Central nervous system (CNS) involvement in mycosis fungoides (MF) usually occurs in the presence of active skin lesions and infiltration of many visceral organs. A case of MF is presented, associated with a neurological syndrome, characterized by ataxia and motor-neuron disease. Autopsy revealed widespread infiltration of the CNS by MF, in the presence of only one minimal skin lesion and with no involvement of visceral organs. The importance of being aware of this discrepancy is stressed in the light of the possibility of radiotherapy in CNS involvement.
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