PDF(Pubmed)
Abstract：
Arthrogryposis multiplex congenita (AMC) is characterized by joint contractures and muscle weakness, which limit daily activities. Youths with AMC require frequent physical therapeutic follow-ups to limit the recurrence of contractures and maintain range of motion (ROM) and muscle strength; however, access to specialized care may be limited because of geographical distance. Telerehabilitation can offer a potential solution for delivering frequent follow-ups for youth with AMC, but research on the use of telerehabilitation in children with musculoskeletal disorders is scarce.
The study aims to evaluate the feasibility of delivering a home exercise program (HEP) by using telerehabilitation for youth with AMC. We also aim to explore the effectiveness of the HEP as a secondary aim.
Youths aged between 8 and 21 years with AMC were recruited at the Shriners Hospitals for Children-Canada. The participants completed baseline and post-HEP questionnaires (the Physical Activity Questionnaire for Adolescents, Pediatrics Outcomes Data Collection Instrument, and Adolescent and Pediatric Pain Tool), and clinicians assessed their active ROM using a virtual goniometer. Clinicians used the Goal Attainment Scale with the participants to identify individualized goals to develop a 12-week HEP and assess the achievement of these goals. Follow-ups were conducted every 3 weeks to adjust the HEP. Data on withdrawal rates and compliance to the HEP and follow-ups were collected to assess the feasibility of this approach. The interrater reliability of using a virtual goniometer was assessed using the intraclass correlation coefficient and associated 95% CI. Nonparametric tests were used to evaluate feasibility and explore the effectiveness of the HEP.
Of the 11 youths who were recruited, 7 (median age: 16.9 years) completed the HEP. Of the 47 appointments scheduled, 5 had to be rescheduled in ≤24 hours. The participants performed their HEP 2.04 times per week (95% CI 1.25-4.08) and reported good satisfaction with the approach. A general intraclass correlation coefficient of 0.985 (95% CI 0.980-0.989) was found for the web-based ROM measurement. Individualized goals were related to pain management; endurance in writing, standing, or walking; sports; and daily activities. In total, 12 of the 15 goals set with the participants were achieved. Statistically significant improvements were observed in the pain and comfort domain of the Pediatrics Outcomes Data Collection Instrument (preintervention: median 71; 95% CI 34-100; postintervention: median 85; 95% CI 49-100; P=.08) and Physical Activity Questionnaire for Adolescents (preintervention: median 1.62; 95% CI 1.00-2.82; postintervention: median 2.32; 95% CI 1.00-3.45; P=.046).
The remote delivery of an HEP for youth with AMC is feasible. Promising results were found for the effectiveness of the HEP in helping youths with AMC to achieve their goals. The next step will be to assess the effectiveness of this exercise intervention in a randomized controlled trial.
RR2-10.2196/18688.
The study aims to evaluate the feasibility of delivering a home exercise program (HEP) by using telerehabilitation for youth with AMC. We also aim to explore the effectiveness of the HEP as a secondary aim.
Youths aged between 8 and 21 years with AMC were recruited at the Shriners Hospitals for Children-Canada. The participants completed baseline and post-HEP questionnaires (the Physical Activity Questionnaire for Adolescents, Pediatrics Outcomes Data Collection Instrument, and Adolescent and Pediatric Pain Tool), and clinicians assessed their active ROM using a virtual goniometer. Clinicians used the Goal Attainment Scale with the participants to identify individualized goals to develop a 12-week HEP and assess the achievement of these goals. Follow-ups were conducted every 3 weeks to adjust the HEP. Data on withdrawal rates and compliance to the HEP and follow-ups were collected to assess the feasibility of this approach. The interrater reliability of using a virtual goniometer was assessed using the intraclass correlation coefficient and associated 95% CI. Nonparametric tests were used to evaluate feasibility and explore the effectiveness of the HEP.
Of the 11 youths who were recruited, 7 (median age: 16.9 years) completed the HEP. Of the 47 appointments scheduled, 5 had to be rescheduled in ≤24 hours. The participants performed their HEP 2.04 times per week (95% CI 1.25-4.08) and reported good satisfaction with the approach. A general intraclass correlation coefficient of 0.985 (95% CI 0.980-0.989) was found for the web-based ROM measurement. Individualized goals were related to pain management; endurance in writing, standing, or walking; sports; and daily activities. In total, 12 of the 15 goals set with the participants were achieved. Statistically significant improvements were observed in the pain and comfort domain of the Pediatrics Outcomes Data Collection Instrument (preintervention: median 71; 95% CI 34-100; postintervention: median 85; 95% CI 49-100; P=.08) and Physical Activity Questionnaire for Adolescents (preintervention: median 1.62; 95% CI 1.00-2.82; postintervention: median 2.32; 95% CI 1.00-3.45; P=.046).
