Indolent T-cell lymphoproliferative disease of the gastrointestinal tract after treatment with adalimumab in resistant Crohn's colitis

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• 作者：Natalia Edison ; PhD^a ; ^b ; Hila Belhanes-Peled ; MSc^b ; Yuval Eitan ; PhD^b ; ^c ; Yifat Guthmann ; MSc^b ; ^c ; Yelena Yeremenko ; MD^d ; Mark Raffeld ; MD^e ; Irit Elmalah ; MD^b ; Philippe Trougouboff ; MD^a ; ^b ; ^{phil_t@clalit.org.il" class="auth_mail" title="E-mail the corresponding author}
• 关键词：Indolent T-cell lymphoproliferative disease ; Immunosuppression ; Inflammatory bowel disease ; Tumor necrosis factor&ndash ; α inhibitor
• 刊名：Human Pathology
• 出版年：2016
• 出版时间：November 2016
• 年：2016
• 卷：57
• 期：Complete
• 页码：45-50
• 全文大小：1396 K

文摘

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 After the TNF-α inhibitor treatment was withdrawn, the T-cell infiltrates regressed, but 2 years later, the same monoclonal T-cell infiltrate reappeared at the only site of active inflammation.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.