Keywords
Celiac disease - failure to thrive - multiple intussusception
Introduction
Intussusception is the most common cause of small bowel obstruction in children between
the ages of 6 months and 4 years. Most (over 90%) have no lead point and are due to
lymphoid hypertrophy, usually following a viral infection. Other lead points are Meckel’s
diverticula, adhesions, adenitis, trauma, celiac disease, duplications, lipoma, and
inflammatory lesions; small bowel malignancy (either primary or metastatic) may account
for less than one-third of adult intussusceptions. The image-guided reduction can
be performed using a pneumatic technique or by contrast enema, under fluoroscopy or
ultrasound guidance.
Case History
A 3-year-old girl was brought to the department of pediatrics with a complaint of
intermittent abdominal pain for 6 months, vomiting, and increased frequency of stool
for 1 month and fever for 7 days. There was a history of similar complaints in the
past.
On examination, the patient was cachexic and underweight (8.6 kg). Tongue, nail, and
conjunctiva were pale with edema on bilateral legs. In the abdomen examination, there
was abdominal distension; however, no evidence of hepatosplenomegaly and central nervous
system (CNS), cardiovascular system (CVS), and respiratory system (RS) examinations
were normal.
Total leucocyte count was 6200/cmm (within normal limits) and hemoglobin was reduced
(10.7 g%). Serum sodium and potassium were reduced (130 mmol/dL and 1.7 mmol/dL, respectively).
Renal and hepatic function tests were within normal limits.
X-ray abdomen erect was showing multiple abnormal air-fluid levels in the abdomen
[Figure 1]. Ultrasound findings were showing excessive gaseous abdomen with few dilated bowel
loops and single intussusception in the left upper abdomen; however, no lead point
of intussusception was identified [Figure 2]. Due to severe abdominal pain, a CT of the abdomen was done to look for bowel ischemia
secondary to bowel obstruction and to look for a lead point of intussusception if
any. CT was showing a total of five small bowel intussusceptions [Figure 3].
Figure 1: X-ray abdomen erect showing multiple abnormal air-fluid levels and gas-filled bowel
loops in the abdomen
Figure 2: Ultrasound showing well-defined round to oval heterogeneously hypoechoic lesion with
internal vascularity giving typical bowel within bowel appearance (Target sign) suggestive
of intussusception
Figure 3: CT abdomen with contrast reformated coronal images showing telescoping of proximal
bowel loops into distal loops at five different sites corresponding with multiple
intussusceptions
Her IgA antibodies to tissue transglutaminase were done to look for the cause of failure
to thrive and its titer raised significantly.
Electrolyte imbalance was corrected, a gluten-free diet with nutritional supplements
for anemia and malnutrition was started and symptoms were resolved within 7 days without
surgical management. A follow-up ultrasound of the patient was done and did not reveal
any abnormality.
Discussion
Intussusception as a presenting symptom of pediatric celiac disease has been very
rarely reported.[1] Although intussusception is the most common cause of intestinal obstruction in children,
celiac disease is a frequent small bowel disease they rarely had been reported in
association with each other.[2]
In most children (90%) with intussusception, the lead point could not be found. But
it is due to lymphoid hypertrophy, usually following a viral infection. Other lead
points in children can be Meckel’s diverticula, adhesions, trauma, celiac disease,
enteric duplications, lipoma, and inflammatory polypoidal lesions.[3]
In the past decades when barium studies were used more commonly in the diagnosis of
malabsorption, transient intussusception was seen in 20% of patients with proven celiac
disease.[4] Reilly et al.[1] also found that intussusception was far more common among children with untreated
celiac disease than in the general pediatric population. Among 254 children with celiac
disease, 1.2% experienced the intussusception <9 months before their diagnosis with
celiac disease compared with 0.07% of children of their institution in the same time
period. They concluded that the diagnosis of celiac disease should be considered in
children with intussusception, even in the absence of growth failure.
Borkar VV et al. has stated that intussusception is frequently (25%) seen in children with newly
diagnosed celiac disease, generally asymptomatic and resolves spontaneously on gluten
free diet. It is often associated with more severe disease.[5]
Conclusion
Workup for diagnosis of celiac disease should be considered in children whose initial
ultrasound or CT scan reveals intussusception especially in the presence of growth
failure. So that unnecessary surgical intervention for the treatment of intussusception
can be prevented.
Declaration of patient consent
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understand that their names and initials will not be published and due efforts will
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