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Collagenous Sprue Successfully Treated with Tioguinine


celiac sprue successfully treated with thioguanine. Photo: CC--Rob Ellis

Celiac.com 07/11/2016 - Collagenous sprue is a rare form of small bowel enteropathy characterized by a thickened basement membrane and is considered to be directly related to celiac disease.

Doctors have numerous treatment strategies for celiac sprue, but there is currently no effective standardized therapy. One medical team recently described four cases of celiac sprue and proposes thioguanine (6-TG) treatment, based on their results.

The research team included Tom van Gils, Tine van de Donk, Gerd Bouma, Foke van Delft, E Andra Neefjes-Borst, and Chris JJ Mulder. They are variously affiliated with the Department of Gastroenterology and Hepatology, VU University Medical Center, Amsterdam, The Netherlands, Department of Pathology, VU University Medical Center, Amsterdam, The Netherlands.

The team reviewed 4 cases of celiac sprue. They got their data from the prospective database of patients referred to their celiac centre. The team had an expert pathologist evaluate the small bowel biopsies.

None of the patients had ever shown celiac-specific antibodies, and all were negative for HLA-DQ2 and HLA-DQ8 phenotype. Three patients were treated with a combination of 6-TG and budesonide, and 1 patient received 6-TG only. All patients improved remarkably.

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They found normalized thickening of the basement membrane in 2 patients, and complete histological improvement, including full recovery of villi, in 1 patient. In the third patient, the thickened basement membrane was only very focally recognized. The thickened membrane remained in the last patient, likely due to the short follow-up time.

Celiac sprue should be separated from celiac disease. Based on the lack of typical HLA phenotyping and the absence of celiac-specific antibodies, there seems to be no relation with celiac disease in these four patients.

A promising treatment option might be 6-TG with or without budesonide. Larger study groups are needed to develop an effective standardized treatment for celiac sprue.

This is exciting for folks with celiac sprue, as they previously had no good treatment options at all.

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