished with mild pallor, no
neck or axillary gland enlargement, with stable vital
parameters. Abdominal examination reveals a mass in the
right lower abdomen involving the lumbar, iliac region
with extension in infra umbilical area; the mass was soft to
cystic in consistency, mobile freely in transverse direction
while less mobile in vertical direction, no shifting dullness
and no thrill noted. Per vaginal examination revealed
anteverted uterus and fullness in the right fornix with
cystic feeling. Per rectal examination revealed a tense
cystic fullness in pouch of Douglas with definite lumpy
feeling. Figure 1: Contrast-enhanced computed tomography scan
On investigations, routine hemogram showed showing cystic lesion in the right iliac fossa region.
hemoglobin 8.6 g/dL with other normal parameters.
Chest radiography was normal. Ultrasonography showed
heterogenous hypoechoic lesion to right of psoas muscle.
Computed tomography (CT) scan revealed a well-
marginated low attenuating SOL with attenuation value
of +13 to +34 HU in right lower abdomen and pelvis,
deforming the right psoas muscle, displacing the aorta
and IVC to left side (Figure 1). There was loss of interface
between right ovary and the cyst. Other pelvic organs are
normal.
The patient was taken up for surgery with a few
possibilities such as retroperitoneal cyst, mesenteric
cyst, ovarian cyst. Under general anesthesia, one 10 mm
umbilical port given and diagnostic laparoscopy was
done. It was found that a cystic mass measuring about
15x15x8 cm in the retroperitoneal region just below the
cecum displacing the bowel loops medially. Two 5 mm
ports given to proceed with the surgery, one in left iliac
fossa and the other in right pararectal area just above Figure 2: Diagnostic laparoscopy showing cystic lesion just
umbilicus level. A large bore (18 Gz) spinal needle was below the cecum, fluid aspirated percutaneously.
passed into the cyst from the abdominal wall under
laparoscopic vision to aspirate the cystic fluid (Figure 2)
for confirmation and complete decompression. Later, the
cyst wall was completely resected out taking care not to
injure the right iliac vessels which were in close proximity
to the cyst (Figure 3). Finally, reperitonealization was
done, after the cyst wall was removed in toto (Figure 4)
and sent for histopathological examination which came
out to be as a lymphatic cyst. There were no intraoperative
or postoperative complications. The postoperative period
was uneventful and the patient discharged on fourth
postoperative day. Follow-up At fifth month, the patient
was good and he is doing fine till date.
DISCUSSION
Embryologically, there are five regional primitive
lymphatic sacs which normally develop into chains of
lymph nodes [2] the paired jugular sacs lateral to the Figure 3: Bed of the cyst after its complete excision.
retroperitoneum or greater omentum [4], where they

are referred to as ‘omental or mesenteric cysts’. The
retroperitoneum is the second-most common location
for the abdominal lymphangiomas after mesentery of the
small bowel.
In Thrupp’s [5] series, 57.2% had asymptomatic
abdominal masses, while 23.8% had infections or
hemorrhagic complications, and 19% were postmortem
or operative findings. Intestinal obstruction, peritonitis,
rupture or infection may also be presenting symptoms.
However, most tumors present with an increasing
abdominal or flank mass and a dull flank pain with a “full
sensation.”
The differential diagnosis of cystic tumor in the
retroperitoneum raises several possibilities. These
include both malignant and benign tumors, such as cystic
Figure 4: Reperitonealization after cyst excision. mesothelioma, teratoma, undifferentiated sarcoma,
cystic metastases (especially from ovarian or gastric
primaries), cysts of urothelial and foregut origin, benign
tumors such as lymphangioma, and other tumors such
as retroperitoneal hematoma, abscesses, duplication
internal jugular veins, an unpaired retroperitoneal sac at cysts, ovarian cysts [6]. Computed tomography scan
the root of the mesentery, and the paired sacs adjacent is ideal for assessing retroperitoneal cysts because it
to the sciatic veins. These sacs form chains of lymph provides discrete sectional images of the organs and
nodes which drain the head, neck, arm, mesentery, hip, retroperitoneal compartments, and in some cases,
back, and leg, respectively. These regional primitive familiarity with the most relevant radiographic features,
lymph sacs are generally thought to be developmental in combination with clinical information, allows adequate
sites of lymphangiomas. Many agree for the note— lesion characterization [7].
lymphangiomas arise at these sites by continued growth The treatment of choice for such retroperitoneal cyst
of congenitally misplaced primitive lymphatic tissue is simple total excision. The recurrence is not commonly
which fails to acquire venous connections or as continued noticed unless you go with treatment options such as
endothelial outgrowth of veins, where as others believe aspiration or drainage procedures or marsupialization.
that its due the lymphatic channel obstruction caused Dissemination in the retroperitoneum is a very rare, but
by trauma, fibrosis, node degeneration, inflammation potentially fatal complication [8].
genetic component and disorders of endothelial lymphatic Cysts arising within the retroperitoneum outside the
vascular secretion or permeability. major organs, within that compartment are very rare and
In 1877, Wegner histologically divided lymphangiomas one-third of them are asymptomatic. Retroperitoneal
into three classifications: (i) lymphangiomas simplex cystic lesions, although are benign, can be technically
(capillary lymphangioma), small, thin-walled lymphatic difficult to excise because of the proximity to major
channels not common or found intra-abdominally, (ii) vessels or other organs. These rare tumors can be cured
cavernous (sometimes malignant), larger thin-walled by complete excision. With advance in minimal access
channels, more common but rare intra-abdominally, (iii) surgeries, we can tackle such diseases by laparoscopic
cystic (always benign) composed of large cystic spaces cysts excision, which is feasible and safe, more on provides
lined with flat endothelium, but common retroperitoneally excellent cosmesis and has all the other advantages of
and intra-abdominally [3]. Based on embryologic origin, laparoscopy. Laparoscopic approach may be attempted
retroperitoneal cysts are classified into (i) urogenital, in selected cases to prevent large scars and morbidity
(ii) mesocolic, (iii) cysts arising in cell inclusions, (iv) associated with it including good results as in our case.
traumatic, (v) parasitic and (vi) lymphatic [1].
Approximately, 50% of lymphangiomas are present
at birth, and almost 90% are diagnosed before the age CONCLUSION
of 2–5 years. These cysts can occur in any part of body
where lymphatics are normally encountered. The most Retroperitoneal lymphatic cysts are uncommon
commonly affected sites are the head and neck (75%), lesions in adults. Since the disease is rare, investigations
where these are commonly referred to as ‘cystic hygromas’ may not provide accurate diagnosis and in such a case
(seen in newborns), followed by the axilla (20%). The diagnostic laparoscopy is helpful. These rare tumors
remainder (approximately 5%) of the lymphangiomas have an excellent prognosis, with symptomatic relief
are intra-abdominal arising from the mesentery, and definite cure after complete surgical excision. In the
modern era of minimal access surgery, this condition has
a definite treatment by laparoscopy, avoiding the need of Attribution License which permits unrestricted use,
laparotomy in selected cases. distribution and reproduction in any medium provided
the original author(s) and original publisher are properly
********* credited. Please see the copyright policy on the journal
website for more information.
Manash Ranjan Sahoo – Conception and design,
Acquisition of data, Analysis and interpretation of data, REFERENCES
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Copyright
© 2014 Manash Ranjan Sahoo et al. This article is
distributed under the terms of Creative Commons

Appendicular abscess in left inguinal hernia mimicking

strangulation
Manash Ranjan Sahoo, Basavaraja C., Kumar A.T., Sunil Jaiswal
Abstract How to cite this article

