16 October 2009
THE FIRST CASE OF CONGENITAL LEISHMANIASIS IN A FEMALE INFANT IN GREECE
Leishmaniasis is a zoonotic infection transmitted by insect vectors, like sandflies, especially by the Phlebotomus species. Other exceptional modes of non-vector transmission include blood transfusion, organ transplantation, sexual intercourse, inoculation of cultures as well as vertical transmission from mother to child.1 Congenital visceral leishmaniasis was first described in a 10-month infant by Low and Cooke in 1926.2 Until now, only sporadic cases of congenital transmitted disease have been reported in literature.2–4 We report the first case of congenital disease described in Greece.
An 11-month infant that came from a rural region of northern Greece was admitted to our department with a 15-day history of high intermittent fever up to 40°C, paleness, gingival bleeding, weakness and weight loss. On admission, physical examination revealed severe malnutrition, anaemia with paleness of the skin and the mucosa, bilaterally enlarged cervical and inguinal lymph nodes, gingival bleeding and hepatosplenomegaly. The mother of the infant was hospitalised a few days before labour because of anaemia and hepatosplenomegaly, and titres for Leishmania antibodies were positive. Laboratory exams revealed pancytopenia with lymphocytosis (Hb: 4.3 g/dL, WBC: 4600/mm3, PLT: 27 000/mm3) and elevated erythrocyte sedimentation rate (65 mm in the first hour). Blood and urine cultures, antibody titres for a wide spectrum of infectious diseases and laboratory exams for autoimmune diseases were negative. Chest radiography and cardiac ultrasonography were within the normal limits. Moreover, abdominal sonography verified hepatosplenomegaly. The immunological studies revealed high hyperglobulinaemia (immunoglobulin G [IgG] 2250 IU/mL), and serological examination showed a positive titre for Leishmania antibodies (indirect immunofluorescence assay [IFA] 1:640). The results were confirmed on a second specimen. A bone marrow aspirate showed no evidence of malignancy, except from a slight decrease of myelopoesis, erytropoesis and thrombopoesis. However, the promastigote form of Leishmania was found, and therefore, diagnosis of leishmaniasis was confirmed.
Although the majority of previous cases reported in literature was treated with antimonial compounds, we administered liposomal amphotericin B (LAmB) at a daily dose of 4 mg/kg on 6 consecutive days and repeated doses at days 14 and 21. Treatment was well tolerated, and no side effects were reported. After the third day of LAmB infusion, the fever subsided. After 8 days of treatment, the clinical manifestations disappeared, and after 5 weeks, blood cell counts reached its normal values.
We conclude that in endemic areas for leishmaniasis, like the Mediterranean regions, the congenital mode of transmission should always be considered in infants presenting with fever, pancytopenia and hepatosplenomegaly. Although the treatment success rates of congenital leishmaniasis with antimonial compounds are high, we believe that LAmB represents a safer alternative treatment and should therefore be adopted as a first-line regimen in cases of congenital leishmaniasis.5,6