• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

患有典型半乳糖血症的成年人的健康与福祉。

Health and well-being of maturing adults with classic galactosemia.

作者信息

Garrett Olivia S, Druss Jared J, Vos E Naomi, Fu Yu-Ting Debbie, Lucia Stephanie, Greenstein Patricia E, Bauer Anna, Sykut-Cegielska Jolanta, Stepien Karolina M, Arbuckle Cameron, Grafakou Olga, Meyer Uta, Vanhoutvin Nele, Pané Adriana, Bosch Annet M, Rubio-Gozalbo Estela, Berry Gerard T, Fridovich-Keil Judith L

机构信息

Department of Human Genetics, Emory University School of Medicine, Atlanta, Georgia, USA.

MosaKids Children's Hospital, Department of Pediatrics, Maastricht University Medical Center, Maastricht, Netherlands.

出版信息

J Inherit Metab Dis. 2025 Jan;48(1):e12786. doi: 10.1002/jimd.12786. Epub 2024 Aug 14.

DOI:10.1002/jimd.12786
PMID:39143820
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11670443/
Abstract

Long-term outcomes in classic galactosemia (CG) have been studied previously, but all prior studies have relied on cohorts of patients that were small in number, or heavily skewed toward children and young adults, or both. Here, we extend what is known about the health and well-being of maturing adults with CG by analyzing the results of anonymous custom surveys completed by 92 affected individuals, ages 30-78, and 38 unaffected sibling controls, ages 30-79. The median age for patients was 38.5 years and for controls was 41 years. These study participants hailed from 12 different countries predominantly representing Europe and North America. Participants reported on their general life experiences and outcomes in seven different domains including: speech/voice/language, cognition, motor function, cataracts, bone health, psychosocial well-being, and gastrointestinal health. We also queried women about ovarian function. Our results indicated a prevalence of long-term complications across all outcome domains that aligned with levels previously reported in younger cohorts. Given the sample size and age range of participants in this study, these findings strongly suggest that the adverse developmental outcomes commonly linked to CG are not progressive with age for most patients. We also tested four candidate modifiers for possible association with each of the outcomes followed, including: days of neonatal milk exposure, rigor of dietary galactose restriction in early childhood, current age, and home continent. We observed no associations that reached even nominal significance, except for the following: cataracts with neonatal milk exposure (p = 2.347e-04), cataracts with age (p = 0.018), and bone health with home continent (p = 0.03).

摘要

经典型半乳糖血症(CG)的长期预后此前已有研究,但所有既往研究均依赖数量较少的患者队列,或严重偏向儿童和青年,或两者皆有。在此,我们通过分析92名年龄在30 - 78岁的受影响个体以及38名年龄在30 - 79岁的未受影响的同胞对照完成的匿名定制调查问卷结果,扩展了对成年CG患者健康状况的认识。患者的中位年龄为38.5岁,对照的中位年龄为41岁。这些研究参与者来自12个不同国家,主要代表欧洲和北美。参与者报告了他们在七个不同领域的一般生活经历和预后情况,包括:言语/声音/语言、认知、运动功能、白内障、骨骼健康、心理社会幸福感和胃肠道健康。我们还询问了女性的卵巢功能。我们的结果表明,所有预后领域的长期并发症患病率与先前在较年轻队列中报告的水平一致。鉴于本研究中参与者的样本量和年龄范围,这些发现强烈表明,对于大多数患者而言,通常与CG相关的不良发育后果不会随着年龄增长而进展。我们还测试了四个候选修饰因素与后续每个预后结果的可能关联,包括:新生儿接触牛奶的天数、幼儿期饮食中半乳糖限制的严格程度、当前年龄和所在大陆。我们未观察到任何达到甚至名义显著性的关联,除了以下情况:白内障与新生儿接触牛奶(p = 2.347e - 04)、白内障与年龄(p = 0.018)以及骨骼健康与所在大陆(p = 0.03)。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/7cdeebed492e/JIMD-48-0-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/2ffa7ab9b5ef/JIMD-48-0-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/147c437ff2a1/JIMD-48-0-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/6c6e52addb9e/JIMD-48-0-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/d44cabf2dc27/JIMD-48-0-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/127976e8f5ed/JIMD-48-0-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/7cdeebed492e/JIMD-48-0-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/2ffa7ab9b5ef/JIMD-48-0-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/147c437ff2a1/JIMD-48-0-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/6c6e52addb9e/JIMD-48-0-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/d44cabf2dc27/JIMD-48-0-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/127976e8f5ed/JIMD-48-0-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8171/11670443/7cdeebed492e/JIMD-48-0-g006.jpg