Introduction: The incidence of Amyand’s hernia Sahoo MR, Basavaraja C, Kumar AT, Jaiswal
varies from 0.5–1%, whereas only 0.1% of all cases S. Appendicular abscess in left inguinal hernia
have appendicitis present in an inguinal hernia. mimicking strangulation. Int J Case Rep Images
Case Report: A 40-year-old male presented 2014;5(9):646–649.
to emergency department with incarcerated
left inguinal hernia and pain over the hernia
since two days. Abdominal examination was doi:10.5348/ijcri-2014115-CR-10426
normal except for tenderness and increased
temperature over the left inguinal hernia.
Through an inguinoscrotal incision opening
of sac revealed pus and flakes with perforated
appendix. Lower midline laparotomy incision Introduction
was given and found the tip of perforated
appendix to lie at the left deep inguinal ring. Amyand’s hernia is defined as the presence of the
Appendicectomy with pelvic toileting was done. appendix within an inguinal hernia. It was Claudius
Bassini repair was done in the inguinal region. Amyand, surgeon to King George II, in 1735, the first
Conclusion: To conclude left sided Amyand’s person to describe the presence of a perforated appendix
hernia is very rare. High index of suspicion is within the inguinal hernial sac of an 11-year-old boy and
required for diagnosis and this should be kept performed a successful transherniotomy appendicectomy
as a differential diagnosis for such a varied [1, 2]. Hence named after him. In this rarer variety, right
presentation. sided are more common than left side. The incidence of
Amyand’s hernia varies from 0.5–1%, whereas only 0.1%
Keywords: Amyand’s hernia, Appendicec- of all cases have appendicitis present in an inguinal hernia.
tomy, Incarcerated left inguinal hernia, An extensive literature search revealed five reported cases
Bassini technique of left sided Amyand’s hernia [3–7]. None have described
appendicular abscess in left sided Amyand’s hernia. We
report a case of appendicular perforation with abscess in
left sided Amyand’s hernia.
Manash Ranjan Sahoo1, Basavaraja C.2, Anil Kumar A.T.2,
Sunil Jaiswal2
Affiliations: 1MS, Associate Professor, Department of CASE REPORT
Surgery, S.C.B. Medical College, Cuttack, Odisha, India;
2
Post Graduate, Department of Surgery, S.C.B. Medical A 40-year-old male presented to emergency
department with incarcerated left inguinal region and
Corresponding Author: Dr. Manash Ranjan Sahoo, Orissa pain over the hernia since two days. His vital parameters
Nursing Home, Medical road, Ranihat, Cuttack, Odisha,
were normal. On examination abdomen was soft, non-
India. Postal Code: 753007; Ph: +919937025779; Fax:
0671-2414034; E-mail: manash67@gmail.com tender, bowel sounds were normal, with an irreducible
hernia on the left inguinal region, with increased pain,
temperature and tenderness over the hernia. There was no
Received: 05 June 2014 history of fever. On investigating straight X-ray abdomen
Accepted: 11 July 2014 was normal. With diagnosis of strangulated inguinal
Published: 01 September 2014 hernia, he was planned for emergency exploration.
Through left inguina scrotal incision cremaster box was

opened. A band was seen constricting near the neck of the
sac which was released which resulted in disappearance
of the bowel contents into abdominal cavity. Opening of
the sac at its distant point revealed pus with tip of the
appendix just lying over the deep ring (Figure 1). The
whole of the structure was pulled out to confirm it as
appendix (Figure 2). This is not just a case of appendicitis
where appendicectomy alone through inguinal incision
would suffice. Since there was abscess formation, we
needed thorough toileting of the pelvis, where there
will be pus collection which will hamper postoperative
recovery. Hence a lower midline incision was given;
appendix perforated at the tip was found lying on the
deep inguinal ring. Appendicectomy was done after
ligating at the base (Figure 3). All other parts of bowel
(ileum, cecum) were normal (Figure 4). Thorough pelvic
toileting was done. Hernia repair was done with Bassini
technique. Abdomen was closed placing a pelvic drain.
Patient was fine postoperatively and was discharged on
fifth postoperative day.
DISCUSSION
Figure 2: Appendix confirmed after pulling it out.
The protrusion of a viscus or part of a viscus through
the walls of its containing cavity is defined as hernia.
Most commonly inguinal hernia contains small bowel or
omentum. It may display very unusual sac contents such
as ovary, fallopian tube, urinary bladder, incarcerated
bladder diverticula, large bowel diverticula with the form
Figure 3: Appendix ligated at the base before appendicectomy.
of diverticulitis or abscess, Meckel’s diverticulum (Littré

hernia) or foreign bodies (e.g., fish bones) [8–10]. The
presence of the appendix within an inguinal hernial sac is
Figure 1: Appendix tip found at the internal ring with pus after an uncommon condition and is referred to as Amyand’s
opening the sac. hernia. Amyand’s hernia is a broad term. It is used
In the absence of inflammation hernioplasty is

advocated. Regarding to remove or leave behind a
normal append is rather based on the patient’s age, life
expectancy, life-long risk of developing acute appendicitis
and the size and overall anatomy of the appendix.
Pediatric or adolescent patients have a significantly
higher risk of developing acute appendicitis and should
therefore have their appendices removed, compared to
middle-aged or elderly individuals in whom the appendix
should probably be left intact [15, 16].
When appendicitis is found, they should be treated
with appendectomy; prosthetic materials should not
be used for hernia repair. On the other hand, acute
appendicitis with peritonitis or acute appendicitis with
other pathology hernioplasty is contraindicated [15, 16].
The presence of pus or perforation of the organ is also
an absolute contraindication to the placement of a mesh
for hernia repair. Associated intra-abdominal abscesses,
if present, may be dealt with either percutaneously or
by open drainage. In our case since gush of pus came
out of hernia sac so the abdomen was opened to drain
out pus that might have trickled into pelvic cavity and
Figure 4: All other bowels were inspected for any pathology. appendicectomy was done. Hernia repair was done
without using a mesh since the environment was
contaminated.
variously to refer to occurrence of an inflamed appendix CONCLUSION

within an inguinal hernia, a perforated appendix within
an inguinal hernia or a non-inflamed appendix within an Left sided Amyand’s hernia is very rare. High index of
irreducible inguinal hernia [11]. The incidence of having a suspicion is required for diagnosis and this should be kept
normal appendix within the hernial sac varies from 0.5– as a differential diagnosis for such a varied presentation
1%, whereas only 0.1% of all cases of appendicitis present so that appropriate timely treatment can be instituted.
in an inguinal hernia [12, 13].
The pathophysiology of Amyand’s hernia is unknown. *********
It is not known whether primary visceral inflammation
(appendicitis) is the initiative feature or compression Author Contributions
of appendix may hamper blood supply and then leads Manash Ranjan Sahoo – Conception and design,
to secondary inflammation. Most of the cases occur on Acquisition of data, Analysis and interpretation of data,
the right side, the reasons being- (1) normal anatomical Drafting the article, Final approval of the version to be
position of the appendix (2) right-sided inguinal hernias published
are more common than left-sided hernias. There have Basavaraja C. – Conception and design, Acquisition of
been five reported cases of left sided Amyand’s hernia data, Analysis and interpretation of data, Drafting the
after an extensive search of literature [3–7]. None have article, Final approval of the version to be published
described appendicular abscess in left sided Amyand’s Anil Kumar T. – Conception and design, Acquisition of
hernia. We report here such a rare case. data, Analysis and interpretation of data, Drafting the
Acute appendicitis or perforation of the appendix article, Critical revision of the article, Final approval of
within the sac simulates perforation of the intestine within the version to be published
the hernia, and does not have specific symptoms or signs. Sunil Jaiswal – Conception and design, Acquisition of
Due to these facts, it is very difficult to reach a clinical data, Analysis and interpretation of data, Drafting the
diagnosis of Amyand’s hernia preoperatively. We can article, Final approval of the version to be published
have such a differential diagnosis for right sided hernia
but when similar thing happens on the left side it is very Guarantor
difficult to diagnose and a very high index of suspicion The corresponding author is the guarantor of submission.
is needed for the diagnosis. Although, preoperative
computed tomography (CT) scan of the abdomen may be Conflict of Interest
helpful in reaching the correct diagnosis [14], however, Authors declare no conflict of interest.
CT scan is not routinely used in such cases.
Copyright 8. Gurer A, Ozdogan M, Ozlem N, Yildirim A, Kulacoglu

© 2014 Manash Ranjan Sahoo et al. This article is H, Aydin R. Uncommon content in groin hernia sac.
distributed under the terms of Creative Commons Hernia 2006;10(2):152–5.
9. Greenberg J, Arnell TD. Diverticular abscess
Attribution License which permits unrestricted use,
presenting as an incarcerated inguinal hernia. Am
distribution and reproduction in any medium provided Surg 2005;71(3):208–9.
the original author(s) and original publisher are properly 10. Casadio G, Chendi D, Franchella A. Fishbone
credited. Please see the copyright policy on the journal ingestion: Two cases of late presentation as pediatric
website for more information. emergencies. J Pediatr Surg 2003;38(9):1399–400.
11. Bakhshi GD, Bhandarwar AH, Govila AA. Acute
appendicitis in left scrotum. Indian J Gastroenterol
REFERENCES 2004;23(5):195.
12. Logan MT, Nottingham JM. Amyand’s hernia: A case
1. Orr KB. Perforated appendix in an inguinal hernial report of an incarcerated and perforated appendix
sac: Amyand’s hernia. Med J Aust 1993;159(11- within an inguinal hernia and review of the literature.
12):762–3. Am Surg 2001;67(7):628–9.
2. Hutchinson R. Amyand’s hernia. J R Soc Med 13. Thomas WE, Vowles KD, Williamson RC.
1993;86(2):104–5. Appendicitis in external herniae. Ann R Coll Surg
3. Gupta S, Sharma R, Kaushik R. Left-sided Amyand’s Engl 1982;64(2):121–2.
hernia. Singapore Med J 2005;46(8):424–5. 14. Luchs JS, Halpern D, Katz DS. Amyand’s hernia:
4. Breitenstein S, Eisenbach C, Wille G, Decurtins M. Prospective CT diagnosis. J Comput Assist Tomogr
Incarcerated vermiform appendix in a left-sided 2000;24(6):884–6.
inguinal hernia. Hernia 2005;9(1):100–2. 15. Losanoff JE, Basson MD. Amyand hernia: What
5. Carey LC. Acute appendicitis occurring in hernias: A lies beneath--a proposed classification scheme to
report of 10 cases. Surgery 1967;61(2):236–8. determine management. Am Surg 2007;73(12):1288–
6. Johari HG, Paydar S, Davani SZ, Eskandari S, Johari 90.
MG. Left-sided Amyand hernia. Ann Saudi Med 16. Losanoff JE, Basson MD. Amyand hernia: A
2009;29(4):321–2. classification to improve management. Hernia
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Physicians Surg Pak 2010;20(7):480–1.

