目的 分析Williams综合征（Williams syndrome，WS）儿童的早期运动发育情况，为临床早期干预提供依据。 方法 对2018年9月至2021年8月间59例0~24月龄的WS儿童的临床资料进行回顾性分析，其中男40例，女19例。根据Peabody运动发育量表（Peabody Developmental Motor Scale Ⅱ）测试结果，分析不同年龄患儿的运动发育情况。 结果 男女儿童月龄和运动商的比较差异均无统计学意义（P>0.05）。<6月龄组、6~<12月龄组、12~<18月龄组和18~24月龄组的粗大运动商分别为94±5、78±11、71±8、63±8，精细运动商分别为94±5、80±10、74±9、65±9，粗大运动商和精细运动商随着月龄增长逐渐下降（P<0.05）。<6月龄组、6~<12月龄组、12~<18月龄组和18~24月龄组的粗大运动发育异常率分别为0%、53%、87%、93%，精细运动发育异常率分别为0%、47%、67%、93%，粗大运动和精细运动发育异常率均随月龄增长而上升（P<0.05）。 结论 6个月以内WS儿童未表现出明显的运动能力落后，但随年龄增长其运动能力逐渐降低，运动发育迟缓率逐渐增加。因此必须做好该人群运动能力的随访工作，早期制订干预策略以减少运动发育迟缓的发生。 中国当代儿科杂志，2022，24（9）：984-987］
Abstract
Objective To study the early motor development of children with William syndrome (WS). Methods The medical data of 59 children with WS (40 males and 19 females) aged 0-24 months from September 2018 to August 2021 were retrospectively analyzed. Based on the test results of the Peabody Developmental Motor Scale II, the motor development ability of the children of different ages was analyzed. Results There was no significant difference in age and motor quotient between boys and girls (P>0.05). For the age groups of <6 months, 6 to <12 months, 12 to <18 months, and 18 to 24 months, the gross-motor quotients were 94±5, 78±11, 71±8, and 63±8, respectively, and the fine-motor quotients were 94±5, 80±10, 74±9, and 65±9, respectively. Both the gross- and fine-motor quotients significantly decreased with age (P<0.05). For the above age groups, the rates of gross-motor abnormalities were 0%, 53%, 87%, and 93%, respectively, and the rates of fine-motor development abnormalities were 0%, 47%, 67%, and 93%, respectively. The rates of gross- and fine-motor development abnormalities increased significantly with age (P<0.05). Conclusions Children with WS have no obvious motor delays within 6 months of age, but present with decreasing motor ability and an increasing incidence of motor delays with age. Therefore, it is necessary to follow up their motor abilities and provide early intervention to decrease the incidence of motor developmental delays.
关键词
Williams综合征 /
Peabody运动发育量表 /
运动发育 /
儿童
Key words
Williams syndrome /
Peabody Developmental Motor Scale II /
Motor development /
Child
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参考文献
1 Ramírez-Velazco A, Aguayo-Orozco TA, Figuera L, et al. Williams-Beuren syndrome in Mexican patients confirmed by FISH and assessed by aCGH[J]. J Genet, 2019, 98(2): 34. PMID: 31204697.
2 Ji C, Yao D, Li MY, et al. A study on facial features of children with Williams syndrome in China based on three-dimensional anthropometric measurement technology[J]. Am J Med Genet A, 2020, 182(9): 2102-2109. PMID: 32706523. DOI: 10.1002/ajmg.a.61750.
3 Wuang YP, Tsai HY. Sensorimotor and visual perceptual functioning in school-aged children with Williams syndrome[J]. J Intellect Disabil Res, 2017, 61(4): 348-362. PMID: 27900816. DOI: 10.1111/jir.12346.
4 Greiner de Magalh?es C, Cardoso-Martins C, Mervis CB. Spelling abilities of school-aged children with Williams syndrome[J]. Res Dev Disabil, 2022, 120: 104129. PMID: 34875548. PMCID: PMC8724450. DOI: 10.1016/j.ridd.2021.104129.
