Journal of Pediatric Gastroenterology and Nutrition
43:256 Y 259 Ó August 2006 Lippincott Williams & Wilkins, Philadelphia
Case Report
Massive Intestinal Bleeding in a Child With Superior Mesenteric
Artery Aneurysm and Gastrointestinal Tuberculosis
Stacy A. Kahn and Barbara S. Kirschner
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Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics,
University of Chicago, Chicago, IL
INTRODUCTION
Superior mesenteric artery (SMA) aneurysms are
exceedingly rare in the pediatric population. They are
most commonly caused by infection or inflammatory
processes and typically present with acute abdominal
pain (1,2). We report the case of a 12-year-old girl with
gastrointestinal tuberculosis (GITB) who presented with
massive lower gastrointestinal (GI) bleeding due to a
bleeding SMA aneurysm. To the best of our knowledge,
this is the only reported case of tuberculosis associated
with an SMA aneurysm in a child and 1 of only 2
reports in patients of any age group (3).
CASE REPORT
A 12-year-old African-American female was trans-
ferred from an outside hospital for evaluation of a
significant painless lower GI bleed of approximately
300 mL bright red blood per rectum. She reported a
2-day history of flank and back pain, and decreased
appetite and oral intake. She had 3 episodes of non-
bloody, nonbilious emesis and nausea. She denied fever,
dysuria or diarrhea. She reported intermittent abdominal
pain for the past several months that occasionally woke
her from sleep. Her past medical history was significant
for a ruptured appendix 4 months prior. Initially, she
was treated conservatively with antibiotics but subse-
quently underwent laparoscopic appendectomy. Her
mother reported intermittent weight loss and gain but
felt this was related to the appendicitis. There was no
family history of inflammatory bowel disease, polyps or
bleeding disorders.
Results of initial laboratory tests were as follows: white
blood cell count 29,000/mm3 with 52% neutrophils, 37%
bands, hemoglobin 4.7 g/dL, platelets 294,000/mm3,
Received September 30, 2005; accepted Febuary 23, 2006.
Address correspondence and reprint requests to Stacy A. Kahn,
Division of Pediatric Gastroenterology, Hepatology, and Nutrition,
Department of Pediatrics, University of Chicago, Chicago, IL. (e-mail:
kahns01@hotmail.com).
256
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prothrombin time 21[, international normalized ratio of
1.6, partial thromboplastin time of 30.6[, bicarbonate
18 mmol/L, calcium of 6.4 mg/dL and an albumin of
1.8 g/dL. Hemoglobin electrophoresis was normal. She
was transfused 3 U of packed red blood cells and given
vitamin K. Blood and urine cultures were negative and
stool studies for Salmonella, Shigella, Campylobacter
and Clostridium difficile were negative but positive for
adenovirus 40/41 and rotavirus. Abdominal x-ray
showed a gasless abdomen and air-fluid levels. Meckel
scan was negative. She was made n.p.o. and started on
famotidine 20 mg IV b.i.d. The patient continued to
have profuse bleeding that required transfusions of
packed red blood cells and fresh frozen plasma.
On hospital day 2, esophagogastroduodenoscopy
(EGD) and flexible sigmoidoscopy were performed.
The EGD was normal. The sigmoidoscopy to 40 cm
showed numerous large clots in the rectum and sigmoid.
The underlying mucosa seemed edematous, and there
were scattered circumscribed ulcers with focal loss of
the normal vascular pattern. She was started on empiric
therapy with ampicillin, gentamicin, metronidazole,
gangcyclovir and methylprednisone 20 mg IV b.i.d. for
possible Crohn disease and cytomegalovirus infection.
A central line was placed and total parenteral nutrition
initiated. Pathology revealed normal antral mucosa that
was Helicobacter pylori negative. Multiple biopsies
from the sigmoid showed mild acute inflammation
without granulomas, architectural distortion, ischemic
changes or viral inclusions. Cultures remained negative
and the antibiotics were discontinued.
The patient continued to have episodic bleeding of 100
to 450 mL daily. Tagged red blood cell scans on hospital
days 6 and 7 were negative. Stool tests for adenovirus 40/
41 and rotavirus were repeated and were positive. On
hospital day 8, on the pediatric surgeon’s recommenda-
tion, she underwent superior mesenteric arteriogram that
demonstrated a small saccular aneurysm from the jejunal
branch in the left upper quadrant fed by the inferior and
superior mesenteric arteries with active bleeding into the
small bowel (Fig. 1). Multiple microcoils were deployed
into the sac, and the feeding branches off the superior
 INTESTINAL BLEEDING IN ANEURYSM AND TUBERCULOSIS 257

