Cyclosporin-responsive enteropathy and protracted diarrhea. 1990

M Coulthard, and J Searle, and M Patrick, and P Ravenscroft, and G Cleghorn, and R Shepherd
Department of Gastroenterology and Nutrition, Royal Children's Hospital, Brisbane, Australia.

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.

UI MeSH Term Description Entries
D007231 Infant, Newborn An infant during the first 28 days after birth. Neonate,Newborns,Infants, Newborn,Neonates,Newborn,Newborn Infant,Newborn Infants
D003524 Cyclosporins A group of closely related cyclic undecapeptides from the fungi Trichoderma polysporum and Cylindocarpon lucidum. They have some antineoplastic and antifungal action and significant immunosuppressive effects. Cyclosporins have been proposed as adjuvants in tissue and organ transplantation to suppress graft rejection. Cyclosporines
D003968 Diarrhea, Infantile DIARRHEA occurring in infants from newborn to 24-months old. Infantile Diarrhea,Diarrheas, Infantile,Infantile Diarrheas
D004751 Enteritis Inflammation of any segment of the SMALL INTESTINE. Enteritides
D005260 Female Females
D006801 Humans Members of the species Homo sapiens. Homo sapiens,Man (Taxonomy),Human,Man, Modern,Modern Man

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