改善结节性硬化症患儿的发育能力:一项先导行为干预研究。

IF 1.1 4区 医学 Q3 EDUCATION, SPECIAL
Nicole M McDonald, Carly Hyde, April Boin Choi, Amanda C Gulsrud, Connie Kasari, Charles A Nelson, Shafali S Jeste
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引用次数: 3

摘要

结节性硬化症(TSC)是一种罕见的遗传综合征,可导致神经发育障碍,包括自闭症谱系障碍和智力残疾。早在9个月大的TSC儿童就可以观察到社会沟通和早期认知能力的延迟;然而,目前尚无对TSC进行早期行为干预的研究。我们进行了一项以证据为基础的、父母介导的行为干预的试点研究,重点是改善5名TSC儿童(1-3岁)的早期社会沟通和游戏技能。在干预后,参与者表现出相对于同龄人的发展能力的维持,有时甚至有所提高。家长普遍认为干预是有帮助的,并且能够忠实地实施干预。初步结果表明,早期以游戏为基础的父母干预的初步可行性,并支持对TSC进行大规模随机临床试验的必要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Improving Developmental Abilities in Infants With Tuberous Sclerosis Complex: A Pilot Behavioral Intervention Study.

Tuberous sclerosis complex (TSC) is a rare genetic syndrome that confers risk for neurodevelopmental disorders, including autism spectrum disorder and intellectual disability. Delays in social communication and early cognitive abilities are observable as early as 9 months of age in children with TSC; however, there have been no studies of early behavioral intervention in TSC. We conducted a pilot study of an evidence-based, parent-mediated behavioral intervention focused on improving early social communication and play skills in 5 children with TSC (aged 1-3 years). Participants showed maintenance and sometimes gains in developmental abilities, relative to peers, following intervention. Parents generally found the intervention to be helpful and were able to administer the intervention with fidelity. Preliminary results demonstrate initial feasibility of an early play-based, parent-mediated intervention and support the need for a large-scale, randomized clinical trial in TSC.

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来源期刊
CiteScore
1.90
自引率
9.10%
发文量
24
期刊介绍: Infants & Young Children is an interdisciplinary journal focusing on vulnerable children from birth to five years of age and their families. Of special interest are articles involving innovative interventions, summaries of important research developments and their implications for practice, updates for high priority topic areas, balanced presentations of controversial issues, and articles that address issues involving policy, professional training, new conceptual models, and related matters. Although data are often presented primarily to illustrate points, some types of data-based articles may be appropriate.
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