A Traumatic Direct Inguinal Hernia from Pelvic Ring Disruption

Kevin L. Chow, 1
Eduardo Smith-Singares, 2 and James Doherty 2

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Abstract

1. Introduction
Acute traumatic abdominal wall hernias (TAWHs) are rare but can be potential injuries seen
after blunt trauma. First reported in 1906, TAWHs are defined by three criteria: (1) herniation
through disrupted musculature and fascia after sufficient trauma, (2) no evidence of skin
penetration, and (3) no evidence of a prior hernia defect [1, 2]. Traumatic inguinal hernias
(TIHs) fall under that definition, but to our knowledge, a case resulting from an open-book
pelvic fracture has not been described.
Pelvic fractures usually involve a high-energy traumatic mechanism and account for
approximately 3% of all blunt traumatic skeletal injuries. Additional intraabdominal and
musculoskeletal injuries are expected to be found in over 80% of unstable pelvic fractures
given the associated high-energy trauma [3]. TAWH is a rarely reported consequence with
fewer than 40 cases of bowel herniation associated with traumatic pelvic fractures reported
over the past century [4, 5].
In this article, we summarize the presentation of an acute TIH due to an open-book pelvic
fracture after blunt abdominal trauma. This injury was managed with a simultaneous open
reduction and internal fixation (ORIF) of the pelvis and abdominal wall reconstruction with
mesh. This case report was written following the recommendations for the conduct, reporting,
editing, and publication of scholarly work in medical journals (contained in the ICJME
statement, http://www.icmje.org/), the work of the Equator Network (http://www.equator-
network.org/) and the SCARE checklist [6].

2. Case Presentation
A 45-year-old male motorcyclist with a history of hypertension, hyperlipidemia, and
coronary artery disease was brought to the emergency department after being struck by
another car on the highway at speeds of at least 40 miles per hour. Upon presentation, the
patient was evaluated using Advanced Trauma Life Support (ATLS) principles. He had a
patent airway on arrival and was breathing spontaneously on room air. His initial heart rate
was 87 beats per minute, and his blood pressure was 124/63 mmHg without signs of
significant hemorrhage. He had an initial Glasgow coma score (GCS) of 15 with equal and
reactive pupils. The patient admitted to consuming alcohol and had a serum alcohol of
243 mg/dL. A later CT of the head demonstrated a subcutaneous hematoma without any
intracranial abnormalities. His remaining physical examination revealed left lower quadrant
abdominal pain without signs of peritonitis, ankle deformities bilaterally, pain with hip range
of motion, and blood at the urethral meatus. Given his physical examination findings,
subsequent imaging confirmed an unstable pelvic fracture with diastasis of the symphysis
pubis of 6 cm, widening of the left sacroiliac joint, a left ischial pubic ramus fracture, and a
urethral injury (Figure 1). He also had a left ankle dislocation and a right compound fracture
of the distal tibia and fibula. No intraabdominal injuries were identified on CT imaging of the
abdomen. The pelvis was stabilized with a binder by the orthopedic surgeons with subsequent
emergency irrigation, debridement, and open reduction and internal fixation (ORIF) of the
open ankle fracture as well as reduction of the left ankle dislocation. He was extubated after
the procedure and monitored in the ICU while the remaining preoperative medical workup
was completed including X-rays and CT scans with 3D reconstructions of the pelvis
reconstructions. A hydromorphone patient-controlled analgesia (PCA) pump was utilized for
pain control.

Figure 1
CT Reconstruction demonstrating open-book pelvic fracture. The white arrow demonstrates the
inguinal hernia.
On hospital day 2, the patient was deemed fit for surgery and was taken to the operating
theater for a combined operation by the orthopedic surgeons for ORIF of the pubic diastasis,
sacral fracture, and sacroiliac joint followed by the trauma surgeons to reconstruct the
abdominal wall and inguinal canal. The trauma team performed the exposure of the pubic
symphyseal region and the pubic diastasis. A Pfannenstiel incision was made, and the planes
were dissected exposing the left spermatic cord. The orthopedic team then performed a gentle
open reduction of the pubic diastasis taking care to ensure that the bladder and urethra were
not incarcerated. The Asnis III cannulated screw system and a Matta pubic symphyseal plate
(Stryker GmbH, Switzerland) were utilized under C-arm fluoroscopic guidance with
appropriate alignment of the AP and inlet and outlet pelvis views. Once the Mata plate was in
place and the orthopedic reduction was completed, we proceeded to reconstruct the anterior
abdominal wall. Since the Cooper ligament was destroyed, it was dissected to allow direct
visualization of the pubic rami. The abdominal wall defect was measured to be 10 × 12 cm.
We then used a modified Stoppa technique by placing the 6 × 6 in Prolene mesh under the
damaged internal inguinal ring, making sure the spermatic cord on the left side was not
injured or pinched, securing it in place using sutures, including direct suturing to the
periosteum of the repaired pubic symphysis and the plate as needed. The medial borders of
the mesh were tucked inside the opened rectus sheath on the right side and secured laterally
with fires of a 5 mm Covidien Endotack (Medtronic, MN, USA) to the remnants of the
conjoint ligament. The midline was then repaired with sutures, including the mesh as
reinforcement. The patient did well postoperatively with postreduction films demonstrating
appropriate alignment (Figure 2). He was discharged to rehab on postoperative day 5. There
were no recurrences during the follow-up period of 10 years.

Figure 2
Postreduction X-rays.

3. Discussion
True acute TIHs are a rare entity that falls within the category of TAWH with some believing
them to be a “myth” [7]. TAWHs are a rare sequelae of blunt abdominal trauma with an
estimated incidence of 0.07% in a large single-center retrospective analysis of CT scans
performed for blunt trauma [8]. The mechanism of TAWH typically involves a combination
of local tangential shearing forces and a sudden rise in intraabdominal pressures resulting in
damage or disruption to the underlying muscle fibers, fascia, and peritoneum. The skin may
or may not be involved as its inherent elasticity is protective [2].
These hernias occur most frequently after motor vehicle collisions (40%) and motorcycle
collisions (16%). The clinical presentation of TAWH typically involves a tender soft tissue
mass on the abdomen (31%) or an associated ecchymosis (49%). They can present acutely in
the case of concomitant intraabdominal injuries and peritonitis or evisceration. A delayed
presentation is also possible with muscle spasm from pain following trauma initially masking
the defect. Delayed herniation is also a possibility following the weakening of the abdominal
wall from a hematoma or wound infection [5]. The CT scan is the diagnostic modality of
choice and allows for the detection and grading of abdominal wall injuries, first described by
Liasis et al. and Dennis et al. [2, 8].
Early reports of patients with TAWH recommended early operative exploration and repair
due to the high risk of concomitant intraabdominal injuries and potential for incarceration
[9, 10]. More recent studies have debated that recommendation by demonstrating that in the
absence of concurrent intraabdominal injuries, delayed repair or a laparoscopic approach can
be considered [10–12]. However, the natural history of undetected hernias is progressive
enlargement that can lead to bowel incarceration and strangulation. Therefore, early
abdominal wall reconstruction with tension-free repairs remains the standard. The use of
mesh repairs is well recognized for its ability to minimize recurrence rates but must be
weighed against the risk of infection particularly with hollow viscus injuries and abdominal
contamination [13].
We presented the unique presentation of a TIH due to an associated open-book pelvic fracture
after blunt abdominal trauma. The hernia was diagnosed based on CT imaging, and there
were no other concomitant intraabdominal injuries found. It was then repaired with a tension-
free, modified Stoppa mesh technique during the same operation as the pelvic ORIF. The
patient had a good outcome without recurrence during the follow-up period of 10 years.

4. Conclusions
In summary, the management of TAWH and TIH is complicated given the high association
with other concomitant intraabdominal and musculoskeletal injuries particularly with pelvic
fractures. Here, we have shown that for selected patients, without severe intraabdominal
injuries or gross contamination, it is possible to perform the abdominal wall and inguinal
canal reconstruction with a tension-free, modified Stoppa mesh technique and reduce the
pelvis simultaneously in the acute setting with durable results.

Ethical Approval
The institutional review board (IRB) of our institution was consulted, and appropriate ethical
approval was granted.

