Introduction: Orofacial granulomatosis represents a group of pathologies characterized by a granulomatous inflammation of the face and the oral mucosa. Its etiology is unknown. Since the macrocheilitis resulting from an inflammation could be associated to different pathologies, an accurate diagnosis has to be established.

Observation: A 74-year old woman developed a swelling of the inferior lip spreading towards the chin. No lesions was present on the oral mucosa. No other symptoms were reported. The biopsy and further exams concluded to an idiopathic orofacial granulomatosis. Triamcinolone 10 mg/ml was used as treatment and has allowed a lasting improvement of the swelling.

Discussion: This rare observation, especially with an older patient, has allowed to illustrate the importance of an accurate diagnosis when facing a macrocheilitis. Indeed, macrocheilitis can be associated to pathologies such as Crohn disease, sarcoidosis or tuberculosis, requiring an appropriate care.

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