1型神经纤维瘤病患儿的运动能力
Motor proficiency in children with neurofibromatosis type 1.
作者信息
Johnson Barbara A, MacWilliams Bruce A, Carey John C, Viskochil David H, D'Astous Jacques L, Stevenson David A
机构信息
Shriners Hospitals for Children Salt Lake City, Salt Lake City, Utah, USA.
出版信息
Pediatr Phys Ther. 2010 Winter;22(4):344-8. doi: 10.1097/PEP.0b013e3181f9dbc8.
Abstract
PURPOSE
Neurofibromatosis type 1 (NF1) is a genetic disorder with associated musculoskeletal abnormalities, tumors, and developmental delays. The purpose of this study was to investigate and characterize the motor proficiency of children with NF1.
METHODS
Children with NF1 were assessed using the Bruininks-Oseretsky Test (BOT 2) instrument. The NF1 group scores were compared with age and sex-matched test norms.
RESULTS
Twenty-six children participated in the study. The NF1 group had statistically significant lower scores (P < .05) for the total motor composite (Z = -1.62) and 7 of the 8 subtests. Nineteen percent (n = 5) scored in the average category, 54% (n = 14) scored in the below-average category, and 27% (n = 7) scored in the well-below-average category.
CONCLUSIONS
Children with NF1 have significantly lower motor proficiency than the BOT 2 normative scores. The results indicate the BOT 2 is useful in identifying and characterizing delays in motor proficiency for children with NF1.
摘要
目的
1型神经纤维瘤病（NF1）是一种伴有肌肉骨骼异常、肿瘤和发育迟缓的遗传性疾病。本研究的目的是调查和描述NF1患儿的运动能力。
方法
使用布鲁因inks-奥塞雷茨基测试（BOT 2）工具对NF1患儿进行评估。将NF1组的分数与年龄和性别匹配的测试常模进行比较。
结果
26名儿童参与了该研究。NF1组在总运动综合得分（Z = -1.62）以及8个分测试中的7个分测试中，得分在统计学上显著较低（P <.05）。19%（n = 5）的儿童得分处于平均水平，54%（n = 14）的儿童得分处于低于平均水平，27%（n = 7）的儿童得分处于远低于平均水平。
结论
NF1患儿的运动能力明显低于BOT 2常模分数。结果表明，BOT 2有助于识别和描述NF1患儿的运动能力延迟情况。
相似文献
1
Motor proficiency in children with neurofibromatosis type 1.1型神经纤维瘤病患儿的运动能力
Pediatr Phys Ther. 2010 Winter;22(4):344-8. doi: 10.1097/PEP.0b013e3181f9dbc8.
2
Ecological validity of the German Bruininks-Oseretsky Test of Motor Proficiency - 2nd Edition.德国布鲁因inks - 奥塞列茨基运动能力测验第二版的生态效度
Hum Mov Sci. 2017 Jun;53:45-54. doi: 10.1016/j.humov.2016.10.005. Epub 2016 Nov 7.
3
Commentary on Motor proficiency in children with neurofibromatosis type 1.关于1型神经纤维瘤病患儿运动能力的评论
Pediatr Phys Ther. 2010 Winter;22(4):349.
4
Review of the Bruininks-Oseretsky Test of Motor Proficiency, Second Edition (BOT-2).《布鲁茵inks-奥泽斯基运动技能测验第二版（BOT-2）综述》
Phys Occup Ther Pediatr. 2007;27(4):87-102.
5
Motor impairment in children with Neurofibromatosis type 1: Effect of the comorbidity with language disorders.1型神经纤维瘤病患儿的运动障碍：与语言障碍合并存在的影响。
Brain Dev. 2016 Feb;38(2):181-7. doi: 10.1016/j.braindev.2015.08.001. Epub 2015 Aug 29.
6
Relations between fine motor skill and parental report of attention in young children with neurofibromatosis type 1.1型神经纤维瘤病幼儿精细运动技能与家长注意力报告之间的关系。
J Clin Exp Neuropsychol. 2014;36(9):930-43. doi: 10.1080/13803395.2014.957166. Epub 2014 Oct 6.