Consent
Written informed consent was obtained from the patient for publication of this case report
and accompanying images.

Conflicts of Interest
The authors declare that there is no conflict of interest regarding the publication of this
article.

References
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wall hernia: is the treatment strategy a real problem? Journal of Trauma and Acute Care
Surgery. 2013;74(4):1156–1162. doi: 10.1097/TA.0b013e318283d88dz.
3. Puzio T., Shah M., Dineen H., Chen J., Caddell K., Charles A. A case of iliac crest
avulsion with peritoneal disruption and bowel herniation after blunt trauma: a case report and
review of literature. The American Surgeon. 2016;82(7):655–658.
4. Walcher F., Rose S., Roth R., Lindemann W., Mutschler W., Marzi I. Double traumatic
abdominal wall hernia and colon laceration due to a pelvic fracture. Injury. 2000;31(4):253–
256. doi: 10.1016/S0020-1383(99)00295-8.
5. Suhardja T. S., Atalla M. A., Rozen W. M. Complete abdominal wall disruption with
herniation following blunt injury: case report and review of the literature. International
Surgery. 2015;100(3):531–539. doi: 10.9738/INTSURG-D-14-00079.1.
6. Agha R. A., Fowler A. J., Saeta A., et al. The SCARE statement: consensus-based surgical
case report guidelines. International Journal of Surgery. 2016;34:180–186. doi:
10.1016/j.ijsu.2016.08.014
7. Dinneen M. D., Wetter L. A., Orr N. W. M. Traumatic indirect inguinal hernia: a seat belt
injury. Injury. 1989;20(3):p. 180. doi: 10.1016/0020-1383(89)90097-1.
8. Dennis R. W., Marshall A., Deshmukh H., et al. Abdominal wall injuries occurring after
blunt trauma: incidence and grading system. American Journal of Surgery. 2009;197(3):413–
417. doi: 10.1016/j.amjsurg.2008.11.015.
9. Esposito T. J., Fedorak I. Traumatic lumbar hernia: case report and literature review. The
Journal of Trauma: Injury, Infection, and Critical Care. 1994;37(1):123–126. doi:
10.1097/00005373-199407000-00022.
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Critical Care. 2006;61(5):1058–1061. doi: 10.1097/01.ta.0000240450.12424.59.
12. Novitsky Y. W. Laparoscopic repair of traumatic flank hernias. Hernia. 2018;22(2):363–
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13. Pardhan A., Mazahir S., Rao S., Weber D. Blunt traumatic abdominal wall hernias: a
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J Surg Case Rep. 2018 Aug; 2018(8): rjy199.

Published online 2018 Aug 7. doi: 10.1093/jscr/rjy199

PMCID: PMC6080051

PMID: 30093997

Conservative management of pelvic sepsis with severe shock and

multiple organ dysfunction syndrome after rubber-band ligation of
internal haemorrhoids: surgery is not the only option
Bojana Misheva,1 Roy Hajjar,2 Frédéric Mercier,3 Frank Schwenter,4 and Herawaty Sebajang4

Author information ► Article notes ► Copyright and License information ► Disclaimer

Abstract

INTRODUCTION
Rubber-band ligation (RBL) has been introduced as a treatment for internal haemorrhoids a
few decades ago by Blaisdell and Barron, and was used since with little to no side effects
[1, 2]. RBL is established as a cost-effective and efficient treatment for first- to third-degree
internal haemorrhoids. It works by inducing fibrosis, retraction and fixation of the
haemorrhoidal cushions [3]. Although it is a safe procedure, some common complications
exist, such as pain, bleeding, thrombosis, prolapse and urinary retention.
Pelvic sepsis is a rare but serious complication of this procedure. The latter usually develops
48 h after the operation and generates dysuria, perineal pain, fever and sometimes systemic
sepsis. Several cases of pelvic sepsis were formally detailed in the literature after RBL, and
many of them were fatal [4]. The mainstay of treatment is not clearly defined as conservative
and surgical approaches both yielded favourable results. Active surgical debridement seems
to be a more logical approach in severely ill patients. However, data on its superiority to
conservative management are lacking.

CASE PRESENTATION
A 58-year-old female patient is diagnosed with internal haemorrhoids and undergoes RBL.
Her past medical history includes osteoporosis, dyslipidemia and depression. Forty-eight
hours after the procedure, she presented back to the emergency room for pelvic pain and
dysuria. Physical examination did not reveal any necrosis or signs of infection. Management
of her symptoms consisted of empirical intravenous antibiotics, ciprofloxacin and
metronidazole, and clinical observation.
Her condition did not improve and her white blood count (WBC) increased significantly (24
000/μL). She was consequently transferred to a referral centre where she was brought to the
operating room for an exploration of the surgical site. The band was removed and a small
superficial abscess was drained. A computed tomography (CT) scan of the abdomen and
pelvis showed a rectosigmoiditis with a moderate to severe amount of ascites. No
pneumatosis was noted. An exploratory laparoscopy was thus performed to rule out rectal
necrosis, and revealed an important amount of serous ascites. The patient was suffering after
the surgical procedure from a severe systemic inflammatory response syndrome (SIRS) and
required several days of intensive care. Due to clinical deterioration and persistent
leukocytosis (25 000/μL), she was transferred to our tertiary care centre with a suspicion of
ischaemic colitis.
Upon arrival, her hemodynamic and respiratory status deteriorated rapidly and ventilator
support was necessary for an acute respiratory distress syndrome (ARDS). The antibiotics’
spectrum was broadened to a carbapenem. A colonoscopy to the mid-transverse colon was
performed and showed a superficially necrotic 2 cm segment in the lower rectum with
petechial mucosa.
After 72 h of inotropic support with vasopressors, aggressive volemic resuscitation, ventilator
support and antibiotics, a modest improvement was noted. WBC count decreased slightly
from 30 000 to 23 000/μL. A repeat abdominopelvic CT scan was performed four days after
the transfer and revealed once again the presence of a circumferential thickening of the
rectosigmoid colon without pneumatosis (Fig. (Fig.1).1). The state of shock subsided
gradually and the patient was extubated after a total of 9 days of ventilator support. She was
transferred to the floor before being discharged from the hospital a few days later. She
recovered progressively from the intensive care unit myopathy and the prolonged intubation,
and benefited afterward from intensive rehabilitation.

Figure 1:
Thickening of the rectal wall.
At follow-up appointments, the patient was clinically better, but still complained of muscular
weakness and poor exercise tolerance up to several months after the episode.
Three years after the events, a colonoscopy was performed and was normal. The patient was
followed by a respirologist for persistent but improving dyspnea that was deemed secondary
to severe deconditioning during the hospital stay. No evidence of a residual cardiorespiratory
injury was objectified.

DISCUSSION
We hereby present the case of a 58-year-old woman with post-RBL severe pelvic sepsis, and
associated shock and multiple organ failure (MOF), which was successfully treated
conservatively.
Pelvic sepsis is an extremely rare complication of internal haemorrhoids RBL. A review of
39 studies including 8060 patients undergoing RBL revealed post-banding complications in
14% of them, including pain (5.8%), haemorrhage (1.7%), anal fissure or fistula (0.4%), and
infection (0.05%) [5, 6].
As expected, severe perineal or abdominal pain, urinary retention, fever and leukocytosis
could be features of perineal sepsis. In previous reports, some patients attended the
emergency room more than once with these symptoms before pelvic sepsis was diagnosed
[4]. Clinical presentation could therefore be sometimes misleading and a severe underlying
complication is not always easy to suspect in patients undergoing this procedure. The scarcity
of this adverse event makes the identification of its most specific and sensitive symptoms
complex.
From what we experienced in our case, the support measures were the mainstay of the
treatment since the capillary leak syndrome was the main issue to address. In the literature,
patients who developed severe septic complications and organ failure were for some human
immunodeficiency virus positive or had an objectified infectious lesion such as a cellulitis or
gangrene [7–9]. Interestingly, despite the excessive systemic response, our patient did not
show any signs of transmural tissue necrosis requiring aggressive surgical debridement.
Hence, the role of the surgeon in such a case is to evaluate if a surgery will benefit the patient
or not. A defunctioning colostomy or a perineal tissue debridement would have probably not
added much to the maximal conservative medical treatment, and possibly complicated further
the hospital stay.
Surgical intervention in a severely ill patient with a postoperative complication may
sometimes be reasonable. This case shows that conservative management could be a valid
approach as surgical intervention with a diverting stoma or an ischiorectal exploration might
not necessarily be more efficient. Early recognition of this complication could probably
prevent significant deterioration and hasten rapid referral to specialized care. Reports on such
morbid cases are lacking, and early manoeuvres to slow the inflammatory response are yet to
be elucidated.

