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    Collagenous Sprue Successfully Treated with Tioguinine


    Jefferson Adams

    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.


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    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.

    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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  • About Me

    Jefferson Adams is a freelance writer living in San Francisco. He has covered Health News for Examiner.com, and provided health and medical content for Sharecare.com. His work has appeared in Antioch Review, Blue Mesa Review, CALIBAN, Hayden's Ferry Review, Huffington Post, the Mississippi Review, and Slate, among others.

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  • Related Articles

    Jefferson Adams
    Celiac.com 09/12/2012 - A team of researchers recently evaluated tioguanine as a treatment for refractory celiac disease type I. The very small study indicates that tioguanine, a thiopurine derivative, offers easy, efficient treatment for refractory celiac disease, compared with current treatment regimens.
    The research team included G. J. Tack; D. P. van Asseldonk; R. L. J. van Wanrooij; A. A. van Bodegraven; and C. J. Mulder.
    Refractory celiac disease type I is a form of celiac disease in which patients show resistance to a gluten-free diet, and suffer persistent or recurring intestinal villous atrophy, along with symptoms of malabsorption.
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    For their study, the research team set out to assess how well patients with refractory celiac disease type I tolerate tioguanine, and how effective it is in relieving symptoms.
    The team studied a group of twelve patients with refractory celiac disease type I, who were treated with tioguanine between June 2001 and November 2010, including a follow-up period of at least 1 year.
    The team assessed and recorded adverse events, laboratory values, 6-thioguanine nucleotide concentrations and rates of both clinical and histological response at baseline and during follow-up.
    They noted that the average tioguanine treatment lasted 14 months. Ten patients tolerated long-term tioguanine treatment, while two patients discontinued treatment due to adverse reactions.
    The team found no nodular regenerative hyperplasia of the liver. Follow-up showed clinical and histological response in 83% and 78% of patients, respectively. Overall, patients decreased corticosteroid reliance by 50%.
    Because of its higher histological and similar clinical response rates compared with current treatments, tioguanine seems to be a good drug for treating refractory celiac disease type I.
    Source:
    Alimentary Pharmacology & Therapeutics. 2012;36(3):274-281.

    Jefferson Adams
    Celiac.com 01/15/2014 - Complicated celiac disease is uncommon, but patients have high death rates, say a team of researchers, who recently set out to better understand the epidemiology of complications in patients with celiac disease.
    The research team included F. Biagi, P. Gobbi, A. Marchese, E. Borsotti, F. Zingone, C. Ciacci, U. Volta, G. Caio, A. Carroccio, G. Ambrosiano, P. Mansueto, G.R. Corazza. They are affiliated with the Coeliac Centre/First Department of Internal Medicine at the Fondazione IRCCS Policlinico San Matteo at the University of Pavia in Italy.
    The team conducted a retrospective multi-center case-control study based on collection of clinical and laboratory data. They looked at incidence of complicated celiac disease among celiac patients directly diagnosed in four Italian centers.
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    Between January of 1999 and October 2011, the team followed-up on 1840 adult celiac patients, for a total of 7364.3 person-years. They found that fourteen patients developed complications to they celiac disease.
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    Source:
    "Dig Liver Dis. 2013 Nov 19. pii: S1590-8658(13)00624-5. doi: 10.1016/j.dld.2013.10.010.

    Jefferson Adams
    Celiac.com 04/07/2014 - Histologically non-responsive celiac disease (NRCD) is a potentially serious condition found in celiac disease patients who suffer persistent villous atrophy despite following a gluten-free diet (GFD).
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    They also found that anti-dGP IgG measurement by ELISA discriminated between NRCD and responsive celiac disease patients with 87% sensitivity and 89% specificity.
    Most importantly, they found that dGP antibody levels correlated with the severity of mucosal damage, meaning that IgG dGP levels may be useful in monitoring small intestinal mucosal recovery on a GFD in NCRD patients.
    The team found that celiac patients with NRCD can be spotted by their increased levels of anti-dGP IgG antibodies even when the patients are following strict gluten-free diets
    Lastly, they feel that anti-dGP IgG assays may be useful for monitoring mucosal damage and histological improvement in celiac disease patients on a strict GFD.
    Source:
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    Jefferson Adams
    Celiac.com 05/24/2016 - People with type II refractory celiac disease (RCD), suffer from severe malabsorption syndrome and face a poor prognosis, as there is currently no effective treatment.
    Prompted by the regenerative and immune-influencing properties of mesenchymal stem cells (MSCs), a research team recently set out to assess the viability, safety, and efficacy of a series of infusions of autologous bone marrow-derived MSCs in a 51-year-old woman with type II RCD.
    The research team included R Ciccocioppo, A Gallia, MA Avanzini, E Betti, C Picone, A Vanoli, C Paganini, F Biagi, R Maccario, and GR Corazza. They are variously affiliated with the Department of Internal Medicine, Fondazione IRCCS Policlinico San Matteo and Università degli Studi di Pavia, the Department of Internal Medicine, Fondazione IRCCS Policlinico San Matteo and Università degli Studi di Pavia, the Cell Factory and Research Laboratory, Department of Pediatrics, Fondazione IRCCS Policlinico San Matteo and Università degli Studi di Pavia, the Clinic Cytometry Laboratory, Department of Hematology, Fondazione IRCCS Policlinico San Matteo and Università degli Studi di Pavia, Department of Molecular Medicine, Fondazione IRCCS Policlinico San Matteo and Università degli Studi di Pavia, all in Pavia, Italy.

    The team began by isolating, expanding, and characterizing mesenchymal stem cells using standard clinical protocols. For each patient, the team arranged to monitor malabsorption indexes, mucosal architecture, and percentage of aberrant intraepithelial lymphocytes at the time of enrollment, at each infusion, and after 6 months.
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    Source:
    Mayo Clin Proc. 2016 Apr 14. pii: S0025-6196(16)30004-0. doi: 10.1016/j.mayocp.2016.03.001.

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    Jefferson Adams
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