The remote delivery of an HEP for youth with AMC is feasible. Promising results were found for the effectiveness of the HEP in helping youths with AMC to achieve their goals. The next step will be to assess the effectiveness of this exercise intervention in a randomized controlled trial.
RR2-10.2196/18688.
摘要：
多发性先天性关节病（AMC）的特征是关节挛缩和肌肉无力，限制日常活动。患有AMC的年轻人需要频繁的物理治疗随访，以限制挛缩的复发并保持活动范围（ROM）和肌肉力量；然而，由于地理上的距离，获得专门护理的机会可能受到限制。远程康复可以提供一种潜在的解决方案，为AMC的青少年提供频繁的随访，但是关于在患有肌肉骨骼疾病的儿童中使用远程康复的研究很少。
该研究旨在评估通过使用远程康复为AMC青少年提供家庭锻炼计划（HEP）的可行性。我们还旨在探索HEP的有效性作为次要目标。
加拿大Shriners儿童医院招募了年龄在8至21岁之间的AMC青年。参与者完成了基线和HEP后问卷(青少年体力活动问卷，儿科结果数据收集仪，和青少年和儿科疼痛工具)，临床医生使用虚拟测角仪评估他们的活动ROM。临床医生与参与者一起使用目标达成量表来确定个性化目标，以制定12周的HEP并评估这些目标的实现情况。每3周进行随访以调整HEP。收集有关退出率和对HEP的依从性以及随访的数据，以评估该方法的可行性。使用虚拟测角仪的评分者之间的可靠性使用组内相关系数和相关的95％CI进行评估。使用非参数检验来评估HEP的可行性并探索其有效性。
在被招募的11名青年中,7（中位年龄：16.9岁）完成了HEP。在预定的47个约会中，5必须在≤24小时内重新安排。参与者每周进行2.04次HEP（95％CI1.25-4.08），并报告对该方法的满意度良好。基于网络的ROM测量的一般组内相关系数为0.985(95%CI0.980-0.989)。个性化目标与疼痛管理有关；写作中的耐力，站立,或步行；运动；和日常活动。总的来说,与参与者一起设定的15个目标中的12个已实现。在儿科结果数据收集工具的疼痛和舒适度方面观察到统计学上的显着改善（干预前：中位数71；95％CI34-100；干预后：中位数85；95％CI49-100；P=.08）和青少年体育锻炼问卷（干预前：中位数1.62；95％CI1.00-2.82；干预后：中位数2.32；95％CI-3.45；P=.
使用AMC为青少年远程交付HEP是可行的。HEP在帮助AMC青少年实现目标方面的有效性得到了有希望的结果。下一步将是在随机对照试验中评估这种运动干预的有效性。
RR2-10.2196/18688。
该研究旨在评估通过使用远程康复为AMC青少年提供家庭锻炼计划（HEP）的可行性。我们还旨在探索HEP的有效性作为次要目标。
加拿大Shriners儿童医院招募了年龄在8至21岁之间的AMC青年。参与者完成了基线和HEP后问卷(青少年体力活动问卷，儿科结果数据收集仪，和青少年和儿科疼痛工具)，临床医生使用虚拟测角仪评估他们的活动ROM。临床医生与参与者一起使用目标达成量表来确定个性化目标，以制定12周的HEP并评估这些目标的实现情况。每3周进行随访以调整HEP。收集有关退出率和对HEP的依从性以及随访的数据，以评估该方法的可行性。使用虚拟测角仪的评分者之间的可靠性使用组内相关系数和相关的95％CI进行评估。使用非参数检验来评估HEP的可行性并探索其有效性。
在被招募的11名青年中,7（中位年龄：16.9岁）完成了HEP。在预定的47个约会中，5必须在≤24小时内重新安排。参与者每周进行2.04次HEP（95％CI1.25-4.08），并报告对该方法的满意度良好。基于网络的ROM测量的一般组内相关系数为0.985(95%CI0.980-0.989)。个性化目标与疼痛管理有关；写作中的耐力，站立,或步行；运动；和日常活动。总的来说,与参与者一起设定的15个目标中的12个已实现。在儿科结果数据收集工具的疼痛和舒适度方面观察到统计学上的显着改善（干预前：中位数71；95％CI34-100；干预后：中位数85；95％CI49-100；P=.08）和青少年体育锻炼问卷（干预前：中位数1.62；95％CI1.00-2.82；干预后：中位数2.32；95％CI-3.45；P=.
使用AMC为青少年远程交付HEP是可行的。HEP在帮助AMC青少年实现目标方面的有效性得到了有希望的结果。下一步将是在随机对照试验中评估这种运动干预的有效性。
RR2-10.2196/18688。
