Lip leishmaniasis: a case series with molecular identification and literature review

BACKGROUND: Mucocutaneous leishmaniasis (MCL), a protozoan infectious disease, is very rare in Iran despite the endemicity of both cutaneous and visceral forms. It is transmitted by the Phlebotomus sand fly. The lip is considered one of the extraordinary sites. Lesions usually initiate with erythematous papules, slowly enlarges and then it ulcerates. The diagnosis of MCL encompasses epidemiological, clinical and laboratory aspects. Usually, the combination of some of these elements is necessary for the final diagnosis. So, lip leishmaniasis lesions can be challenging to diagnose. CASE PRESENTATION: We presented seven rare cases of lip leishmaniasis. Tissue impression smear, culture, PCR and phylogenetic analysis were carried out for explicit diagnosis. Skin scraping investigation showed several Leishmania spp. amastigotes in the cytoplasm of macrophages. Culture examination was positive for Leishmania spp. PCR was positive for L. major, L. tropica, and L. infantum. Differential diagnosis includes orofacial granulomatosis, basal cell carcinoma, squamous cell carcinoma, and mesenchymal tumors. The cases were treated with systemic meglumine antimoniate (Glucantime(®)). No relapses were observed during 1 year of follow-up. Early detection of the infection are necessary in order to start effective treatment and prevent more serious complications. CONCLUSIONS: In this paper, we reported seven rare cases of lip leishmaniasis in Iran, emphasized the importance of clinical and diagnostic features of lesions, characterized the phylogenetic kinship of isolated parasites, and reviewed the literature on lip leishmaniasis.