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Background: Lymphadenopathy is one of the earliest and commonest manifestations in HIV patients. Fine needle aspiration cytology is an accurate, common procedure in the evaluation of lymphadenopathy in HIV-positive patients. The most frequent etiology of this clinical manifestation, in Western studies, is the presence of reactive hyperplasia due to the HIV itself and infectious diseases with opportunistic agents, namely Mycobacterium. The diagnosis of other microorganisms, such as fungi, helminthes and protozoa, is less likely, and most cases are reported as curiosities., Case: Chronic visceral leishmaniasis occurred in an HIV-1 patient. Fine needle aspiration biopsy was performed in an axillary lymph node during the course of follow-up. The lymph node aspirates showed numerous macrophages, carrying several intracellular microorganisms (Leishmania amastigotes)., Conclusion: The cytologic diagnosis offered no major challenge, but the differential diagnosis with other intracellular infectious agents that can also affect HIV patients should always be considered. In this context, we reviewed the HIV patients with lymphadenopathy seen in our hospital and who underwent fine needle biopsy in the last 5 years. From a series of 201 patients and 250 fine needle aspiration biopsy samples, this was the only case of leishmaniasis to date.