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Appendicular bleeding: An exceptional cause of lower hemorrhage

Article  in  Revista espanola de enfermedades digestivas: organo oficial de la Sociedad Espanola de Patologia Digestiva · July 2015
DOI: 10.17235/reed.2015.3828/2015 · Source: PubMed

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Accepted Article
Appendicular bleeding: An exceptional cause of lower
hemorrhage

MARTA MAGAZ MARTINEZ, JUAN DE LA REVILLA NEGRO,


JAVIER MARTÍN LÓPEZ, irene gonzalez partida, TANIA DE LAS
HERAS, MARIA ROSARIO SANCHEZ YUSTE, ROBERTO RÍOS
GARCÉS, CLARA SALAS ANTON, Luis Esteban Abreu García

DOI: 10.17235/reed.2015.3828/2015
Link: PDF

Please cite this article as: MAGAZ MARTINEZ M, DE LA


REVILLA NEGRO J, MARTÍN LÓPEZ J, gonzalez partida I, DE
LAS HERAS T, SANCHEZ YUSTE M R, RÍOS GARCÉS R, SALAS
ANTON C, Abreu García L E. Appendicular bleeding: An
exceptional cause of lower hemorrhage. Rev Esp Enferm Dig
2015. doi: 10.17235/reed.2015.3828/2015.

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NC 3828 inglés

Appendicular bleeding: An excepcional cause of lower hemorrhage

Marta Magaz-Martínez1, Javier Martín-López2, Juan de-la-Revilla-Negro1, Irene


González-Partida1, Tania de-las-Heras3, María Rosario Sánchez-Yuste2, Roberto Ríos-
Garcés4, Clara Salas-Antón2 and Luis Abreu-García1
Departments of 1Digestive Diseases, 2Pathology, 3Radiology and 4Internal Medicine.
Hospital Universitario Puerta de Hierro Majadahonda. Madrid, Spain

Received: 28/04/2015
Accepted: 06/05/2015
Correspondence: Marta Magaz Martínez. Department of Digestive Diseases. Hospital
Universitario Puerta de Hierro Majadahonda. C/ Manuel de Falla, 1. 28222
Majadahonda, Madrid. Spain
e-mail: martamagazm@gmail.com

ABSTRACT
Chronic complications of acute appendicitis managed in a conservative manner are not
frequent. We present a case of acute lower gastrointestinal hemorrhage in a young
patient with a previous acute appendicitis without surgical intervention. The
colonoscopy detected an appendicular bleeding which was surgically treated. The
anatomopathological diagnosis was granulomatous appendicitis. The clinical evolution
of the patient was favorable without bleeding recurrence. Appendicular hemorrhage
can be an unusual complication —however potentially severe— of acute appendicitis
not treated surgically.
Key words: Lower gastrointestinal hemorrhage. Granulomatous appendicitis.

INTRODUCTION
Severe lower gastrointestinal bleeding (LGIB) accounts for 15% of all cases of acute
LGIB (1). The most frequent causes of rectorrhagia without hemodynamic repercussion
are hemorrhoidal pathology and neoplasias. Acute lower gastrointestinal bleeding is
usually related to bleeding of diverticular origin or with vascular ectasias. Patients aged
< 30 that debut with an important LGIB episode usually present lesions due to vascular
malformations (Dieulafoy’s lesion) or intestinal malformation (Meckel’s diverticulum),
and their etiological diagnosis is rather difficult sometimes.
Surgical management of appendicitis has been a subject of debate. Many surgeons are
in favor of an immediate appendectomy. However, the potential morbility, or even
mortality, as a result of this technique lead other group of experts to defend a more
conservative management (2,3).
Long term complications of this therapeutical non-aggressive attitude are not well
known. Hereunder we describe a case of acute appendicitis that was not treated
surgically and its long term unusual and potentially severe evolution (4).