7
Movement difficulties in children with ADHD: Comparing the long- and short-form Bruininks-Oseretsky Test of Motor Proficiency-Second Edition (BOT-2).儿童注意缺陷多动障碍的运动困难：比较长式和短式布鲁金斯-奥塞尔斯基运动技能测试第二版（BOT-2）。
Aust Occup Ther J. 2020 Apr;67(2):153-161. doi: 10.1111/1440-1630.12641. Epub 2020 Jan 16.
8
Comparing the Movement Assessment Battery for Children with the Bruininks-Oseretsky Test of Motor Proficiency in adolescents with and without schizophrenia.比较用于儿童的运动评估量表与布鲁因inks - 奥塞瑞斯基运动技能测验在患精神分裂症和未患精神分裂症青少年中的应用。
Physiother Res Int. 2023 Apr;28(2):e1980. doi: 10.1002/pri.1980. Epub 2022 Nov 9.
9
Fine motor skill proficiency in typically developing children: On or off the maturation track?正常发育儿童的精细运动技能熟练程度：是否遵循成熟轨迹？
Hum Mov Sci. 2016 Apr;46:78-85. doi: 10.1016/j.humov.2015.12.011. Epub 2015 Dec 28.
10
Deficient motor timing in children with neurofibromatosis type 1.1型神经纤维瘤病患儿的运动时间缺陷。
Res Dev Disabil. 2014 Nov;35(11):3131-8. doi: 10.1016/j.ridd.2014.07.059. Epub 2014 Aug 20.
引用本文的文献
1
Unraveling neuronal and metabolic alterations in neurofibromatosis type 1.解析神经纤维瘤病 1 型中的神经元和代谢改变。
J Neurodev Disord. 2024 Aug 31;16(1):49. doi: 10.1186/s11689-024-09565-6.
2
NF1 miniswine model the cellular disruptions and behavioral presentations of NF1-associated cognitive and motor impairment.NF1 小型猪模型模拟了 NF1 相关认知和运动障碍的细胞紊乱和行为表现。
Clin Transl Sci. 2024 Jun;17(6):e13858. doi: 10.1111/cts.13858.
3
Motor Function and Physiology in Youth With Neurofibromatosis Type 1.青少年 1 型神经纤维瘤病的运动功能和生理学。
Pediatr Neurol. 2023 Jun;143:34-43. doi: 10.1016/j.pediatrneurol.2023.02.014. Epub 2023 Mar 3.
4
Cerebellum-dependent associative learning is not impaired in a mouse model of neurofibromatosis type 1.小脑依赖的联想学习在神经纤维瘤病 1 型的小鼠模型中没有受损。
Sci Rep. 2022 Nov 9;12(1):19041. doi: 10.1038/s41598-022-21429-4.
5
The skeletal muscle phenotype of children with Neurofibromatosis Type 1 - A clinical perspective.神经纤维瘤病 1 型患儿的骨骼肌表型——临床视角。
J Musculoskelet Neuronal Interact. 2022 Mar 1;22(1):70-78.
6
Atypical Microdeletions: Challenges and Opportunities for Genotype/Phenotype Correlations in Patients with Large Deletions.非典型微缺失：大缺失患者基因型/表型相关性的挑战与机遇。
Genes (Basel). 2021 Oct 19;12(10):1639. doi: 10.3390/genes12101639.
7
Longitudinal Investigation of Early Motor Development in Neurofibromatosis Type 1.神经纤维瘤病 1 型早期运动发育的纵向研究。
J Pediatr Psychol. 2022 Feb 14;47(2):180-188. doi: 10.1093/jpepsy/jsab090.
8
Attention and Motor Learning in Adult Patients with Neurofibromatosis Type 1.注意力和运动学习在成年 1 型神经纤维瘤病患者中的作用。
J Atten Disord. 2022 Feb;26(4):563-572. doi: 10.1177/10870547211012035. Epub 2021 May 12.
