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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.

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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5
Celiac disease prevalence and predisposing-HLA in a cohort of 93 Williams-Beuren syndrome patients.
Am J Med Genet A. 2023 Jan;191(1):84-89. doi: 10.1002/ajmg.a.62990. Epub 2022 Oct 18.
6
Williams syndrome.
Nat Rev Dis Primers. 2021 Jun 17;7(1):42. doi: 10.1038/s41572-021-00276-z.
7
Anesthetic Considerations for Patients With Williams Syndrome.
J Cardiothorac Vasc Anesth. 2021 Jan;35(1):176-186. doi: 10.1053/j.jvca.2020.01.022. Epub 2020 Jan 21.
8
Health Care Supervision for Children With Williams Syndrome.
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9
Global Prevalence of Celiac Disease: Systematic Review and Meta-analysis.
Clin Gastroenterol Hepatol. 2018 Jun;16(6):823-836.e2. doi: 10.1016/j.cgh.2017.06.037. Epub 2018 Mar 16.
10
Diverticulitis in a child with Williams syndrome: a case report and review of the literature.
J Pediatr Surg. 2012 Sep;47(9):E33-5. doi: 10.1016/j.jpedsurg.2012.05.036.

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