5 Farran EK, Bowler A, Karmiloff-Smith A, et al. Cross-domain associations between motor ability, independent exploration, and large-scale spatial navigation; attention deficit hyperactivity disorder, Williams syndrome, and typical development[J]. Front Hum Neurosci, 2019, 13: 225. PMID: 31333435. PMCID: PMC6618048. DOI: 10.3389/fnhum.2019.00225.
6 Mervis CB, Greiner de Magalh?es C, Cardoso-Martins C. Concurrent predictors of word reading and reading comprehension for 9-year-olds with Williams syndrome[J]. Read Writ, 2022, 35(2): 377-397. PMID: 35221525. PMCID: PMC8827302. DOI: 10.1007/s11145-021-10163-4.
7 解雅春, 姚天红, 胡小沙, 等. 儿童早期发展训练对婴儿神经心理发育影响的分析[J]. 中国儿童保健杂志, 2013, 21(11): 1228-1230.
8 王美琪, 梅花, 刘春枝, 等. Peabody运动发育量表在早产儿神经发育障碍评估中的应用[J]. 中国小儿急救医学, 2017, 24(10): 760-763. DOI: 10.3760/cma.j.issn.1673-4912.2017.10.010.
9 杨红, 史惟, 王素娟, 等. Peabody运动发育量表在婴幼儿评估中的信度和效度研究[J]. 中国儿童保健杂志, 2010, 18(2): 121-123.
10 FolioMR, FewellRR. Peabody 运动发育量表-上-检查者手册 项目测试指导[M]. 李明, 黄真, 译. 北京: 北京大学医学出版社, 2006: 3-60.
11 杨玉凤, 王惠珊, 洪琦, 等. 儿童发育行为心理评定量表[M]. 北京: 人民卫生出版社, 2016: 115-118.
12 Kozel BA, Barak B, Kim CA, et al. Williams syndrome[J]. Nat Rev Dis Primers, 2021, 7(1): 42. PMID: 34140529. DOI: 10.1038/s41572-021-00276-z.
13 Miezah D, Porter M, Batchelor J, et al. Cognitive abilities in Williams syndrome[J]. Res Dev Disabil, 2020, 104: 103701. PMID: 32554266. DOI: 10.1016/j.ridd.2020.103701.
14 Braga AC, Carreiro LRR, Tafla TL, et al. Cognitive and behavioral profile of Williams syndrome toddlers[J]. Codas, 2018, 30(4): e20170188. PMID: 30043830. DOI: 10.1590/2317-1782/20182017188.
15 Sauna-Aho O, Bjelogrlic-Laakso N, Sirén A, et al. Cognition in adults with Williams syndrome—a 20-year follow-up study[J]. Mol Genet Genomic Med, 2019, 7(6): e695. PMID: 31033253. PMCID: PMC6565587. DOI: 10.1002/mgg3.695.
16 Morris CA, Braddock SR, Council on Genetics. Health care supervision for children with Williams syndrome[J]. Pediatrics, 2020, 145(2): e20193761. PMID: 31964759. DOI: 10.1542/peds.2019-3761.
17 陈维军, 季钗. 儿童Williams综合征行为问题研究[J]. 中国儿童保健杂志, 2013, 21(12): 1309-1312.
18 Tsai SW, Wu SK, Liou YM, et al. Early development in Williams syndrome[J]. Pediatr Int, 2008, 50(2): 221-224. PMID: 18353064. DOI: 10.1111/j.1442-200X.2008.02563.x.
19 Mayall LA, D'Souza H, Hill EL, et al. Motor abilities and the motor profile in individuals with Williams syndrome[J]. Adv Neurodev Disord, 2021, 5(1): 46-60. DOI: 10.1007/s41252-020-00173-8.
20 王翠, 李一芳, 黄真, 等. Alberta婴儿运动量表与Peabody粗大运动发育量表在高危儿中的预测能力研究[J]. 中国康复医学杂志, 2019, 34(3): 293-296. DOI: 10.3969/j.issn.1001-1242.2019.03.008.
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