Int J Case Rep Images 2014;5(9):650–655. Katewa et al. 650
Mixed connective tissue disorder: A case report

Rishi Katewa, Jakhar R. S., Barala G.L.
Abstract aggressively as most deaths in mixed connective

tissue disorders are due to heart failure caused
Introduction: Mixed connective tissue disorder by pulmonary arterial hypertension.
is rarely reported in India. It is a disease with
overlapping features of many connective tissue Keywords: Mixed connective tissue disorder
disorders and the presence of anti-U1RNP. We (MCTD), Raynaud’s phenomenon, Sclerodactyly,
present a case with dyspnea and generalized Anti-U1RNP antibody, Scleroderma
swelling as the initial presentation that led to the
diagnosis of mixed connective tissue disorder. How to cite this article
Case report: A 60-year-old female came with
initial complains of progressive dyspnea limiting Katewa R, Jakhar RS, Barala GL. Mixed connective
her daily activities, palpitations, generalized tissue disorder: A case report. Int J Case Rep Images
body swelling since 15 days and fever since one 2014;5(9):650–655.
week. Detailed history and physical examination
revealed bluish discoloration of fingers on doi:10.5348/ijcri-2014116-CR-10427
exposure to low temperature, dysphagia and
difficulty opening the mouth and sclerodactyly.
Laboratory workup and radiography concluded
the diagnosis of mixed connective tissue
disorder. Pulmonary function tests suggested Introduction
restrictive lung disease and two-dimensional
echo showed pulmonary hypertension. Our Mixed connective tissue disease (MCTD) is an
patient followed both the Alarcon-Segovia’s autoimmune disease which was first described in
criteria and Kasukawa diagnostic criteria 1972 by Sharp et al. as a disease syndrome with
for mixed connective tissue disease (MCTD). overlapping features of systemic sclerosis, systemic lupus
Conclusion: Pulmonary hypertension presents erythematosus (SLE) and polymyositis [1]. Therefore,
late in the illness when other clinical signs MCTD is sometimes referred to as an overlap disease. The
are easily recognizable and should be treated initial presentation of the patients usually comprises non-
specific signs such as swollen digits, arthralgia, myalgia
or muscle weakness, acid reflux or dysphagia, Raynaud’s
Rishi Katewa1, Jakhar RS2, Barala GL3
phenomenon, shortness of breath on activity, a general
Affiliations: 1MBBS, Medical Officer, General Medicine,
malaise and fatigue. Over a period of time the symptoms
Barala Hospital and Research Center, Jaipur, Rajasthan,
India; 2MBBS, MD, Attending Physician, General Medicine,
are dominated by symptoms of either of one of the three
Barala Hospital and Research Center, Jaipur, Rajasthan, illness along with high titers of anti-U1RNP antibody.
India; 3MBBS, MD, Attending Physician and Director, Barala The etiology of the mixed connective tissue disorder
Hospital and Research Center, Jaipur, Rajasthan, India. is unknown but being an autoimmune disease MCTD
Corresponding Author: Dr. Rishi Katewa, 117, Everest Vihar, can run within families and is known to affect women
Kings Road, Jaipur, Rajasthan, Postal Code- 302019, India; more than men. No causal association and the varied
Ph: +919571069175; Email: rishikatewa88@gmail.com presentation, makes the diagnosis of this rare condition
difficult.
We encountered one such case with dyspnea and
Received: 23 May 2014 generalized swelling as the initial presentation that
Accepted: 27 June 2014 eventually led to the diagnosis of MCTD disorder with
Published: 01 September 2014 restrictive lung disease and pulmonary hypertension.
CASE REPORT The management of the patient included:

The patient was started on injection amoxicillin/
A 60-year-old female was admitted with complaints clavulinic acid 1.2 g 1-1-1(three times a day) and
of dyspnea on exertion, intermittent fever without chills paracetamol SOS for fever. Later the following drugs
since last seven days. Dyspnea was limiting her daily were added to the management:
activities and fever was mild, low grade, without chills • A combination of furosemide 20 mg +
and rigors, responded to antipyretics. Since 15 days, she spironolactone 40 mg 1-1-0
also had swelling all over the body and pedal edema used • Sildenafil citrate 50 mg bid
to increase with her daily activities. • Syrup muciane gel 2 tsp 1-1-1 (three times a day)
On enquiry she gave history of painful bluish for dyspeptic symptoms.
discoloration of fingers while washing hands with water • Oral steroids. Prednisone 40 mg tapered every
or in cold temperature (Figure 1) since last two years, five days to 30 mg, 20 mg, 10 mg, 5 mg. 1-0-0.
difficulty in opening mouth from last one year and • Metoprolol 12.5 mg 1-0-0.
dysphagia with dyspeptic symptoms since two months. • Supplemental oxygen was given for dyspnea.
On examination there was mild pallor and The patient was discharged after 22nd day of
temperature of 98.90F. She had generalized edema with hospitalization and is currently on sildenafil citrate 50
diffuse alopecia. Facial skin was taut with pinched up mg bid.
nose. Mouth orifice was small with difficulty in opening.
Fingers showed peripheral cyanosis and sclerodactyly.
Physical examination per abdomen showed
hepatomegaly 3 cm below costal margin, non-tender,
firm. Cardiovascular examination showed normal heart
sounds with soft systolic murmur at apex and pulmonary
area with same intensity. However, respiratory and
central nervous system examination provided no physical
findings.
On investigating hemoglobin was 9.8 g/dL with
platelet count 1.2x105 mm3. Urine (routine and
microscopic) was normal. Liver function test revealed
ALT of 94 IU/L, however, renal function test was normal
(Table 1). Serum CPK levels were raised (942 IU/L).
Two-dimensional echo showed moderate to severe
pulmonary hypertension and X-ray showed cardiomegaly
(Figure 2). Pulmonary function test was suggestive of
severe restrictive disease. Esophagogastroduodenoscopy
revealed multiple small gastric ulcers with evidence of old
hemorrhages in the centre at the antrum suggestive of
vasculitic lesion. Antral biopsy was suggestive of a single
superficial erosive patch. Skin biopsy showed features
of scleroderma with dermis showing mild increase in
the amount of collagen with few congested blood vessels
(Figure 3). Our patient followed the Alarcon-Segovia
diagnostic criteria (Table 2) with positive serology and
three of the five clinical criteria’s namely Raynaud’s
phenomenon, edema of hands and myositis depicted
by high serum CPK value. Our patient also followed the
Kasukawa diagnostic criteria (Table 3) with both the
common symptoms of Raynaud’s and swollen hands
being present associated with positive serology and mixed Figure 1: (A, B): Bluish discoloration of fingers on exposure to
findings of thrombocytopenia, esophageal dysmotility cold water.
(Figure 4), sclerodactyly and high CPK values.
Serological examination showed anti-mRNP/Sm and
anti-Ro52 positive. However anti-nuclear antibody and
ScL-70, Pm-Scl, Jo-1, CENP-B, dsDNA, anti-histone, DISCUSSION
anti-nucleosome, anti-ribosomal-P protein all were
negative (Table 2). The initial history of Raynaud’s phenomenon are
A diagnosis of MCTD was made. generalized swelling and dysphagia. Physical examination
findings of sclerodactyly and microstomia guided our
Table 1: Laboratory examination of the patient Table 3: Kasukawa diagnostic criteria for mixed connective
tissue disease
Hb 9.8 g/dL
1) Common Symptoms
TLC 6500/mm3 Raynaud´s Phenomenon
Swollen fingers or hands
Peripheral blood smear Normocytic normochromic 2) Presence of Anti U1 RNP
3) Mixed findings
MCV 94 fl
A. Systemic lupus erythematosus (SLE) like
MCH 30.7 pg Polyarthritis
Pericarditis/pleuritis
MCHC 32.7 g/dL Lymphadenopathy
Facial erithema
Reticulocyte count 0.5% Leucopenia/thrombocytopenia
B. Scleroderma like
Platelet count 120000/mm3
Sclerodactyly
Serum bilirubin Pulmonary fibrosis
Esophageal dysmotility
Total 1.0 mg% C. Polymyositis like
Muscle weakness
Direct 0.4 mg% High creatine phosphokinase (CPK)
Myophatic electromyogram (EMG)
ALT 94 IU/L
Requirement for diagnosis: At least one common symptom,
with positive U1RNP antibodies and one or more findings
ALP 43 IU/L
in at least two of the three categories A, B, and C.
Urea 35 mg%
Serum Creatinine 1.2 mg%