9
Cell autonomous requirement of neurofibromin (Nf1) for postnatal muscle hypertrophic growth and metabolic homeostasis.神经纤维瘤病基因（Nf1）在出生后肌肉肥大生长和代谢稳态中的细胞自主需求。
J Cachexia Sarcopenia Muscle. 2020 Dec;11(6):1758-1778. doi: 10.1002/jcsm.12632. Epub 2020 Oct 19.
10
Brain-wide structural and functional disruption in mice with oligodendrocyte-specific deletion is rescued by inhibition of nitric oxide synthase.脑内少突胶质细胞特异性敲除小鼠的全脑结构和功能障碍可被一氧化氮合酶抑制剂挽救。
Proc Natl Acad Sci U S A. 2020 Sep 8;117(36):22506-22513. doi: 10.1073/pnas.2008391117. Epub 2020 Aug 24.
本文引用的文献
1
The effectiveness of exercise intervention on inhibitory control in children with developmental coordination disorder: using a visuospatial attention paradigm as a model.运动干预对发展性协调障碍儿童抑制控制能力的影响：以视空间注意范式为模型。
Res Dev Disabil. 2009 Nov-Dec;30(6):1268-80. doi: 10.1016/j.ridd.2009.05.001. Epub 2009 Jun 3.
2
Rasch analysis of the Bruininks-Oseretsky Test of Motor Proficiency-Second Edition in intellectual disabilities.智力障碍者第二版布鲁因克斯-奥塞尔斯基运动技能测试的 Rasch 分析。
Res Dev Disabil. 2009 Nov-Dec;30(6):1132-44. doi: 10.1016/j.ridd.2009.03.003. Epub 2009 Apr 22.
3
Muscular force is reduced in neurofibromatosis type 1.1型神经纤维瘤病患者的肌肉力量会减弱。
J Musculoskelet Neuronal Interact. 2009 Jan-Mar;9(1):15-7.
4
Reliability and responsiveness of the Bruininks-Oseretsky Test of Motor Proficiency-Second Edition in children with intellectual disability.布鲁因inks - 奥塞瑞斯基运动技能测试第二版在智力残疾儿童中的信度与反应度
Res Dev Disabil. 2009 Sep-Oct;30(5):847-55. doi: 10.1016/j.ridd.2008.12.002. Epub 2009 Jan 31.
5
Task-specific training, learning, and memory for children with developmental coordination disorder: a pilot study.针对发育性协调障碍儿童的特定任务训练、学习与记忆：一项初步研究。
Percept Mot Skills. 2008 Oct;107(2):473-80. doi: 10.2466/pms.107.6.473-480.
6
Bone health and fracture rate in individuals with neurofibromatosis 1 (NF1).1型神经纤维瘤病（NF1）患者的骨骼健康与骨折率
J Med Genet. 2009 Apr;46(4):259-65. doi: 10.1136/jmg.2008.061895. Epub 2008 Dec 9.
7
Impact of neurofibromatosis type 1 on school performance.1型神经纤维瘤病对学业成绩的影响。
J Child Neurol. 2008 Sep;23(9):1002-10. doi: 10.1177/0883073808316366.
8
Review of the Bruininks-Oseretsky Test of Motor Proficiency, Second Edition (BOT-2).《布鲁茵inks-奥泽斯基运动技能测验第二版（BOT-2）综述》
Phys Occup Ther Pediatr. 2007;27(4):87-102.
9
Bone mineral density in children and adolescents with neurofibromatosis type 1.1型神经纤维瘤病患儿及青少年的骨矿物质密度
J Pediatr. 2007 Jan;150(1):83-8. doi: 10.1016/j.jpeds.2006.10.048.
10
Learning disabilities in children with neurofibromatosis type 1: subtypes, cognitive profile, and attention-deficit-hyperactivity disorder.1型神经纤维瘤病患儿的学习障碍：亚型、认知特征及注意力缺陷多动障碍
Dev Med Child Neurol. 2006 Dec;48(12):973-7. doi: 10.1017/S0012162206002131.
Zotero 插件