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西班牙儿童全国样本中Ⅰ型脊髓性肌萎缩症新表型的临床和功能特征:一项横断面研究

Clinical and Functional Characteristics of a New Phenotype of SMA Type I among a National Sample of Spanish Children: A Cross-Sectional Study.

作者信息

de-Andrés-Beltrán Beatriz, Güeita-Rodríguez Javier, Palacios-Ceña Domingo, Rodríguez-Fernández Ángel Luis

机构信息

Department of Physical Therapy, Centro RIE (Rehabilitación Infantil Especializada), 28050 Madrid, Spain.

International Doctorate School, Rey Juan Carlos University, 28008 Madrid, Spain.

出版信息

Children (Basel). 2023 May 16;10(5):892. doi: 10.3390/children10050892.

DOI:10.3390/children10050892
PMID:37238440
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10216906/
Abstract

Spinal Muscular Atrophy (SMA) type I has classically presented extremely severe clinical features. New pharmacological treatments have led to a new phenotype of SMA. The aim of this study was to describe the current health and functional status of children with SMA. A cross-sectional study was conducted based on the STROBE guidelines. Patient questionnaires and standardized tools were used. A descriptive analysis was conducted establishing the proportions of subjects for each of the characteristics of interest. In total, 51 genetically confirmed SMA type I subjects were included. Fifty-seven percent received oral feeding, 33% received tube feeding and 10% combined both. Moreover, 21.6% had tracheostomies, and 9.8% needed more than 16 h/d ventilatory support. Regarding orthopedic status, 66.7% had scoliosis, and 68.6% had hip subluxation or dislocation. Up to 67% were able to sit independently, 23.5% walked with support and one child walked independently. Current SMA type I is a different entity from the classic phenotype but also from types II and III. In addition, no differences were found between SMA type I subgroups. These findings may enable the professionals involved in the care of these patients to improve their interventions in terms of prevention and rehabilitation measures for these children.

摘要

I型脊髓性肌萎缩症(SMA)传统上呈现出极其严重的临床特征。新的药物治疗导致了SMA的一种新表型。本研究的目的是描述SMA患儿目前的健康和功能状况。根据STROBE指南进行了一项横断面研究。使用了患者问卷和标准化工具。进行了描述性分析,确定了感兴趣的每个特征的受试者比例。总共纳入了51名基因确诊的I型SMA受试者。57%接受口服喂养,33%接受管饲,10%两者结合。此外,21.6%进行了气管切开术,9.8%每天需要超过16小时的通气支持。关于骨科状况,66.7%有脊柱侧弯,68.6%有髋关节半脱位或脱位。高达67%的患儿能够独立坐立,23.5%在支撑下行走,一名患儿能够独立行走。目前的I型SMA与经典表型不同,也与II型和III型不同。此外,I型SMA亚组之间未发现差异。这些发现可能使参与这些患者护理的专业人员能够在针对这些儿童的预防和康复措施方面改进他们的干预措施。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c491/10216906/5ff63dfb2300/children-10-00892-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c491/10216906/5ff63dfb2300/children-10-00892-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c491/10216906/5ff63dfb2300/children-10-00892-g001.jpg

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本文引用的文献

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Neurol Clin Pract. 2022 Aug;12(4):279-287. doi: 10.1212/CPJ.0000000000001179.
2
Natural history of Type 1 spinal muscular atrophy: a retrospective, global, multicenter study.1 型脊髓性肌萎缩症的自然病史:一项回顾性、全球性、多中心研究。
Orphanet J Rare Dis. 2022 Jul 29;17(1):300. doi: 10.1186/s13023-022-02455-x.
3
Type I SMA "new natural history": long-term data in nusinersen-treated patients.I 型 SMA 的“新自然病史”:用 nusinersen 治疗的患者的长期数据。
拷贝数与脊髓性肌萎缩严重程度的关联:来自哥伦比亚患者的见解
J Clin Med. 2024 Oct 25;13(21):6402. doi: 10.3390/jcm13216402.
4
Exploring variability in cognitive functioning in patients with spinal muscular atrophy: a scoping review.探索脊髓性肌萎缩症患者认知功能的变异性:范围综述。
Neurol Sci. 2024 Aug;45(8):3699-3710. doi: 10.1007/s10072-024-07503-x. Epub 2024 Apr 6.
5
Treatment of Symptomatic Spinal Muscular Atrophy with Nusinersen: A Prospective Longitudinal Study on Scoliosis Progression.用诺西那生治疗有症状的脊髓性肌萎缩症:关于脊柱侧弯进展的前瞻性纵向研究。
J Neuromuscul Dis. 2024;11(2):349-359. doi: 10.3233/JND-230077.
6
Pain in Children and Adolescents with Spinal Muscular Atrophy: A Longitudinal Study from a Patient Registry.脊髓性肌萎缩症患儿及青少年的疼痛:一项来自患者登记处的纵向研究
Children (Basel). 2023 Nov 30;10(12):1880. doi: 10.3390/children10121880.
Ann Clin Transl Neurol. 2021 Mar;8(3):548-557. doi: 10.1002/acn3.51276. Epub 2021 Feb 6.
4
Powered mobility interventions for very young children with mobility limitations to aid participation and positive development: the EMPoWER evidence synthesis.助力行动能力受限的非常年幼儿童参与和积极发展的电动移动干预措施:EMPOWER 证据综合研究。
Health Technol Assess. 2020 Oct;24(50):1-194. doi: 10.3310/hta24500.
5
Natural history in spinal muscular atrophy Type I in Taiwanese population: A longitudinal study.台湾人群中脊髓性肌萎缩症 I 型的自然病史:一项纵向研究。
Brain Dev. 2021 Jan;43(1):127-134. doi: 10.1016/j.braindev.2020.07.012. Epub 2020 Aug 30.
6
Stander Use in Spinal Muscular Atrophy: Results From a Large Natural History Database.脊柱肌肉萎缩症中站立架的使用:来自大型自然史数据库的结果
Pediatr Phys Ther. 2020 Jul;32(3):235-241. doi: 10.1097/PEP.0000000000000713.
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Int J Mol Sci. 2020 May 7;21(9):3297. doi: 10.3390/ijms21093297.
8
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Neuromuscul Disord. 2019 Nov;29(11):842-856. doi: 10.1016/j.nmd.2019.09.007. Epub 2019 Sep 12.
9
Long-term progression in type II spinal muscular atrophy: A retrospective observational study.《Ⅱ型脊髓性肌萎缩症的长期进展:一项回顾性观察研究》
Neurology. 2019 Sep 24;93(13):e1241-e1247. doi: 10.1212/WNL.0000000000008166. Epub 2019 Aug 26.
10
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Neurology. 2019 Jul 9;93(2):e149-e158. doi: 10.1212/WNL.0000000000007742. Epub 2019 Jun 4.