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DOI: 10.48087/BJMS.2026.130317
Authors: Amine RAHOU, Saliha BOUKHEMACHA, Najib TALEB, Malika BACHAOUI
Affiliations: Faculté de Médecine de l’Université Oran 1 Ahmed Ben Bella. Service de Médecine Interne, EHU 1er Novembre 1954, Oran, Algérie.
Abstract
Melkersson–Rosenthal syndrome (MRS) is a rare orofacial granulomatosis that primarily affects young adults, with an estimated incidence of less than 0.1% and a slight female predominance. The diagnosis relies on a characteristic clinical triad: recurrent orofacial swelling, fissured tongue, and recurrent peripheral facial palsy; however, this complete triad is observed in only 8 to 25% of cases. Labial biopsy may reveal non-caseating epithelioid granulomas, which are essential for excluding other granulomatous conditions. We report two cases of young women presenting with the complete MRS triad. Both cases were characterized by recurrent lip swelling and successive episodes of facial palsy, resulting in significant functional and aesthetic impairment. Biopsy, imaging, and immunological workup excluded alternative diagnoses. Initial systemic corticosteroid therapy proved ineffective in both patients, necessitating the introduction of methotrexate, which led to notable clinical improvement. The etiopathogenesis of MRS remains poorly understood, with genetic, environmental, and immunological factors potentially implicated. Diagnosis is frequently delayed, particularly in monosymptomatic presentations. Treatment is primarily based on corticosteroid therapy, whereas immunosuppressants and reconstructive surgery are reserved for severe or disfiguring forms. Given the considerable aesthetic and psychosocial burden of the disease, a multidisciplinary approach — including psychological support — is warranted.
Keywords: Melkersson–Rosenthal syndrome, peripheral facial paralysis, fissured tongue, orofacial swelling, non-caseating granuloma.