Visceral leishmaniasis (VL), caused by Leishmania infantum, is endemic in Brazil. In people living with HIV (PLHIV), coinfection may present with atypical manifestations, severe course, and higher risk of relapse. Among the rare presentations is post- or para-kala-azar dermal leishmaniasis (PKDL), characterized by a maculopapular or nodular rash predominantly on the face, upper limbs, and trunk that occurs during or shortly after the visceral disease. Most cases described in the literature come from areas with higher prevalence of L. donovani; presentation with L. infantum is rare. We report the case of a 50-year-old Black woman from Chapada Diamantina (Bahia), diagnosed with VL in 2000 and treated with meglumine antimoniate. In 2013, she was diagnosed with HIV, with CD4 count 102 cells/mm³ and viral load 470,717 copies/mL. Initially with poor ART adherence, she had virologic failure and was later rescued with a new regimen. In 2019, with undetectable viral load and CD4 134 cells/mm³, she presented bicytopenia and gastrointestinal symptoms. Amastigotes were identified on duodenal biopsy, confirming the first VL relapse, treated with liposomal amphotericin B. She then started biweekly secondary prophylaxis with amphotericin. In 2020, during the COVID-19 pandemic, prophylaxis became irregular. In 2021, a new relapse occurred with pancytopenia and hepatosplenomegaly, confirmed by bone-marrow examination. In 2023, even on regular secondary prophylaxis, she developed epistaxis and bicytopenia associated with multiple cutaneous nodules on the face, limbs, and back. Workup for VL relapse was undertaken. Bone-marrow examination showed Leishmania spp. amastigotes. Histopathology of skin lesions revealed a nodular hypodermal lesion with epithelioid macrophages containing Leishmania spp. amastigotes, compatible with PKDL. She received liposomal amphotericin B (40 mg/kg) plus miltefosine, with clinical resolution of lesions within three months. She currently remains on biweekly prophylaxis, without further relapses. HIV/VL coinfection is a clinical challenge, particularly in patients with CD4 < 200 cells/mm³. PKDL is rare in areas with L. infantum and is more prevalent in PLHIV. This case underscores the importance of continuous prophylaxis, ART adherence, and recognition of PKDL as a manifestation of VL relapse.
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