Celiac Disease: An Uncommon Cause of Recurrent Intussusception

Abstract

Celiac disease, one of the most common causes of intestinal malabsorption in childhood, is a mucosal reaction of the small intestine to gluten (1). The characteristic mucosal lesions, degeneration of epithelium and the cuticular layer, villous atrophy, and lymphoplasmocytic infiltration result in a diminished hydrolytic and resorbent function of the small intestine. Typical symptoms of general malabsorption are failure to thrive, large pungent stools, and a distended abdomen. The results of malnutrition are hypoproteinemia, iron deficiency, coagulopathy, and vitamin D deficiency. The symptoms differ in severity. Whereas constipation or intermittent ileus-like symptoms such as vomiting and stool retention with typical radiologic findings can occur in celiac disease, intussusception is rare (2,3). CASE REPORT A 3½-year-old girl with abdominal distension and diarrhea for 6 months had recurrent colicky abdominal pain for 2 weeks that always stopped spontaneously after several hours. On clinical examination during which the patient felt well, there was distended abdomen with increased intestinal peristalsis without other pathologic signs. Growth and weight were on the 10th percentile. Endomysium-immunoglobulin (Ig)A and IgG-antigliadin-antibodies were elevated (4-6). Other parameters were normal. During ultrasound performed while the patient was in abdominal pain, general thickening of the intestinal wall with high echogenicity (7) was seen as well as increased intestinal peristalsis and multiple presumably ileo-ileal intussusceptions (Figs. 1 and 2). These intussusceptions were seen for several days and reduced spontaneously. The mucosa of the small intestine showed villous atrophy and diminished disaccharidases and fructaldolases. Two weeks after being placed on a gliadin-free diet the girl was free of colicky complaints. Subsequently, on repeated ultrasound there was no sign of intussusception. DISCUSSION In childhood, intussusception usually occurs without obvious cause. Common triggers are: Meckel diverticulum, polyps, tumors, swelling of lymph nodes, cystic fibrosis, and Schoenlein-Henoch purpura. A correlation between intussusception and celiac disease is hardly mentioned in pediatric literature, whereas in adulthood, this combination is repeatedly described (3,8). Typical for intussusception of the small intestine seems to be a transient course with mild symptoms. One cause appears to be a disturbed peristalsis in the hypotonic intestinal loop (8). In this case, the correlation between celiac disease and repeated intussusception in different parts of the small intestine is obvious, especially because the recurrent intussusception stopped after starting the patient on a gliadin-free diet. Particularly in recurrent intussusception, celiac disease should be considered as a cause.FIG. 1.: Ultrasound showing thickening of the intestinal wall with high echogenicity and hyperperistalsis.FIG. 2.: Ultrasound showing three intussusceptions.