University of California San Diego, Division of Respiratory Medicine, Department of Pediatrics, Rady Children's Hospital San Diego, San Diego, California.
Division of Respiratory Medicine, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
J Clin Sleep Med. 2022 Feb 1;18(2):609-616. doi: 10.5664/jcsm.9678.
Individuals with Duchenne muscular dystrophy (DMD) frequently develop sleep-disordered breathing. Noninvasive ventilation is often prescribed for sleep-disordered breathing treatment based on the American Academy of Sleep Medicine (AASM) criteria. In 2018, DMD disease-specific criteria for sleep-disordered breathing were established. Our study aimed to examine the clinical interpretation differences using these different criteria.
We performed a multicenter, retrospective chart review of children with DMD followed at The Hospital for Sick Children, Toronto, Canada, and Rady Children's Hospital, San Diego, California, who underwent polysomnography from August 1, 2012, to February 29, 2020. Baseline characteristics and polysomnography data were summarized using descriptive statistics. Agreement for the diagnosis of sleep-disordered breathing evaluated by kappa statistics and sensitivity/specificity analysis was assessed.
One hundred five male children with DMD (mean ± SD age: 12.1 ± 3.8 years; body mass index score: 0.2 ± 2.3) were included. The proportions of children with DMD that met at least 1 AASM criterion and at least 1 DMD criterion were 45.7% and 67.6%, respectively. We found that 32.4% of children met neither AASM nor DMD criteria. Overall agreement between AASM and DMD criteria was moderate (k = 0.57). There was almost perfect agreement in sleep apnea diagnosis (k = 0.90); however, there was only slight agreement in hypoventilation diagnosis (k = 0.12) between AASM and DMD criteria.
There were more children with DMD diagnosed with nocturnal hypoventilation and prescribed noninvasive ventilation using DMD criteria compared with AASM criteria. Future studies should address whether the prescription of noninvasive ventilation for children with DMD based on both criteria is associated with different clinical outcomes.
Hurvitz MS, Sunkonkit K, Massicotte C, Li R, Bhattacharjee R, Amin R. Characterization of sleep-disordered breathing in children with Duchenne muscular dystrophy by the American Academy of Sleep Medicine criteria vs disease-specific criteria: what are the differences? . 2022;18(2):609-615.
杜氏肌营养不良症（DMD）患者常发生睡眠呼吸障碍。根据美国睡眠医学学会（AASM）标准，常为睡眠呼吸障碍患者开具无创通气治疗。2018 年，建立了 DMD 睡眠呼吸障碍特异性标准。本研究旨在通过使用这些不同的标准来检查临床解释差异。
我们对加拿大安大略省多伦多 SickKids 医院和加利福尼亚州圣地亚哥 Rady 儿童医院的 105 名 DMD 患儿进行了多中心、回顾性图表审查，这些患儿在 2012 年 8 月 1 日至 2020 年 2 月 29 日期间进行了多导睡眠图检查。使用描述性统计数据总结了基线特征和多导睡眠图数据。通过 kapp 统计和敏感性/特异性分析评估了睡眠呼吸障碍诊断的一致性。
纳入 105 名男性 DMD 患儿（平均年龄±标准差：12.1±3.8 岁；体重指数 z 评分：0.2±2.3）。至少符合 1 项 AASM 标准和至少符合 1 项 DMD 标准的 DMD 患儿比例分别为 45.7%和 67.6%。我们发现，32.4%的患儿既不符合 AASM 标准，也不符合 DMD 标准。AASM 和 DMD 标准之间的总体一致性为中度（k=0.57）。在睡眠呼吸暂停诊断方面具有近乎完美的一致性（k=0.90）；然而，在 AASM 和 DMD 标准之间，在呼吸不足的诊断方面只有轻微的一致性（k=0.12）。
与 AASM 标准相比，使用 DMD 标准诊断 DMD 患儿夜间呼吸不足和开具无创通气的患儿更多。未来的研究应探讨根据这两个标准为 DMD 患儿开具无创通气治疗是否与不同的临床结果相关。
Hurvitz MS, Sunkonkit K, Massicotte C, Li R, Bhattacharjee R, Amin R. Characterization of sleep-disordered breathing in children with Duchenne muscular dystrophy by the American Academy of Sleep Medicine criteria vs disease-specific criteria: what are the differences?. 2022;18(2):609-615.
