Abnormal layering of muscularis propria as a cause of chronic intestinal pseudo-obstruction: A case report and literature review.

Napat Angkathunyakul, Suporn Treepongkaruna, Sani Molagool, Nichanan Ruangwattanapaisarn

Journal: World journal of gastroenterology 2016;21(22):7059-64

PMID: 26078585

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Abstract

Visceral myopathy is one of the causes of chronic intestinal pseudo-obstruction. Most cases pathologically reveal degenerative changes of myocytes or muscularis propia atrophy and fibrosis. Abnormal layering of muscularis propria is extremely rare. We report a case of a 9-mo-old Thai male baby who presented with chronic intestinal pseudo-obstruction. Histologic findings showed abnormal layering of small intestinal muscularis propria with an additional oblique layer and aberrant muscularization in serosa. The patient also had a short small bowel without malrotation, brachydactyly, and absence of the 2(nd) to 4(th) middle phalanges of both hands. The patient was treated with cisapride and combined parenteral and enteral nutritional support. He had gradual clinical improvement and gained body weight. Subsequently, the parenteral nutrition was discontinued. The previously reported cases are reviewed and discussed.

Address: Napat Angkathunyakul, Department of Pathology, Ramathibodi Hospital, Mahidol University, Bangkok 10400, Thailand.

DOI: 10.3748/wjg.v21.i22.7059

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MeSH Terms: Biomarkers, Biopsy, Brachydactyly, Chronic Disease, Cisapride, Digestive System Abnormalities, Enteral Nutrition, Fingers, Gastrointestinal Agents, Humans, Immunohistochemistry, Infant, Intestinal Pseudo-Obstruction, Intestine, Small, Male, Muscle, Smooth, Parenteral Nutrition, Short Bowel Syndrome, Treatment Outcome, Weight Gain