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[Long-term collagenous sprue--remission with a gluten-free diet]
M Holtmann1, A von Herbay, P R Galle
1Medizinische Klinik IV, Abteilung für Gastroenterologie, Universität Heidelberg.
Abstract:
We report on a 56-year-old caucasian female, 154 cm, 38 kg, who was admitted to the hospital in extremely poor, cachectic condition with pretibial edemas, ascites and pleural effusion with a history of severe watery diarrhea up to 15 times daily. The patient's history with permanent meteorism, recurrent episodes of watery diarrhea and dystrophy dates back to her early childhood. Multiple biopsies from the distal part of the duodenum showed a complete atrophy of villi with an extensive subepithelial layer of collagenous fibers. No antigliadin or antiendomysial antibodies could be detected. Under a strictly gluten-free diet, the patient's symptoms quickly improved. During the follow-up of one year, no recurrence of diarrhea and meteorism has been seen. The patient showed a continuous improvement of her general condition and nutritional state with a weight gain to 50 kg. Repeated control biopsies still revealed a pronounced villous atrophy. However, no subepithelial collagenous fibers were detectable. A diagnosis fitting to the patient's clinical symptoms of severe malabsorption, would be the rare "collagenous sprue", which is considered to be gluten-insensitive. However, the patient's good response to a gluten-free diet raises the possibility, that the collagenous layer represents a manifestation of more severe forms of common celiac disease. In this case, it would not be necessary to regard collagenous sprue as a distinct entity.