Language and Executive Function in Females with ASD or FXS
患有 ASD 或 FXS 的女性的语言和执行功能
基本信息
- 批准号:10657280
- 负责人:
- 金额:$ 67.74万
- 依托单位:
- 依托单位国家:美国
- 项目类别:
- 财政年份:2023
- 资助国家:美国
- 起止时间:2023-09-01 至 2028-06-30
- 项目状态:未结题
- 来源:
- 关键词:12 year oldAcademic achievementAcademic skillsAdaptive BehaviorsAddressAgeAreaAttentionBiological ModelsChildClinicalCoffeeCognitionCognitiveCommunicationCommunication impairmentComparative StudyComplexDataDevelopmentDiagnosisDiseaseEducational ModelsFemaleFoundationsFragile X SyndromeGenesGoalsImpairmentIndividualInheritedIntellectual functioning disabilityInterventionKnowledgeLanguageLanguage DevelopmentMeasuresMissionNational Institute on Deafness and Other Communication DisordersNatureNeurodevelopmental DisorderOutcomeParentsPerformancePhenotypePopulationPrevalencePublic HealthReportingResearchSamplingSchool-Age PopulationSchoolsSeveritiesSpecificityStandardizationSymptomsSyndromeTestingTimeWorkautism spectrum disorderautisticbehavioral phenotypingclinical practicecognitive abilitycourse developmenteducation planningexecutive functionexperiencefunctional outcomesimprovedindividualized medicinelanguage impairmentliteracylongitudinal designmalenon-verbalpeerprogramspsychiatric comorbidityrecruitskillssymptomatology
项目摘要
PROJECT SUMMARY/ABSTRACT
Autistic females (iASD) and females with fragile X syndrome (FXS) are two clinical groups that are significantly
understudied in research. Despite the fact that, like their male peers, females experience pervasive, lifelong
difficulties with language, adaptive functioning, psychiatric comorbidities, and academic achievement, there
remains a dearth of research regarding phenotypic development in iASD females and females with FXS. FXS
is the most common inherited cause of intellectual disability and the most common single gene cause of ASD,
with 14-45% of females with FXS having a co-occurring ASD diagnosis. Research on males has identified key
areas of overlap and distinction between FXS and iASD, particularly in language and cognition. Thus, studies
comparing the phenotypes of FXS and iASD in females, focusing on areas of overlap and divergence, are
particularly important because they provide the foundation for clinical and educational planning and elucidating
the mechanisms producing the phenotypes of FXS and iASD. Executive functions are one aspect of cognition
that has received significant attention in FXS and iASD, since they are an important contributor to real-world
outcomes and particularly amenable to treatment. The hierarchical competing systems model of executive
functions describes the interrelationships between language and executive functions in neurotypical
development, but these relationships have yet to be explored in iASD females and females with FXS. Both
language and executive functions are known to have a significant and pervasive impact on critical
developmental outcomes including academic skills (i.e., literacy), adaptive behavior, and psychiatric
symptomatology. The impact of language and executive functions on these outcomes has not been explored in
iADS females and females with FXS. There is an urgent need to characterize the language and cognitive
phenotypes of school-age iASD females and females with FXS within a developmental framework to
understand the nature and course of development, and impact over time on important outcomes. Through
three specific aims, we will determine the extent, nature and syndrome specificity of impairments in language
and executive functions in iASD females and females with FXS, and test their causal relationships through
longitudinal associations. We will recruit 8-12 year-old females with FXS (n = 60) and iASD females (n = 60).
The proposed study will include standardized assessments, expressive language samples, experimental tasks,
and parent report measures taken at two time points. We will determine the impact of language and executive
functions on key developmental outcomes including literacy, adaptive functioning, and psychiatric
symptomatology. The data from this study will provide critical information for clinicians and educators working
with iASD females and females with FXS and will provide data necessary for the development of targeted
interventions for cognitive and language skills directly impacting developmental outcomes.
项目总结/摘要
孤独症女性(iASD)和脆性X综合征女性(FXS)是两个临床组,
在研究中被忽视。尽管事实上,像他们的男性同龄人一样,女性经历了普遍的,终身的
语言困难,适应功能,精神病合并症和学业成绩,
仍然缺乏关于iASD女性和FXS女性表型发育的研究。FXS
是智力残疾最常见的遗传原因,也是ASD最常见的单基因原因,
14-45%患有FXS的女性患有共同发生的ASD诊断。对男性的研究发现了
FXS和iASD之间的重叠和区别,特别是在语言和认知方面。因此,研究
比较女性FXS和iASD的表型,重点是重叠和分歧的领域,
特别重要,因为它们为临床和教育规划提供了基础,
FXS和iASD表型的产生机制。执行功能是认知的一个方面
这在FXS和iASD中受到了极大的关注,因为它们是现实世界的重要贡献者。
结果,特别适合治疗。经理人的递阶竞争系统模型
功能描述了语言和执行功能之间的相互关系,
但是这些关系还有待于在iASD女性和FXS女性中进行探索。两
众所周知,语言和执行功能对关键的
发展成果,包括学术技能(即,识字)、适应行为和精神病学
医学。语言和执行功能对这些结果的影响尚未探讨,
iADS雌性和FXS雌性。迫切需要对语言和认知进行表征
学龄期iASD女性和FXS女性在发育框架内的表型,
了解发展的性质和进程,以及随着时间的推移对重要成果的影响。通过
三个具体目标,我们将确定语言障碍的程度,性质和综合征特异性
iASD女性和FXS女性的执行功能,并通过以下方式测试其因果关系:
纵向关联我们将招募患有FXS(n = 60)和iASD(n = 60)的8-12岁女性。
拟议的研究将包括标准化评估,表达语言样本,实验任务,
和父母报告在两个时间点采取的措施。我们将确定语言和执行的影响
关键发展结果的功能,包括识字,适应功能和精神
医学。这项研究的数据将为临床医生和教育工作者提供关键信息。
患有iASD女性和FXS女性,并将提供制定目标所需的数据
认知和语言技能的干预直接影响发展成果。
项目成果
期刊论文数量(0)
专著数量(0)
科研奖励数量(0)
会议论文数量(0)
专利数量(0)
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Audra Marie Sterling其他文献
Audra Marie Sterling的其他文献
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{{ truncateString('Audra Marie Sterling', 18)}}的其他基金
Grammatical Development in Boys with Fragile X Syndrome and Autism
患有脆性 X 综合征和自闭症的男孩的语法发展
- 批准号:
8443403 - 财政年份:2011
- 资助金额:
$ 67.74万 - 项目类别:
Grammatical Development in Boys with Fragile X Syndrome and Autism
患有脆性 X 综合征和自闭症的男孩的语法发展
- 批准号:
8103637 - 财政年份:2011
- 资助金额:
$ 67.74万 - 项目类别:
Grammatical Development in Boys with Fragile X Syndrome and Autism
患有脆性 X 综合征和自闭症的男孩的语法发展
- 批准号:
8259150 - 财政年份:2011
- 资助金额:
$ 67.74万 - 项目类别:
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