Background: Syringolymphoid hyperplasia with alopecia is considered to be a rare variant of cutaneous T-cell lymphoma. Current therapy for cutaneous T-cell lymphoma limited to the skin favors topical treatment, chemo-, and photochemotherapy. Case Report: We report on a 52-year-old patient presenting with disseminated, reddish-brown scaly papules on both soles and erythematous papules with alopecia on his thighs. First skin lesions arose 5 years ago and subsequently a continuous progression was noted. Finally, the patient was severely limited due to pressure-induced pain on the soles. Topical glucocorticosteroids, local PUVA bath therapy, and systemic interferon-α administration failed to improve skin lesions. Thus, a local radiotherapy with 60Co γ-rays was administered. A total dose of 30 Gy was delivered in 2-Gy fractions over a 3-week period. Clinically, there was a complete regression of irradiated skin lesions. Conclusion:Though there is limited experience regarding the therapy of syringolymphoid hyperplasia with alopecia, radiotherapy has to be considered in recalcitrant cases.