Chilaiditi syndrome in pediatric patients - Symptomatic hepatodiaphragmatic interposition of colon: A case report and review of literature

Abstract

Background: Chilaiditi syndrome is a rare disorder characterized by the hepatodiaphragmatic interposition of the intestine.

Case summary: Here we report a case of a 12-year-old male who was admitted to the pediatric intensive care unit secondary to abdominal pain and severe respiratory distress. He was treated conservatively but the symptoms persisted requiring a surgical approach. While there have been several cases of Chilaiditi syndrome reported in adults, there is a scarcity of cases reported in the pediatric population. Our review of the literature found only 30 pediatric cases, including our reported case, with Chilaiditi syndrome, 19 (63%) of which were male. The median age of diagnosis was 4.5 years old with an interquartile range of 2.0-10.0 years. In our review, we found that the most common predisposing factors in children are aerophagia (12/30 cases) and constipation (13/30 cases). Ninety percent of the cases presented with complete intestinal interposition, in 100% of which, the colon was involved. Three of the 30 cases were associated with volvulus.

Conclusion: In the pediatric population, conservative (21/30 cases) and surgical (8/30 cases) treatment approaches have produced satisfactory outcomes for all the patients, regardless of approach.

Keywords: Abdominal pain; Case report; Colon; Constipation; Dyspnea; Rare diseases; Respiratory insufficiency.

Figure 1

Imaging of abdomen and pelvis of a 12-year-old male with Chilaiditi syndrome and constipation. A: Computerize tomography. Marked air and fecal retention of the entire colon with colonic interposition above the liver with displacement of the liver leftwardly. This phenomenon indicates segmental agenesis of the right lobe of the liver and relaxation of the hepatic suspensory ligament; B: Plain X-ray. Markedly greater than average amount of fecal material particularly in the rectosigmoid colon. Few air fluid levels in the distal small bowel and air filled colonic loops that reach the right hemidiaphragm.