We present a rare case of adult T cell leukemia/lymphoma (ATL) in which leu
kemic T cells expressed CD4 and CD25 surface antigens and infiltrated mamma
ry glands during clinical course of the disease. A 40-year-old male was adm
itted with long-standing skin lesions and leukocytosis. Peripheral blood ly
mphocytes were highly pleomorphic and presented CD2, CD4, CD25, CD38 membra
ne surface antigens. The patient proved to be seropositive for human T-cell
lymphotropic virus type I (HTLV-I) antibodies. Monoclonal expansion of lym
phoid cells integrated with HTLV-I genome was observed, and the diagnosis o
f AIL chronic type was made. He underwent a chemotherapy regimen, and skin
lesions and leukocytosis improved markedly. He progressed with an indolent
clinical course of ATL, when he was admitted with bilateral hyperplasia of
breast, recurrent skin lesions, and leukocytosis. Breast biopsy revealed bi
lateral gynecomasty, extensive leukemic infiltration of typical ATL cells i
n the mammary glands, and the presence of mammary epithelial cells producti
vely infected with HTLV-I. This is the first report describing invasion of
the mammary tissue with HTLV-l-transformed T-cells and HTLV-l-associated br
east disease. (C) 2000 Wiley-Liss, Inc.