Cutaneous T-cell lymphoma includes a heterogeneous group of lymphomas that share the unique feature of T lymphocytes which are tropic to the skin. Clinically, the presentation of this disease is protean and can range from a single lesion to generalized erythroderma. Unilesional mycosis fungoides, a rare variant, is presented here. Its morphology mimicked squamous cell carcinoma in situ, and was refractory to conventional therapies. A review of the treatment of this subset is also presented.