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Enteric neuropathology of congenital intestinal obstruction:A case report

World Journal of Gastroenterology 2006年第32期12卷 5229-5233页

作者： Giovanni Di Nardo Vincenzo Stanghellini Salvatore Cucchiara Giovanni Barbara Gianandrea Pasquinelli Donatella Santini Cristina Felicani Gianluca Grazi Antonio D Pinna Rosanna Cogliandro cesare cremon Alessandra Gori Roberto Corinaldesi Kenton M Sanders Roberto De Giorgio Department of Pediatrics Pediatric Gastroenterology Unit University of Rome 'La Sapienza' Rome Italy Department of Internal Medicine and Gastroenterology and Centro di Ricerca Biomedica Applicata (CRBA) Rome Italy 不详 Department of Pathology and Department of Surgery and Transplantation University of Bologna Bologna Italy Department of Physiology and Cell Biology University of Nevada School of Medicine Reno Nevada United States

Experimental evidence indicates that chronic mechanical sub-occlusion of the intestine may damage the enteric nervous system （ENS）, although data in humans are lacking. We here describe the first case of enteric degenerative neuropathy related to a congenital obstruction of the gut. A 3-year and 9-mo old girl began to complain of vomiting, abdominal distension, constipation with air-fluid levels at plane abdominal radiology. Her subsequent medical history was characterized by 3 operations： the first showed dilated duodeno-jejunal loops in the absence of occlusive lesions; the second （2 years later） was performed to obtain full-thickness biopsies of the dilated intestinal loops and revealed hyperganglionosis at histopathology; the third （9 years after the hyperganglionosis was identified） disclosed a Ladd＇s band which was removed and the associated gut malrotation was corrected. Repeated intraoperative full-thickness biopsies showed enteric degenerative neuropathy along with reduced interstitial cells of Cajal network in dilated loops above the obstruction and a normal neuromuscular layer below the Ladd＇s band. One year after the latest surgery the patient tolerated oral feeding and did well, suggesting that congenital （partial） mechanical obstruction of the small bowel in humans can evoke progressive adaptive changes of the ENS which are similar to those found in animal models of intestinal mechanical occlusion. Such ENS changes mimic neuronal abnormalities observed in intestinal pseudoobstruction.

关键词： Enteric neuropathy Chronic intestinal pseudo-obstruction Congenital intestinal obstruction Ladd's band Enteric nervous system

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