CONFLICT OF INTEREST STATEMENT

None declared.

REFERENCES
1. Blaisdell PC. Prevention of massive hemorrhage secondary to hemorrhoidectomy. Surg
Gynecol Obstet1958;106:485–8.
2. Barron J. Office ligation treatment of hemorrhoids. Dis Colon Rectum 1963;6:109–13.
3. ASGE Technology Committee Siddiqui UD, Barth BA, Banerjee S, Bhat YM, Chauhan
SS, Gottlieb KT, et al. Devices for the endoscopic treatment of hemorrhoids. Gastrointest
Endosc 2014;79:8–14.
4. McCloud JM, Jameson JS, Scott AN. Life-threatening sepsis following treatment for
haemorrhoids: a systematic review. Colorectal Dis 2006;8:748–55. Review.
5. Wechter DG, Luna GK. An unusual complication of rubber band ligation of
hemorrhoids. Dis Colon Rectum 1987;30:137–40.
6. Shiller M, Reshef R. An unusual complication of rubber band ligation of
hemorrhoids. Gastrointest Endosc 1988;34:490.
7. Buchmann P, Seefeld U. Rubber band ligation for piles can be disastrous in HIV-positive
patients. Int J Colorectal Dis 1989;4:57–8.
8. Quevedo-Bonilla G, Farkas AM, Abcarian H, Hambrick E, Orsay CP. Septic complications
of hemorrhoidal banding. Arch Surg 1988;123:650–1.
9. Albuquerque A. Rubber band ligation of hemorrhoids: a guide for complications. World J
Gastrointest Surg 2016;8:614–20. Review.
Medicine (Baltimore). 2018 Apr; 97(17): e0532.

Published online 2018 Apr 27. doi: 10.1097/MD.0000000000010532

PMCID: PMC5944502

PMID: 29703028

Osteoporosis and fracture after gastrectomy for stomach cancer

A nationwide claims study
Gi Hyeon Seo, MD,a Hae Yeon Kang, MD, PhD,b,∗ and Eun Kyung Choe, MD, PhDc

Monitoring Editor: Ziyuan Zhou.

Author information ► Article notes ► Copyright and License information ► Disclaimer

Abstract

1. Introduction
Globally, stomach cancer is the fourth most common cause of cancer,[1,2] and the early
detection and postoperative survival rates are also increasing.[3] In Korea, approximately
20,000 people undergo gastrectomy for gastric cancer every year. Osteoporosis and fractures
commonly occur after gastrectomy for stomach cancer.[4–6] In previous studies, the incidence
of osteoporosis was reportedly 32% to 42%, and the incidence of fracture was approximately
40% after gastrectomy.[4,6,7] Osteoporotic fractures interfere with patients’ quality of life and
increase the socioeconomic burden on individuals and society.[8]
Despite the high prevalence of osteoporosis, no program has been established after
gastrectomy for stomach cancer. In addition, the precise incidence is unknown, because many
people are diagnosed with osteoporosis and fractures at hospitals outside of the hospital that
performed the gastrectomy. The purpose of this study was to evaluate the incidence and risk
factors of osteoporosis and fracture after gastrectomy for stomach cancer using the
nationwide claims database in South Korea.

2. Patients and methods

2.1. Data acquisition

All South Koreans are obliged to enroll in the National Health Insurance Corporation; thus,
approximately 98% of the Korean people are registered. Claims data from the Health
Insurance Review and Assessment Service (HIRA) are collected when medical providers
provide services to patients and request reimbursement from the HIRA. The patient records in
the HIRA database include gender, age, diagnoses, treatments, and prescriptions.[9] The data
used in this study were obtained from all claims data registered between January 2007 and
December 2015. Data prior to 2007 were inaccessible.
2.2. Study population
Patients who underwent gastrectomy and had a diagnosis of stomach cancer (C160–C169
according to the ICD-10 classification) between January 2008 and December 2010 were
identified from the HIRA database. These 54,146 patients were defined as having undergone
index gastrectomy. Among these patients, individuals 50 to 79 years of age were selected for
the analysis (n = 41,512). The average age of menopausal women in Korea is 50 years old;
therefore, this study included only postmenopausal women 50 years of age or older. We
excluded patients with a record of stomach surgery (n = 9), patients diagnosed with
osteoporosis and prescribed bisphosphonates or raloxifene (n = 2458), or patients diagnosed
with osteoporotic fractures (n = 2484) prior to the index gastrectomy. Finally, 37,076 patients
were included in the final analysis (Fig. (Fig.11).

Figure 1

A flow chart of the study population. Patients were classified into the osteoporosis (+) group or
fracture (+) group if they were diagnosed with operationally defined osteoporosis or fracture during
the follow-up period after the index gastrectomy; otherwise, the patients were assigned to the
osteoporosis (−) group or fracture (−) group.
Because the HIRA data did not include bone mineral density (BMD) measurements using
dual energy X-ray absorptiometry (DXA), an accurate osteoporosis diagnosis could not be
obtained using T-scores.[10,11]In Korea, there is a risk of overestimation when the diagnosis is
considered, because the diagnostic code of osteoporosis can be used even in the diagnostic
work-up process to apply for medical insurance. To avoid overestimation, we operationally
defined osteoporosis as a diagnostic code for osteoporosis (M80–M82 according to the ICD-
10 classification) plus a prescription for bisphosphonate or raloxifene or the occurrence of
osteoporotic fractures after index gastrectomy. We included only bisphosphonates and
raloxifene as the drugs for osteoporosis, because these drugs are most commonly used to treat
osteoporosis in Korea. Moreover, hormone therapy or calcium agents could be prescribed for
other indications. Osteoporotic fracture was defined as a fracture of a common osteoporotic
fracture site, such as the spine, pelvis, hip, forearm, or rib.
We included the patient age at the time of the index gastrectomy (i.e., 50–64 years was
considered young, and 65–79 years was considered elderly), patient gender, surgical method
(subtotal gastrectomy [STG] vs. total gastrectomy [TG]), chemotherapy use (within 180 days
after index gastrectomy or until osteoporosis or a fracture occurred), and diabetes mellitus
status. Each patient underwent a follow-up period of at least 5 to 8 years, because we used
data registered in the HIRA database until December 2015. The end of follow-up date was
defined as the last claims data registered in the HIRA database, because most cancer patients
were monitored for recurrence at least once yearly after the index gastrectomy for as long as
the patients survived. Person-years were counted from the date of gastrectomy until the end
of follow-up.

2.3. Ethics statement

The study protocol was approved by the Institutional Review Board of Seoul National
University Hospital (IRB number 1706-054-858), and the study was conducted in agreement
with the Declaration of Helsinki. Informed consent was waived by the board.

2.4. Statistical analysis

We expressed categorical variables as frequencies with percentages. The chi-squared test was
used to assess differences between independent groups. The annual cumulative incidence rate
for osteoporosis or fracture among the at-risk patients was calculated using Kaplan–Meier
plots. Cox proportional hazard analyses were also performed to identify the risk factors for
osteoporosis or fracture development. The statistical analyses were performed using the R
statistical software, version 3.2.2 (R development Core Team; R Foundation for Statistical
Computing, Vienna, Austria). Statistical significance was established for 2-sided P values <
.05.

