BACKGROUND Visceral Leishmaniasis (VL) is seen commonly in eastern parts of India, while occurrences of cutaneous forms of the disease have been reported recently from Western Ghats of southern India. The clinical manifestations of VL resembles many other chronic illnesses and its diagnosis and management requires added attention in southern parts of the country. The clinical, epidemiological and entomological investigations related to the occurrence of a case of VL in Kerala, south India are described in detail. MATERIALS AND METHODSThis study is a descriptive study. The patient suffered from various clinical manifestations which were investigated repeatedly at various primary, secondary and tertiary care institutions. Several laboratory tests for multiple diseases turned out to be negative, even though the symptoms were persisting. Accurate diagnosis was made from the bone marrow cytological examination, which indicated the presence of Leishman-Donovan bodies. Further epidemiological and entomological investigations confirmed the presence of vector (Phlebotomus argentipes) from multiple locations around the residence of the patient. Evidence of Leishmaniasis or its source could not be diagnosed in contacts, vectors or suspected animal reservoirs in the area. RESULTSAccurate diagnosis was made from the bone marrow cytological examination, which indicated the presence of Leishman-Donovan bodies. Epidemiological and entomological investigations confirmed the presence of vector (Phlebotomus argentipes) from multiple locations around the residence of the patient. Evidence of Leishmaniasis or its source could not be diagnosed in contacts, vectors or suspected animal reservoirs in the area. CONCLUSIONEarly and accurate diagnosis of VL is difficult in south India due to unawareness about the manifestations of disease and also due to unavailability of reliable laboratory kits. The management of such cases is even more challenging due to the lack of availabi lity of proper drugs. The recent reports of emergence of resistant cases should be a pointer in creating improved awareness and facilities for the rapid identification and management of cases of visceral leishmaniasis. KEYWORDS