Acute segmental obstructing ileitis in a Caucasian boy.

Abstract

To the Editor: Acute segmental obstructing enteritis (ASOE) is an uncommon inflammatory condition, usually involving the jejunum, limited to the Third World and usually related to malnutrition (1,2). To the best of our knowledge, this condition has been described mostly by Asiatic authors from China, Goa, India, and Thailand (1,2). Hung-Chang Lee et al. have recently reported a group of 15 children with ASOE (3). All of their cases had a good nutritional status. We have recently observed an acute enteritis with selective involvement of the ileum, causing partial upper intestinal obstruction in a 3-year-old French boy with a good nutritional status. Case report. A 3-year-old boy was admitted in April 1992 because of acute severe abdominal pain, bilious vomiting, and hypothermia (35°C). Before the onset of his illness, he was in good health, well nourished, and had no socioeconomic problems. On admission to another hospital, his physical examination revealed a distended silent abdomen. The boy was dehydrated and showed signs of an infection. He had no rectal bleeding. Laboratory results showed a hemoglobin of 13.4 g/100 ml and a white blood cell count of 33,000/mm3, with 67% polymorpho-nuclear cells. Serum electrolyte, glucose, protein, and immunoglobulin analyses were within normal limits. The coagulation profile including prothrombin time, partial thromboplastin time, fibrinogen, platelets, coagulation factors, proteins C and S, and antithrombin 3 was normal. An abdominal x-ray showed dilatation and thickening of the small bowel. Stool cultures failed to grow any pathogenic bacteria and examination of stool for virus (rotavirus, adenovirus) and parasites was negative. Culture of aspirate was also negative for Giardia lamblia. Acute and convalescent serum titers for Salmonella and Campylobacter jejuni were also negative. In addition, Clostridium difficile toxin and culture were negative. Cow's milk allergy was ruled out. Treatment began with intravenous fluids, parenteral antibiotics, and nasogastric aspiration. Due to increasing signs of toxemia, diffuse peritonitis, and subacute obstruction, surgical intervention was necessary. The preoperative diagnosis was acute appendicitis or intestinal obstruction associated with peritonitis. Exploratory laparotomy revealed a few distinct zones of inflammation and necrosis in the ileum. Ischemic changes were noted throughout the ileum, sparing only the distal 15 cm. The jejunum was not involved. The affected segment was resected (1 m15) and jejunostomy performed. The remaining small bowel was made of 100 cm of jejunum and 15 cm of distal ileum. The mesenteric arteries were found to be pulsatile, with no evidence of arterial or venous thrombosis. The mesenteric lymph nodes throughout the small bowel were normal in size and appearance. Microscopic examination of serial sections of the ileum showed ulceration loss of mucosa and revealed an acute inflammatory infiltrate in the mucosa and superficial layers of the submucosa (Fig. 1). There were areas of focal inflammation as well as diffuse necrosis of the mucosa extending to a variable depth to the submucosa and the muscularis. There was no evidence of chronic inflammatory or granulomatous changes and no evidence of vascular involvement. After a 2 week-period of total parenteral nutrition, oral feeding with an elemental diet was introduced and gradually increased. Because of an hyperproductive ileostomy outflow, the patient was started on a home parenteral nutrition program at 3.5 years of age. Since improvement of digestive function progressively occurred within a few months, jejunostomy could be closed out at 4.5 years of age and parenteral nutrition stopped at the age of 5 years. This case can be included in the group of acute segmental obstructing enteritis. In fact, the clinical presentation was acute intestinal obstruction, and abdominal exploration revealed segmental zones of congestion and necrosis in the ileum. Other causes of enteritis in children were ruled out such as Crohn's disease, sarcoidosis, chronic granulomatous disease, Behcet disease, and lymphoma. Histological findings of the involved intestine revealed nonspecific inflammation and necrosis without granulomatous changes. Our patient was healthy before the onset of the disease. He has been followed for 2 years, remains entirely asymptomatic, and appears to have completely recovered. The present case illustrates some interesting points. This disease affects predominantly the jejunum, and the terminal ileum is the second most likely segment to be affected (1-3). In our case, inflammatory lesions involved the ileum and did not affect the jejunum, the terminal ileum, or the colon. The disease was very extensive and severe, requiring a very long follow-up and a 2-year period of home parenteral nutrition. This case confirms that ASOE is not limited to the Third World or to Asia, nor is it always associated with malnutrition, and it can occur in developed countries (4). L. Michaud; D. Turck; F. Gottrand; S. Ategbo; G. Dubar; J. P. Farriaux Pediatric Gastroenterology and Nutrition Unit Department of Pediatrics Hôpital Huriez Lille, FranceFIG. 1.: Gross section of the affected ileum wall, showing erosion of the epithelium, necrosis, edema, and diffuse inflammation of the mucosa and submucosa.