3. Results

3.1. Baseline characteristics

A total of 37,076 patients (9915 females, 26.7%) were analyzed in this study. The mean
patient age was 63.4 ± 7.8 years, and 38.2% had diabetes. STG was performed in 76.7% of the
patients, and 40.3% patients received chemotherapy. The patients were followed for 179,421
person-years. Our study had a mean follow-up period of 5.0 ± 2.3 years (median 5.7 years). Of
these patients, 3690 (10.0%) were prescribed bisphosphonates or raloxifene, and 5175
patients (14.0%; 12.3% of the males and 18.4% of the females) had identified osteoporotic
fractures after gastrectomy during the follow-up period (mean ± standard deviation: 3.3 ± 2.0
years, median 3.1 years, interquartile range 1.6–4.8). A total of 7514 (20.3%) of the patients
were operationally diagnosed with osteoporosis after the index gastrectomy in this study.
3.2. Cumulative incidences of osteoporosis or fractures during the follow-up
after the index gastrectomy
The 5-year cumulative incidence rates of osteoporosis for older age, female gender, TG,
chemotherapy, and diabetes were 25.0% (vs. 13.7%, younger age), 31.2% (vs. 14.0%, male),
19.3% (vs. 18.6%, STG), 18.3% (vs. 18.9%, without chemotherapy), and 20.9% (vs. 17.4%,
nondiabetes), respectively (Table (Table1).1). The multivariate cox regression analysis
showed that older age (hazard ratio [HR] 1.88; 95% confidence interval [CI] 1.79–1.96),
female gender (HR 2.46; 2.35–2.58), TG (HR 1.10; 1.04–1.16), and diabetes (HR 1.16; 1.11–
1.22) were significantly associated with osteoporosis. Chemotherapy was not significantly
related to osteoporosis.

Table 1
Annual cumulative osteoporosis incidence after gastrectomy calculated from Kaplan–Meier
plots and cox proportional hazard analysis for each risk factor.

The 5-year cumulative incidence rates of osteoporotic fractures for older age, female gender,
TG, chemotherapy, and diabetes were 17.1% (vs. 9.1%, younger age), 16.3% (vs. 11.3%,
male), 14.0% (vs. 12.3%, STG), 13.1% (vs. 12.4%, without chemotherapy), and 14.8% (vs.
11.4%, nondiabetes), respectively (Table (Table2).2). The multivariate cox regression
analysis showed that older age (HR 1.90; 95% CI 1.80–2.01), female gender (HR 1.50; 1.41–
1.58), TG (HR 1.17; 1.10–1.25), chemotherapy (HR 1.06; 1.00–1.12), and diabetes (HR 1.26;
1.19–1.33) were significantly associated with fractures.

Table 2
Annual cumulative osteoporotic fracture incidence after gastrectomy calculated from
Kaplan–Meier plots and cox proportional hazard analysis for each risk factor.
3.3. Locations of fractures and multisite fractures after gastrectomy
Osteoporotic fractures were common in the spine (2031, 5.3%), rib (1966, 5.3%), forearm
(1318, 3.6%), and femur (753, 2.0%). When we divided the patients by gender, rib fractures
were the most common sites in males (5.5%), and spine fractures were the most common
sites in females (13.1%). Among the total of 5175 fracture patients, 780 (15.1%) experienced
multisite fractures (Table (Table3).3). Significantly more multisite fractures occurred in the
elderly and chemotherapy groups (P < .05).

Table 3
Multisite fracture (%).
4. Discussion and conclusion
Stomach cancer is particularly common in East Asia.[12] Recently, a large proportion of
stomach cancer cases have been diagnosed and treated early; as a result, the 5-year survival
rate for early gastric cancer in Korea is over 90%.[13,14] Therefore, more attention should be
directed toward preventing osteoporosis to improve patients’ quality of life and reduce
socioeconomic costs during the long survival period after gastrectomy. To the best of our
knowledge, this study is the first to use nationwide claims data to investigate the incidence
and risk factors of osteoporosis and fractures in patients who have undergone gastrectomy for
gastric cancer. Older age, female gender, TG, and diabetes were significantly associated with
osteoporosis and fractures. The elderly and chemotherapy groups were significantly
associated with multisite fractures.
Although the pathogenesis is still unclear, malabsorption is known to be a common cause of
osteoporosis after gastrectomy.[15,16] Gastrointestinal physiology could be altered after
gastrectomy and reconstruction. The primary sites affected after gastrectomy are the
duodenum and proximal jejunum, which are the main sites of calcium
absorption.[4,17] Previous experimental studies have suggested that hypovitaminosis D and a
subsequent increase in parathyroid hormone may contribute to increased rates of bone loss
after gastrectomy.[18–20] After gastrectomy, the patients experienced rapid weight loss of 5% to
15% during the immediate postoperative period, which also affected the risk of osteoporosis
and fractures due to a lack of appetite, dyspepsia, and altered intestinal motility.[21,22]
In a previous study, the prevalence of osteoporosis among patients 50 years of age or older
was 32.9% to 35.5% for females and 7.5% to 12.2% for males according to the BMD, and the
treatment rate was 44% to 58% for physician-diagnosed osteoporosis patients in Korea.[23–
25]
A previous study also reported a higher incidence of osteoporosis, because the authors
examined every patient who underwent gastrectomy for osteoporosis regardless of the
symptoms.[4] Osteoporosis diagnoses were made using the World Health Organization
(WHO) T-score of <−2.5, which was calculated from the Asian reference data. However,
prior to October 2011, the South Korean reimbursement guidelines for osteoporosis treatment
required that patients to have a T-score of <−3.0 or a history of osteoporosis-related fractures
of the spine or hip. Thus, based on the WHO and reimbursement guidelines, some real
patients may not have been included in our study. Bisphosphonates and raloxifene are the
most commonly used drugs to treat osteoporosis, and their market share among nonhormonal
osteoporotic drugs reaches approximately 90% in Korea.[26,27]Therefore, osteoporosis might be
underestimated in this study, because many people were not diagnosed due to the lack of
symptoms. In addition, many diagnosed patients were not treated, and people who used drugs
other than bisphosphonate or raloxifene were excluded. However, various factors, such as the
definition of osteoporosis, the methods used to determine the BMD, and the reference
population used to calculate the T-scores, affect the incidence rate of osteoporosis.[24,28,29] In
this study, we used an operational definition of a patient with newly diagnosed osteoporosis
(with a prescription) after index gastrectomy.
In the present study, osteoporotic fractures were identified in 12.3% of males and 18.4% of
females after index gastrectomy. Female gender is a risk factor for both osteoporosis and
fracture. Fracture may also be underestimated, because fractures are included only in cases
diagnosed in a hospital. In fact, the fracture incidence rates were relatively low compared to
the rates found in a previous single-center study that reported a cumulative incidence of
fracture up to 40.6%, with most fractures occurring within the first 6 postoperative years.[6] In
that study, annual bone scintigraphy with additional computed tomography, magnetic
resonance imaging, or X-ray examinations were performed with or without trauma; thus,
those results differ from our research findings. In our study, most patients might have
clinically important symptoms or trauma to receive medical treatment. Moreover, fractures
were identified within a median of 3.1 years after the index gastrectomy in this study because
a bone remodeling imbalance reportedly occurs early within the first postoperative year.[30]
In this study, diabetes, TG, and chemotherapy were associated with a higher risk of fracture.
Type 2 diabetes is a well-known cause of secondary osteoporosis.[31] Patients who underwent
TG were more likely to have more advanced disease, receive more chemotherapy, and
consequently have more severe weight loss than patients who underwent STG. Chemotherapy
can also be related to osteoporotic fractures because it induces physical inactivity and weight
loss due to the lack of appetite and reduced oral intake and the effects of the
chemotherapeutic drugs themselves. Systemic chemotherapeutic agents, such as 5-
fluorouracil and cisplatin, which are the most commonly used drugs for gastric cancer, have
been reported to contribute to bone loss by inducing apoptosis of osteoblasts and increasing
osteoclast activity.[32–34]
The American Gastroenterological Association has recommended DXA in patients who are at
least 10 years postgastrectomy, particularly postmenopausal females and males over 50 years
of age, based on reports involving patients with peptic ulcer disease.[35–38] However, the
application of this recommendation to patients with gastric cancer is inappropriate because
cancer patients tend to be older and have worse general conditions than peptic ulcer patients.
Moreover, the risk of osteoporosis or fracture in the short term after surgery should be
considered, as described our study. A program designed to diagnose and prevent osteoporosis
and fractures should be established shortly after surgery for gastric cancer patients who are
expected to survive long term.
This study has several limitations. First, an accurate osteoporosis diagnosis using T-scores
was not possible, because the HIRA data did not include the BMD. Thus, the incidence of
osteoporosis reported in this study might differ from the incidence reported in studies that
defined osteoporosis based on BMD scores. Second, the HIRA database does not include
laboratory data, BMI, weight change, menopause, smoking, or the cancer stage, which are
known risk factors for osteoporosis. The average age of menopause in Korean women is 50
years old; therefore, this study included only patients older than 50 years of age at the time of
the index gastrectomy. Third, fractures in this study might include bone metastasis or trauma,
other than osteoporotic fracture, even if we only included common osteoporotic fracture sites.
Fourth, the duration after gastrectomy is not sufficient, because the HIRA database has been
available only since 2007. Therefore, our study had a mean follow-up period of only 5.0
years. Nevertheless, our results confirmed that the fracture incidence rate was high during the
initial follow-up years.
In conclusion, this study demonstrated a high prevalence of osteoporosis and fractures in
stomach cancer patients in the early postgastrectomy years. A careful surveillance program is
needed early after gastrectomy to prevent and detect osteoporosis early and to prevent
fractures for the improvement of the long-term quality of life of gastric cancer survivors.
Previous studies have suggested that bisphosphonate therapy shows effectiveness in
increasing the BMD and reducing the risk of fracture in gastric cancer patients after
gastrectomy.[39,40] Further studies are needed to compare the incidence of fractures between
osteoporosis patients treated with bisphosphonate or other active agents and those who are
not treated. A systematic program should be developed to prevent and manage osteoporosis
and fractures through periodic follow-up after gastrectomy for stomach cancer.

