de-Andrés-Beltrán Beatriz, Güeita-Rodríguez Javier, Palacios-Ceña Domingo, Rodríguez-Fernández Ángel Luis
Department of Physical Therapy, Centro RIE (Rehabilitación Infantil Especializada), 28050 Madrid, Spain.
International Doctorate School, Rey Juan Carlos University, 28008 Madrid, Spain.
Children (Basel). 2023 May 16;10(5):892. doi: 10.3390/children10050892.
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.
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。