Suppr超能文献

患有和未患自闭症的脆性X综合征患儿的运动能力

Motor abilities of children diagnosed with fragile X syndrome with and without autism.

作者信息

Zingerevich C, Greiss-Hess L, Lemons-Chitwood K, Harris S W, Hessl D, Cook K, Hagerman R J

机构信息

Occupational Therapy Department, Rady Children Hospital, San Diego, CA, USA.

出版信息

J Intellect Disabil Res. 2009 Jan;53(1):11-8. doi: 10.1111/j.1365-2788.2008.01107.x. Epub 2008 Sep 3.

Abstract

BACKGROUND

Previous studies suggested that children diagnosed with fragile X syndrome (FXS) often meet criteria for autism or PDD. This study describes the fine motor abilities of children diagnosed with FXS with and without autism spectrum disorder, and compares the motor scores of those groups controlling for cognitive level.

METHOD

Forty-eight children, ages 12-76 months (SD = 16) diagnosed with FXS were assessed with the Mullen Scales of Early Learning, and the Autism Diagnostic Observation Schedule. Their parents were interviewed with the Autism Diagnostic Interview-Revised. We used a one-way analysis of variance to determine if the fine motor scale of the Mullen would show group differences based on autism classifications for the sample. In addition, we used Pearson correlation coefficient to examine the relationship between the cognitive level, the autism severity and the motor abilities. Lastly, we conducted a one-way analysis of covariance to determine the difference between the motor abilities of the Autism Spectrum Disorder groups controlling for cognitive level.

RESULTS

We found that 60% of the children with FXS met criteria for autism or Pervasive Developmental Disorder - Not otherwise specified (PDD-NOS). Children with FXS with autism and PDD-NOS had lower fine motor scores than those without. However, there was no significant association between degree of motor impairment and communication and social impairments after controlling for cognitive level, indicating that cognitive level contributes to impaired motor abilities of children diagnosed with FXS and autism, more than the severity of autism symptoms.

CONCLUSION

children with FXS and autism are at risk for impaired motor abilities. Implications for development and intervention are discussed.

摘要

背景

先前的研究表明,被诊断为脆性X综合征(FXS)的儿童通常符合自闭症或广泛性发育障碍的标准。本研究描述了患有和未患有自闭症谱系障碍的FXS患儿的精细运动能力,并比较了在控制认知水平的情况下这些组别的运动得分。

方法

对48名年龄在12 - 76个月(标准差 = 16)被诊断为FXS的儿童进行了早期学习穆伦量表和自闭症诊断观察量表的评估。他们的父母接受了自闭症诊断访谈修订版的访谈。我们使用单因素方差分析来确定穆伦精细运动量表是否会根据样本的自闭症分类显示出组间差异。此外,我们使用皮尔逊相关系数来检验认知水平、自闭症严重程度和运动能力之间的关系。最后,我们进行了单因素协方差分析,以确定在控制认知水平的情况下自闭症谱系障碍组之间运动能力的差异。

结果

我们发现60%的FXS患儿符合自闭症或未特定的广泛性发育障碍(PDD-NOS)的标准。患有自闭症和PDD-NOS的FXS患儿的精细运动得分低于未患这些疾病的患儿。然而,在控制认知水平后,运动障碍程度与沟通和社交障碍之间没有显著关联,这表明认知水平对被诊断为FXS和自闭症患儿运动能力受损的影响大于自闭症症状的严重程度。

