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  • Title: Large granular lymphocytic leukemia: a treatable form of refractory celiac disease.
    Author: Malamut G, Meresse B, Verkarre V, Kaltenbach S, Montcuquet N, Duong Van Huyen JP, Callens C, Lenglet J, Rahmi G, Samaha E, Ranque B, Macintyre E, Radford-Weiss I, Hermine O, Cerf-Bensussan N, Cellier C.
    Journal: Gastroenterology; 2012 Dec; 143(6):1470-1472.e2. PubMed ID: 22922421.
    Abstract:
    Large granular lymphocyte leukemia (LGL) is characterized by clonal expansion of CD3+ T cells or CD3(-) natural killer cells and frequently is associated with autoimmune diseases. We describe 2 patients with celiac disease who no longer responded to gluten-free diets after they developed T-cell LGL, with intestinal localization of malignant lymphocytes. Flow cytometry phenotyping of isolated intestinal intraepithelial and lamina propria cells eliminated type II refractory celiac disease, identifying large-sized CD8(+)CD57(+) T cells. Treatment with a combination of cyclosporine and methotrexate restored the patients' sensitivity to gluten-free diets. LGL therefore might be a cause of refractory celiac disease that is sensitive to immunosuppressive therapy.
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