We would like to report a case of visceral leishmaniasis (VL) or Kala-azar with an atypical course in a liver transplant recipient, who benefited from a prompt diagnosis and successful treatment. A 34-year-old man underwent orthotopic liver transplantation in December 2003 due to cryptogenic cirrhosis. Serology was positive (low-level) for Chagas disease. Immunosuppression following transplantation was done with prednisone and tacrolimus. The patient was admitted to the University Hospital in September 2005 complaining of night sweats, fatigue, weakness, and weight loss for four months. He denied fever, coughing, dyspnea, or diarrhea. At that time, he did not present splenomegaly. Laboratory exams results were: hemoglobin, 8.5 g/dL; white blood cell count, 2,100/mm3; platelet count, 99,000/ mm3; normal coagulation parameters; normal liver biochemistries; albumin, 2.3 g/dL; polyclonal hypergammaglobulinemia, 3.5 g/dL; and normal renal function. Anti-Leishmania serum titers by indirect immunofluorescence were 1/320 (cutoff 1/80). Leishmania amastigotes were found in the liver biopsy and in the bone marrow aspirate. At the time of diagnosis, the serum level of tacrolimus was 7.5 ng/dL and was decreased to 5.0 ng/dL. The patient was treated with liposomal amphotericin B (AmBisome), at 3 mg/kg once a day for seven days, and recovered well with no cytopenia. Twelve months later, no relapse has occurred. Brazil has the highest leishmaniasis rate in South America with 1,980 new cases per year. The majority (90%) of domestic cases occur in northeast Brazil (20.4 cases/100,000 habitants) where VL is endemic and is usually caused by Leishmania chagasi. Animal reservoirs are dogs, foxes, and skunks. Brazil is also the leading country for solid-organ transplantation in South America. However, there are no reported cases of VL in transplant recipients prior to this paper. Leishmania infection may occur as a reactivation of a previously acquired infection, or it may be transmitted from the graft or acquired after transplantation. The full-blown clinical picture is characterized by fever, weight loss, organomegaly, blood cytopenia, and hypergammaglobulinemia. The clinical diagnosis of VL in immunocompromised patients is difficult, and the common signs may be subtle, being disease fatal if left untreated (1–5). We highlight the fact that although we are in an endemic area, no serological test for leishmaniasis is done routinely in recipients or donors. Asymptomatic infections are much more frequent than patent cases even in non endemic areas. The geographical origin of the donor may not always be a valid criterion, as travel is common, and infection may occur even during a short stay in endemic areas (1, 5). VL is uncommon in liver recipients and few cases have been published. Three cases have recently been reported in southern France (1). Fever was the most common symptom (84.3%), followed by splenomegaly (58.8%) (1). Our patient presented an atypical course due to the absence of fever and splenomegaly. The clues for prompt diagnosis were a cross-reaction with Chagas disease, cytopenia, hypergammaglobulinemia, and a positive epidemiology. We recommend routine serological testing for Leishmaniasis in donors and recipients from endemic areas due to the possibility of disease infection and reactivation. Wanessa Trindade Clemente Department of Laboratory Medicine, Faculty of Medicine Alfa Gastroenterology Institute Federal University of Minas Gerais Belo Horizonte, Brazil Cláudia Alves Couto Department of Internal Medicine, Faculty of Medicine Alfa Gastroenterology Institute Federal University of Minas Gerais Belo Horizonte, Brazil Daniel Dias Ribeiro Marcelo de Medeiros Chaves França Alfa Gastroenterology Institute Federal University of Minas Gerais Belo Horizonte, Brazil Marcelo Dias Sanches Department of Surgery, Faculty of Medicine Alfa Gastroenterology Institute Federal University of Minas Gerais Belo Horizonte, Brazil