相似文献

1
Health and well-being of maturing adults with classic galactosemia.患有典型半乳糖血症的成年人的健康与福祉。
J Inherit Metab Dis. 2025 Jan;48(1):e12786. doi: 10.1002/jimd.12786. Epub 2024 Aug 14.
2
Patterns of Penetrance and Expressivity of Long-Term Outcomes in Classic Galactosemia.经典型半乳糖血症长期预后的外显率和表现度模式
J Inherit Metab Dis. 2025 May;48(3):e70020. doi: 10.1002/jimd.70020.
3
Rigor of non-dairy galactose restriction in early childhood, measured by retrospective survey, does not associate with severity of five long-term outcomes quantified in 231 children and adults with classic galactosemia.通过回顾性调查衡量的婴幼儿时期非乳制品半乳糖限制的严格程度,与 231 名经典半乳糖血症患儿和成人中量化的 5 种长期结局的严重程度无关。
J Inherit Metab Dis. 2017 Nov;40(6):813-821. doi: 10.1007/s10545-017-0067-x. Epub 2017 Jul 10.
4
Long-term complications in classic galactosemia are not progressive.经典半乳糖血症的长期并发症并非进行性的。
Mol Genet Metab. 2023 Nov;140(3):107708. doi: 10.1016/j.ymgme.2023.107708. Epub 2023 Oct 11.
5
Modifiers of ovarian function in girls and women with classic galactosemia.经典半乳糖血症女性和女童卵巢功能的调节剂。
J Clin Endocrinol Metab. 2013 Jul;98(7):E1257-65. doi: 10.1210/jc.2013-1374. Epub 2013 May 20.
6
A multinational study of acute and long-term outcomes of Type 1 galactosemia patients who carry the S135L (c.404C > T) variant of GALT.一项关于携带 GALT 基因 S135L(c.404C>T) 变异的 1 型半乳糖血症患者急性和长期结局的多国研究。
J Inherit Metab Dis. 2022 Nov;45(6):1106-1117. doi: 10.1002/jimd.12556. Epub 2022 Sep 26.
7
Acute and long-term outcomes in a Drosophila melanogaster model of classic galactosemia occur independently of galactose-1-phosphate accumulation.在经典半乳糖血症的果蝇模型中,急性和长期结果的出现与1-磷酸半乳糖的积累无关。
Dis Model Mech. 2016 Nov 1;9(11):1375-1382. doi: 10.1242/dmm.022988. Epub 2016 Aug 24.
8
Transient developmental delays in infants with Duarte-2 variant galactosemia.患有 2 型达尔特变异半乳糖血症婴儿的短暂性发育迟缓。
Mol Genet Metab. 2021 Sep-Oct;134(1-2):132-138. doi: 10.1016/j.ymgme.2021.07.009. Epub 2021 Jul 30.
9
Neonatal GALT gene replacement offers metabolic and phenotypic correction through early adulthood in a rat model of classic galactosemia.新生儿 GALT 基因替换通过经典半乳糖血症大鼠模型在成年早期提供代谢和表型校正。
J Inherit Metab Dis. 2022 Mar;45(2):203-214. doi: 10.1002/jimd.12471. Epub 2022 Jan 14.
10
Hand fine motor control in classic galactosemia.经典半乳糖血症患者手部精细运动控制能力。
J Inherit Metab Dis. 2021 Jul;44(4):871-878. doi: 10.1002/jimd.12376. Epub 2021 Mar 23.

