A 38-year-old male with a past history of intravenous drug use, AIDS and Visceral Leishmaniasis in 2000, was hospitalized after presenting with fever, myalgias and arthralgias, fatigue, hepatosplenomegaly and oedema of the inferior limbs. On the tenth day of admission, the patient developed painful subcutaneous nodules of the thighs and a Dermatology consultation was requested. A clinical and histological diagnosis of Leishmania Panniculitis was made, in the setting of Visceral Leishmaniasis recurrence. Leishmania Panniculitis is rarely found simultaneously with Visceral Leishmaniasis, and it is more frequently seen in HIV co-infected patients. In this case, the skin involvement allowed for an early diagnosis and histological confirmation of Leishmaniasis recrudescence to be made.