Author contributions
Conceptualization: Gi Hyeon Seo, Hae Yeon Kang, Eun Kyung Choe.
Data curation: Gi Hyeon Seo, Hae Yeon Kang, Eun Kyung Choe.
Formal analysis: Gi Hyeon Seo, Hae Yeon Kang.
Investigation: Gi Hyeon Seo, Hae Yeon Kang.
Methodology: Gi Hyeon Seo, Hae Yeon Kang.
Project administration: Hae Yeon Kang.
Resources: Hae Yeon Kang.
Software: Gi Hyeon Seo, Hae Yeon Kang.
Supervision: Hae Yeon Kang, Eun Kyung Choe.
Validation: Hae Yeon Kang, Eun Kyung Choe.
Visualization: Hae Yeon Kang, Eun Kyung Choe.
Writing-original draft: Hae Yeon Kang.
Writing-review and editing: Gi Hyeon Seo, Hae Yeon Kang, Eun Kyung Choe.

Footnotes
Abbreviations: BMD = bone mineral density, CI = confidence interval, DXA = dual energy X-ray
absorptiometry, HIRA = Health Insurance Review and Assessment Service, HR = hazard ratio, STG =
subtotal gastrectomy, TG = total gastrectomy, WHO = World Health Organization.
The authors have no funding and conflicts of interest to disclose.

References
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[2] Fitzmaurice C, Allen C, et al. Global Burden of Disease Cancer Collaboration. Global, regional, and
national cancer incidence, mortality, years of life lost, years lived with disability, and disability-
adjusted life-years for 32 cancer groups, 1990 to 2015: a systematic analysis for the Global Burden of
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[3] Chan BA, Jang RW, Wong RK, et al. Improving outcomes in resectable gastric cancer: a review of
current and future strategies. Oncology (Williston Park) 2016;30:635–45.

[4] Lim JS, Kim SB, Bang HY, et al. High prevalence of osteoporosis in patients with gastric
adenocarcinoma following gastrectomy. World J Gastroenterol 2007;13:6492–7.

[5] Krupski W, Tatara MR, Bury P, et al. Negative effects of total gastrectomy on bone tissue
metabolism and volumetric bone mineral density (vBMD) of lumbar spine in 1-year study in
men. Medicine (Baltimore) 2016;95:e2817.

[6] Oh HJ, Lim CH, Yoon BH, et al. Fracture after gastrectomy for gastric cancer: a long-term follow-
up observational study. Eur J Cancer 2017;72:28–36.

[7] Inoue K, Shiomi K, Higashide S, et al. Metabolic bone disease following gastrectomy: assessment
by dual energy X-ray absorptiometry. Br J Surg 1992;79:321–4.

[8] Mohd-Tahir NA, Li SC. Economic burden of osteoporosis-related hip fracture in Asia: a systematic
review. Osteoporos Int 2017;28:2035–44.

[9] Kim L, Kim JA, Kim S. A guide for the utilization of Health Insurance Review and Assessment
Service National Patient Samples. Epidemiol Health 2014;36:e2014008.

[10] Park C, Jang S, Lee A, et al. Incidence and mortality after proximal humerus fractures over 50
years of age in South Korea: national claim data from 2008 to 2012. J Bone Metab 2015;22:17–21.

[11] Yoon HK, Lee YK, Ha YC. Characteristics of patients diagnosed with osteoporosis in South Korea:
results from the National Claim Registry. J Bone Metab 2017;24:59–63.

[12] Torre LA, Siegel RL, Ward EM, et al. Global cancer incidence and mortality rates and trends—an
update. Cancer Epidemiol Biomarkers Prev 2016;25:16–27.

[13] Lee HJ, Yang HK, Ahn YO. Gastric cancer in Korea. Gastric Cancer 2002;5:177–82.

[14] Jeong O, Park YK. Clinicopathological features and surgical treatment of gastric cancer in South
Korea: the results of 2009 nationwide survey on surgically treated gastric cancer patients. J Gastric
Cancer2011;11:69–77.

[15] Liedman B, Bosaeus I, Mellstrom D, et al. Osteoporosis after total gastrectomy. Results of a
prospective, clinical study. Scand J Gastroenterol 1997;32:1090–5.

[16] Kwon SJ, Hahm JS, Cho YJ, et al. The influence of gastrectomy on the change of bone
metabolism and bone density. Korean J Intern Med 2000;15:25–31.
[17] Bernstein CN, Leslie WD, Leboff MS. AGA technical review on osteoporosis in gastrointestinal
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[18] Piert M, Machulla HJ, Jahn M, et al. Coupling of porcine bone blood flow and metabolism in
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[19] Piert M, Zittel TT, Jahn M, et al. Increased sensitivity in detection of a porcine high-turnover
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[20] Glatzle J, Piert M, Meile T, et al. Prevalence of vertebral alterations and the effects of calcium
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[21] Liedman B. Symptoms after total gastrectomy on food intake, body composition, bone
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diseases. Gastroenterology2003;124:791–4.

[36] Adachi Y, Shiota E, Matsumata T, et al. Osteoporosis after gastrectomy: bone mineral density of
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[37] Heiskanen JT, Kroger H, Paakkonen M, et al. Bone mineral metabolism after total
gastrectomy. Bone2001;28:123–7.

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[39] Lim JS, Jin SH, Kim SB, et al. Effect of bisphosphonates on bone mineral density and fracture
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[40] Iwamoto J, Uzawa M, Sato Y, et al. Effect of alendronate on bone mineral density and bone
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Radiol Case Rep. 2018 Oct; 13(5): 993–998.

Published online 2018 Aug 11. doi: 10.1016/j.radcr.2018.07.006

PMCID: PMC6092483

PMID: 30116461

Pelvic tuberculosis: a forgotten diagnosis – case report

Natacha Abreu, MD, Resident in Radiology,a,⁎ Maria Ana Serrado, MD, Resident in Radiology,a Rosário
Matos, MD, Senior Consultants in Pediatric Radiology,b Rita Carneiro, MD, Senior Consultants in Pediatric
Radiology,b andAna Nunes, MD, Senior Consultants in Pediatric Radiologyb

Author information ► Article notes ► Copyright and License information ► Disclaimer

Abstract
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Introduction
Tuberculosis (TB) may be considered in 2 forms: pulmonary and extrapulmonary (kidneys,
bone, central nervous system, gastrointestinal tract, female genital tract, peritoneum, etc).
Extrapulmonary TB accounts for 15%-20% of all cases of TB, but among them, female
pelvic TB is rare (about 5%). In children and adolescents, extrapulmonary forms are rarely
reported in the pelvis, most reports being localized to lymph nodes, bone, and
peritoneum [1], [2]. Due to its insidious nature and nonspecific signs and symptoms,
radiological investigation plays a crucial role in the early and correct identification of the
disease. However, owing to its rarity and the fact that it is restricted to some epidemiologic
contexts, imaging findings are easily misdiagnosed as advanced ovarian malignancy or pelvic
inflammatory disease [3]. With this case report [4], we aim at promoting recognition and
understanding of the imaging findings spectra associated with female pelvic TB and
emphasize the need for integration with clinical and laboratory data that are crucial to avoid
misdiagnosis, delay of treatment (allowing disease progression), and surgical exploration.

