Department of Medicine, Division of Neurology, University of Toronto, Toronto, Ontario, Canada.
Department of Pediatrics, Division of Neurology, University of Toronto, Toronto, Ontario, Canada.
J Child Neurol. 2020 Aug;35(9):600-606. doi: 10.1177/0883073820924213. Epub 2020 Jun 2.
The purpose of this study was to prospectively evaluate sleep patterns and the presence of sleep-disordered breathing in children with myasthenia gravis. We further aimed to examine the relationship between sleep and daytime respiratory function using spirometry tests including upright and supine forced vital capacity, sniff nasal inspiratory pressure, and maximal inspiratory pressure.
Eleven children between 3 and 18 years old with confirmed myasthenia gravis were recruited from The Hospital for Sick Children Neuromuscular Clinic in this prospective observational study. After informed consent was obtained, patients underwent a comprehensive clinical assessment with collection of anthropometric data. Following this, all subjects performed pulmonary function tests, overnight polysomnography and completed the Epworth Sleepiness Scale questionnaire.
Two of eleven children who reported no symptoms of sleep disordered breathing were diagnosed with mild to moderate obstructive sleep apnea. Pulmonary function tests showed abnormal maximal inspiratory pressure in 6 of 11 patients, whereas seated forced vital capacity as well as seated to supine forced vital capacity ratios were normal in the entire group.
In our small group of pediatric myasthenia gravis subjects, there was an unexpected finding of obstructive sleep apnea in 2 of the 11 patients studied. Maximal inspiratory pressure appears to be a more sensitive method of detecting abnormalities compared to upright or seated forced vital capacity. A larger multicenter study is needed to validate our findings and to determine the impact of obstructive sleep apnea in the pediatric myasthenia gravis population as well as risk factors associated with sleep disordered breathing.
本研究旨在前瞻性评估重症肌无力患儿的睡眠模式和睡眠呼吸障碍的发生情况。我们还旨在通过包括直立和仰卧位用力肺活量、嗅探鼻吸气压力和最大吸气压力在内的肺功能测试来检查睡眠与日间呼吸功能之间的关系。
在这项前瞻性观察研究中,我们从 SickKids 神经肌肉诊所招募了 11 名年龄在 3 至 18 岁之间的确诊重症肌无力患儿。在获得知情同意后,患者接受了全面的临床评估并收集了人体测量数据。之后,所有受试者均进行了肺功能测试、整夜多导睡眠图检查,并完成了 Epworth 嗜睡量表问卷调查。
在报告无睡眠呼吸障碍症状的 11 名儿童中,有 2 名被诊断为轻度至中度阻塞性睡眠呼吸暂停。肺功能测试显示 11 名患者中有 6 名存在异常最大吸气压力,而整个组的坐位用力肺活量以及坐位与仰卧位用力肺活量比值均正常。
在我们的一小部分儿科重症肌无力患者中,令人意外的是,在 11 名研究患者中有 2 名患有阻塞性睡眠呼吸暂停。与直立位或坐位用力肺活量相比,最大吸气压力似乎是一种更敏感的检测异常的方法。需要更大的多中心研究来验证我们的发现,并确定阻塞性睡眠呼吸暂停对儿科重症肌无力人群的影响以及与睡眠呼吸障碍相关的危险因素。