and inferior mesenteric arteries were embolized. There

were no further episodes of bleeding, and she was
discharged home on hospital day 12 on a general diet
with famotidine and a prednisone taper. She was given
the steroid taper for a presumptive diagnosis of
inflammatory bowel disease based on her history of
recurrent abdominal pain, anorexia, weight loss, bleed-
ing, bowel wall thickening on CT and biopsies that
demonstrated mild acute inflammation in the sigmoid.
Initial immunomodulatory medications were withheld
given her recent history of an extended antibiotic
course for a perforated appendix and multiple intra-
abdominal abscesses. We had planned to complete her
work-up as an outpatient; however, she was not com-
pliant with follow-up.
The patient returned to our pediatric emergency depart-
ment 4 months later with a temperature of 38.9-C,
epigastric pain, 2 episodes of hematemesis and 1 episode
of hematochezia. She had completed her steroid taper 2
weeks prior. Result of the laboratory tests were as follows:
white blood cell count 18,500/mm3 with 60% neutrophils
and 19% bands, hemoglobin 10.2 g/dL, platelets 573,000/ FIG. 2. Upper GI demonstrating 2 irregular sinus tracts (arrows)
mm3, mean corpuscular volume of 65 Kmc, C-reactive extending from the second portion of the duodenum without
protein 76 ng/mL, erythrocyte sedimentation rate 54 mm/ evidence of inflammatory bowel disease.
h and bicarbonate 21 mmol/L. Liver function tests,
amylase and lipase were normal. Stool was negative for follow through on hospital day 2 showed 2 irregular
occult blood. AntiYSaccharomyces cerevisiae IgG was sinus tracts extending from the second portion of the
41.2 EU/mL (normal G40), antiYSaccharomyces cerevi- duodenum and no evidence of inflammatory bowel disease
siae IgA 18.8 EU/mL, perinuclear antineutrophilic (Fig. 2). Abdominal ultrasound (US) demonstrated 2
cytoplasmic antibody negative and anti-OmpC IgA hyperechoic masses, 1 on the head and 1 on the tail of
was18.5 EU/mL (normal G16.5). the pancreas, multiple small lymph nodes and hypo-
Additional family history revealed 2 grandparents with echoic tubular structures near the hilum of the liver. A
a history of intracranial aneurysms. Computed tomog- purified protein derivative (PPD) skin test was placed
raphy (CT)Yangiogram of the brain showed no evidence and was 15 mm at 36 hours. Upon direct questioning,
of aneurysm formation. Upper GI and small bowel the family now reported several family members with a
history of tuberculosis, including one who died from
related complications. However, the patient had a history
of multiple negative PPD and a normal chest x-ray. Chest
x-ray was repeated and was without evidence of tuber-
culosis. Gastric aspirates were positive for Mycobacte-
rium tuberculosis, and the patient was started on
quadruple therapy with isoniazid, rifampin, pyrazinamide
and ethambutol. A CT performed on hospital day 8
demonstrated numerous retroperitoneal, porta hepatic,
mesenteric and pericecal lymph nodes, some of which
were necrotic, multiple loculated abdominal and pelvic
fluid collections, intraperitoneal free fluid and bowel wall
thickening with fluid-filled loops. The patient’s symp-
toms began to abate, and she was discharged home on
hospital day 10 with quadruple antituberculous therapy.
She continues to be followed by the Pediatric Infectious
Disease service and is asymptomatic.

DISCUSSION

FIG. 1. Superior mesenteric arteriogram with aneurysm (arrow) This case highlights a unique presentation in the
from jejunal branch in left upper quadrant. pediatric population of gastrointestinal tuberculosis

