Abstract：
OBJECTIVE: The objective was to identify clinical and radiological factors associated with sleep-disordered breathing (SDB) in children with Chiari type I malformation (CIM) and to evaluate the efficacy of foramen magnum decompression (FMD) in resolving SDB.
METHODS: A retrospective chart review was conducted for all children evaluated for CIM at a single institution from 2002 to 2022, identifying all children who had undergone nocturnal polysomnography (PSG). Apnea-hypopnea index (AHI) score, sleep apnea type (obstructive, central, mixed, and unspecified), clinical manifestations, and radiological measurements were recorded. SDB was considered present when officially diagnosed in the PSG report. Logistic regression was performed to identify factors correlating with the presence of SDB. For children with SDB who underwent FMD, the Wilcoxon signed-rank test was used to assess AHI improvement.
RESULTS: Of the 997 children referred for CIM, 310 completed PSG. SDB was diagnosed in 147 patients (overall prevalence 14.7%, 95% CI 12.7%-17.1%; prevalence among children with PSG 47.4%, 95% CI 41.9%-53%). Specific SDB diagnosis consisted of 33% of patients with central sleep apnea, 27% with obstructive sleep apnea, 9% mixed, and 31% unspecified. Lower cranial nerve (CN) dysfunction (OR 3.891, p = 0.009), tonsillar position (OR 1.049, p = 0.017), Chiari type 1.5 malformation (OR 1.862, p = 0.044), and BMI (OR 1.039, p = 0.036) were significantly associated with presence of SDB. Of the 310 patients who underwent PSG, 47 were originally categorized as asymptomatic: 27 (57%) of these asymptomatic patients were diagnosed with SDB on PSG. Of children diagnosed with SDB, 34 completed PSG before and after FMD. Median AHI score decreased from 6.5 preoperatively to 1.8 postoperatively, with a median (IQR) difference of -2.3 (-11.9 to 0.1) (p = 0.001). Twelve (35%) had resolution of SDB.
CONCLUSIONS: The authors\' findings suggest that the prevalence of SDB in children with CIM is high (15%-47%). Furthermore, lower CN dysfunction, Chiari type 1.5, lower tonsillar position, and higher BMI may be risk factors. Notably, SDB can be present even in the absence of clinical symptoms. This study also demonstrates that surgical intervention has the potential to reduce the severity of SDB. These results could help clinicians identify CIM patients at risk for SDB and those who may benefit from surgical decompression.
METHODS: A retrospective chart review was conducted for all children evaluated for CIM at a single institution from 2002 to 2022, identifying all children who had undergone nocturnal polysomnography (PSG). Apnea-hypopnea index (AHI) score, sleep apnea type (obstructive, central, mixed, and unspecified), clinical manifestations, and radiological measurements were recorded. SDB was considered present when officially diagnosed in the PSG report. Logistic regression was performed to identify factors correlating with the presence of SDB. For children with SDB who underwent FMD, the Wilcoxon signed-rank test was used to assess AHI improvement.
RESULTS: Of the 997 children referred for CIM, 310 completed PSG. SDB was diagnosed in 147 patients (overall prevalence 14.7%, 95% CI 12.7%-17.1%; prevalence among children with PSG 47.4%, 95% CI 41.9%-53%). Specific SDB diagnosis consisted of 33% of patients with central sleep apnea, 27% with obstructive sleep apnea, 9% mixed, and 31% unspecified. Lower cranial nerve (CN) dysfunction (OR 3.891, p = 0.009), tonsillar position (OR 1.049, p = 0.017), Chiari type 1.5 malformation (OR 1.862, p = 0.044), and BMI (OR 1.039, p = 0.036) were significantly associated with presence of SDB. Of the 310 patients who underwent PSG, 47 were originally categorized as asymptomatic: 27 (57%) of these asymptomatic patients were diagnosed with SDB on PSG. Of children diagnosed with SDB, 34 completed PSG before and after FMD. Median AHI score decreased from 6.5 preoperatively to 1.8 postoperatively, with a median (IQR) difference of -2.3 (-11.9 to 0.1) (p = 0.001). Twelve (35%) had resolution of SDB.
CONCLUSIONS: The authors\' findings suggest that the prevalence of SDB in children with CIM is high (15%-47%). Furthermore, lower CN dysfunction, Chiari type 1.5, lower tonsillar position, and higher BMI may be risk factors. Notably, SDB can be present even in the absence of clinical symptoms. This study also demonstrates that surgical intervention has the potential to reduce the severity of SDB. These results could help clinicians identify CIM patients at risk for SDB and those who may benefit from surgical decompression.
