Mucosal leishmaniasis of the lip: an emerging clinical form in Tunisia.

BACKGROUND: Mucosal leishmaniasis (ML), which mostly occurs in the New World, is mainly associated with Leishmania braziliensis. Primary lip ML is very rare in the Mediterranean basin and particulary in Tunisia despite the endemicity of both cutaneous and visceral leishmaniasis in this area. OBJECTIVES: To highlight a recent emergence of primary lip ML in Tunisia, to describe its epidemiological and clinical features and to identify the causative Leishmania species. METHODS: Epidemiological, clinical and therapeutic data of 10 cases presenting a ML of the lip were collected. Diagnosis confirmation of leishmaniasis was obtained by microscopic examination of Leishmania parasites in Giemsa stained smears of the lesion sampling and in cutaneous biopsies. Polymerase chain reaction (PCR) detecting Leishmania DNA directly from dermal scraping was also performed for diagnosis and species identification. RESULTS: Seven men and three women with lip ML were diagnosed during the last 6 years (2008-2013). The mean age was 29.7 years. Clinical presentation was characterized by an infiltrated and ulcerated plaque leading to macrocheilitis involving the upper lip in eight cases and the lower lip in two cases. Mean diagnosis delay was 6.9 months. PCR identified L. infantum in seven cases and L. major in two cases. Seven patients received intramuscular injections of meglumine antimoniate (MA) and three patients received both MA intralesional injections of MA and cryotherapy. A clinical remission was rapidly observed in all cases (on average in 2.2 months). CONCLUSIONS: Primary lip ML is emerging in Tunisia. Macrocheilitis of the upper lip is the main clinical presentation. PCR revealed more sensitive than direct examination in the diagnosis of such form (P < 0.01). Leishmania infantum was the most identified species (7 cases) while L major was involved in only two lesions. A benign local evolution and a rapid recovery were observed in all cases after MA treatment.