Table 4: Serological examination of the patient
Serum Sodium 130 mmol/L ANTIBODY RESULT
ANA negative
Serum Potassium 4.0 mmol/L
SS-a negative
Urine albumin +1 SS-b negative
Total CPK 942 IU/L Scl-70 negative

Anti-mRNP/Sm positive
Anti HCV negative Pm-Scl negative
HIV/HBsAg negative Jo-1 negative
CENP-B negative
Antimicrosomal antibody 31 IU/mL (negative-<34 IU/
mL) Anti-Ro52 positive
PNCA negative
T3 54 ng/dL (60-200)
ds-DNA negative
T4 6.3 mg/dL (4.5-12.0) Anti-histone negative
Anti-nucleosome negative
TSH 11.79 uIU/mL (0.30-5.5)
Anti-ribosomal P Protein negative
AMA-M2 negative
Table 2: Alarcon-Segovia diagnostic criteria for mixed

connective tissue disease
Serologic Criteria Positive anti-U1RNP at hemagglutination

titer >1:1600
Clinical Criteria Edema of hands

Synovitis
Myositis
Raynaud’s
Acrosclerosis
Figure 4: Barium study. Filled phase showing dilatation of

esophagus with barium in thoracic oesophagus, however no
peristaltic wave was seen on dynamic study. No evidence of any
filling defect was seen. Suggestive of hypomotility disorder of
esophagus, likely to be scleroderma.
Figure 2: X-ray of chest posterior-anterior view had the following

findings. Soft tissues and bony thorax are normal. The trachea A 2011 Norwegian study showed a 3.8 per 100,000
is central. Left costophrenic and cardiophrenic angles are clear. prevalence of MCTD among adults, with an incidence
Right costophrenic angle appears blunted. Both domes of
of 2.1 per million per year [4]. Mean age of onset was
diaphragm are normal in position and contour. Both the hila
are normal in size, shape, position and density. The visualized 31.9 years and more than three quarters of patients were
lung fields are normal. Cardiac shadow is enlarged in size (CT females [5]. This confirms the rarity of the disease and a
ratio 54%). Impression: Cardiomegaly. predilection for female sex.
MCTD should be kept as a differential diagnosis
when overlapping signs of autoimmune diseases are
present such as swollen digits, arthralgia, myalgia or
muscle weakness, acid reflux or dysphagia, Raynaud’s
phenomenon, shortness of breath on activity, a general
malaise and fatigue. Many criteria have been described
to classify MCTD. Table 3 gives the Alarcon-Segovia’s
criteria and Table 4 gives the Kasukawa diagnostic
criteria for mixed connective tissue disease. Alarcon-
Segovia’s criteria are simple and comprises five clinical
manifestations in addition to the serological status [6].
Our patient showed the Alarcon-Segovia diagnostic
criteria with positive serology and three of the five clinical
criteria’s namely Raynaud’s phenomenon, edema of
hands and myositis depicted by Serum CPK value of 942
Figure 3: Skin biopsy was suggestive of mild hyperkeratosis of
IU/L.
the epidermis (thick arrow). The dermis showing mild increase
in the amount of collagen with few congested blood vessels (thin
Our patient also followed the Kasukawa diagnostic
arrow). No evidence of perivascular infiltrates and fibrinoid criteria with both the common symptoms of Raynaud’s
necrosis. and swollen hands being present associated with positive
serology and mixed findings of thrombocytopenia,
esophageal dysmotility, sclerodactyly and high CPK
values.
laboratory and radiologic workup and thereafter helped Myositis clinically presenting as muscle weakness
us to reach the diagnosis. is more common than laboratory increase in muscle
An MCTD is a rare disease with unknown etiology, enzymes but in our patient a frank increase is CPK was
but few cases have been reported after occupation of seen.
vinyl chloride [2], and even after breast augmentation Lung function tests, especially the single breath
surgeries [3]. There are familial cases of MCTD with diffusing capacity may be abnormal in many patients
increased instance of HLADR4 compared with controls. but only a small fraction of those are symptomatic,
meanwhile our patient had dyspnea as the presenting Conflict of Interest

complaint. Pulmonary artery hypertension is not Authors declare no conflict of interest.
common in early MCTD [7]. Pulmonary hypertension,
occurring secondary to interstitial lung disease is an Copyright
increasingly recognized complication and our patient was © 2014 Rishi Katewa et al. This article is distributed
suffering from pulmonary hypertension as diagnosed by under the terms of Creative Commons Attribution
two-dimensional echo. License which permits unrestricted use, distribution
Membranous glomerulopathy and mesangial and reproduction in any medium provided the original
proliferative glomerulonephritis are the main renal author(s) and original publisher are properly credited.
manifestations. Less common renal manifestations Please see the copyright policy on the journal website for
include diffuse proliferative glomerulonephritis, vascular more information.
or glomerular sclerosis. It is clinically manifested mainly
with proteinuria, rarely with hematuria [8]. Our patient
had +1 hematuria on urine examination. REFERENCES
The therapy for MCTD would combine a cocktail of
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Guarantor

Int J Case Rep Images 2014;5(9):656–660. Ibrahim 656
Primary ciliary dyskinesia (Kartagener syndrome) in a

38-year-old Egyptian male: A rare case
Motaz Badr Abdellatif Ibrahim
Abstract to remind physicians by Kartagener syndrome

despite being rare because early diagnosis is the
Introduction: Kartagener syndrome is a rare, key for those patients to avoid complications and
ciliopathic, autosomal recessive genetic disorder to give those patients a better lifestyle, another
with an estimated incidence of about 1 case conclusion is that our case is a fertile male which
per 32,000 live births. It always causes a triad in uncommon in such syndrome.
which is situs inversus, chronic sinusitis and
bronchiectasis. Case Report: We present a case Keywords: Kartagener syndrome, Bronchiectasis,
of a 38-year-old male patient complaining of Primary ciliary dyskinesia
productive cough and hemoptysis since 15 days,
headaches since one month and easily fatigability How to cite this article
two years ago. He suffered from similar complains
during childhood with episodic fever, worsening Ibrahim MB. Primary ciliary dyskinesia (Kartagener
of symptoms and he was wrongly diagnosed with syndrome) in a 38-year-old Egyptian male: A rare
tuberculosis. After 28 years, he complicated by case. Int J Case Rep Images 2014;5(9):656–660.
severe empyema leading to surgical intervention.
Laboratory workup was unremarkable except
doi:10.5348/ijcri-2014117-CR-10428
for leukocytosis (>12,000/mm3) and hypoxia,
semen analysis showed defective motility
(asthenozoospermia) with grade c while other
parameters were found normal including sperm
count (35 million per milliliter), morphology and Introduction
volume, chest X-ray and computed tomography
(CT) scan were done, and he was diagnosed Siewert described primary ciliary dyskinesia as a
with Kartagener syndrome. He took medical combination of situs inversus, chronic sinusitis, and
treatment in the form of antibiotics (augmantin, bronchiectasis in 1904 [1]. However, the clinical triad
40 mg/kg/day PO), antipyretics (ibuprofen, 1 of Kartagener syndrome was recognized by Manes
tablet PO q8h), mucolytics (guaifenesin, 100–400 Kartagener [1] in 1933 and that is why it bears his
mg PO q4h), inhaled bronchodilators (albuterol/ name until now. Kartagener syndrome is an autosomal
ipratropium, nebulizer solution: 3 mL inhaled recessive pattern in which its symptoms and signs are due
q6h) and a regular follow-up is done with chest to defective cilia motility. The frequency of Kartagener
X-ray. Conclusion: In this case report, we intend syndrome is 1 case per 32,000 live births.
Motaz Badr Abdellatif Ibrahim

