Visceral Leishmaniasis Revealed by an Atypical Oral Location in A PLWHIV

Objectives: There is an increase of reported coinfection with leishmania and HIV. The HIV-related immunodepression is associated with atypical manifestations of leishmaniasis. The aim of the study was to report a case of atypical oral leishmaniasis (OL) that revealed a visceral location in an HIV/HBV coinfected patient and to review the literature data.
Methods: A 43-year-old man living with HIV and under antiretroviral therapy was referred for a labial macrocheilitis. Clinical, radiological and biopsy of the labial lesion were performed. The review of all comparable reported cases in the literature was conducted using PubMed database.
Results: The patient’s T lymphocytes CD4 count was 81/mm³. Oral examination revealed a diffuse, painless and nodular swelling of both the upper and lower lip that extended to the right and left mucosa cheek, with no detected cervical lymphadenopathy. Poor oral hygiene with multiple dental infectious foci and apical periodontitis were confirmed by a panoramic radiography. The diagnosis of OL was confirmed by the presence in the labial biopsied specimen of multiple leishmania amastigotes in the cytoplasm of macrophages. Clinical examination revealed splenomegaly, blood tests noted pancytopenia and anemia: total white blood cell count of 900/mm³, neutrophils of 600/mm³, hemoglobin of 7.2 g/dl and platelet count of 62,000/mm³/l. Leishmania infantum was both identified by culture and PCR. The treatment was initiated using liposomal Amphotericin B for 6 weeks. The oral lesion has markedly decreased with a 6-month favorable outcome. Nine publications of OL associated with a visceral location were reported in the literature. In most cases, the lesion was described as erosive or ulcerated. The tongue and the palate were mostly involved.

Conclusions: To our best knowledge, this is the first reported condition of visceral leishmaniasis revealed by an atypical oral location in Tunisia. The role of the dentist in making an accurate diagnosis is crucial.