A 53-year-old man was evaluated for snoring, dysphagia for solid foods and difficulty of breathing and a polysomnographic recording was consistent with a diagnosis of obstructive sleep apnoea syndrome (OSAS). A flexible fiberoptic bronchoscopy (FFB) showed the presence of a nodular lesion of the posterior ventral surface of the tongue strictly connected to the left lateral border of the epiglottis. The biopsy specimen taken from the lesion was consistent with sarcoidosis. No involvement of pulmonary parenchyma, lymph nodes or other organs was recognized. After two months of steroid treatment, symptoms disappeared and resolution of the nodular lesion at the FFB and normalization of the polysomnographic recording were observed. This is the first report of orolaryngeal sarcoidosis associated with OSAS as the only clinical presentation of the disease.