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Unresponsive Enteropathy Associated with Circulating Enterocyte Autoantibodies in a Boy with Common Variable Hypogammaglobulinemia and Type I Diabetes

1988; Lippincott Williams & Wilkins; Volume: 7; Issue: 4 Linguagem: Inglês

10.1097/00005176-198807000-00022

1536-4801

Carlo Catassi, Rita Mirakian, G Natalini, Andrea Sbarbati, Saverio Cinti, Giovanni V. Coppa, P. L. Giorgi,

Celiac Disease Research and Management

Summary: A 13-year-old boy with common variable hypogammaglobulinemia and type I diabetes developed a severe enteropathy that proved to be unresponsive to any treatment, including total parenteral nutrition. No evidence of known etiologies of malabsorption and/or secretory diarrhea was found in this subject. A high titer of complement-fixing enterocyte autoantibody was persistently found in the patient's serum. These features suggest that the enteropathy of this primarily immunodeficient subject had an autoimmune origin. A cycle of cyclophosphamide failed to show any amelioration of the diarrhea or a significant decrease in the autoantibody titer.