Mucosal leishmaniasis of the lips and cheeks: a first concomitant presentation of visceral and mucosal leishmaniasis in a patient living with HIV/AIDS in Monastir, Tunisia

Abstract

Background: Visceral Leishmaniasis (VL) is the most severe and fatal disease if left untreated. In people living with HIV/AIDS (PLHA), VL is considered an emerging opportunistic infection. The aim of this manuscript was to report a first case in Tunisia of a concomitant presentation of visceral and oral leishmaniasis in a patient LHA. A systematic review of the literature was performed according to PRISMA guidelines, as well.

Case presentation: The patient, a 43-year-old heterosexual man, treated for HIV/AIDS was referred for macrocheilitis of the upper and lower lips. A noticeable nodular and painless swelling extending to the cheeks' mucosa was noted. The patient's poor oral hygiene was evident due to the presence of multiple dental caries. Histological analysis of the biopsied lower lip sample revealed the presence of numerous Leishmania amastigotes. The diagnosis of VL was clinically confirmed by the presence of a mild splenomegaly and pancytopenia and biologically by the identification of the parasite using PCR Lei and the species L. infantum involved using RFLP-PCR and culture. The treatment consisted of an intravenous administration of liposomal Amphotericin B (Ambisome®, 40 mg/kg/weight) for a period of 6 weeks. A favorable outcome was noted after one year with the resolution of clinical symptoms and a negative Leishmania blood PCR test. After 2 years, the patient remained asymptomatic but showed a positive Leishmania blood PCR test. Dolutegravir® was introduced in the patient's ART regimen.

Conclusions: To the best of our knowledge, this is the first case report in Tunisia of atypical VL diagnosed through an uncommon oral location in an HIV/AIDS co-infected patient . Since VL is a severe and potentially fatal disease, it is essential for dentists to perform a thorough clinical examination and adopt a multidisciplinary approach in order to ensure an early diagnosis and an effective treatment outcome.

Fig. 1

A–C Clinical views showing diffuse swelling of both upper and lower lips extending to the mucosa of the cheeks. Note the presence of fissures in the lower lip and cheeks. D Poor oral hygiene

Fig. 2

Panoramic radiography shows multiple dental caries, infected dental roots, and periapical lesions. related to root

Fig. 3

A, B Histological finding of the biopsied lower lip show a significant lymphocyte inflammatory infiltrate with multiple round to oval bodies in the cytoplasm of macrophages, which were consistent with Leishmania amastigotes (hematoxylin and eosin staining, ×400 magnification)

Fig. 4

A–E Identification of Leishmania. A MGG from peripheral blood leucocytoconcentration shows intracellular Leishmania amastigotes. B Immunoprinting using Leishmania Western Blot IgG LDBIO Diagnostics® yielded positive results and showed the presence of two Leishmania protein bands: p14 and p16 (band 23: positive control and band 24: patients’ results). C Agarose gel electrophoresis of 18S rRNA PCR product. Lane 1: negative control; Lane 2 patient blood, Lane 3: patient lip biopsy, Lane 4: positive control 343 bp; Lane MW: Molecular Weight marker (100 bp DNA ladder). D Restriction pattern of the amplified ribosomal Internal Transcribed Spacer 1 using HaeIII. Reference strains used were: Leishmania infantum MHOM/DZ/82/LIPA59 (LI) (three fragments of 187 bp, 72 bp and 55 bp), Leishmania killicki MHOM/TN/LEM163 (LK) (three fragments of 188 bp, 57 bp and 26 bp) and L. major MHOM/MA/81/LEM265 (LM) (two fragments of 206 bp and 132 bp). Lanes 1, 2, and 3: L. infantum identified from blood, lip biopsy and lip scraping product. MP: Molecular Weight marker (25-bp DNA ladder). E Culture using biphasic medium (blood horse based) from the peripheral blood leukocyte layer shows the promastigote form of Leishmania

Fig. 5

A, B Clinical views after treatment showing resolution of the swelling of the lips and cheeks

Fig. 6

PRISMA flowchart