Case report
An African14-year-old girl was admitted to the pediatric disease department for investigation
of an adnexal mass.
Five months before admission, she immigrated to Portugal from Angola. One month after
being in Portugal, she started feeling fatigue, loss of appetite, daily somnolence, and a
persistent dry cough. She was medicated with an antihistaminic with some improvement of
the cough. As the symptoms persisted, she had some laboratory tests, which showed
microcytic hypochromic anemia that was medicated with iron replacement therapy. Two
months later, she went to the emergency department due to aggravating symptoms, fever (of
irregular pattern), weight loss (4 kg in 1 month), a low abdominal pain, and pain with
inspiration in the right hemithorax. There was no associated past medical or surgical history
and no history of recent disease in her family circle. She had no other signs or symptoms,
namely, no menstrual disturbances (menarcha at 10 years), no urinary, respiratory (the cough
subsided), or gastrointestinal symptoms. On physical examination, a palpable painful
hypogastric mass was found, hard on palpation and of irregular contours. A BCG vaccination
scar was visible.
She had an ultrasound (Fig. 1) that showed a large pelvic mass (about 12 × 8 cm), centered in
the right adnexal area, predominantly cystic, complex, encapsulated, with multiple thin septa
and a solid component that exhibited some vascularity in the Color Doppler study (Fig. 1b),
creating a mass effect on the uterus and bladder. The left ovary was heterogeneous and mildly
enlarged (not shown). There were associated terminal ileum, peritoneal thickening (Fig. 2),
and mild ascitis. Pelvic magnetic resonance (MR; Fig. 3) documented a complex cystic mass,
apparently having its epicenter on the right adnexal area, with high signal in T2W sequences
and intermediate-to-low signal in T1W sequence, with no saturation on fat-suppressed
sequences. The multiple septa were thin, irregular, and had no definite enhancement on post
gadolinium sequences. A solid enhancing component was not confirmed. Upon scrolling the
sagittal images, the lesion suggested to be contiguous with the uterine horn and terminating at
the right ovary, hence a hydropyosalpingitis was neither confirmed nor excluded. There was
some distortion of ileal loops that were hard to characterize as they were retracted and
adherent close together and in apposition with the mass. Despite this, the lesion seemed to be
encapsulated and well defined. Some peritoneal enhancement was noted. No adenopathies
were visible in the pelvis or in the lomboaortic chains. Chest radiograph (Fig. 4a) revealed an
obliteration of the right costophrenic angle extending upwards along the lateral wall,
suggesting a loculated effusion. The corresponding chest ultrasound (Fig. 4b and c) exhibited
a pleural effusionat the right lung base, anechogenic, with some thin septa, extending
superiorly to the middle lung and causing passive atelectasis of the adjacent lung
parenchyma.

Fig. 1
Ultrasound of the pelvis showing a complex lesion, predominantly cystic, centered in the right
adnexal area (a). It appears to be encapsulated with defined margins. There are multiple thin septa and
an apparent solid component in a gravity-dependent location, which exhibited some vascularity in
Color Doppler study (b).
Fig. 2
Ultrasound of the right iliac fossa showing associated ileal bowel wall (a) and peritoneal thickening
(c) with inflammatory features (Color Doppler signal in b and c). MR image of the corresponding area
(d) depicts the thickened ileal bowel loops adjacent to the lesion.

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Fig. 3
MR images of the pelvic lesion. (a) Axial T2-weighted image of the complex lesion, with high signal
intensity and some thin septa. (b) Axial T1-weighted image does not show areas of hyperintensity. (c)
Axial contrast-enhanced T1-weighted image with fat suppression exhibits prominent enhancement of
the lesion's pseudocapsule and peritoneum. (d) Subtraction image does not document any enhancing
solid component within the mass.

Fig. 4
(a) Chest radiograph shows a right loculated pleural effusion. (b, c) Corresponding ultrasound
showing a predominantly anechogenic effusion with some thin septa (b). A large pouch of loculated
pleural fluid (c), with 96 × 45 mm, shows that the fluid is clear despite the overall complex nature of
the effusion.
Laboratory investigations elicited a normal leucogram and C-reactive protein (CRP),
persistence of a microcytic hypochromic anemia (9.1 g/dL) and an elevated CA-125 (576
U/mL, normal values <35 U/mL).
As an ovarian malignant tumor was suspected, she was submitted to an exploratory
laparotomy with the intent of an eventual resection. A large right adnexal whitish mass was
found, occupying the small pelvis, with an uninterrupted capsule, from which adhesions
emanated to the bladder and ileon bowel walls, preventing it from resection. There was
extensive perilesional inflammatory reaction, adherent to the peritoneal fat and lateral wall of
the pelvic region, and multiple lesions with morphology suggestive of implants to the wall of
the ileal loops in contact with the mass and in the peritoneum adjacent to the mass.
The biopsy of the lesion revealed numerous granulomata with giant cells but no caseous
necrosis and no evidence of neoplastic cells. Further laboratory testing revealed an elevated
seric ADA (83 U/L, normal ranges being 4.8-23 U/L). As the results of the initial mass
sample testing were nonspecific, she was submitted to an ultrasound guided fine-needle
aspiration of the cystic lesion and a diagnostic thoracocentesis (also ultrasound guided).
The tuberculin skin testing (TST/Mantoux) was marginally positive (13 mm).
Interferon-gamma release assay (IGRA) QuantiFERON testing was negative but further
testing with IGRA T-SPOT was positive, substantiating the clinical suspicion of tuberculosis.
Pleural liquid analysis revealed a very elevated LDH (1083 U/L, 5 times the normal seric
value) and elevated ADA (188 U/L, normal being < 45 U/L), with normal leucocyte count.
No BK was identified either by PCR or cultural exam.
The mass aspirate was PCR positive for Mycobacterium tuberculosis (MTB). Almost 1
month later, cultural examination became positive for M. tuberculosis.
She started antibacillary therapy with progressive symptomatic and overall improvement.
After 8 months on combined antibacillary therapy, ultrasonography (Fig. 5) exhibited a
residual complex cystic lesion in the right adnexal area, with some distortion of normal
ovarian morphology. CA-125 returned to normal values.

Fig. 5
Pelvic ultrasound, 8 months after antibacillary therapy showing almost complete regression of the
lesion, with a residual 65 mm biloculated lesion.

