Abstract Introduction OSA is one of the most common comorbidities seen in pediatric patients with Trisomy 21 (T21) with a prevalence of up to 79% compared to 1-5% seen in the general pediatrics population. Uncontrolled OSA can have serious clinical consequences, including cognitive and behavioral impairment and cardio-respiratory sequelae such as pulmonary hypertension, making OSA particularly important to treat. Unfortunately, OSA can be difficult to manage in patients with T21 due to a multitude of anatomic risk factors and difficulties with adherence to standard therapy (i.e. positive airway pressure). However, hypoglossal nerve stimulation (HNS) has recently emerged as a promising therapy for adolescents with T21. In this study, we aimed to evaluate the safety and efficacy of HNS in adolescents at Oklahoma Children’s Hospital. Methods In a retrospective chart review, we evaluated the tolerance, subjective changes, and apnea-hypopnea index (AHI) differences before and after implantation of HNS in adolescents with T21. Baseline data included demographics, patient/parental experience, and polysomnography (PSG) results. Only adolescents (ages 13-19) with T21 were included. The primary outcome measurement was AHI following HNS implantation compared to AHI prior to HNS implantation. Secondary outcomes included tolerance to the HNS device and subjective differences in symptoms after implantation. Results We evaluated 4 adolescents who received HNS implantation. Each patient had improvement in AHI with absolute decreases ranging from 7.8-35.64 (average of 17.5) and relative decreases ranging from 51.7-85.7% (average of 72.0%). Furthermore, caretakers of each patient also reported subjective improvement in sleep symptoms and quality of life, including improvements in snoring, nighttime restlessness, daytime somnolence, and school performance. Conclusion While OSA can be a difficult disease to control in patients with T21, HNS appears to be a viable and effective option for management of treatment-resistant OSA in adolescents with T21 with this case series showing improvements in both subjective and objective outcomes for OSA in every patient. This study was limited by the small number of pediatric patients with T21 and HNS along with a lack of formal tools to assess tolerance and improvements in quality of life. Future research is needed to assess long-term safety and efficacy of HNS in children. Support (if any)
Sittner et al. (Fri,) studied this question.