结论

患有FXS和自闭症的儿童存在运动能力受损的风险。文中讨论了对发育和干预的影响。

相似文献

1
Motor abilities of children diagnosed with fragile X syndrome with and without autism.
J Intellect Disabil Res. 2009 Jan;53(1):11-8. doi: 10.1111/j.1365-2788.2008.01107.x. Epub 2008 Sep 3.
3
Motor skills and calibrated autism severity in young children with autism spectrum disorder.
Adapt Phys Activ Q. 2014 Apr;31(2):95-105. doi: 10.1123/apaq.2013-0068.
4
Autism spectrum disorder in fragile X syndrome: a longitudinal evaluation.
Am J Med Genet A. 2009 Jun;149A(6):1125-37. doi: 10.1002/ajmg.a.32848.
5
Expressive language in male adolescents with fragile X syndrome with and without comorbid autism.
J Intellect Disabil Res. 2010 Mar;54(3):246-65. doi: 10.1111/j.1365-2788.2010.01255.x. Epub 2010 Feb 8.
6
Psychophysiological responses to emotional stimuli in children and adolescents with autism and fragile X syndrome.
J Clin Child Adolesc Psychol. 2015;44(2):250-63. doi: 10.1080/15374416.2013.843462. Epub 2013 Oct 24.
8
Autism in fragile X syndrome: a category mistake?
J Am Acad Child Adolesc Psychiatry. 2010 Sep;49(9):921-33. doi: 10.1016/j.jaac.2010.07.001. Epub 2010 Aug 1.
10
Consistency between research and clinical diagnoses of autism among boys and girls with fragile X syndrome.
J Intellect Disabil Res. 2014 Oct;58(10):940-52. doi: 10.1111/jir.12121. Epub 2014 Feb 17.

引用本文的文献

2
Motor Influences on Communication: Comparisons Between Down Syndrome and Fragile X Syndrome.
Am J Intellect Dev Disabil. 2021 Nov 1;126(6):460-476. doi: 10.1352/1944-7558-126.6.460.
3
Speech Impairments Explain Unique Variance in Adaptive Behavior Skills in Young People With Down Syndrome.
Am J Speech Lang Pathol. 2021 Jan 27;30(1):253-259. doi: 10.1044/2020_AJSLP-20-00054. Epub 2020 Nov 16.
5
What's missing in autism spectrum disorder motor assessments?
J Neurodev Disord. 2018 Dec 13;10(1):33. doi: 10.1186/s11689-018-9257-6.
6
Parents' initial concerns about the development of their children later diagnosed with fragile X syndrome.
J Intellect Dev Disabil. 2017;42(2):114-122. doi: 10.3109/13668250.2016.1228858. Epub 2016 Sep 18.
8
Association of fragile X syndrome, Robertsonian translocation (13, 22) and autism in a child.
Clujul Med. 2017;90(4):445-448. doi: 10.15386/cjmed-763. Epub 2017 Oct 20.
9
Decreased home cage movement and oromotor impairments in adult Fmr1-KO mice.
Genes Brain Behav. 2017 Jun;16(5):564-573. doi: 10.1111/gbb.12374. Epub 2017 Mar 29.
10
Brief Report: Autism Symptoms in Infants with Fragile X Syndrome.
J Autism Dev Disord. 2016 Dec;46(12):3830-3837. doi: 10.1007/s10803-016-2903-5.

本文引用的文献

1
Autism profiles of males with fragile X syndrome.
Am J Ment Retard. 2008 Nov;113(6):427-38. doi: 10.1352/2008.113:427-438.
2
Secondary medical diagnosis in fragile X syndrome with and without autism spectrum disorder.
Am J Med Genet A. 2008 Aug 1;146A(15):1911-6. doi: 10.1002/ajmg.a.32290.
3
The Prader-Willi phenotype of fragile X syndrome.
J Dev Behav Pediatr. 2007 Apr;28(2):133-8. doi: 10.1097/01.DBP.0000267563.18952.c9.
6
Fragile X syndrome and autism at the intersection of genetic and neural networks.
Nat Neurosci. 2006 Oct;9(10):1221-5. doi: 10.1038/nn1765.
8
Autistic behavior in children with fragile X syndrome: prevalence, stability, and the impact of FMRP.
Am J Med Genet A. 2006 Sep 1;140A(17):1804-13. doi: 10.1002/ajmg.a.31286.
9
Autism spectrum disorders and attention-deficit/hyperactivity disorder in boys with the fragile X premutation.
J Dev Behav Pediatr. 2006 Apr;27(2 Suppl):S137-44. doi: 10.1097/00004703-200604002-00012.
10
Video analysis of sensory-motor features in infants with fragile X syndrome at 9-12 months of age.
J Autism Dev Disord. 2005 Oct;35(5):645-56. doi: 10.1007/s10803-005-0008-7.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验