CASE REPORT
We present the case of a Caucasian male aged 22 that comes to Emergency Service in
our hospital with rectal bleeding. The only relevant personal background relates to
appendicitis a year before, managed in a conservative manner with antibiotics. The
patient shows a 3-4 day evolution consisting of bright red blood emanating from the
rectum, not accompanied by defecation or other symptoms.
Several weeks before admission in the Emergency Service he had undergone an
analytic control including a complete blood count (CBC) with normal levels of
hemoglobin (13.3 g/dl).
Upon admission in Emergency the patient presented with hemodynamic stability,
blood pressure 138/61 mmHg and a heart rate of 84 bpm. The physical exploration
highlighted a pallor of the skin and the mucous membranes, perspiration and mucosal
dryness, whereas the rest of the exploration was anodyne. The rectal examination
showed plain rectal bleeding, but rectal masses could not be detected. The inspection
around the annus showed no fissures or hemorrhoids.
During his stay in Emergency two peripheral lines were secured, blood tests were
crossmatched and we started with the infusion of 500 cc of physiological saline at
0.9%. An urgent blood test highlighted a normocytic anemia (hemoglobin 7.3 g/dL and
mean corpuscular volume MCV of 87.40 fL). The rest of the blood count, biochemistry
and coagulation maintained values within the range of normality. Considering the
hemodynamic stability of the patient we decided to schedule an elective colonoscopy
prior colonic preparation with polyethylene glycol and transfusion of 4 etrytrocyte
concentrates. The colonoscopy was carried out 24 hours after and it reached the
terminal ileum. Blood remnants were only detected in the transverse and ascending
colon. Ileoscopy was normal, showing bile remains. In the absence of conclusive
findings, we carried out an oral panendoscopy, exploring the esophagus, the stomach
and the duodenum until the second duodenal portion, but did not observe traces of
blood or potentially bleeding injuries. We decided to complete the study by means of a
capsule endoscopy. Nevertheless, 12 hours after the endoscopic examinations, the
patient presented with a new episode of rectorrhagia with hemodynamic repercussion
and new anemia to a 7.6 g/dl hemoglobin (in spite of the transfusion of six erytrocyte
concentrates, what led us to carry out an urgent CT angiography. Thereby we found
hyperdensity at the cecum level, findings that suggested active endoluminal bleeding.
(Figs. 1 and 2). After the hemodynamic stabilization of the patient we repeated the
colonoscopy. The colonoscope was introduced until the terminal ileon, that showed a
normal mucosa without blood remnants. We examine the cecum where abundant
traces of blood remnants and blood clots are detected, but no potentially bleeding
mucosal lesions. At the base of the appendiceal orifice we observe an irregular
structure with active bleeding at the base (Fig. 3), which suggests that it might
correspond to a blood vessel.
With the clinical suspicion that it may be a severe acute LGIB of appendicular origin we
decided to carry out an urgent surgical intervention, performing an appendectomy
with associated removal of the cecum. The postoperative course was uneventful and
the patient is discharged from hospital 72 hours after surgery. The clinical follow-up
three and six months after discharge shows that the patient presents no further
symptoms. He has not suffered new episodes of macroscopic bleeding and the
recovery from anemia is confirmed as well.
Upon examination of the pieces removed the only significant thing was a cecal
appendix with diffuse enlargement. Under the microscope we could observe non-
necrotizing epitheliod granulomas, scattered along the wall, ulcers, fissures and cryptal
abscesses in the mucosa. The diagnosis thereof was chronic granulomatous
appendicitis (Figs. 4 and 5).

