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Williams 综合征的胃肠道表现:成人和儿科队列的前瞻性分析。

Gastrointestinal manifestations in Williams syndrome: A prospective analysis of an adult and pediatric cohort.

机构信息

Department of Pediatrics, Walter Reed Army Medical Center, Bethesda, Maryland, USA.

Tripler Army Military Medical Center, Honolulu, HI, USA.

出版信息

Am J Med Genet A. 2024 Dec;194(12):e63827. doi: 10.1002/ajmg.a.63827. Epub 2024 Jul 29.

DOI:10.1002/ajmg.a.63827
PMID:39073239
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11540717/
Abstract

Williams syndrome (WS) is a multi-system condition caused by the deletion of 25-27 coding genes on human chromosome 7. Irritability, gastrointestinal (GI) reflux and slow growth are commonly reported in infants with WS, but less data exist regarding GI concerns in older children and adults with the condition. This study evaluates 62 individuals with WS (31 children aged 3-17, and 31 adults aged 18-62) as well as 36 pediatric and adult controls to assess current and historical rates of common GI symptoms. Data were evaluated using a regression model including age, sex, self-reported race, and diagnosis. Symptoms including food intolerance, reflux, dysphagia, choking/gagging, vomiting, constipation, bloating, diarrhea, hematochezia, rectal prolapse, abdominal pain, and weight loss are more common in those with WS relative to controls. In addition, people with WS utilize more GI medications, specialty care, procedures, and supplemental feeds. Among those with WS, symptoms were present at similar rates in children and adults, except for diverticular disease, which was not noted until adulthood. GI symptoms are frequent in people with WS and serve as a significant source of morbidity.

摘要

威廉姆斯综合征(WS)是一种由人类 7 号染色体上 25-27 个编码基因缺失引起的多系统疾病。易激惹、胃肠道(GI)反流和生长缓慢在患有 WS 的婴儿中较为常见,但关于患有该病的大龄儿童和成人的 GI 问题的数据较少。本研究评估了 62 名 WS 患者(31 名 3-17 岁的儿童和 31 名 18-62 岁的成年人)以及 36 名儿科和成人对照组,以评估常见 GI 症状的当前和既往发生率。数据使用包括年龄、性别、自我报告种族和诊断在内的回归模型进行评估。与对照组相比,WS 患者更容易出现食物不耐受、反流、吞咽困难、呛咳、呕吐、便秘、腹胀、腹泻、血便、直肠脱垂、腹痛和体重减轻等症状。此外,WS 患者使用更多的 GI 药物、专科治疗、手术和补充喂养。在 WS 患者中,除了成年后才会出现的憩室病外,儿童和成人的症状发生率相似。GI 症状在 WS 患者中较为常见,是发病率的重要来源。

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Biomaterials. 2023 Dec;303:122405. doi: 10.1016/j.biomaterials.2023.122405. Epub 2023 Nov 17.
2
Loss of Baz1b in mice causes perinatal lethality, growth failure, and variable multi-system outcomes.在小鼠中缺失 Baz1b 会导致围产期致死、生长发育迟缓,并伴有多种系统的可变结局。
Dev Biol. 2024 Jan;505:42-57. doi: 10.1016/j.ydbio.2023.09.007. Epub 2023 Oct 11.
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Body mass index variation in adults with Williams syndrome: associations with predicted dietary intake and food behaviors.
屈曲形态发生过程中胚胎小肠的材料特性
bioRxiv. 2024 Aug 9:2024.08.07.606927. doi: 10.1101/2024.08.07.606927.
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Food Nutr Res. 2023 Jun 23;67. doi: 10.29219/fnr.v67.9321. eCollection 2023.
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Analysis of gut microbiota in patients with Williams-Beuren Syndrome reveals dysbiosis linked to clinical manifestations.分析威廉姆斯-贝伦综合征患者的肠道微生物群,揭示与临床表现相关的失调。
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