Discussion
Differential diagnosis of adnexal masses is based on their imaging appearance (cystic vs
solid, enhancement, fat and blood products content, among others) further supported by
ancillary findings suggesting local or distant spread of a malignant process and tumor
markers. Neoplastic etiologies (either benign or malignant) are first considered given the
ominous prognosis usually associated with malignant lesions. Although diagnostic “decision
trees” do include infectious causes (usually in the context of inflammatory pelvic disease),
tuberculosis is a forgotten “infection” not only because extrapulmonary, pelvic tuberculosis is
rare (about 1.5% of TB affected females; Ref. 5) and related to specific epidemiologic
contexts, but also because it rarely presents in a recognizable imaging pattern. Pelvic
tuberculosis has been reported in several case reports coming from the developing countries.
The striking feature in the case we present is the fact that it presented in a child (although
post pubertal, but the majority of literature reports come from women at childbearing age)
and all investigations had no definite suggestion of a diagnosis: the imaging features either in
ultrasound and MR were hard to categorize into a most probable diagnosis, although a very
high suspicion of malignancy was present (eventually a cystadenoma borderline); laboratory
tests had difficulty unmasking the culprit, all together with an elevated CA-125, a misleading
sum of events that led to an unnecessary laparotomy because of the reasonable suspicion of
an ovarian malignancy. The result could have been more devastating if hysterectomy and
salpingo-oophorectomy had been undertaken, as it has occurred in many others cases
reported [7]. This is one further argument stressing the importance of this case promoting
awareness in considering tuberculosis as part of the diagnostic workup of adnexal masses,
especially in patients with a long potential fertile/reproductive life ahead. As presentation is
often so insidious and nonspecific (majority presenting with abdominal pain, infertility, or
abnormal menstrual cycles/bleeding), the diagnosis is highly supported in imaging findings,
but they can be misleading as granulomata spreading throughout the peritoneal surfaces and
omenta may appear as enhancing nodular thickening, suggesting carcinomatosis [3].
Furthermore, the complex adhesions elicited by the inflammatory response distort the
anatomical planes and surrounding structures, hampering the Radiologist's task. In regard to
pelvic tuberculosis, salpingitis is the most frequent form [6] and indeed several features (in
MR) in our case could point to that, although severe pelvic anatomy distortion prevented us
from understanding the nature/origin of the mass (a tubo-ovarian mass being the most correct
label). At ultrasound, tubo-ovarian masses do produce a very complex appearance with wall
thickening, and pseudosolid areas and Doppler interrogation may suggest a solid neoplastic
component. The imaging criteria in which Radiologists rely upon to suspect a malignant
process when presented with a pelvic mass are well represented in pelvic tuberculosis, which
is why the definitive diagnosis relies on histopathology analysis of biopsy specimens.
However, tuberculosis can be overlooked if tuberculoid granulomata are minimally or
noncaseating as observed in our case, so a high index of suspicion must be held to account
and persisted on. Some features could have reinforced an underlying chronic ongoing
infection, such as the persistence of microcytic anemia. Culture and identification of MTB is
still a lengthy process to yield results (2-8 weeks, and with a wide range of sensibility
reported, from 20% to 80%), limiting its clinical utility in an acute setting. IGRA was
particularly helpful overcoming the interference of BCG vaccination and further supporting
the diagnosis. A great confounding factor is still CA-125, whose increased levels are readily
attributed to a malignant ovarian process, and this report also serves to emphasize the fact
that elevated levels of CA-125 can be produced by inflammation of epithelial cells
(peritoneum, pleura and pericardium) explaining why it can be increased in varied situations
such as infections, tuberculosis, endometriosis, Meigs syndrome, menstruation, pericarditis,
pneumonia, and acute pancreatitis [3], [6]. Normalization of CA-125 levels has been
associated with the response to anti-TB therapy [4], which we also verified in our case.
Retrospectively analyzing, the elicited clinical history suggests a primary pulmonary focus
(substantiated by a very elevated ADA in pleural fluid), probably spread by hematogeneous
route to the pelvis, where it had its major expression.
Thus, pelvic tuberculosis remains very deceitful in all approaches, either by clinical, imaging,
histopathology, or laboratory tests. In order to find it, it should be thought of first, and it will
require a comprehensive and integrated data analysis to reach a consistent diagnosis. It
presents as one of a kind but it actually is kind of a lot (of presentations).
Overall, this case reminds us that proficient Radiologists should look at patients as a whole,
integrating clinical and laboratory data with imaging features in order to avoid disconnect
care.
Informed consent: Informed consent was obtained from the parents of the child included in
this case report.
Go to:

Reference
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tuberculosis at a referral TB's Hospital in Tehran, Iran. Mediterr J Hematol Infect
Dis. 2016;8(1)
2. Baghaie N, Khalilzade S, Boloursaz MR, Khodayari AA, Velayati AA. Extra pulmonary
tuberculosis in children: two years study. Acta Med Iran. 2010;48(4):239–243. [PubMed]
3. Sah SK, Shi X, Du S, Li X, Li CH, Shah S. CT findings and analysis for misdiagnosis of
female pelvic tuberculosis. Radiol Inf Dis. 2017;4:19–25.
4. Gagnier JJ, Kienle G, Altman DG, Moher D, Sox H, Riley D. The CARE guidelines:
consensus-based clinical case reporting guideline development. Glob Adv Health
Med. 2013;2(5):38–43.
5. Oge T, Ozalp SS, Yalcin OT, Kabukcuoglu S, Kebapci M, Arik D. Peritoneal tuberculosis
mimicking ovarian cancer. Eur J Obst Gyn Reprod Biol. 2012;162:105–108.
6. Delgado RE, Fernandes DS, Ahouagi AC, Almeida LJ. Peritoneal tuberculosis mimicking
advanced ovarian carcinoma with peritoneal carcinomatosis. Brazilian National Cancer
Institute, Rio de Janeiro - Single Center. JSM Surg Oncol Res. 2017;2(2):1016.
7. Gatongi DK, Gitau G, Kay V, Ngwenya S, Lafong C, Hasan A. Female genital
tuberculosis. Obstet Gynaecol. 2005;7:75–79.
hieme Medical Publishers 333 Seventh Avenue, New York, NY 10001, USA.

Ruptured Abdominal Aortic Aneurysm in a Patient with

Congenital Fused Pelvic Kidney: A Case Report of
Emergency Endovascular Treatment

Claudio Bianchini Massoni , Matteo Azzarone, Danilo Barbarisi, Paolo Perini ,Antonio Freyrie

Abstract

A 90-year-old male developed acute onset of abdominal and lumbar pain due to the rupture of an
11-cm abdominal aortic aneurysm. A congenital fused pelvic kidney perfused by three renal
arteries arising from iliac axes was detected. In an emergent setting, an aorto-uni-iliac endograft
was deployed through right femoral surgical access with occlusion of the upper right renal artery.
An occluder device was placed in the common iliac artery above the renal artery through left
femoral access. A femorofemoral crossover bypass completed the procedure. The patient
developed acute renal failure, with no dialysis necessity. One-month computed tomography
angiography showed procedure success.

Keywords

fused pelvic kidney - abdominal aortic aneurysm - aortic rupture - endovascular procedures

Introduction

Congenital renal abnormalities occur in 3.3 to 11% of the population[1] and include alterations in
the number, shape, and/or site of the organ.[2] In patients with abdominal aortic aneurysm
(AAA), the incidence of congenital renal variants is not well known. In a case series over a 20-year
period, AAAs with congenital kidney anomalies were seen in 25 patients.[3]

In this population, the number and site of origin of renal arteries are extremely variable and, for
this reason, the treatment of AAA is more complex. Assessment of renal arteries (dimension,
stenosis, and thrombus) and renal function (creatinine, serum urea level, and glomerular filtration
rate) is mandatory for optimal planning and to obtain technical and clinical success after any type
of procedure (surgical, endovascular, and hybrid).

Management of ruptured AAA is challenging and perioperative mortality after emergency open or
endovascular treatment reaches 53%.[4]

The aim of this article is to report a case of emergency endovascular treatment of a ruptured AAA
with congenital fused pelvic kidney (CFPK).

Case Presentation

A 90-year-old male presented to the emergency department complaining of severe abdominal and
back pain. The symptoms began 3 hours earlier. The patient was severely obese and under chronic
pharmacological therapy for arterial hypertension and coronary artery disease (previous coronary
artery bypass graft). The abdominal computed tomography angiogram (CTA) showed an 11-cm
AAA with a retroperitoneal hematoma and a posterior wall rupture ([Fig. 1]). The aortic rupture
was partially contained by the vertebral bodies and retroperitoneum. A unique lobulated renal
mass was detected in pelvic region (CFPK or pancake kidney[2]) ([Fig. 2]). Three renal arteries
were detected arising from both iliac axes: one (8 mm in diameter) from the proximal portion of
the right common iliac artery, the second (9 mm in diameter) from the right hypogastric artery,
and the third (4 mm in diameter) from the distal segment of the left common iliac artery ([Fig. 3]).
The proximal neck (between the superior mesenteric artery and the AAA) was 20 mm in length and
23 to 24 mm in diameter with no intraluminal thrombus or wall calcification. The patient was
conscious and hemodynamically stable (systolic blood pressure: 90–100 mm Hg). Considering the
presence of a favorable proximal aortic neck, the partial hemodynamic stability, the age, and the
obesity, an endovascular treatment was preferred. Only aorto-uni-iliac endografts were available
for emergency procedures.

