Diercks Gillian R, Wentland Carissa, Keamy Donald, Kinane Thomas Bernard, Skotko Brian, de Guzman Vanessa, Grealish Ellen, Dobrowski John, Soose Ryan, Hartnick Christopher J
Department of Otolaryngology, Massachusetts Eye and Ear Infirmary, Boston.
Department of Otology and Laryngology, Harvard Medical School, Boston, Massachusetts.
JAMA Otolaryngol Head Neck Surg. 2018 Jan 1;144(1):37-42. doi: 10.1001/jamaoto.2017.1871.
Obstructive sleep apnea (OSA) affects up to 60% of children with Down syndrome (DS) and may persist in half of patients after adenotonsillectomy. Children with DS who have persistent OSA often do not tolerate treatment with positive pressure airway support devices or tracheotomy for their residual moderate to severe OSA. The hypoglossal nerve stimulator is an implantable device that delivers an electrical impulse to anterior branches of the hypoglossal nerve in response to respiratory variation, resulting in tongue base protrusion that alleviates upper airway obstruction in adults.
To determine whether hypoglossal nerve stimulation is safe and effective in children with DS.
DESIGN, SETTING, AND PARTICIPANTS: Case series of the first 6 adolescents with DS to undergo hypoglossal nerve stimulator implantation. Participants were 6 children and adolescents (12-18 years) with DS and severe OSA (apnea hypopnea index [AHI] > 10 events/h) despite prior adenotonsillectomy.
Inspire hypoglossal nerve stimulator placement.
Patients were monitored for adverse events. Adherence to therapy was measured by hours of use recorded by the device. Efficacy was evaluated by comparing AHI and OSA-18, a validated quality-of-life instrument, scores at baseline and follow-up.
In 6 patients (4 male, 2 female; aged 12-18 years), hypoglossal nerve stimulator therapy was well tolerated (mean use, 5.6-10.0 h/night) and effective, resulting in significant improvement in OSA. At 6- to 12-month follow-up, patients demonstrated a 56% to 85% reduction in AHI, with an overall AHI of less than 5 events/h in 4 children and less than 10 events/h in 2 children. Children also demonstrated a clinically significant improvement (mean [SD] overall change score, 1.5 [0.6]; range, 0.9-2.3) on the OSA-18, a validated quality-of-life instrument.
Hypoglossal nerve stimulation was well tolerated and effective in the study population, representing a potential therapeutic option for patients with DS and refractory OSA after adenotonsillectomy who are unable to tolerate positive pressure airway devices.
clinicaltrials.gov Identifier: NCT2344108.
阻塞性睡眠呼吸暂停（OSA）影响多达60%的唐氏综合征（DS）患儿，并且在腺样体扁桃体切除术后，半数患者的该症状可能会持续存在。患有持续性OSA的DS患儿通常无法耐受使用正压气道支持装置或气管切开术来治疗其残留的中度至重度OSA。舌下神经刺激器是一种可植入装置，它能根据呼吸变化向舌下神经的前支传递电脉冲，从而使舌根前突，缓解成人的上气道阻塞。
确定舌下神经刺激对DS患儿是否安全有效。
设计、地点和参与者：对首批6名接受舌下神经刺激器植入的DS青少年患者进行的病例系列研究。参与者为6名DS患儿及青少年（12 - 18岁），尽管之前接受了腺样体扁桃体切除术，但仍患有严重OSA（呼吸暂停低通气指数[AHI]＞10次/小时）。
植入Inspire舌下神经刺激器。
监测患者的不良事件。通过设备记录的使用时长来衡量治疗依从性。通过比较基线和随访时的AHI以及OSA - 18（一种经过验证的生活质量工具）评分来评估疗效。
6例患者（4例男性，2例女性；年龄12 - 18岁）对舌下神经刺激器治疗耐受性良好（平均使用时长为5.6 - 10.0小时/晚）且有效，OSA有显著改善。在6至12个月的随访中，患者的AHI降低了56%至85%，4名儿童的总体AHI低于5次/小时，2名儿童低于10次/小时。在经过验证的生活质量工具OSA - 18上，儿童也表现出临床上的显著改善（平均[标准差]总体变化评分，1.5[0.6]；范围为0.9 - 2.3）。
在该研究人群中，舌下神经刺激耐受性良好且有效，对于腺样体扁桃体切除术后无法耐受正压气道装置且患有难治性OSA的DS患者来说，这是一种潜在的治疗选择。
clinicaltrials.gov标识符：NCT2344108。
