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Am J Geriatr Psychiatry. 2023 Nov;31(11):943-952. doi: 10.1016/j.jagp.2023.05.010. Epub 2023 May 28.
2
Pediatric Obstructive Sleep Apnea and Sickle Cell Disease: Demographic and Polysomnographic Features.儿科阻塞性睡眠呼吸暂停与镰状细胞病:人口统计学和多导睡眠图特征。
Laryngoscope. 2023 Jul;133(7):1766-1772. doi: 10.1002/lary.30638. Epub 2023 Mar 8.
3
The association between sleep disturbances and neurocognitive function in pediatric sickle cell disease.儿童镰状细胞病中睡眠障碍与神经认知功能的关系。
Sleep Med. 2022 Sep;97:27-35. doi: 10.1016/j.sleep.2022.05.015. Epub 2022 May 26.
4
Polisomographic Investigation of Sleep Disorders in Patients with Sickle Cell Anemia and Evaluation of the Effect on the Prognosis.镰状细胞贫血患者睡眠障碍的多导睡眠图调查及对预后影响的评估
Turk Thorac J. 2022 May;23(3):218-224. doi: 10.5152/TurkThoracJ.2022.21199.
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单基因疾病镰状细胞病患儿多导睡眠图结果的多中心比较研究。

Multicenter comparative study of polysomnography outcomes in children with the monogenic disorder sickle cell disease.

作者信息

Alishlash Ammar Saadoon, Nourani Anis Rabbani, Lebensburger Jeffrey, Rothman Jennifer A, Ali-Dinar Tarig, Ezmigna Dima

机构信息

Department of Pediatrics, Heersink School of Medicine, University of Alabama at Birmingham, Birmingham, Alabama.

Department of Pediatrics, Duke University, Durham, North Carolina.

出版信息

J Clin Sleep Med. 2025 Feb 1;21(2):297-304. doi: 10.5664/jcsm.11372.

DOI:10.5664/jcsm.11372
PMID:39347561
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11789244/
Abstract

STUDY OBJECTIVES

Sleep-disordered breathing is prevalent in children with sickle cell disease (SCD) and is associated with worse outcomes. This study aimed to compare the outcomes of polysomnography (PSG) performed for pediatric patients with SCD at 3 US centers.

METHODS

We included patients with SCD aged 0-21 years who underwent PSG at 3 American Academy of Sleep Medicine-accredited centers, the University of Alabama at Birmingham, the University of Florida, and Duke University Hospital, between 2012 and 2022. Descriptive statistics were used as appropriate to compare the baseline characters and PSG outcomes among the different centers.

RESULTS

A total of 210 children with SCD from the 3 centers were included, with comparable sex, SCD genotypes, hemoglobin, hematocrit levels, and chronic transfusion. Children from the different centers exhibited variations in age ( < .001), body mass index ( < .05), mean corpuscular volume ( < .05), and hydroxyurea usage ( < .05) at the time of the PSG. Overall, the 3 centers showed significantly different PSG outcomes. Patients from the University of Florida had worse obstructive sleep apnea, oxygenation, and periodic leg movement events, together with lower hydroxyurea usage, and those from Duke University Hospital showed higher hypoventilation and arousal indices.

CONCLUSIONS

This multicenter study underscores variations in PSG outcomes among pediatric SCD patients at different centers in the southeastern United States. These findings emphasize the need for standardized approaches to screen for sleep-disordered breathing, refer to PSG, and interpret the results in children with SCD. These conclusions may apply to other genetic disorders associated with an increased risk of sleep-disordered breathing.

CITATION

Alishlash AS, Nourani AR, Lebensburger J, Rothman JA, Ali-Dinar T, Ezmigna D. Multicenter comparative study of polysomnography outcomes in children with the monogenic disorder sickle cell disease. 2025;21(2):297-304.

摘要

研究目的

睡眠呼吸障碍在镰状细胞病(SCD)患儿中很常见,且与更差的预后相关。本研究旨在比较美国3家中心对儿科SCD患者进行多导睡眠图(PSG)检查的结果。

方法

我们纳入了2012年至2022年间在3家美国睡眠医学学会认可的中心(阿拉巴马大学伯明翰分校、佛罗里达大学和杜克大学医院)接受PSG检查的0至21岁SCD患者。采用描述性统计方法,适当比较不同中心之间的基线特征和PSG结果。

结果

共纳入了来自3家中心的210名SCD患儿,他们在性别、SCD基因型、血红蛋白、血细胞比容水平和慢性输血方面具有可比性。不同中心的儿童在进行PSG检查时,年龄(<.001)、体重指数(<.05)、平均红细胞体积(<.05)和羟基脲使用情况(<.05)存在差异。总体而言,3家中心的PSG结果存在显著差异。佛罗里达大学的患者阻塞性睡眠呼吸暂停、氧合和周期性腿部运动事件更严重,同时羟基脲使用量较低;而杜克大学医院的患者低通气和觉醒指数较高。

结论

这项多中心研究强调了美国东南部不同中心儿科SCD患者PSG结果的差异。这些发现强调了需要采用标准化方法来筛查睡眠呼吸障碍、转诊进行PSG检查以及解读SCD患儿的检查结果。这些结论可能适用于其他与睡眠呼吸障碍风险增加相关的遗传疾病。

引用文献

Alishlash AS, Nourani AR, Lebensburger J, Rothman JA, Ali-Dinar T, Ezmigna D. 单基因疾病镰状细胞病患儿多导睡眠图结果的多中心比较研究。2025;21(2):297 - 304。