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《Williams-Beuren 综合征患儿生长评估:系统综述》

Growth assessment in children with Williams-Beuren syndrome: a systematic review.

机构信息

Department of Pediatrics, School of Medical Sciences (FCM), State University of Campinas (UNICAMP), Campinas, Sao Paulo, Brazil.

Prof. Dr. José Aristodemo Pinotti Women's Hospital, UNICAMP, Sao Paulo, Campinas, Brazil.

出版信息

J Appl Genet. 2020 May;61(2):205-212. doi: 10.1007/s13353-020-00551-x. Epub 2020 Mar 10.

Abstract

Williams-Beuren syndrome (WBS) is a rare genetic disease caused by a sporadic heterozygous microdeletion in 7q11.23. It is characterized by distinctive facial appearance, cardiopathy, short stature, intellectual disability, and endocrine abnormalities. To evaluate the growth pattern of patients with WBS and to identify the prevalence of malnutrition, overweight, and obesity in this population, a systematic review of studies published in English, between 1987 and 2018, was performed following the PRISMA protocol using the PubMed, Cochrane, and BIREME databases. Original articles and articles that evaluated growth status using weight, or height, or head circumference (HC), or body mass index (BMI) of individuals with WBS were included. Case reports, articles with data from other syndromes, and articles that did not present as a central theme the evaluation of growth were not included. WBS presented specific growth pattern, characterized by intrauterine growth restriction, low weight, length, and HC at birth. This global growth delay persisted during childhood and adolescence. BMI was not different to the reference population, and obesity was not observed in childhood. The mechanisms that determine this typical growth pattern are not totally clear; however, the typical pubertal development of these patients and the intrinsic and secondary lesions caused by microdeletion at 7q11.23 seem to be the major factors involved. Conclusion: Patients with WBS have a growth pattern different from the general reference population. The reference charts for normal population should not be used for WBS patients because it often underestimate their growth. Specific growth charts for WBS patients are necessary.

摘要

威廉姆斯-比伦综合征(WBS)是一种罕见的遗传性疾病,由 7q11.23 处的散发性杂合性微缺失引起。其特征为独特的面部外观、心脏病、身材矮小、智力障碍和内分泌异常。为了评估 WBS 患者的生长模式,并确定该人群中营养不良、超重和肥胖的患病率,我们按照 PRISMA 方案,使用 PubMed、Cochrane 和 BIREME 数据库,对 1987 年至 2018 年间发表的英文文献进行了系统评价。纳入了使用 WBS 个体的体重、身高、头围(HC)或体重指数(BMI)评估生长状况的原始文章和文章。排除了病例报告、来自其他综合征的数据的文章以及未将生长评估作为主要主题的文章。WBS 表现出特定的生长模式,其特征为宫内生长受限、出生时体重、长度和 HC 较低。这种全身性生长延迟在儿童期和青春期持续存在。BMI 与参考人群无差异,且在儿童期未观察到肥胖。决定这种典型生长模式的机制尚不完全清楚;然而,这些患者典型的青春期发育以及 7q11.23 处微缺失引起的内在和继发损伤似乎是涉及的主要因素。结论:WBS 患者的生长模式与一般参考人群不同。不应将正常人群的参考图表用于 WBS 患者,因为它常常低估了他们的生长。需要为 WBS 患者制定特定的生长图表。

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