儿童神经肌肉疾病患者的睡眠呼吸障碍特征分析

杨琴, 鲍燕敏, 路新国, 贠国俊, 刘爱良, 郑跃杰, 文飞球

中国当代儿科杂志 ›› 2021, Vol. 23 ›› Issue (2) : 158-163.

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中国当代儿科杂志 ›› 2021, Vol. 23 ›› Issue (2) : 158-163. DOI: 10.7499/j.issn.1008-8830.2011030
论著·临床研究

儿童神经肌肉疾病患者的睡眠呼吸障碍特征分析

  • 杨琴1, 鲍燕敏1, 路新国2, 贠国俊3, 刘爱良1, 郑跃杰1, 文飞球1
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Clinical features of sleep-disordered breathing in children with neuromuscular disease

  • YANG Qin1, BAO Yan-Min1, LU Xin-Guo2, YUN Guo-Jun3, LIU Ai-Liang1, ZHENG Yue-Jie1, WEN Fei-Qiu1
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摘要

目的 分析神经肌肉疾病(NMD)患儿的睡眠呼吸障碍(SDB)特征,提高对儿童NMD合并SDB的诊治认识。方法 回顾性分析既往确诊NMD且行多导睡眠监测(PSG)的18例患儿(NMD组)的病例资料,选取同期睡眠习惯异常、无神经肌肉疾病且PSG显示睡眠结构正常的11例儿童作为对照组。比较两组患儿的SDB日夜间症状、阻塞性睡眠呼吸暂停(OSA)发生率、肺功能及呼气末二氧化碳分压数值、睡眠结构特征、睡眠呼吸事件等。结果 NMD组中16例(89%)存在SDB相关日、夜间临床症状,发生症状最小年龄为1岁。与对照组相比,NMD组总睡眠时间减少、睡眠效率降低(P < 0.05),快速动眼(REM)期睡眠比例下降(P < 0.05),OSA和低通气事件增加(P < 0.05),REM期氧减事件增多,该期血氧饱和度降低(P < 0.05)。NMD组中确诊OSA 17例(94%);所有患儿肺功能及呼气末二氧化碳分压均正常。结论 NMD儿童发生SDB比例高,病程早期即可出现SDB,其睡眠结构受损,睡眠效率降低,呼吸事件以阻塞型为主,氧减多发生REM期。

Abstract

Objective To study the clinical features of sleep-disordered breathing (SDB) in children with neuromuscular disease (NMD). Methods A retrospective analysis was performed on the medical data of 18 children who were diagnosed with NMD and underwent polysomnography (PSG) (NMD group). Eleven children without NMD who had abnormal sleeping habit and normal sleep structure on PSG were enrolled as the control group. The two groups were compared in terms of the daily and nocturnal symptoms of SDB, incidence rate of obstructive sleep apnea (OSA), pulmonary function, end-tidal partial pressure of carbon dioxide (PetCO2), features of sleep structure, and sleep respiratory events. Results In the NMD group, 16 children (89%) had related daily and nocturnal symptoms of SDB, and the youngest age was 1 year at the onset of such symptoms. Compared with the control group, the NMD group had significant reductions in total sleep time and sleep efficiency (P < 0.05), a significant reduction in the proportion of rapid eye movement (REM) sleep (P < 0.05), significant increases in obstructive apnea and hypopnea events (P < 0.05) and oxygen reduction events during REM sleep (P < 0.05), and a significant reduction in blood oxygen saturation during REM sleep (P < 0.05). In the NMD group, 17 children (94%) were diagnosed with OSA, and all children had normal lung function and PetCO2. Conclusions There is a high proportion of children with SDB among the children with NMD, and SDB can be observed in the early stage of NMD, which results in the damage of sleep structure and the reduction in sleep efficiency. Respiratory events are mainly obstructive events, and oxygen reduction events are mainly observed during REM sleep.

关键词

神经肌肉疾病 / 睡眠呼吸障碍 / 多导睡眠监测 / 儿童

Key words

Neuromuscular disease / Sleep-disordered breathing / Polysomnography / Child

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杨琴, 鲍燕敏, 路新国, 贠国俊, 刘爱良, 郑跃杰, 文飞球. 儿童神经肌肉疾病患者的睡眠呼吸障碍特征分析[J]. 中国当代儿科杂志. 2021, 23(2): 158-163 https://doi.org/10.7499/j.issn.1008-8830.2011030
YANG Qin, BAO Yan-Min, LU Xin-Guo, YUN Guo-Jun, LIU Ai-Liang, ZHENG Yue-Jie, WEN Fei-Qiu. Clinical features of sleep-disordered breathing in children with neuromuscular disease[J]. Chinese Journal of Contemporary Pediatrics. 2021, 23(2): 158-163 https://doi.org/10.7499/j.issn.1008-8830.2011030

