Use of continuous positive airway pressure to treat obstructive sleep apnea with an orofacial vascular malformation and bilateral tonsillar enlargement.

摘要

Craniofacial structures such as tonsillar enlargement and reduced lateral pharyngeal diameter can contribute to the development of obstructive sleep apnea (OSA) . Tumors of the soft tissues of the pharynx are uncommon risk factors . We report the first case to our knowledge of OSA with an orofacial vascular malformation and bilateral tonsillar enlargement that was treated with continuous positive airway pressure (CPAP).A 43-year-old man presented with snoring and witnessed apneas. His Epworth Sleepiness Scale score (ESS) was 20/24. Oral exam showed low-flow venous vascular malformation with bluish discoloration on the left side of the face, involving the cheek with extension to the upper lip, palate, left orbit and left side of the oral cavity (Figs. 1 and 2). Present since birth, this vascular malformation was compressible without tenderness. He underwent surgical resection about 25 years ago, but it recurred and had slowly grown in size over the last few years, coinciding with the onset of his symptoms. He also had grade 3-4 bilateral tonsillar enlargement (Fig. 3).A split-night polysomnogram revealed an apnea-hypopnea index (AHI) of 67 with a minimum oxygen desaturation of 69%. CPAP was introduced via a small Respironics ComfortClassic~(TM) nasal mask (Murraysville, PA), which was tolerated without discomfort, leaks, or mouth breathing. At 9 cm of water pressure, AHI wasreduced to 2 and snoring was completely eliminated with improvement in oxygen saturation above 89%. At one-month follow-up, the patient was compliant with CPAP and ESS dropped to 10.