Atypical Presentation of Visceral Leishmaniasis

Fever occurring twice daily, accompanied by profuse sweating, anorexia, and malaise of 15 days' duration resulted in hospitalization of a 31-year-old woman. At this time, her temperature was 37.6°C (99.7°F); blood pressure, 110/70 mm Hg; pulse rate, 90 beats per minute; and respiratory rate, 18 breaths per minute. A palpable spleen was about 3 to 5 cm below the left subcostal margin. Neither lymphadenopathy nor hepatomegaly was noted.

A complete blood cell count revealed pancytopenia: hematocrit, 29%; white blood cell count, 3,450/µL with 44% neutrophils, 38% lymphocytes, 15% monocytes, and 3% eosinophils; and platelet count, 110,000/µL. The C-reactive protein level was 32 mg/dL, and the erythrocyte sedimentation rate was 55 mm/h. Polyclonal hypergammaglobulinemia was shown on serum protein electrophoresis.

No abnormalities were found in results of biochemical tests, urinalysis, chest films, and electrocardiogram (ECG). A test was negative for purified protein derivative, as were serologic tests for syphilis, brucellosis, cytomegalovirus infection, Epstein-Barr virus infection, HIV infection, and hepatitis.

An abdominal ultrasonogram confirmed the splenomegaly. Bone marrow aspiration disclosed intracellular amastigotes of Leishmania species within marrow mononuclear phagocytes (arrows). At that time, the patient's serum direct agglutination test for leishmaniasis was positive (1:360). She was treated with meglumine antimoniate, 20 mg/kg/d for 20 days, with periodic ECG monitoring. After 3 months, she was feeling well, and the hematologic abnormalities were resolved.

Drs Haralampos Milionis, Mary Bai, and Moses Elisaf of Ioannina, Greece, report this case of visceral leishmaniasis not only because of its atypical presentation (mild pyrexia and moderate-rather than massive-splenomegaly), but also to emphasize the importance of bone marrow aspiration and biopsy as a valuable tool in the clinical diagnosis of fever of unknown origin.