J Pediatr Gastroenterol Nutr, Vol. 43, No. 2, August 2006

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258
causing bleeding from an SMA aneurysm, presenting
with a massive lower GI bleed. SMA aneurysms are
uncommon and even more so in pediatric patients. They
are life threatening when they rupture and cause
profound bleeding with mortality rates close to 50% (1).
Infections such as bacterial endocarditits are the most
common cause of SMA aneurysms in patients less than
50 years old (1). Extensive review of the literature
yielded only a few isolated reports of SMA aneurysms
in children. Michalsky reported a previously healthy
12-year-old girl who presented with severe abdominal
pain and emesis and who was found to have an SMA
aneurysm (4). She ultimately required arterial graft;
however, no underlying cause of the aneurysm was
identified. Volpe et al reported the case of a 14-year-old
girl who developed fever, abdominal pain and a precipi-
tous drop in hemoglobin after mitral valve repair. Ultra-
sound was suggestive of an SMA aneurysm that was
confirmed by surgery. Her aneurysm was due to bacterial
endocarditis (5). Christophe et al also describe an SMA
aneurysm due to mitral valve disease and bacterial
endocarditis in a 6-year-old girl. She presented with a
slow-growing retro-umbilical mass that on laparotomy
was found to be a ruptured SMA aneurysm with a large
intramesenteric hematoma (6).
There are a limited number of reports of tubercu-
lous aneurysms, most of which describe abdominal
aortic aneurysms (7,8). The only pediatric report of a
mycotic aneurysm due to tuberculosis was a 14-year-old
who presented with abdominal pain, chills and weight
loss and was found to have an abdominal aortic
aneurysm. That patient underwent insertion of an aortic
graft but died from postoperative Bhypovolemia[ (9).
We found a single reported case of tuberculosis causing
an SMA aneurysm and catastrophic GI bleeding in the
adult literature. Oran et al described a case of a 41-year-
old woman who presented massive lower GI bleed due
to a mesenteric artery aneurysm caused by GITB (3).
However, this patient’s case was complicated by signifi-
cant immune suppression for orthotopic liver trans-
plantation due to primary biliary cirrhosis. Massive
lower GI bleeding in intestinal tuberculosis is uncommon
and most often due to ulcerations in the terminal ileum,
cecum or colon (10). There have been no reports of
massive lower GI bleeding in children due to GITB.
Although the antral biopsy did not demonstrate patho-
logical abnormality, the CT, US and UGI all reflect
diffuse GI tract involvement.
Among the extrapulmonary manifestations of tuber-
culosis, gastrointestinal presentation is quite uncommon.
A retrospective study by Maltezou et al in 2000
reviewed 102 cases of extrapulmonary tuberculosis in
children and found that the most common presentation
was lymphadenitis (n = 48) at 47%. Only 2% of the
cases (n = 2) were gastrointestinal (11). In addition,
most children with GITB present with vague symptoms
such as abdominal pain, distension, weight loss, fever
J Pediatr Gastroenterol Nutr, Vol. 43, No. 2, August 2006
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KAHN AND KIRSCHNER
and emesis. One small review of GITB in children over
a 16-year period found 8 of 13 cases presented with
abdominal pain and only 4 presented with fever or
vomiting (12). Patients can present with GITB localized
to the small bowel, colon and/or peritoneal cavity. Most
often, GITB is located in the ileocecal area, often
leading to further difficulty in differentiating it from
Crohn disease (13). Endoscopic findings in GITB vary
but include circular ulcers as were seen in our patient.
Other descriptions include nodules, sessile firm polyps
and small diverticula (14,15). Pathological diagnosis of
GITB is difficult because stains are often negative for
the organism and may reveal both caseating and
noncaseating granulomas. Gastrointestinal tuberculosis
may exist without pulmonary disease as evidenced by
our patient. A review by Horvath and Whelan in 1998
found only 20% of cases of GITB are associated with
active pulmonary tuberculosis. In addition, patients with
GITB may have a negative PPD. Up to 14% patients
with GITB have negative PPDs (14). In these cases,
diagnosis is made by gastric aspirates for acid-fast
bacilli, tissue culture, tissue with positive Ziehl-Neelsen
stain or polymerase chain reaction for tubercle bacilli
DNA (15).
Our patient had no significant past medical history,
was immunocompetent and was HIV negative. It is
unclear why an otherwise healthy child would present
with such an aggressive case of gastrointestinal tuber-
culosis. From her endoscopy and imaging tests, we
know that her disease involved portions of the small
bowel and colon from the duodenum to the rectosig-
moid. She also had extensive peritoneal involvement
with lymphadenopathy and fluid collections. In our
opinion, it is likely that her ruptured appendix may have
been the first presentation of disease. Ruptured ap-
pendix has been described as the initial presentation of
GITB (16). Review of the pathology from our patient’s
appendix revealed necrotizing granulomas, although
special stains for acid-fast bacilli were negative. We
believe the positivity for adenovirus 40/41 and rotavirus
was an incidental finding and most likely had little
clinical impact on her course.
Gastrointestinal tuberculosis is known as the great
mimic and is difficult to diagnose. It is well supported in
the literature that Crohn disease is misdiagnosed as
GITB and vice versa; this is of serious consequence
because inappropriate treatment can be detrimental. The
use of common immunosuppressive agents for Crohn
disease can cause significant morbidity in a patient with
tuberculosis. Timely diagnosis remains a crucial issue
and is of great consequence in countries where tuber-
culosis is endemic and in the United States, where rates
of tuberculosis peaked in the 1990s and have remained
relatively high (17). A study by Patel et al of 260
patients in India found that 3.9% of the patients with
Crohn disease were initially diagnosed with GITB (18).
Diagnosis of GITB remains difficult because of the
 INTESTINAL BLEEDING IN ANEURYSM AND TUBERCULOSIS
nonspecific signs and symptoms at presentation and its
overlap with other more common GI diseases. Radio-
logical imaging does not provide a definitive diagnosis,
although CT, US or barium contrast studies may detect
changes consistent with GITB. In addition, false-
negative PPD tests or the lack of pulmonary involve-
ment may further delay the diagnosis.
In essence, this case serves as a reminder that GITB
must be considered early in the differential diagnosis
for patients with severe abdominal pain and GI
bleeding of unclear etiology that may resemble Crohn
disease. GITB should also be included in the differ-
ential diagnosis of a patient with GI bleeding due to a
vascular aneurysm. Furthermore, this case illustrates
that children can develop mycotic SMA aneurysms
that present with massive life-threatening gastrointesti-
nal bleeding.
Acknowledgments: The authors thank Dr Stefano Guanda-
lini for his helpful suggestions and assistance in editing the
article and Dr Jiri Nevoral for his assistance in translating one
of the references into English. We also thank Dr Kate
Feinstein and Dr David Yousefzadeh for their interpretation
of the radiological images.
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