Affiliations: Sixth Year Medical Student, Alexandria Faculty CASE REPORT
of Medicine, Alexandria, Egypt.
Corresponding Author: Motaz Badr Abdellatif Ibrahim, 293 A 38-year-old male presented to our hospital with a
Port Said Street, apartment 14, Sidigaber, Alexandria, Egypt. few days’ history of chest pain, productive cough with
21131; Ph: +201002356766; Email: motazmd@hotmail.com yellowish sputum and significant shortness of breath.
He retired two years ago from his job because of his easy
fatigability and persistent productive cough forced him
Received: 10 June 2014 to do so. He denied any history of smoking. No family
Accepted: 11 July 2014 history of such a case in his first degree relatives.
The condition started since he was 10-year-old with

recurrent pneumonia and bronchiectasis in which he was
wrongly diagnosed with tuberculosis so the doctor gave
him antituberculous drugs (a 6-month course of isoniazid
(INH) and rifampin, supplemented during the first two
months with pyrazinamide) and antibiotics (augmantin)
after which he was sent home after relieving.
After 28 years, he suffered from worsening of the
symptoms and hemoptysis in which he was admitted
to the hospital and diagnosed with severe empyema in
which he underwent surgical decortication on the right
side and application of chest tube for drainage to treat his
severe empyema.
On general examination, it was found that his vital signs
were: respiratory rate 28/min, blood pressure 110/70
mmHg, and temperature 37.4°C. Chest examination
revealed on inspection limited chest movement more
on the right side, apical pulsations on the right fifth Figure 1: Chest X-ray revealed a shifted mediastinum to the
intercostals space in the midclavicular line no any other right side, old bronchiectatic areas at the bases of the right lung,
hyperinflated left lung and dextrocardia.
pulsations noticed. By palpation a tracheal shift to the
right side was found with a tactile vocal fremitus more on
the right side. By auscultation diffuse ronchi, diminished
air movement in both lungs and heart sounds were more
pronounced at the right sternal border. Other physical
examination findings were unremarkable.
Laboratory workup was unremarkable except for
leukocytosis (>12,000 per mm3) and hypoxia, semen
analysis showed defective motility (asthenozoospermia)
with grade c (non-progressive motility because they
do not move forward despite the fact that they move
their tails. Sometimes also denoted motility II), other
parameters were found normal including sperm count
(35 million per milliliter), morphology and volume.
The patient is fertile with three children by the help of
assisted reproductive techniques.
Chest X-ray and computed tomography (CT) scan
revealed a shifted mediastinum to the right side; the
right lung field is totally replaced by cystic bronchiectatic
changes showing thickened bronchial wall, hyperinflated
left lung, dextrocardia and the stomach was normally on
the left side due to the presence of a stomach bubble on
X-ray which is confirmed by CT scan of abdomen (Figures
1 and 2).
The clinical diagnosis of Kartagener syndrome was
made and the condition was explained to the patient
and he was treated with antibiotics (augmantin, 40 mg/
kg/day PO), antipyretics (ibuprofen, 1 tablet PO q8h),
mucolytics (guaifenesin, 100–400 mg PO q4h), inhaled
bronchodilators (albuterol/ipratropium, nebulizer
solution: 3 mL inhaled q6h) and a regular follow-up is
done with chest X-rays.
The follow-up showed a good response to the medical
treatment and the patient went back to his job and he is
having a better lifestyle. Figure 2: Computed tomography scan of chest revealed a shifted
mediastinum to the right side, the right lung field is totally
replaced by cystic bronchiectatic changes showing thickened
bronchial wall; hyperinflated left lung, scattered areas of
consolidative changes in the left lung, dilated pulmonary artery
(3–4 cm) and dextrocardia.
DISCUSSION Multiple new genes were discovered related to this

syndrome based on the theory which states the existence
Kartagener syndrome is a primary ciliary motility of additional ciliary mutations which do not manifest
disorder in which the majority of the patients present in those patients. This is why future genetic testing will
with situs inversus, chronic sinusitis and bronchiectasis be one of the main diagnostic tools in diagnosing those
caused by pseudomonal infection [2, 3]. patients [6].
When taking a history, the patient mainly complains The treatment regimen for those patients includes:
of chronic cough with unexplained respiratory distress antibiotics with good pseudomonal coverage, daily
and he/she always describes this cough as wet and chest physiotherapy. Dnase, hypertonic saline and
productive, found in nearly all of infants [4]. In addition acetylcysteine may be tried in patients with recurrent
to the defective drainage of the sinonasal system in those infections and respiratory symptoms [7]. Surgical
patients, this will lead to congestion, rhinorrhea, and intervention for those patients can be beneficial in
chronic middle ear effusions with suppurative otorrhea. complicated bronchiectasis and when the disease is
Imaging for those patients will reveal situs inversus localized as in our case [8].
abnormalities which are relatively specific for Kartagener
syndrome [5].
The initial diagnostic workup is started after proper CONCLUSION
history taking and physical examination.
In Kartagener syndrome, postpubescent males will Kartagener syndrome must be in the differential
have abnormal sperm motility (asthenozoospermia) diagnostic list of conditions leading to bronchiectasis
which will be an obstacle in the future for those patients in pediatric age group including (foreign body, tumor,
if they want children. However, this problem is solved by lymphadenopathy, chronic obstructive pulmonary
the new techniques of assisted reproductive techniques disease (COPD), immunodeficiency states with recurrent
nowadays as in our case. infections, IgG and IgA deficiencies, alpha-1antitrypsin
Despite the newly multiple diagnostic tests that deficiency, recurrent aspiration pneumonia, inhalation
emerged there is no full standard test to diagnose this of poisonous dust and fumes) despite being rare because
syndrome and as an example of those tests: early diagnosis is the key for those patients to avoid
1. Electron microscopy, which visualizes the ciliary complications and to give those patients a better lifestyle,
ultrastructure. The samples are obtained by nasal another conclusion from our case is that our patient is a
scrape or brush biopsy. fertile male which is uncommon in such syndrome.
2. High-speed videomicroscopy is used in some
research centers for a better observation of the *********
ciliary beats [5].
3. Nasal nitric oxide measurement is measurable in Acknowledgements
nasal air of normal subjects and found to be low The pulmonology department staff members in Faculty
in cystic fibrosis and very low in primary ciliary of medicine Alexandria University for their cooperation
dyskinesia. Recently, it was suggested to play an with us by allowing us to take the history and physical
important role in regulating ciliary motility. examination of the admitted patient.
4. The immunofluorescence diagnosis of primary
ciliary dyskinesia, various components (proteins) Author Contributions
of cilia structure is stained by specific antibodies. Motaz Badr Abdellatif Ibrahim – Substantial
The cross section of a normal respiratory tract contributions to conception and design, Acquisition of
cilium shows the typical anatomy of the axoneme, data, Analysis and interpretation of data, Drafting the
which consists of nine peripheral microtubule article, Revising it critically for important intellectual
doublets that surround two centrally located single content, Final approval of the version to be published
microtubules (9+2 structure). The peripheral
microtubule doublets are interconnected with Guarantor
each other by inner and outer dynein arms and The corresponding author is the guarantor of submission.
to the central microtubules by radially oriented
spokes. The outer and inner dynein arms are Conflict of Interest
protein complexes of various dynein chains and Authors declare no conflict of interest.
associated proteins. A mutation in one of the genes
that encodes a component of the dynein arms often Copyright
results in a total loss of dynein arm structure. © 2014 Motaz Badr Abdellatif Ibrahim. This article
In addition to the previously mentioned diagnostic is distributed under the terms of Creative Commons
tests, there is a large expansion into the field of genetic Attribution License which permits unrestricted use,
testing and isolation of Kartagener syndrome mutations. distribution and reproduction in any medium provided
the original author(s) and original publisher are properly
credited. Please see the copyright policy on the journal SUGGESTED READINGS
website for more information.
• Afzelius BA, Stenram U. Prevalence and genetics of
immotile-cilia syndrome and left-handedness. Int J
REFERENCES Dev Biol. 2006;50(6):571–573.
• Kartagener M. Zur pathogenese der bronchiectasien.
1. Kartagener M. Zur pathogenese der bronchiectasien. I Mitteilung:bronchiectasien bei situs viscerum
I Mitteilung: Bronchiectasien bei situs viscerum inversus. Betr Klin Tuberk. 1933;83:498–501.
inversus. Betr Klin Tuberk 1933;83:498–501. • Knowles MR, Daniels LA, Davis SD, Zariwala MA,
2. Kollberg H, Mossberg B, Afzelius BA, Philipson K, Leigh MW. Primary ciliary dyskinesia. Recent
Camner P. Cystic fibrosis compared with the immotile- advances in diagnostics, genetics, and characterization
cilia syndrome. A study of mucociliary clearance, of clinical disease. Am J Respir Crit Care Med. Oct 15
ciliaryultrastructure, clinical picture and ventilatory 2013;188(8):913–22.
function. Scand J Respir Dis 1978;59(6):297–306. • Kollberg H, Mossberg B, Afzelius BA, Philipson K,
3. Afzelius BA, Stenram U. Prevalence and genetics of Camner P. Cystic fibrosis compared with the immotile-
immotile-cilia syndrome and left-handedness. Int J cilia syndrome. A study of mucociliary clearance,
Dev Biol 2006;50(6):571–3. ciliary ultrastructure, clinical picture and ventilatory
4. Knowles MR, Daniels LA, Davis SD, Zariwala MA, Leigh function. Scand J Respir Dis.1978;59(6):297–306.
MW. Primary ciliary dyskinesia. Recent advances in • Leigh MW, O’Callaghan C, Knowles MR. The
diagnostics, genetics, and characterization of clinical challenges of diagnosing primary ciliary dyskinesia.
disease. Am J Respir Crit Care Med 2013;188(8):913– Proc Am Thorac Soc. Sep 2011;8(5):434–7.
22. • Leigh MW, Pittman JE, Carson JL, Ferkol TW, Dell
5. Leigh MW, Pittman JE, Carson JL, et al. Clinical SD, Davis SD, et al. Clinical and genetic aspects of
and genetic aspects of primary ciliary dyskinesia/ primary ciliary dyskinesia/Kartagener syndrome.
Kartagener syndrome. Genet Med 2009;11(7):473– Genet Med. Jul 2009;11(7):473–87.
87. • Mossberg B, Afzelius B, Camner P. Mucociliary
6. Leigh MW, O’Callaghan C, Knowles MR. The clearance in obstructive lung diseases. Correlations to
challenges of diagnosing primary ciliary dyskinesia. the immotile cilia syndrome. Eur J Respir Dis Suppl.
Proc Am Thorac Soc 2011;8(5):434–7. 1986;146:295–301.
7. Ten Berge M, Brinkhurst G, Kroon AA, de Jongste • Ten Berge M, Brinkhurst G, Kroon AA, de Jongste
JC. DNase treatment in primary ciliary dyskinesia- JC. DNase treatment in primary ciliary dyskinesia
-assessment by nocturnal pulse oximetry. Pediatr – assessment by nocturnal pulse oximetry. Pediatr
Pulmonol 1999;27(1):59–61. Pulmonol.1999;27(1):59–61.
8. Mossberg B, Afzelius B, Camner P. Mucociliary • http://emedicine.medscape.com
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the immotile cilia syndrome. Eur J Respir Dis Suppl PMC3056330/#b10-ijgm-4-041
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About the Author
Article citation: Ibrahim MB. Primary ciliary dyskinesia (Kartagener syndrome) in a 38-year-old Egyptian male:
A rare case. Int J Case Rep Images 2014;5(9):656–660.
Motaz Badr Abdellatif Ibrahim is sixth year medical student at Alexandria Faculty of medicine,
Alexandria, Egypt. He is interested in the pediatrics research fileds especially in pediatric pulmonology,
gastroenterology, cardiology and congenital disorders.

