Mucocutaneous Leishmaniasis as a Manifestation of Immune Reconstitution Inflammatory Syndrome in Plhiv/AIds

A 77-year-old White man, born in Guarantã and residing in São Bernardo do Campo, a retired machine operator, widower, with a vaccinated dog. He lived during childhood in a rural area of Londrina and in Tobatí, Paraguay, for two years, where he received injections for an ulcerated lesion of the right lower limb of undefined etiology. Diagnosed with HIV/AIDS in June 2024 during evaluation of a consumptive syndrome, with viral load 1,860 copies and CD4+ T cells 288. After ART initiation, viral load 228 copies and CD4+ T cells 447, he developed pruritic erythematous papules and plaques progressing to crusts over the entire body, increasing in number and size. Workup showed positive LF-LAM and a skin biopsy with ulcerated granuloma and focal caseous necrosis; treatment for cutaneous tuberculosis was initiated. After 4 months on rifampicin and isoniazid, there was little improvement and a new lesion appeared on the nasal alae. Facial CT showed a 0.9-cm discontinuity in the left maxillary sinus wall. A new skin biopsy revealed chronic granulomatous dermatosis, amastigotes within superficial macrophages, and immunohistochemistry positive for Leishmania spp. Blood testing for leishmania was negative. Liposomal amphotericin was initiated to a cumulative dose of 2.8 g, with lesion regression and cicatricial aspect at the end of treatment. Disseminated mucocutaneous leishmaniasis, although rare, should be considered in people living with HIV, particularly with severe immunosuppression and in the setting of immune reconstitution inflammatory syndrome, as an important differential diagnosis – specially with relevant epidemiology. In this condition, partial restoration of immune responses induced by ART may trigger exuberant inflammatory manifestations, either by unmasking subclinical infections or by paradoxical worsening of diagnosed conditions. Given the clinical and diagnostic complexity of leishmaniasis in this context, an individualized approach is required, with active investigation of coinfection and therapeutic choice tailored to the degree of immunosuppression, lesion extent, and systemic inflammatory response. Early recognition of this interaction between HIV and leishmaniasis is fundamental to avoid unfavorable outcomes.