Introduction: dermatofibroma is a benign mesenchymal lesion of uncertain etiology, centered in the dermis, with fibroblastic and histiocytic differentiation. The atypical variant can mimic superficial sarcoma histologically. Objective: to describe the clinical and histopathological findings of atypical dermatofibroma in a patient undergoing treatment for leprosy with multiple therapeutic failures. Case Description: the report concerns a 39-year-old woman with multiple therapeutic failures for Virchowian leprosy, who developed an atypical dermatofibroma on the upper limb, which clinically resembled a leproma. Discussion: molecular analyses suggest dermatofibroma may be associated with a neoplastic process. However, several studies correlate it to trauma, insect bites, folliculitis, immunological changes, and treated or reactive leprosy. The association of dermatofibroma and leprosy, especially in the Virchowian pole, is documented in the literature. However, there are no reports of atypical dermatofibroma associated with leprosy. Final Considerations: this case adds to other studies regarding a possible association between the pathogenesis of dermatofibroma and the immunological aspects present in leprosy.