Title : Mucocutaneous leishmaniasis: A rare entity in algeria
Abstract:
Introduction: Leishmaniasis is a parasitic disease that can manifest as skin, mucosal, mucocutaneous, and visceral disorders. The cutaneous form is the most prevalent and endemic in the Mediterranean region, while the mucocutaneous form is exceptionally rare, with only a few reported cases in Tunisia, primarily presenting as labial lesions caused by Leishmania major. This report describes a unique case of mucocutaneous leishmaniasis in Algeria, initially misdiagnosed as granulomatous macrocheilitis of Miescher.
Material/methods: A male patient, 28 years of age, had been under the care of the dermatology department for a duration of two years due to the presence of macrocheilitis, which was initially diagnosed as Miescher's granulomatous disease. The patient presented with asymmetrical, dry, painless, non-pruritic macrocheilitis on the right edge of the lower lip, accompanied by wrinkled and scaly skin appearance. Ipsilateral jugal edema was also observed(Fig1.A). Although the patient mentioned residing in an endemic Leishmanian zone, no history of insect bite was reported. A PCR test on a biopsy fragment was performed to reassess the diagnosis(Fig2), confirming leishmaniasis. The identified species was leishmania major. The patient was receiving Meglumine antimoniate at 60mg/kg/day for 15 days, followed by a second course of the same duration a month later. Long-term oral antiparasitic treatment (fluconazole at 100mg/day) was also prescribed. Subsequently, a significant improvement in macrocheilitis and a decrease in cheek edema were observed.
Results (and discusssion): Mucocutaneous leishmaniasis, primarily caused by L. braziliensis, is endemic in Central and South America and often results in disfiguring and mutilating lesions, which
are challenging to treat. However, such cases are rare in the Mediterranean region, with a few documented instances in Tunisia characterized by the absence of mutilating lesions and a favorable response to treatment. The diagnosis of mucocutaneous leishmaniasis can be made through direct examination, histological analysis, culture in NNN medium, or PCR. Leishmania major is the predominant species reported, followed by leishmania infantum. To our knowledge, no cases of mucocutaneous leishmaniasis have been previously reported in Algeria. Our patient's case aligns with the Tunisian cases, showing a lack of mutilating lesions and a positive response to treatment. However, the mucosal involvement could be secondary to contiguous cutaneous involvement, and the associated cheek edema may be attributed to lymphatic compression from macrocheilitis.
Conclusion: Algeria, clinicians should consider mucocutaneous leishmaniasis as a differential diagnosis for atypical chronic mucosal lesions with an unknown etiology. This consideration should prompt parasitological examinations to determine the leishmanian origin. Awareness of this rare entity and its potential presentations is crucial for accurate diagnosis and appropriate management.
