Crohn Disease in Early Childhood

To the Editor: In a short communication (J Pediatr Gastroenterol Nutr 2000;30:461–3), Cohen et al. reported the occurrence in one Bedouin family of four cases of infantile Crohn disease. We think that such reports are important. First, they increase the attention of clinicians to the possibility of Crohn disease in infants with serious gastrointestinal problems. Second, they may lead to elucidation of the relative importance of inheritance and environmental factors in the etiology of Crohn disease. We report a recent similar case occurring in a 3-month-old breast-fed infant. MNP, a male infant of Danish ethnic origin, was admitted to our department because of intermittent fever and diarrhea starting at 3 months of age. Failure to thrive was evident at the clinical examination, and aphthous ulcerations were noticed in his mouth. Blood biochemistry revealed anemia, hypoalbuminemia, and elevated erythrocyte sedimentation rate. Stool specimens for bacterial pathogens and parasites including electron microscopy were negative. A normal nitroblue tetrazolium test ruled out chronic granulomatous disease. At the age of 4.5 months, colonoscopy showed pancolitis with increased vascularity and aphthous ulcerations. Histologically, a mixed acute and chronic inflammation and crypt deformations were found, suggesting inflammatory bowel disease. Upper endoscopy including a small intestinal biopsy was normal. A small intestinal follow-through barium study was normal. He was treated with antibiotics (ciprofloxacin) and subsequently with 2 mg/kg/day prednisolone, but the clinical symptoms persisted and failure to thrive continued. When the patient was 5 months old, a control colonoscopy was complicated by a perforation of the sigmoid colon, and a laparotomy was performed. Because of severe macroscopic inflammation, a resection including the left transverse colon and the descending colon, but excluding the sigmoid part, was performed. The rest of the intestinal tract appeared normal. An end transversostomy with closure of the sigmoid colon was performed. Microscopy showed severe, ulcerative, transmural inflammation with deep fissures. The remaining mucosa revealed alteration in crypt architecture and crypt abscesses. Nerve plexuses in the tunica muscularis were hyperplastic. No granulomas were demonstrated. Histologic diagnosis was Crohn disease-like inflammatory bowel disease (Fig. 1). Micrograph showing severe ulcerative transmural inflammation with fissures and pseudopolyposis. Hematoxyleine Eosin (×25). Postoperatively, the patient was given prophylaxis with mesalazine by mouth. He has remained well and showed complete catch-up on the growth chart. One year later, endoscopy showed normal mucosa in the proximal as well as distal colon and, subsequently, the stoma was closed. He is now 24 months old, has remained well, and passes normal stools. Inflammatory bowel disease in very young children is very rare. Apart from the report of Cohen et al. and the case reports referenced by them, we are aware of two epidemiologic studies from Scandinavia giving population-based data on the subject. In a Swedish study (1) that included 639 children with inflammatory bowel disease, three children (0.5%) were younger than 2 years of age. One of these children had Crohn disease. In a similar Danish report (2) of 103 children, five (5%), including one with Crohn disease, were reported to be younger than 2 years. Recent Canadian studies found that 1% of pediatric Crohn disease patients experienced their initial symptoms during their first year of life (3), and symptoms may begin while the infants are breast-fed exclusively (4). An Italian report included an infant with apparent Crohn disease starting at 1 month of age (5). The causes of Crohn disease remain unknown, but genetic and environmental factors are believed to act in concert for clinical disease to develop. We think that reports like ours may be helpful in future attempts to elucidate the pathogenesis of inflammatory bowel disease and to identify the aggravating factors that determine early onset in infancy. Zitta Barrella Harboe Anders Paerregaard Birgit Fischer Hansen Peter Hesselfeldt

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