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Paraneoplastic eosinophilic fasciitis with generalized morphea and vitiligo in a patient working with organic solvents

Dear Editor, Eosinophilic fasciitis is occasionally associated with hematological malignancies. We herein describe a case with eosinophilic fasciitis, generalized morphea and vitiligo, which developed into multiple myeloma. A 61-year-old man complained of skin thickening of the extremities. He had been working in the printing industry, cleaning the ink printer using organic solvents containing thinner, for over 10 years. His hobby was climbing mountains, and skin hardening had appeared a few years previously. He had vitiligo from the age of 12 years. Physical examination showed skin hardening of the upper and lower extremities, and vitiligo on the face, hands, trunk and lower extremities (Fig. 1a). Diffuse sclerosis was observed on the abdomen and trunk (Fig. 1b). Neither sclerodactyly nor Raynaud’s phenomenon was recognized. Laboratory examination showed a slightly elevated eosinophil subset (9%), increased aldolase (12.3 U/L), immunoglobulin (Ig) G (4425 mg/dL) and normal creatine kinase levels. Antinuclear antibody was positive (1:2560, specificity), whereas anti-dsDNA was normal and neither anticentromere nor Topo-I antibody was detected. T2-weighted magnetic resonance images showed significant thickening of the fascia of the thigh. Bone marrow biopsy showed normocellular marrow with slight proliferation of IgG-k cells. A biopsy specimen taken from the forearm revealed inflammatory infiltrates of mononuclear cells and eosinophils in the thickened fascia (Fig. 1c,d). Another specimen taken from the abdomen showed dermal sclerosis, corresponding to morphea. Lymph node biopsy denied malignant lymphoma. Our patient was treated with oral prednisolone (30 mg/day), which was gradually tapered. One year later, skin sclerosis of the extremities was sufficiently improved and that of the trunk had almost completely disappeared, whereas vitiligo was unchanged. Three years later, he developed splenomegaly (Fig. 1e), and the second bone marrow biopsy revealed plasma cell myeloma with plasma cell infiltration into the spleen. No flares of skin sclerosis were observed. Our patient showed increased levels of IgG and M-protein in the sera, but initial bone marrow aspiration biopsy denied multiple myeloma. Thereafter, he interrupted the follow up in the hematological department, and developed splenomegaly, and plasma cell myeloma 3 years later. So far, association of eosinophilic fasciitis and multiple myeloma is extremely rare, and interleukin-6 and transforming growth factor-b are suggested to play an important role.