Frank Porter Graham Child Development Institute, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.
J Intellect Disabil Res. 2013 Sep;57(9):837-49. doi: 10.1111/j.1365-2788.2012.01608.x. Epub 2012 Sep 14.
Fragile X syndrome (FXS) is known to be associated with a range of developmental challenges, yet the occurrence and intensity of therapy services along with associated factors have not been determined.
In a US national survey, caregivers provided information regarding the therapy services received by their sons (n = 1013) and daughters (n = 283) with FXS (from birth to 63 years; mean = 15.6 years, SD = 10.6). Caregivers reported (1) type, (2) amount, (3) location, and (4) overall satisfaction with services. Associations with other child variables and family income were also examined.
Key findings included that 72% of males and 47% of females were currently receiving at least one type of therapy service; the most common services for both males and females were speech-language therapy (ST) and occupational therapy (OT). Overall, males were more likely to receive therapy services as well as a greater number of services than females. Autism status was significantly associated with both males and females receiving ST and males receiving OT and behaviour management therapy. Therapies were provided in a variety of locations, and parents were generally satisfied with the amount and quality of therapy services. Age-related declines were evident in the use of services for both males and females, with very few individuals receiving any therapy services after 20 years of age.
This study provides a baseline description of the current state of therapy services for children with FXS, laying a foundation for future research and recommendations for service provision and policy.
脆性 X 综合征（FXS）与一系列发育挑战有关，但尚未确定治疗服务的发生和强度以及相关因素。
在美国全国性调查中，护理人员提供了其患有 FXS 的儿子（n=1013）和女儿（n=283）从出生到 63 岁（平均=15.6 岁，SD=10.6）接受的治疗服务信息。护理人员报告了（1）服务类型、（2）服务数量、（3）服务地点和（4）对服务的总体满意度。还检查了与其他儿童变量和家庭收入的关联。
主要发现包括，72%的男性和 47%的女性目前正在接受至少一种类型的治疗服务；男女最常见的服务都是言语治疗（ST）和职业治疗（OT）。总体而言，男性比女性更有可能接受治疗服务，接受的服务数量也更多。自闭症状态与男性和女性接受 ST 以及男性接受 OT 和行为管理治疗均显著相关。治疗服务在各种地点提供，父母通常对治疗服务的数量和质量感到满意。男女的服务使用都出现了与年龄相关的下降，很少有 20 岁以上的人接受任何治疗服务。
本研究提供了 FXS 儿童治疗服务现状的基线描述，为未来的研究和服务提供及政策建议奠定了基础。