Int J Case Rep Images 2014;5(9):661–664. Jehangir et al. 661
Clinical images OPEN ACCESS
Differentiating a simple cyst or metastatic breast cancer:

A medical dilemma
Waqas Jehangir, Zorawar Singh, Abdul I. Mahmad, Teena Mathew
CASE REPORT
A 60-year-old American Hispanic female presented
to the emergency room for gradual onset dizziness,
associated with lightheadedness, double vision and
nausea. She described her symptoms as vertigo-like.
She has a known past medical history of breast cancer
and was treated with surgery and chemotherapy. The
patient had been in remission for over six years. Patient
denied any recent travel outside United States. Physical
examinations of the patient were within normal limits,
apart from the dizziness. Patient did not complain of any
weight changes, vision changes, or any focal neurological
deficits.
Computed tomography (CT) scan of head (Figure
1) showed a large, 4.3 cm cystic mass projecting in the
mid-posterior aspect of the cerebellum causing anterior
displacement of the fourth ventricle. It was repeated again
and the results were similar. Her last CT scan of head
was done a year before which was completely normal. Figure 1: Computed tomography scan of head without contrast
Magnetic resonance imaging (MRI) scan of the brain showing a 4.3-cm cystic mass projecting in the mid-posterior
with and without contrast (Figure 2) showed multiple aspect of the cerebellum causing anterior displacement of the
ring enhancing intra-axial masses, the largest centered fourth ventricle.
at the vermis measuring nearly 4 cm and compressing
the fourth ventricle mildly. In the differential was either
a metastatic neoplasm from primary breast cancer or a
Waqas Jehangir1, Zorawar Singh2, Abdul I Mahmad1, Teena

Mathew3
Affiliations: 1MD, Raritan Bay Medical Center – Resident,
Internal Medicine, Raritan Bay Medical Center, Perth Amboy,
New Jersey, USA; 2Medical Student, Ross University School
of Medicine – 4th Year Medical Student, Ross University
School of Medicine, Portsmouth, Dominica, West Indies;
3
MD, Raritan Bay Medical Center – Attending Physician,
Internal Medicine, Raritan Bay Medical Center, Perth Amboy,
New Jersey, USA.
Corresponding Author: WaqasJehangir, MD, 530 New
Brunswick Ave, Perth Amboy, New Jersey, USA. 08861; Ph:
267-844-7119; Email: wjehangir@hotmail.com
Received: 05 May 2014 Figure 2: Magnetic resonance imaging scan of brain without
Accepted: 24 May 2014 contrast showing multiple ring enhancing intra-axial masses,
Published: 01 September 2014 the largest centered at the vermis measuring nearly 4 cm and
compressing the fourth ventricle mildly.
parasitic infection (Table 1). Since the patient did not had a normal head CT scan a year ago, this case shows
have any likely risk factors for parasitic infections, such how quickly a metastatic lesion can resurface and present
as any recent traveling or interactions with cats or cat itself. Breast cancer patients who achieve a pathological
feces, the likelihood of a parasitic infection was low. The complete response after neoadjuvant chemotherapy
patient was subsequently treated with radiotherapy with usually have a favorable prognosis [1–3]. From this
complete resolution of symptoms. case, we can learn that the breast cancer may have been
eradicated from the original source, but we should always
be suspicious of the malignancy re-emerging in other parts
DISCUSSION of the body. As physicians, we also must be aware of many
of the symptoms of metastasis in women who have been
This case illustrates the importance of suspecting eradicated of breast cancer, those symptoms include, but
a metastatic neoplasm in the brain in a patient who are not limited to, severe headaches, generalized fatigue,
presents with neurological abnormalities and has a past dizziness, vertigo, blurry vision, hypotension, difficulties
history of a malignancy. Although the patient had been in with balance and coordination, and even polyuria and
remission from breast cancer for more than six years, and polydipsia [4, 5].
Table 1: Differential diagnosis of cystic brain lesion

Differential Diagnosis Computed tomography Magnetic resonance imaging
T1 T2
Primary tumor/Neoplasm Hypodense with rim enhancement Hypointense Hypointense
Metastasis Hypodense with rim enhancement Hypointense Hypointense
Cerebral toxoplasma Ring enhancing lesions with Core hyperintense; Cavity hypointense; surrounding
abscess surrounding edema. Multilobulated surrounding hypointense hperintense edema
lesions may be seen edema
Neurocysticercosis Ring enhancement of capsule, Core, iso-to hypointense Core iso-to hypointense with
eccentric dot sign, thin regular outline hypointense rim
Hydatid cyst Well demarcated, isodense/hypodense Isointense Isointense
Arachnoid cyst Well circumscribed, same signal No contrast No contrast enhancement
intensity enhancement
Ependymal cyst Well-defined, thin-walled and do not Isointense nonenhancing Isointense nonenhancing wall
contrast enhance wall
Colloid cyst Hyperdense, hypo/isodense Hyperintense Hypointense
Epidermoid cyst Well-demarcated hypodense, enhance Isointense Isointense/Hyperintense
with contrast agents
Dermoid cyst Well-circumscribed, hypodense Hyperintense Hypo to hyper intense
masses
Neuroenteric cyst Iso-/hypodense lesions Iso-/slightly Hyperintense
hyperintense
CONCLUSION How to cite this article
For our patient, who presented with dizziness, we Jehangir W, Singh Z, Mahmad AI, Mathew T.
should always keep in mind that this simple symptom Differentiating a simple cyst or metastatic breast
warrants concern. As physicians, we must always cancer: A medical dilemma. Int J Case Rep Images
counsel our patients that they may not have the breast 2014;5(9):661–664.
cancer physically at the moment but there is always a
slight possibility for it to resurface in other parts of the
doi:10.5348/ijcri-201461-CL-10049
body.
*********
Acknowledgements License which permits unrestricted use, distribution

