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a systematic review of the literature on VL unusually presenting with spleen focal lesions, including three previously unpublished cases.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.
Keywords: Atypical; Focal spleen lesions; Haematologic disorder; Spleen; Visceral leishmaniasis.