Large granular lymphocytic leukemia: a treatable form of refractory celiac disease.
case_report · Level V
Where this comes from
- Record sourced from PubMed, PMID 22922421.
- Also identified by DOI 10.1053/j.gastro.2012.08.028.
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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.
Medical subject headings
- Celiac Disease
- Diet, Gluten-Free
- Immunosuppressive Agents
- Leukemia, Large Granular Lymphocytic