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Morbihan syndrome is a rare condition and its pathogenesis is not fully known. This entityh is characterized by facial edema and facial erythema, with a slow, symmetrical appearance, affecting the upper portion of the face. This case report is about a patient with an atypical case of Morbihan syndrome, with asymmetric facial involvement. Clinical and histopathological aspects were highlighted aiming the diagnosis of this rare pathology, as well as possible differential diagnoses and treatment options. It has also been aimed to call attention to less typical forms of this disease.