A Dyshidrosis-Like Variant of Adult T-Cell Leukemia/Lymphoma With Clinicopathological Aspects of Mycosis Fungoides. A Case Report

Abstract

Adult T-cell leukemia/lymphoma (ATL) is an aggressive type of leukemia/lymphoma associated with the human T-cell lymphotropic virus (HTLV-I). We describe an adult male patient clinically and pathologically diagnosed as mycosis fungoides and treated with chemotherapy after which complete involution of the lesions occurred. The disease relapsed with confluent dyshidrosis-like vesicles on the palmoplantar regions, followed by disseminated vesiculopapules and associated lymphocytosis. A serological test performed at this time revealed HTLV-I infection, and a diagnosis of chronic ATL was made. Monoclonal integration of HTLV-I was detected in peripheral blood mononuclear cells by inverse long polymerase chain reaction. A skin biopsy revealed spongiosis, Pautrier abscesses, and intraepidermal vesicles with atypical lymphocytes and an infiltration of small and atypical CD4 lymphocytes in the superficial dermis. Proliferative index (Ki-67) was 70%. This is the first reported vesicular cutaneous ATL with confirmation of HTLV-I proviral integration. The delay that occurred in diagnosing ATL was due to the fact that mycosis fungoides and ATL may present the same clinical, histopathological, and immunohistochemical features.

Cutaneous manifestations associated with HTLV‐1 infection

“…1b), nodules, tumors (Fig. 15 They are always multiple and are generalized in 50% of cases. 6 Less commonly, the lesions may appear as purpuric plaques or in the form of vesicles (Fig.…”

Dyshidrotic mycosis fungoides

“…To our knowledge, reports of dyshidrotic or pomphylox‐like MF (vesiculobullous MF involving the palms and/or soles) are limited, and of these all but two 2,6 involved known lesions of MF outside the palms and soles at presentation. A dyshidrosis‐like vesicular eruption has also been reported in association with adult T‐cell leukemia/lymphoma 7,8 . The absence of any underlying hematologic disorder or systemic symptoms in our patient mitigates against adult T‐cell leukemia with secondary cutaneous involvement as a potential consideration.…”

“…A dyshidrosis-like vesicular eruption has also been reported in association with adult T-cell leukemia/lymphoma. 7,8 The absence of any underlying hematologic disorder or systemic symptoms in our patient mitigates against adult Tcell leukemia with secondary cutaneous involvement as a potential consideration.…”

Pompholyx‐like skin manifestation of adult T‐cell leukemia/lymphoma

“…A unique clinical aspect of our case was the distribution of vesicles limited to the palmoplantar areas, simulating pompholyx, although as many as 50% of patients with ATLL show skin manifestations such as nodulotumoral, plaque, multipapular, patch, erythrodermic, and purpuric manifestations . There have been only a few reports describing pompholyx‐like vesicular eruptions in patients with ATLL . Michael et al .…”