AbdallaYousif, MD, and reproduction in any medium provided the original
author(s) and original publisher are properly credited.
Author Contributions Please see the copyright policy on the journal website for
Waqas Jehangir – Substantial contributions to more information.
it critically for important intellectual content, Final REFERENCES
Zorawar Singh – Analysis and interpretation of data, 1. Tallet A. Brain metastases from breast cancer. Cancer
Revising it critically for important intellectual content, Radiother 2013;17(7):708–14. [Article in French].
2. Murrell DH, Foster PJ, Chambers AF. Brain
metastases from breast cancer: Lessons from
Abdul I. Mahmad – Analysis and interpretation of data, experimental magnetic resonance imaging studies
Revising it critically for important intellectual content, and clinical implications. J Mol Med (Berl)
Final approval of the version to be published 2014;92(1):5–12.
Teena Mathew – Analysis and interpretation of data, 3. Shimada K, Ishikawa T, Yoneyama S, et al.
Revising it critically for important intellectual content, Early-onset Brain Metastases in a Breast Cancer
Final approval of the version to be published Patient after Pathological Complete Response to
Neoadjuvant Chemotherapy. Anticancer research
Guarantor 2013;33(11):5119–21.
4. Peppa M, Papaxoinis G, Xiros N, Raptis SA,
Economopoulos T, Hadjidakis D. Panhypopituitarism
due to metastases to the hypothalamus and the
Conflict of Interest pituitary resulting from primary breast cancer:
Authors declare no conflict of interest. A case report and review of the literature. Clinical
Breast Cancer 2009;9(4):E4–7.
Copyright 5. Malin D, Strekalova E, Petrovic V, et al. aß-Crystallin:
© 2014 Waqas Jehangir et al. This article is distributed A Novel Regulator of Breast Cancer Metastasis to the
under the terms of Creative Commons Attribution Brain. Clinical Cancer Research 2014;20(1):56–7.
About the Authors
Article citation: Jehangir W, Singh Z, Mahmad AI, Mathew T. Differentiating a simple cyst or metastatic breast
cancer: A medical dilemma. Int J Case Rep Images 2014;5(9):661–664.
Waqas Jehangir is Internal Medicine Resident at Raritan Bay Medical Center, Perth Amboy, NJ. He
earned the Medical degree MBBS from Nishtar Medical College/University of Health Sciences, Lahore,
Pakistan. He has published 10 research papers in national and international academic journals. His
research interests include hematology and oncology. He intends to pursue fellowship in hematology/
oncology.
Zorawar Singh is a fourth year medical student at Ross University School of Medicine. He earned a
Bachelor’s of Science in Human Biology from Michigan State University and will receive his MD Degree
in May 2015 from Ross University. This is his first research paper in national journals. His research
interests include hematologic and oncologic disease states, and cardiovascular disease. He intends to
pursue a residency in Internal Medicine in the future.
Abdul I. Mahmad is Internal Medicine Resident at Raritan Bay Medical Center Perth
Amboy New Jersey. He earned the undergraduate degree MBBS from Deccan College
of Medical Sciences, Hyderabad, India and postgraduate degree MD Dermatology from
Kakatiya Medical College, Warangal, Telangana, India. His research interests include hepatology,
gastroenterology and dermatology. He intends to pursue fellowship in gastroenterology in future.

clinical images OPEN ACCESS
Multidrug resistant pyogenic liver abscesses: A rare but

fatal complication of a life-saving procedure
Waqas Jehangir, Shilpi Singh, Andrea A Lewis, Shuvendu Sen
CASE REPORT blood cell count of 35.7x103/mm3, platelets 3.27x105/mm3,

lactic acidosis, elevated AST, ALT, alkaline phosphatase,
An 84-year-old Korean female, nursing home resident, and total bilirubin. Additionally, a chest X-ray showed
was sent to the emergency department after she was bilateral basilar pneumonia and a small pleural effusion.
discovered to have an altered mental state, lethargy, and Based on the laboratory and radiographic findings,
low-grade fever. This patient recently underwent a biliary patient was admitted to ICU with an assessment of sepsis
stent placement secondary to ascending cholangitis. secondary to pneumonia and to rule out intra-abdominal
Since then, the patient had multiple uncomplicated infection and biliary tract infection. Additionally, patient
recurrent infections. Patient had a past medical history was started on linezolid and primaxin and a full septic
significant for hypertension, dementia, dysphagia, workup was initiated. Computed tomography scan of
deep vein thrombosis (DVT), and cerebral vascular the abdomen revealed multiple hypodense lesions along
accident (CVA) with right paresis. Her past surgical the dome/right hepatic lobe with the largest appearing
history included a G-tube placement, inferior vena cava to be multiloculated measuring 6.8x6 cm in the greatest
(IVC) filter placement, right ureteral stent placement, dimension (Figure 1). Hepatic abscesses versus metastatic
craniotomy, cholecystectomy, ventriculoperitoneal disease was in consideration. A possible removal of the
shunt, and left cataract extraction. Patient’s vital signs biliary stents appeared to be technically not feasible
included tachycardia, tachypnea, with a blood pressure because of the patient’s hemodynamic instability. A
of 134/71 mmHg. Patient was slightly febrile, and with a
saturation of 95% on room air. On physical examination,
patient was toxic appearing, awake, yet drowsy and
confused. Additionally, patient grimaced to tenderness
over the right upper quadrant and had generalized
weakness in all extremities, more pronounced on right
side. Furthermore, patient was found to have bilateral
harsh breath sounds, basilar crackles, and decrease
breath sounds. Her extremities showed trace edema.
Laboratory tests revealed hemoglobin 12.4 g/dL, white
Waqas Jehangir1, Shilpi Singh1, Andrea A Lewis2, Shuvendu

Sen3
Affiliations: 1MD, Resident, Department of Internal Medicine,
Raritan Bay Medical Center, Perth Amboy, NJ, USA; 2BS,
Physician Assistant Student, Rutgers-School of Health
Related Professions, Piscataway, NJ, USA; 3MD, Associate
Program Director Department of Internal Medicine, Raritan
Bay Medical Center, Perth Amboy, NJ, USA.
Corresponding Author: Waqas Jehangir, 530 New Brunswick
Ave, Perth Amboy, NJ, USA 08861; Ph: +1 267-844-7119;
Email: wjehangir@hotmail.com
Figure 1: Computed tomography scan of the abdomen without

Received: 28 April 2014
contrast showing multiple hypodense lesions along the dome/
right hepatic lobe with the largest appearing to be multi-
loculated measuring 6.8x6 cm in the greatest dimension.
computed tomography scan-guided drainage of large it critically for important intellectual content, Final
abscesses was then scheduled. Cultures of the abscess approval of the version to be published
drainage showed Enterococcus faecium-Group D, Shilpi Singh – Analysis and interpretation of data,
Vancomycin-Resistant (VRE), Morganella morganii, and Revising it critically for important intellectual content,
Pseudomonas aeruginosa. Patient was then started on Final approval of the version to be published
tigecycliine, amikacin, and linezolid based on sensitivity. Andrea A Lewis – Analysis and interpretation of data,
However, the patient did not survive and died of septic Revising it critically for important intellectual content,
shock secondary to multidrug resistant pyogenic liver Final approval of the version to be published
abscess, a complication of biliary stent placement. Shuvendu Sen – Analysis and interpretation of data,
DISCUSSION
Guarantor
Pyogenic liver abscesses (PLA) are rare but can be The corresponding author is the guarantor of submission.
a potentially life-threatening condition [1]. The most
common cause in recent data of PLA is biliary disease, Conflict of Interest
which is more prevalent in women. Additional underlying Authors declare no conflict of interest.
or concomitant causes include but are not limited to:
hypertension, intra-abdominal infection, diabetes, Copyright
malignancy, cardiovascular disease, alcohol abuse and © 2014 Waqas Jehangir et al. This article is distributed
cirrhosis, diverticulitis, and inflammatory disease [2]. under the terms of Creative Commons Attribution
Patients with PLA often present to the emergency License which permits unrestricted use, distribution
department with a fever of unknown origin and after and reproduction in any medium provided the original
a thorough assessment, radiographic studies may be author(s) and original publisher are properly credited.
initiated to aid in finding the origin of the fever [3]. Once Please see the copyright policy on the journal website for
a PLA is diagnosed, treatment via percutaneous drainage more information.
is indicated. Surgery can be an alternative choice.
Isolated pathogens of PLA are typically gram-negative
bacteria in which Escherichia coli was thought to be the REFERENCES
most common usually culprit of pyogenic liver abscesses
detected by computed tomography, but new data suggest 1. Gungor G, Biyik M, Polat H, Ciray H, Ozbek O,
Klebsiella pneumonia has become the principal etiology Demir A. Liver abscess after implantation of dental
of PLA [2]. prosthesis. World J Hepatol 2012;4(11):319–21.
2. Rahimian J, Wilson T, Oram V, Holzman RS.
Pyogenic liver abscess: Recent trends in etiology and
mortality. Clin Infect Dis 2004;39(11):1654–9.
CONCLUSION 3. Golia P, Sadler M. Pyogenic liver abscess:
Klebsiella as an emerging pathogen. Emerg Radiol
In our case, the patient developed a multidrug 2006;13(2):87–8.
resistant pyogenic liver abscess, which eventually proved
to be fatal.
How to cite this article
Jehangir W, Singh S, Lewis AA, Sen S. Multidrug

