Open AccessCyclosporin-Responsive Enteropathy and Protracted Diarrhea
Author(s) -
Mark G. Coulthard,
Jeffrey Searle,
M. K. Patrick,
Peter J. Ravenscroft,
G. J. Cleghorn,
R. W. Shepherd
Publication year - 1990
Publication title -
journal of pediatric gastroenterology and nutrition
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.206
H-Index - 131
eISSN - 1536-4801
pISSN - 0277-2116
DOI - 10.1097/00005176-199002000-00021
Subject(s) - medicine , enteropathy , diarrhea , parenteral nutrition , malabsorption , gastroenterology , enteral administration , immunology , villous atrophy , disease , coeliac disease
We describe a child born to unrelated parents who developed severe protracted secretory type diarrhea associated with subtotal villus atrophy and intestinal inflammation at the age of 19 months. No infectious, metabolic, or anatomical basis for this condition was identified and the child required total parenteral nutrition for a period of 18 months despite trials of special enteral formulas, steroids, and anti-inflammatory agents. This refractory "enteropathy" responded dramatically to the introduction of cyclosporin, with cessation of the secretory diarrhea, recovery from the enteropathy, and cessation of parenteral nutrition. The symptoms relapsed when cyclosporin was briefly discontinued and improved following reintroduction of this drug. This experience suggests a role for immune factors in the pathogenesis of the enteropathy in this case and that a trial of cyclosporin is worthy of consideration in similar cases.