Martinique Island is not a known endemic area of leishmaniasis but several cases of autochtonous cutaneous leishmaniasis have been described due to a divergent Leishmania strain recently named as Leishmania martiniquensis (1). We report here the first case of visceral leishmaniasis due to L. martiniquensis in a HIV patient.

A 61-year-old man was diagnosed with HIV-1 infection in 2006. He worked as a painter and as a coconut picker and seller. Two years after introduction of cART, viral load was undetectable but CD4+ cell count had also paradoxically decreased. A normochromic normocytic aregenerative anemia of <10 g/dL appeared progressively. The patient had no fever but reported permanent fatigue. A voluminous hepatosplenomegaly developed. Microscopic examination of a bone marrow aspiration performed in 2011 revealed intrahistiocytic parasites consistent with amastigote forms of Leishmania spp. Diagnosis was confirmed by Leishmania PCR realized on whole blood. Molecular identification gave 100% identity with MHOM/MQ/92/MAR1 strain, newly named as L. martiniquensis. Symptoms improved after treatment with liposomal amphotericin B. Significant increases of hemoglobin level and CD4+ cell count above 350/mm3 followed. The patient did not receive secondary prophylaxis. PCR was negative in blood 20 months later and the patient is asymptomatic more than one year later.

Misknowledge of visceral tropism of this strain, associated with clinical presentation in HIV patients, may lead to delay the diagnosis of this life-threatening disease, uncommon in the Caribbean. Recent entomologic surveys have been carried out in Martinique Island to identify vectors and get a better understanding of this strain ecology.