Indolent T-cell lymphoproliferative disease of the gastro-intestinal tract following treatment with Adalimumab in resistant Crohn's colitis

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, TCR-βF1 was associated with the foci of active inflammation. T-Cell Receptor (TCR) gene clonality studies (BIOMED-2) demonstrated monoclonality. After the TNF-α inhibitor treatment was withdrawn, the T-cell infiltrates regressed, but two years later, the same monoclonal T-cell infiltrate reappeared at the only site of active inflammation.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.