DISCUSSION
Granulomatous appendicitis is a rare clinicopathological finding histologically
characterized by the presence of appendicular granulomas. Granulomas are chronic
inflammatory lesions consisting of clusters of epithelioid histiocytes, occasionally
accompanied by multinucleated giant cells and lymphocytes as well as plasmatic cells
(5). This entity accounts for less that 2% of all appendicitis. It was thought at first that
it was a manifestation of Crohn’s disease. However, only 5-10% of all granulomatous
appendicitis develops this disease. Other etiologies are varied, highlighting the
sarcoidosis, the reaction of foreign-body granuloma and intestinal infectious diseases
such as tuberculosis, parasitic disease and more frequently the Yersinina species.
Recently subacute appendicitis has been described as the most common cause of this
entity. This condition produces a granulomatous reaction in relation to a secondary
inflammatory response to acute appendicitis managed conservatively, with a
postponed appendectomy (4). Unlike our case, the common clinical presentation is the
abdominal pain (6), whereas rectal bleeding is a truly rare event. We are therefore
faced with an exceptional case. The diagnosis is usually made by imaging, while the
definitive diagnosis based on histology. Since in our case the presentation was rectal
bleeding, the differential diagnosis should be made with other causes of severe
gastrointestinal bleeding in young patients. The definitive management of
granulomatous appendicitis is based on the appendectomy (7) and as a whole the
cases reviewed in the literature have a good long term prognosis (8). To date, less than
30 cases of appendiceal bleeding have been reported. Those cases of bleeding of
appendicular origin included inflammatory bowel disease, tuberculosis, cases of
intussusception or angiodysplasias among other etiologies. It is really difficult to make
a diagnosis with endoscopic visualization. And in most cases it is still necessary to
conduct a removal of the cecum (or sometimes an associated hemicolectomy) to
control the bleeding (9).
To conclude, granulomatous appendicitis in the context of a subacute appendicitis
may appear clinically as severe lower gastrointestinal bleeding due to appendicular
bleeding with important clinical and analytical repercussion. The combination of
dynamic radiological tests and endoscopy during the acute bleeding process increases
the diagnostic yield, but in most cases it requires a surgical management for the final
resolution of the case (10).

REFERENCES
1. García Sánchez N, González Galilea A, López Vallejos P, et al. Role of early
colonoscopy in severe acute lower gastrointestinal bleeding. Gastroenterol Hepatol
2001;24:327-32.
2. Varadhan KK, Neal KR, Lobo DN. Safety and efficacy of antibiotics compared with
appendicectomy for treatment of uncomplicated acute appendicitis: Meta-analysis of
randomised controlled trials. BMJ 2012;344:e2156. DOI: 10.1136/bmj.e2156
3. Wilms IM, de Hoog DE, de Visser DC, et al. Appendectomy versus antibiotic
treatment for acute appendicitis. Cochrane Database Syst Rev 2011;(11):CD008359.
DOI: 10.1002/14651858.CD008359.pub2
4. Bronner MP. Granulomatous appendicitis and the appendix in idiopathic
inflammatory bowel disease. Semin Diagn Pathol 2004;21(2):98-107. DOI:
10.1053/j.semdp.2004.12.001
5. Yilmaz M, Akbulut S, Kutluturk K, et al. Unusual histopathological findings in
appendectomy specimens from patients with suspected acute appendicitis. World J
Gastroenterol 2013;19:4015-22. DOI: 10.3748/wjg.v19.i25.4015
6. Bianchi A, Hidalgo LA, Fantova MJ, et al. Granulomatous disease of the cecal
appendix. Clinical significanc. Med Clin (Barc) 2004;122:798. DOI: 10.1157/13062677
7. Alhambra Rodríguez de Guzmán C, Morales Marín VJ, Salvelio Picazo Yeste J, et al.
Granulomatous appendicitis: An uncommon cause of acute abdomen. Cir Esp 2014. pii:
S0009-739X(13)00381-3. DOI: 10.1016/j.ciresp.2013.09.019
8. Tucker ON, Healy V, Jeffers M, et al. Granulomatous appendicitis. Surgeon
2003;1:286-9. DOI: 10.1016/S1479-666X(03)80047-1
9. Konno Y, Fujiya M, Tanaka K, et al. A therapeutic barium enema is a practical option
to control bleeding from the appendix. BMC Gastroenterol 2013;13:152. DOI:
10.1186/1471-230X-13-152
10. Gu J, Allan C. Idiopathic granulomatous appendicitis: a report of three consecutive
cases. ANZ J Surg 2010;80:201. DOI: 10.1111/j.1445-2197.2010.05237.x

Fig. 1. Axial plane-CT angiography. Hyperdensity at the cecum level that suggested
active endoluminal bleeding (blue narrow).
Fig. 2. Sagittal plane-CT angiography. Hyperdensity at the cecum level that suggested
active endoluminal bleeding (blue narrow).

Fig. 3. At the base of the appendicular orifice we observe an irregular structure with
active bleeding which suggests a blood vessel.

Fig. 4. Coronal cross-section of cecal appendix. Transmural lymphoid aggregates.


(HEX20).
Fig. 5. Scattered along the wall, ulcers, fissures and cryptal abscesses in the mucosa.
Non-necrotizing epithelioid granulomas (HEX200).

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