摘要：
目的：目的是确定与ChiariI型畸形（CIM）儿童睡眠呼吸障碍（SDB）相关的临床和放射学因素，并评估大孔减压（FMD）在解决SDB中的疗效。
方法：对2002年至2022年在单一机构接受CIM评估的所有儿童进行回顾性图表审查，确定了所有接受夜间多导睡眠图（PSG）的儿童。呼吸暂停低通气指数(AHI)评分,睡眠呼吸暂停类型(阻塞性，中央,混合,并且未指定)，临床表现，和放射学测量记录。在PSG报告中正式诊断时，SDB被认为存在。进行Logistic回归以确定与SDB存在相关的因素。对于接受口蹄疫的SDB儿童，使用Wilcoxon符号秩检验评估AHI改善情况.
结果：在CIM转介的997名儿童中，310完成PSG。在147例患者中诊断出SDB（总体患病率为14.7％，95%CI12.7%-17.1%；PSG患儿的患病率为47.4%，95%CI41.9%-53%）。具体的SDB诊断包括33%的中心性睡眠呼吸暂停患者，27%患有阻塞性睡眠呼吸暂停，9%混合，和31％未指定。下颅神经（CN）功能障碍（OR3.891，p=0.009），扁桃体位置（OR1.049，p=0.017），Chiari型1.5畸形（OR1.862，p=0.044），BMI（OR1.039，p=0.036）与SDB的存在显着相关。在接受PSG的310名患者中，最初将47例归类为无症状：这些无症状患者中有27例（57％）在PSG上被诊断为SDB。在被诊断为SDB的儿童中，34在FMD前后完成PSG。AHI评分中位数从术前的6.5下降到术后的1.8，中位数（IQR）差异为-2.3（-11.9至0.1）（p=0.001）。12人(35%)具有SDB的分辨率。
结论：作者的研究结果表明，CIM儿童中SDB的患病率很高（15％-47％）。此外，较低的CN功能障碍，Chiari1.5型，扁桃体下部位置，较高的BMI可能是危险因素。值得注意的是,即使在没有临床症状的情况下也可以存在SDB。这项研究还表明，手术干预有可能降低SDB的严重程度。这些结果可以帮助临床医生识别有SDB风险的yCIM患者以及可能从手术减压中受益的患者。
方法：对2002年至2022年在单一机构接受CIM评估的所有儿童进行回顾性图表审查，确定了所有接受夜间多导睡眠图（PSG）的儿童。呼吸暂停低通气指数(AHI)评分,睡眠呼吸暂停类型(阻塞性，中央,混合,并且未指定)，临床表现，和放射学测量记录。在PSG报告中正式诊断时，SDB被认为存在。进行Logistic回归以确定与SDB存在相关的因素。对于接受口蹄疫的SDB儿童，使用Wilcoxon符号秩检验评估AHI改善情况.
结果：在CIM转介的997名儿童中，310完成PSG。在147例患者中诊断出SDB（总体患病率为14.7％，95%CI12.7%-17.1%；PSG患儿的患病率为47.4%，95%CI41.9%-53%）。具体的SDB诊断包括33%的中心性睡眠呼吸暂停患者，27%患有阻塞性睡眠呼吸暂停，9%混合，和31％未指定。下颅神经（CN）功能障碍（OR3.891，p=0.009），扁桃体位置（OR1.049，p=0.017），Chiari型1.5畸形（OR1.862，p=0.044），BMI（OR1.039，p=0.036）与SDB的存在显着相关。在接受PSG的310名患者中，最初将47例归类为无症状：这些无症状患者中有27例（57％）在PSG上被诊断为SDB。在被诊断为SDB的儿童中，34在FMD前后完成PSG。AHI评分中位数从术前的6.5下降到术后的1.8，中位数（IQR）差异为-2.3（-11.9至0.1）（p=0.001）。12人(35%)具有SDB的分辨率。
结论：作者的研究结果表明，CIM儿童中SDB的患病率很高（15％-47％）。此外，较低的CN功能障碍，Chiari1.5型，扁桃体下部位置，较高的BMI可能是危险因素。值得注意的是,即使在没有临床症状的情况下也可以存在SDB。这项研究还表明，手术干预有可能降低SDB的严重程度。这些结果可以帮助临床医生识别有SDB风险的yCIM患者以及可能从手术减压中受益的患者。
