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

Adult T-cell leukemia/lymphoma (ATLL) is an aggressive T-cell neoplasm caused by human T-cell lymphotropic virus type 1 (HTLV-1). Although ATLL can show various skin symptoms, we present here a case of ATLL showing pompholyx-like skin manifestations. A 76-year-old Japanese woman with a 2-month history of painful and pruritic vesicular eruptions on the palms and soles was referred to our hospital (Fig. 1a,b). Viral eruption or neutrophilic dermatosis was suspected. Neither topical corticosteroids nor antibiotics were effective, and after six weeks, she worsened dramatically with numerous painful papulonodules, some with vesicles and erosions, scattered on the palms and soles (Fig. 1c). Laboratory data showed leukocytosis (10 020/ll with 24% lymphocytes, 22% atypical lymphoid cells) and elevated lactase dehydrogenase (313 U/l; normal range: 115–245) and soluble interleukin-2 receptor (4070 U/ml; normal range: 145–519). Anti-HTLV-1 antibody was positive (by particle agglutination), and flower cells were identified by peripheral blood smear (Fig. 1d). The skin biopsy from a red papule on the back of her left hand showed dense infiltration of atypical lymphoid cells with epidermotropism. The papillary dermis was edematous. Pautrier’s microabscesses were not seen (Fig. 2a,b). Immunohistochemistry of the atypical lymphoid cells were CD3, CD4, and CD25 positive and CD20 negative (Fig. 2c,d). Through these findings, we suspected the case as ATLL, and the eruptions on her hands and feet were regarded as direct skin infiltration of ATLL tumor cells. We referred her to a hematologist. Bone marrow aspiration demonstrated the infiltration of 7.6% atypical cells, and flow cytometry analysis on both peripheral blood and bone marrow identified atypical lymphoid cells expressing the same CD markers (CD4 and CD25 double positive) as those found in the tissue biopsy. Based on findings from whole body CT scans, elevated lactase dehydrogenase, and large amounts of atypical lymphoid cells in peripheral blood, this case was diagnosed as chronic ATLL with poor prognostic factors (Shimoyama’s classification). Oral VP-16 (etoposide) and prednisolone (20 mg/day) was administered, and the eruptions on the hands and feet largely resolved. 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