Background: Coinfection with HIV and Leishmania represents a major health problem worldwide. Recent climate changes can favour in part the spread of Leishmania infection even in some Italian Northern regions, traditionally considered non-endemic areas. \ud Methods: We report a case of a multi drug resistant HIV positive patient on antiretroviral (ARV) therapy with multiple failures, suffering from fever, diarrhea and several skin ulcerations on the limbs and trunk. The initial physical examination revealed several large, ovoid skin ulcerations, ranging from 5 to 15 mm in diameter, with raised erythematic borders and a clear exudate. \ud The patient lived in the Italian Northern region Emilia-Romagna and hadn’t never traveled abroad. The CD4+ cells count was equal to 127/l (CD4+/CD8+ ratio 0.11) and HIV-RNAq . , as the patient did not assume ARV correctly. Histopathological examination of the colonic mucosa by biopsy revealed a colic leishmaniasis, while a qualitative PCR performed on a sample aspirate of the skin lesions disclosed the presence of L. infantum DNA. \ud Results: An intravenous liposomal amphotericin B (LAmB) regimen was promptly started, at 3 mg/kg per day for 7 days, and, subsequently, 3 mg/kg once a week for 5 weeks with a rapid improvement of fever and diarrhea, while the cutaneous lesions of the limbs and trunk began to heal 2 weeks after starting treatment. Six months following therapy, the patient developed a visceral leishmaniasis (VL) and cutaneous leishmaniasis (CL) relapse despite a secondary prophylactic regimen with LAmB was maintained. The patient was then treated with miltefosine x OS 30 mg/kg for 15 days, with a rapid clinical improvement but a progressive deterioration of the renal function which required hemodialysis treatment. During hospitalization the patient died due to refractory pancytopenia and acute renal failure.\ud Conclusion: In HIV-coinfected patients with Leishmania, the impaired immune function may: (i) favour the reactivation of latent Leishmania infection and induce, as in this case, a simultaneous presentation of VL and CL with a poorer therapeutic response; (ii) increase the risk of relapse after treatment, although miltefosine could represent in certain cases, an interesting option alone or in combination therapy. Continental Italian Northern regions are now considered at risk for Leishmanial disease, especially for HIV patients. Molecular methods, not yet well validated for cutaneous leishmaniasis, could be a reliable diagnosis tool
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