参考文献

[1] Shieh PB. Advances in the genetic testing of neuromuscular diseases[J]. Neurol Clin, 2020, 38(3):519-528.
[2] Albdewi MA, Liistro G, El Tahry R. Sleep-disordered breathing in patients with neuromuscular disease[J]. Sleep Breath, 2018, 22(2):277-286.
[3] Suresh S, Wales P, Dakin C, et al. Sleep-related breathing disorder in Duchenne muscular dystrophy:disease spectrum in the paediatric population[J]. J Paediatr Child Health, 2005, 41(9-10):500-503.
[4] Al-Shamrani A, Alharbi AS. Diagnosis and management of childhood sleep-disordered breathing. Clinical approach[J]. Saudi Med J, 2020, 41(9):916-929.
[5] Polat M, Sakinci O, Ersoy B, et al. Assessment of sleep-related breathing disorders in patients with Duchenne muscular dystrophy[J]. J Clin Med Res, 2012, 4(5):332-337.
[6] 徐曼婷, 方方, 许志飞, 等. 杜氏肌营养不良患儿睡眠呼吸参数特征和危险因素分析[J]. 中国循证儿科杂志, 2016, 11(5):327-331.
[7] Sheikh O, Yokota T. Advances in genetic characterization and genotype-phenotype correlation of Duchenne and Becker muscular dystrophy in the personalized medicine era[J]. J Pers Med, 2020, 10(3):111.
[8] Atwal PS, Midei M, Adams D, et al. The Infantile Neuroaxonal Dystrophy Rating Scale (INAD-RS)[J]. Orphanet J Rare Dis, 2020, 15(1):195.
[9] Prior TW, Bayrak-Toydemir P, Lynnes TC, et al. Characterization of reference materials for spinal muscular atrophy genetic testing:a genetic testing reference materials coordination program collaborative project[J]. J Mol Diagn, 2021, 23(1):103-110.
[10] Verhulst SL, Schrauwen N, Haentjens D, et al. Reference values for sleep-related respiratory variables in asymptomatic European children and adolescents[J]. Pediatr Pulmonol, 2007, 42(2):159-167.
[11] Traeger N, Schultz B, Pollock AN, et al. Polysomnographic values in children 2-9 years old:additional data and review of the literature[J]. Pediatr Pulmonol, 2005, 40(1):22-30.
[12] 李生慧, 金星明, 沈晓明, 等. 儿童睡眠习惯问卷中文版制定及测量性能考核[J]. 中华儿科杂志, 2007, 45(3):176-180.
[13] Gurbani N, Pascoe JE, Katz S, et al. Sleep disordered breathing:assessment and therapy in the age of emerging neuromuscular therapies[J]. Pediatr Pulmonol, 2020. DOI:10.1002/ppul.24988. Online ahead of print.
[14] Hull J, Aniapravan R, Chan E, et al. British Thoracic Society guideline for respiratory management of children with neuromuscular weakness[J]. Thorax, 2012, 67(Suppl 1):i1-i40.
[15] Berry RB, Budhiraja R, Gottlieb DJ, et al. Rules for scoring respiratory events in sleep:update of the 2007 AASM Manual for the Scoring of Sleep and Associated Events. Deliberations of the Sleep Apnea Definitions Task Force of the American Academy of Sleep Medicine[J]. J Clin Sleep Med, 2012, 8(5):597-619.
[16] 中国儿童OSA诊断与治疗指南制订工作组, 中华医学会耳鼻咽喉头颈外科学分会小儿学组, 中华医学会儿科学分会呼吸学组, 等. 中国儿童阻塞性睡眠呼吸暂停诊断与治疗指南(2020)[J]. 中华耳鼻咽喉头颈外科杂志, 2020, 55(8):729-747.
[17] Aboussouan LS, Mireles-Cabodevila E. Sleep-disordered breathing in neuromuscular disease:diagnostic and therapeutic challenges[J]. Chest, 2017, 152(4):880-892.
[18] Birnkrant DJ, Bushby K, Bann CM, et al. Diagnosis and management of Duchenne muscular dystrophy, part 2:respiratory, cardiac, bone health, and orthopaedic management[J]. Lancet Neurol, 2018, 17(4):347-361.
[19] Nozoe KT, Moreira GA, Tolino JR, et al. The sleep characteristics in symptomatic patients with Duchenne muscular dystrophy[J]. Sleep Breath, 2015, 19(3):1051-1056.
[20] Boentert M. Sleep disturbances in patients with amyotrophic lateral sclerosis:current perspectives[J]. Nat Sci Sleep, 2019, 11:97-111.
[21] Jacobs TL, Brown DL, Baek J, et al. Trial of early noninvasive ventilation for ALS:a pilot placebo-controlled study[J]. Neurology, 2016, 87(18):1878-1883.
[22] Hoque R. Sleep-disordered breathing in Duchenne muscular dystrophy:an assessment of the literature[J]. J Clin Sleep Med, 2016, 12(6):905-911.
[23] Bushby K, Finkel R, Birnkrant DJ, et al. Diagnosis and management of Duchenne muscular dystrophy, part 2:implementation of multidisciplinary care[J]. Lancet Neurol, 2010, 9(2):177-189.
[24] Hussain SN, Cornachione AS, Guichon C, et al. Prolonged controlled mechanical ventilation in humans triggers myofibrillar contractile dysfunction and myofilament protein loss in the diaphragm[J]. Thorax, 2016, 71(5):436-445.
[25] Bernardes Neto SCG, Torres R, Lima Í, et al. Weaning from mechanical ventilation in people with neuromuscular disease:protocol for a systematic review[J]. BMJ Open, 2019, 9(11):e029890.
[26] Lechtzin N, Wiener CM, Shade DM, et al. Spirometry in the supine position improves the detection of diaphragmatic weakness in patients with amyotrophic lateral sclerosis[J]. Chest, 2002, 121(2):436-442.
[27] 中华医学会儿科学分会呼吸学组睡眠协作组, 《中华实用儿科临床杂志》编辑委员会. 无创正压通气治疗儿童阻塞性睡眠呼吸暂停综合征专家共识(草案)[J]. 中华实用儿科临床杂志, 2016, 31(19):1451-1455.

基金

深圳市医疗卫生三名工程项目(SZSM201512030);深圳市医学重点学科建设经费(SZXK032)。


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