Relapsing oral and colonic ulcers with monoclonal T-cell infiltration. A low grade mucosal T-lymphoproliferative disease of the digestive tract

Abstract

BACKGROUND: Some cutaneous T-cell lymphoproliferative diseases (LPD), such as lymphomatoid papulosis and pityriasis lichenoides et varioliformis acuta, are characterized by an indolent or waning and waxing clinical course. However, such T-cell LPD are rarely documented in other organs. METHODS: A patient with T-cell LPD of the digestive tract characterized by repetitive episodes of self-healing ulcers in the oral and intestinal mucosa over the course of 17 years is reported. Biopsy specimens from oral and intestinal mucosa were studied by conventional pathology, immunocytochemistry, and Southern blot analysis of T-cell receptor (TCR)-beta and -gamma gene rearrangement. RESULTS: Immunocytochemically, the infiltrating lymphocytes were lamina propria T cells with a dominant phenotype CD3+, CD4+/-, CD8-, and HML-1-. DNA study revealed the same rearranged configuration of TCR-beta and -gamma genes in specimens from both oral and colonic lesions. CONCLUSIONS: The present case may represent a novel T-cell lymphoproliferative disease (i.e., a digestive-tract mucosal counterpart of cutaneous dysplastic LPD).