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Background: The Mediterranean area is endemic for the zoonotic form of visceral leishmaniasis (VL) caused by Leishmania infantum, a species which has been recently associated with unexpected epidemiological and clinical features., Method: We report the result of a systematic review of the literature on VL unusually presenting with spleen focal lesions, including three previously unpublished cases., Results: A total of 28 cases of VL with multiple spleen focal lesions were retrieved. Twenty-six (93%) were acquired in the Mediterranean area, where L. infantum is endemic. Thirteen cases were diagnosed in children, and the remaining 15 cases in middle-aged adults, of whom 9 were immunosuppressed. Four patients (14%) underwent diagnostic splenectomy, while the diagnosis was confirmed by less invasive techniques in the remaining patients. The most common radiological patterns were: multiple subcentimetric or centimetric spleen lesions (size ≤ 1 cm in 14 of 19 cases, 74%), hypoechoic at ultrasonography (25 of 26 cases, 99%) and hypodense at CT scan (9 of 10 cases, 90%). PET-CT (available for four patients) showed an intense FDG spleen uptake. MRI and contrast-enhanced ultrasonography, available for two and one cases each, showed complex patterns., Conclusions: VL must be added to the list of possible differential diagnosis of spleen focal lesions and health care provider awareness about this condition will avoid unnecessary invasive diagnostic procedures.

Visceral leishmaniasis is a severe disease caused by the intracellular protozoa Leishmania donovani. Diagnosis is based on examination of bone marrow or serology. The role of imaging techniques as diagnostic tools remains to be established in visceral leishmaniasis. We report multiple nodular lesions in the liver and spleen on ultrasonography and computed tomography in a patient with visceral leishmaniasis. To our knowledge, this is the first reported case of multiple nodular hepatosplenic lesions in visceral leishmaniasis.