resistant pyogenic liver abscesses: A rare but fatal
complication of a life-saving procedure. Int J Case
Rep Images 2014;5(9):665–667.
doi:10.5348/ijcri-201462-CL-10050
*********
Waqas Jehangir – Substantial contributions to
About the Authors
Article citation: Jehangir W, Singh S, Lewis AA, Sen S. Multidrug resistant pyogenic liver abscesses: A rare but
fatal complication of a life-saving procedure. Int J Case Rep Images 2014;5(9):665–667.
Waqas Jehangir is Internal Medicine Resident at Raritan Bay Medical Center, Perth Amboy, NJ. He
earned the Medical degree MBBS from Nishtar Medical College/University of Health Sciences, Lahore,
Pakistan. He has published 10 research papers in national and international academic journals. His
research interests include hematology and oncology. He intends to pursue fellowship in hematology/
oncology.
Shilpi Singh is Internal Medicine Resident and Chief Resident at Raritan Bay Medical Center, Perth
Amboy, New Jersey, USA. She earned the undergraduate degree MBBS from Himalayan Institute of
Medical Sciences, Dehradun, India. She also has Masters in Public Health from University of New
England Maine. She has published multiple research papers in national and international academic
journals. Her research interest includes, endocrinology, pulmonary, cardiology and hematology, and
oncology. She Intends to pursue fellowship in pulmonary/critical care.
Andrea Lewis is enrolled in the dual master degree program of Physician Assistant/Public Health and
Rutgers University in Piscataway, New Jersey. She earned her BS in Microbiology from the University
of Louisiana at Lafayette. Her area of interest includes weight loss and nutrition. Her masters project
studied weight loss advertisements in women’s health and fitness magazines. After graduation, she
plans on working in primary care and being over a community outreach program.
Shuvendu Sen is MD, Associate Program Director Department of Internal Medicine, Raritan Bay
Medical Center, Perth Amboy, NJ, USA.

Int J Case Rep Images 2014;5(9):668–670. Boufettal et al. 668
Letter to Editors OPEN ACCESS
Trapezo-metacarpal dislocation diagnosed as sprain

Monsef Boufettal, Rida-Allah Bassir, Mohamed S. Berrada,
Moradh El Yaacoubi
To the Editor,
The trapezo-metacarpal dislocation is a rare injury,
sometimes misunderstood, of which the diagnosis is
essentially radiographic. We report a case of a 22-year-
old athlete presented following a fall onto an outstretched
hand during a handball match,pain with edema of the right
thumb without obvious deformation. Clinically, we found
pain on palpation and mobilization of the right thumb
making examination very difficult. Plain radiography of
the right hand failed to reveal a fracture. However, the
trapezo-metacarpal dislocation went unnoticed (Figure
1). The diagnosis of a severe sprain trapezo-metacarpal
was unfortunately retained and the patient has benefited
from an immobilization with a thumb orthesis associated Figure 1: The trapezo-metacarpal dislocation went unnoticed in
with an analgesic and an anti-inflammatory. Due to the the first plain radiograph.
persistence of pain and the appearance of a deformation,
another plain radiography were performed one week
later which objectified an unstable trapezo-metacarpal
dislocation (Figure 2) requiring surgical management
by the realization of a pinning, associated with
immobilization for three weeks (Figure 3). The pin was
withdrawn after three weeks and reeducation was started
quickly with a good clinical outcome.
The trapezo-metacarpal dislocations are not
uncommon but deserve special attention, and may
sometimes go unnoticed. Their diagnosis is essentially
Monsef Boufettal1, Rida-Allah Bassir1, Mohamed S. Berrada2,

Moradh El Yaacoubi2
Affiliations: 1MD, Orthopedic Surgery Department of Ibn
Sina Hospital, University Mohamed V, Rabat, Morocco;
2
MD, Orthopedic Surgery Department of Ibn Sina Hospital,
University Mohamed V, Rabat, Morocco.
Corresponding Author: Dr. Monsef Boufettal, Postal Address:
N2 Bloc T, Rue Babiana, Secteur 19 Hay Riad Rabat,
Morocco. Institution: Orthopedic Surgery Department of Ibn
Sina Hospital, Rabat, Morocco; Tel: (00212) 668270041
Email: moncef.bof@gmail.com
Received: 23 May 2014

Accepted: 26 June 2014 Figure 2: Plain radiography of the right thumb showing trapezo-
Published: 01 September 2014 metacarpal dislocation.
IJCRI – International Journal of Case Reports and Images, Vol. 5 No. 9, September 2014. ISSN – [0976-3198]
Rida-Allah Bassir – Substantial contributions to

Mohamed S. Berrada – Substantial contributions to
Moradh El Yaacoubi – Substantial contributions to
Guarantor
Figure 3: Postoperative radiographic control after pinning of
the trapezo-metacarpal joint. Conflict of Interest
Authors declare no conflict of interest.
Copyright
radiological; we must insist on the rigor of radiological © 2014 Monsef Boufettal et al. This article is distributed
incidences and slightest doubt, we should have recourse under the terms of Creative Commons Attribution
to a dynamic test [1]. The complexity of the joint system, License which permits unrestricted use, distribution
capsular ligament and neuromuscular system of the and reproduction in any medium provided the original
trapezo-metacarpal joint complicates diagnosis and author(s) and original publisher are properly credited.
requires introducing therapies that do not suffer of any Please see the copyright policy on the journal website for
approximation [2]. The clinical signs are discreet, rapidly more information.
masked by edema and moderate functional impotence
which makes a clinical diagnosis very difficult [3]. The
concept of trapezo-metacarpal dislocation easily reducible REFERENCES
but extremely unstable with iterative dislocation is classic.
1. Pequignot JP, Giordano P, Boatier C, Allieu Y.
The aim of treatment is the restoration of congruency and Luxation traumatique de la trapézo-métacarpienne.
stability of joint. Any imperfection may induce to chronic In Annales de Chirurgie de la Main 1988;7:14–24.
instability and a secondary to rhizarthrosis [4]. 2. Aude Q, Jean Claude C, Raymond-Gilbert D. 23
ENTORSES ET LUXATIONS. Rééducation de
How to cite this article l’appareil locomoteur: Membre supérieur 2008;2:329.
3. Jaaouane M. “La luxation trapézo-métacarpienne (A
Boufettal M, Bassir RA, Berrada MS, Yaacoubi ME. propos de 06 cas) 2010.
4. Amar MF, Loudyi D, Chbani B, Daoudi A, Boutayeb F.
Trapezo-metacarpal dislocation diagnosed as sprain.
Acute traumatic dislocation of the trapeziometacarpal
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doi:10.5348/ijcri-201455-LE-10015
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Contents Vol. 5, No. 9 (September 2014)

Cover Image 642 A huge retroperitoneal lymphatic cyst

Review Article 646 Appendicular abscess in left inguinal hernia

Review Article OPEN ACCESS

Abstract to diagnose or treat a disease based on the

Table 1: Reactor isotopes used for diagnostic purpose [9, 10]

Isotope Half -life Uses

Table 2: Reactor isotopes used in therapeutics [9, 10]

Dysprosium-165 2 hours Used as an aggregated hydroxide for synvectomy treatment of arthritis.

Phosphorus-32 14 days Used in the treatment of polycythemia vera.

Table 3: Cyclotron Radioisotopes

********* 8. Agarwal BS and Kedar Nath Ram Nath. Meerut (1996);

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CASE series OPEN ACCESS

Laparoscopic plication and mesh repair for diastasis recti:

Abstract Keywords: Diastasis recti, Laparoscopic plication,

previous pregnancies. Two females had three children and

Figure 4: Reinforcing the plication using tissue separating mesh

Figure 2: Continuous suturing of the two separated rectus

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CASE REPORT OPEN ACCESS

Cutaneous metastatic breast adenocarcinoma twenty years

Abstract Keywords: Cutaneous metastasis, Metastatic

CASE REPORT prominent nucleoli in a background of desmoplastic

Table 1: Summary of immunoprofile of biopsied sample

occurs at a greater rate than with any other solid organ

to prognosis should justify a confirmatory excisional

Figure 4: Immunohistochemical stain results showed that tumor *********

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CASE REPORT OPEN ACCESS

Aortoduodenal fistula after transperitoneal repair of an

Abstract How to cite this article

Keywords: Aortoenteric, Aortoduodenal fistu- CASE REPORT

angiography (CTA) was performed (Figure 1) because of

Author Year of Number of Time of Fistula after Initial Type of Procedure

Yabu [9] 1998 1 10 years Conventional repair

Constans [10] 1999 7 4-7 years Conventional repair

Hauseggers [11] 1999 1 20 months Endovascular repair

Makar [12] 2000 1 4 months Endovascular repair

Lau, [13] 2001 1 5 years Conventional repair

Ohki, [14] 2001 1 9 months Endovascular repair

Parry, [15] 2001 1 12 months Endovascular repair

Chang, [16] 2002 1 20 days Conventional repair

Kar, [17] 2002 1 22 months Endovascular repair

Berges, [18] 2003 1 2 years Endovascular repair

Elkouri, [19] 2003 1 17 months Endovascular repair

Probst, [20] 2006 1 8 months Conventional repair

Geraci [21] 2008 1 5 years Conventional repair

Tanaka [22] 2009 1 13 years Conventional repair

Tromp [23] 2009 1 6 weeks Endovascular repair