Abstract Introduction Obstructive sleep apnea (OSA) is characterized by daytime sleepiness, fatigue, hypertension, and morning headaches. Treatment of severe OSA may include continuous (CPAP) or biphasic positive airway pressure (BiPAP), tracheostomy, and hypoglossal nerve stimulation (HNS). This case highlights a unique combination therapy using BiPAP and HNS to manage severe OSA in a patient with craniofacial abnormalities likely due to Crouzon syndrome, characterized by coronal craniosynostosis, brachycephaly, orbital proptosis, and maxillary hypoplasia. The case illustrates an alternative approach that may prevent the need for tracheostomy. Case Presentation A 53-year-old man with a longstanding history of severe OSA and craniofacial abnormalities consistent with Crouzon syndrome had undergone three prior tracheostomies, the last maintained for 13 years. Following successful HNS device implantation and tracheostomy decannulation, he presented with three months of hoarseness, dyspnea, fatigue, headaches, and excessive daytime sleepiness. High-resolution chest CT was negative for interstitial lung disease but revealed tracheal stenosis. The patient declined a repeat tracheostomy after unsuccessful BiPAP and oxygen therapy trials. Combination therapy with the previously implanted HNS device and high-pressure BiPAP (IPAP 16 cm H2O, EPAP 8 cm H2O) resulted in significant symptomatic improvement and avoidance of tracheostomy. Discussion OSA management ranges from lifestyle modifications and CPAP/BiPAP to surgical options such as HNS or tracheostomy for refractory disease. Decannulation after tracheostomy is rarely successful, with early complications including granulation tissue, infection, and stoma revision. HNS has demonstrated durable efficacy beyond five years in selected patients. While combination CPAP and bilateral HNS use has been reported in OSA secondary to Treacher Collins syndrome, literature describing BiPAP with HNS is extremely limited. This case demonstrates the potential of combined BiPAP and HNS therapy to control severe OSA in craniofacial abnormality, preventing repeat tracheostomy and improving quality of life. Conclusion Severe OSA in patients with craniofacial abnormalities requires individualized therapy. This case suggests that BiPAP combined with HNS may represent an effective alternative to tracheostomy in complex or refractory cases. Further studies are needed to determine long-term outcomes and to define whether this dual-modality approach can serve as a sustained therapy or bridge to definitive management. This abstract is funded by: None
Herrera et al. (Fri,) studied this question.