title: Indolent T-cell lymphoproliferative disease of the gastrointestinal tract after treatment with adalimumab in resistant Crohn's colitis.
abstract: We report a case of intestinal indolent T-cell lymphoproliferative disease (TCLPD) occurring after the initiation of tumor necrosis factor-α (TNF-α) inhibitor therapy for resistant Crohn's disease. A prominent T-cell infiltrate positive for CD8, TIA-1, and T-cell receptor-βF1 was associated with the foci of active inflammation. T-cell receptor gene clonality studies (BIOMED-2) demonstrated monoclonality. After the TNF-α inhibitor treatment was withdrawn, the T-cell infiltrates regressed, but 2 years later, the same monoclonal T-cell infiltrate reappeared at the only site of active inflammation. To the best of our knowledge, this report is the first to show a link between active inflammation and the TCLPD. In addition, it suggests a possible influence of the TNF-α inhibitor treatment on the evolution of the TCLPD. A high degree of suspicion is required in the presence of any unusual lymphoid infiltrate in inflammatory bowel disease to avoid overlooking an indolent TCLPD or misdiagnose an aggressive lymphoma.
authors.affiliation: Hematopathology Unit, The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel; The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel.
authors.forename: Natalia
authors.initials: N
authors.lastname: Edison
authors.affiliation: The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel.
authors.forename: Hila
authors.initials: H
authors.lastname: Belhanes-Peled
authors.affiliation: The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel; Molecular Pathology Unit, The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel.
authors.forename: Yuval
authors.initials: Y
authors.lastname: Eitan
authors.affiliation: The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel; Molecular Pathology Unit, The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel.
authors.forename: Yifat
authors.initials: Y
authors.lastname: Guthmann
authors.affiliation: Gastroenterology Institute, Emek Medical Center, Afula, 1834111, Israel.
authors.forename: Yelena
authors.initials: Y
authors.lastname: Yeremenko
authors.affiliation: Molecular Diagnostics Section, NCI/NIH, Bethesda, MD, 20892, USA.
authors.forename: Mark
authors.initials: M
authors.lastname: Raffeld
authors.affiliation: The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel.
authors.forename: Irit
authors.initials: I
authors.lastname: Elmalah
authors.affiliation: Hematopathology Unit, The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel; The Institute of Tissue Diagnostics and Cancer Research, Emek Medical Center, Afula, 1834111, Israel. Electronic address: phil_t@clalit.org.il.
authors.forename: Philippe
authors.initials: P
authors.lastname: Trougouboff
mesh_terms: D000068879:Adalimumab / Q000009:adverse effects*; D000328:Adult; D000893:Anti-Inflammatory Agents / Q000009:adverse effects*; D001706:Biopsy; D018414:CD8-Positive T-Lymphocytes / Q000187:drug effects* / Q000276:immunology; D049109:Cell Proliferation / Q000187:drug effects*; D003424:Crohn Disease / Q000175:diagnosis / Q000188:drug therapy* / Q000276:immunology; D005260:Female; D005765:Gastrointestinal Agents / Q000009:adverse effects*; D005819:Genetic Markers; D006801:Humans; D007150:Immunohistochemistry; D008232:Lymphoproliferative Disorders / Q000139:chemically induced* / Q000175:diagnosis / Q000235:genetics / Q000276:immunology; D025202:Molecular Diagnostic Techniques; D011237:Predictive Value of Tests; D013997:Time Factors; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors / Q000276:immunology
chemical_list: D000893:Anti-Inflammatory Agents; D005765:Gastrointestinal Agents; D005819:Genetic Markers; D014409:Tumor Necrosis Factor-alpha; D000068879:Adalimumab
keywords: Immunosuppression; Indolent T-cell lymphoproliferative disease; Inflammatory bowel disease; Tumor necrosis factor–α inhibitor