Leishmaniasis is a complex group of parasitic infectious diseases caused by intracellular protozoa of the genus . It is a zoonosis mainly transmitted by the bite of infected female or sandflies. Clinical manifestations of leishmaniasis are diverse and can range from asymptomatic presentations to disseminated systemic disease. Cutaneous leishmaniasis is endemic in more than 80 countries in the world, having a predominance in tropical and subtropical regions. Although the majority of cases follow a classic development, an increasing number of new and rare variants of cutaneous leishmaniasis have been reported. These variants should be suspected as a cause of diverse clinical presentations, especially in endemic regions and travelers, being a diagnostic challenge for physicians. We present a case of atypical cutaneous leishmaniasis found as a single verrucous plaque of eight months of evolution in the left posterior thigh of a 35-year-old man, who presented mild pruritus. The patient reported shrimp farming as his main occupational activity and was living in a rural region surrounded by forest on the Pacific coast of Ecuador. On dermatological examination, a single 4 x 5 cm verrucous plaque with irregular borders and a scaly erythematous violaceous aspect was found. Histopathological analysis revealed the presence of lymphohistiocytic inflammatory infiltrate with plasmocytes and granulomatous inflammation. On the Giemsa stain, intracellular amastigotes (Leishman-Donovan bodies) were observed. The treatment consisted of intramuscular meglumine antimoniate, presenting significant improvement on follow-up.