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Case of lipodystrophia centrifugalis abdominalis infantilis successfully treated with topical application of tacrolimus

Dear Editor, Lipodystrophia centrifugalis abdominalis infantilis (LCAI) is a localized lipodystrophic disease in childhood in which depressive skin develops in the groin and axillary areas, and extends into the abdomen during the first 4 years of life. Although various therapies are challenging for LCAI, no effective treatment has been recommended. We treated a case of LCAI with application of tacrolimus ointment, which dramatically improved the erythema, and abrogated enlargement of the affected lesion thereafter. This is the first case to be successfully treated with topical application of tacrolimus. A 3-year-old boy visited our hospital for depression of abdominal skin that developed from bilateral inguinal areas and extended in approximately 1 year. There were no mechanical stimulations triggering the skin lesion. He had no previous medical history, except for mild atopic eczema. The depressive abdominal skin was surrounded by a slightly elevated erythematous boarder without pruritus or pain (Fig. 1a). Bilateral inguinal lymph nodes were swollen. A routine blood and chemistry examination disclosed no abnormal findings. No antibody to nucleus, ssDNA or dsDNA was detected. Anti-Borrelia antibody was not checked. A skin biopsy of the erythematous lesion showed dense cellular infiltrates around vessels, sweat glands and adipose tissue, intermingled with lymphocytes and plasma cells (Fig. 1b, inset). There was a dystrophic change in adipose tissue. A direct immunofluorescence study was negative. The diagnosis of LCAI was made based on the clinical and histological features distinguishable from panniculitis including lupus profundus and morphea profunda. Partial efficacy of systemic corticosteroids has been reported in several cases. However, we hesitated to use systemic corticosteroid treatment, which can have a partial effect on abrogation of this disease, because of the disadvantage in growth for his age. We decided to use topical treatment of 0.03% tacrolimus ointment for this case instead. Interestingly, the erythematous border disappeared 1 week after initiation of this treatment and abrogated enlargement of the affected area thereafter (Fig. 1b). Bilateral lymphadenopathy remained swollen. More than 160 cases of LCAI have been reported since it was first described by Imamura et al. in 1971. The majority of cases were Japanese patients, but some Asian and Caucasian patients have also been reported. The mechanism of LCAI remains unknown, although mechanical stimulation triggered this disease in several previous cases. No effective treatments have been described for LCAI. However, a recent report showed efficacy of topical application of pimecrolimus in a case of LCAI. We decided to use topical treatment of 0.03% tacrolimus ointment because of availability and safety of this agent in Japan. Tacrolimus is used as a therapeutic option for localized scleroderma with histological similarity to LCAI. Surprisingly, dramatic improvement was observed in our patient just after topical application of tacrolimus. We cannot exclude