Fig. 1 Computed tomography angiogram showing a voluminous

abdominal aortic aneurysm (11.5 cm in diameter) with rupture of posterior aortic wall (white arrow)
and intra-abdominal hematoma (white asterisk) on the left retroperitoneal region.

Fig. 2 Computed tomography angiogram imaging of congenital

fused pelvic kidney or pancake kidney. (A and B) axial images; red line: renal parenchyma. (C)

Multiplanar reconstruction; red line: renal parenchyma. Fig. 3 (A)

Volume rendering reconstruction of preoperative computed tomography angiogram
(anteroposterior view). (B) Aortic outline and origin of renal arteries.
Preoperative planning was performed using 3mensio Vascular (Pie Medical Imaging) during patient
transport to the operating theater and anesthetic induction.

Under general anesthesia, inguinal longitudinal cut-down was performed bilaterally and the
common femoral arteries were isolated. An Endurant aorto-uni-iliac endograft (Medtronic
Endovascular) was introduced through the right access. The C-arm was rotated at 50°-left anterior
oblique and the preoperative angiogram was performed. The endograft was deployed below the
origin of superior mesenteric artery, using the inframesenteric nondilated aorta as proximal sealing
zone (∼20 mm in length). Two Endurant endograft extensions (Medtronic) were necessary to
obtain sufficient sealing at the right common iliac artery with the occlusion of the upper renal
artery. Through the left access, a Talent occluder was deployed in the proximal left common iliac
artery, above the origin of left renal artery. The completion angiogram showed patency of the
endografts and no endoleak from left iliac axis or renal or lumbar arteries ([Fig. 4]). The procedure
was completed with an extra-anatomical femorofemoral suprapubic bypass graft (8 mm-Silver
Dacron). Operative procedure time was 220 minutes and 120cc of contrast agent was injected
(iomeprol, Iomeron 300; Bracco).

Fig. 4 Anteroposterior digital subtraction angiogram after deployment of

aortouniiliac endograft, two iliac extensions (through right femoral access) and occluder (through
left femoral access); no endoleaks are detected.
Ventilator support was removed after 24 hours and the abdominal and lumbar pain disappeared.
Acute renal failure developed ([Fig. 5] [5]) but no dialysis was necessary. The patient returned to
the ward on the 9th postoperative day and started mobilization. The postoperative duplex
ultrasound showed endograft patency with no endoleak. Renal function gradually improved ([Fig.
5]) and the patient was discharged on the 12th postoperative day.

Fig. 5 Serum levels of creatinine, urea, and estimated glomerular

filtration rate (according to the Chronic Kidney Disease Epidemiology Collaboration Eq.[5]) during
hospitalization.
At 1 month, the renal function remained mildly altered (creatinine serum level: 1.9 mg/dL). After
intravenous hydration, the abdominal CTA showed patency of the aorto-uni-iliac endograft and
femorofemoral bypass graft, sac shrinkage, and no evidence of endoleaks. The pelvic kidney
presented no ischemic lesions and was perfused by two renal arteries (lower right and left) ([Fig.
6]).

Fig. 6 One-month computed tomography angiogram. (A) Volume

rendering shows patency of celiac trunk, superior mesenteric artery, endograft, and femorofemoral
crossover bypass. (B) Patency of lower right renal artery (arising from right hypogastric artery)
(white arrow). (C) Patency of renal artery arising from left common iliac artery, below occluder
device (white arrow).
#
Discussion

Congenital pelvic kidney (CPK) is defined as the presence of one ectopic kidney low lying in the
abdomen.[2] This variant has been reported to be present in 1 to 2,200 to 1 in 3,000
individuals.[6]

Congenital fused pelvic kidney, also called “pancake kidney,” is a rare congenital anatomic variant
consisting of unique lobulated pelvic renal mass of dual parenchymatous systems.[2] During
autopsies, this variant is observed in 1 out 22,000 cases.[7]

Considering patients who underwent major aortic surgery, ∼0.18% presented a CPK.[8] Thus, the
association between AAA and CFPK is extremely rare.

These two anatomical variants are often associated with multiple aberrant renal arteries arising
from the aortic bifurcation and iliac arteries. AAA treatment is technically challenging, and has a
higher risk of kidney failure.

In the current case, endovascular treatment was preferred because of the advanced age and the
severe obesity of the patient.

No bifurcated endografts were available in our center for procedures in an urgent/emergent

setting, so aorto-uni-iliac endografting and femorofemoral bypass were obligatory choices.
Considering the sites of origin of the three renal arteries, the coverage of upper right renal artery
was inevitable to obtain an adequate distal sealing zone. Also in case of left-side deployment of
the aorto-uni-iliac endograft, the upper right renal artery would have been excluded by the
occluder device deployment in the right common iliac artery.

To reduce the risk of persistent type 2 endoleak, a sac embolization may be intraoperatively
performed. This adjunctive procedure was not executed to reduce time of intervention and was not
considered strictly necessary. Indeed, no patent lumbar arteries had been detected at
preoperatory CTA, and the upper right renal artery arises from a nondilated segment making
unlikely a back flow into the aneurysmal sac.

In case of renal anomalies, assessment of preoperative CTA is essential to define their number,
origin, and size. In the literature, sacrifice of accessory renal arteries with diameter < 3 mm is
considered noninfluential on renal function.[9] In the current case, we excluded an 8-mm renal
artery, sparing the other two renal arteries (9 mm and 4 mm in diameter). Transient renal function
worsening was an expected event. Ultimately, no dialytic treatment was required. After 1 month,
mild renal insufficiency persisted.

Several cases of treatment of AAA associated with CPK or CFPK have been reported in literature
([Table 1]).[8] [9] [10] [11] [12] [13] [14] [15] [16] [17] [18] [19] [20] [21] [22] [23] [24] [25] [26] [27] [28] [29] To our
knowledge, the current case is the first reporting of the treatment of a ruptured AAA associated
with the presence of CFPK.

Table 1 Cases of treated abdominal aortic aneurysm associated with pelvic kidney (congenital
pelvic kidney or congenital fused pelvic kidney)

Abbreviations: CFPK, cross-fused pelvic kidney; CPK, congenital pelvic kidney; EV, endovascular;
EVAR, endovascular abdominal aortic aneurysm repair; NA, not available.

Surgical treatment of AAA in patients with pelvic kidney (CPK or CFPK) is more complex than in
the general AAA population. Perfusion of the kidney must be maintained during aortic clamping
and graft sewing. Renal arteries with diameter > 3mm should be reimplanted[9] and perfusion with
cold Ringer solution is necessary in case of cross-clamp time > 40 minutes.[8]
Total endovascular treatment has also been described. Morales and Greenberg[23] treated a
patient with a type IV thoracoabdominal aortic aneurysm associated with a left CPK, deploying a
custom-made fenestrated endograft. A fenestration near the distal bifurcation of the endograft
permitted the perfusion of the CPK through a covered bridging stent. Also, a parallel stent graft
technique has been described to perfuse an ectopic renal artery originating from the AAA in a
patient with a crossover ectopic right kidney fused with the left kidney.[30] A Viabahn–Gore (W. L.
Gore) endograft reinforced with balloon-expandable stents was deployed in a parallel fashion with
an Endurant II–Medtronic endograft iliac limb.

Hybrid treatment has also been reported. In a case of CFPK with renal arteries arising from the
AAA, a double iliac-renal bypass was primarily sutured and an aorto-bifurcated endograft was
deployed to exclude the aneurysm.[28]

In conclusion, in case of CPK or CFPK, endovascular, open surgical, and hybrid treatments of AAA
have been described in literature and are potentially feasible. The most suitable treatment must be
chosen according to the patient's clinical condition and arterial anatomy. Our case demonstrates
that accurate preoperative planning is vital to achieve technical success and to preserve renal
function in this complex subset of patients.

#
#

Conflict of Interest

All authors certify that they have NO affiliations with or involvement in any organization or entity
with any financial interest (such as honoraria; educational grants; participation in speakers'
bureaus; membership, employment, consultancies, stock ownership, or other equity interest; and
expert testimony or patent-licensing arrangements), or nonfinancial interest (such as personal or
professional relationships, affiliations, knowledge, or beliefs) in the subject matter or materials
discussed in this manuscript.

Acknowledgements

None.

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