引用本文的文献

1
Patterns of Penetrance and Expressivity of Long-Term Outcomes in Classic Galactosemia.经典型半乳糖血症长期预后的外显率和表现度模式
J Inherit Metab Dis. 2025 May;48(3):e70020. doi: 10.1002/jimd.70020.
2
Galactose Impairs Motor Performance and Cerebellar Signaling in Young Male Wistar Rats.半乳糖损害雄性幼年Wistar大鼠的运动能力和小脑信号传导。
Mol Neurobiol. 2025 Jun;62(6):7542-7556. doi: 10.1007/s12035-024-04684-6. Epub 2025 Feb 6.

本文引用的文献

1
Neuropsychological stability in classical galactosemia: A pilot study in 10 adult patients.经典型半乳糖血症患者的神经心理稳定性:10例成年患者的初步研究
JIMD Rep. 2024 Jan 9;65(2):110-115. doi: 10.1002/jmd2.12410. eCollection 2024 Mar.
2
Brain function in classic galactosemia, a galactosemia network (GalNet) members review.经典型半乳糖血症中的脑功能,半乳糖血症网络(GalNet)成员综述。
Front Genet. 2024 Feb 15;15:1355962. doi: 10.3389/fgene.2024.1355962. eCollection 2024.
3
Long-term complications in classic galactosemia are not progressive.
经典半乳糖血症的长期并发症并非进行性的。
Mol Genet Metab. 2023 Nov;140(3):107708. doi: 10.1016/j.ymgme.2023.107708. Epub 2023 Oct 11.
4
Racial and ethnic diversity of classic and clinical variant galactosemia in the United States.美国经典型和临床变异型半乳糖血症的种族和民族多样性。
Mol Genet Metab. 2023 Apr;138(4):107542. doi: 10.1016/j.ymgme.2023.107542. Epub 2023 Feb 21.
5
The hypergonadotropic hypogonadism conundrum of classic galactosemia.经典半乳糖血症的促性腺激素性性腺功能减退症难题。
Hum Reprod Update. 2023 Mar 1;29(2):246-258. doi: 10.1093/humupd/dmac041.
6
A multinational study of acute and long-term outcomes of Type 1 galactosemia patients who carry the S135L (c.404C > T) variant of GALT.一项关于携带 GALT 基因 S135L(c.404C>T) 变异的 1 型半乳糖血症患者急性和长期结局的多国研究。
J Inherit Metab Dis. 2022 Nov;45(6):1106-1117. doi: 10.1002/jimd.12556. Epub 2022 Sep 26.
7
Gray and white matter are both affected in classical galactosemia: An explorative study on the association between neuroimaging and clinical outcome.经典型半乳糖血症中灰质和白质均受影响:神经影像学与临床结局相关性的探索性研究。
Mol Genet Metab. 2020 Dec;131(4):370-379. doi: 10.1016/j.ymgme.2020.11.001. Epub 2020 Nov 6.
8
Infants with Congenital Disorders Identified Through Newborn Screening - United States, 2015-2017.通过新生儿筛查发现的先天性疾病患儿 - 美国,2015-2017 年。
MMWR Morb Mortal Wkly Rep. 2020 Sep 11;69(36):1265-1268. doi: 10.15585/mmwr.mm6936a6.
9
Identification of neuronal structures and pathways corresponding to clinical functioning in galactosemia.鉴定半乳糖血症中与临床功能相对应的神经元结构和途径。
J Inherit Metab Dis. 2020 Nov;43(6):1205-1218. doi: 10.1002/jimd.12279. Epub 2020 Aug 3.
10
Global and regional prevalence of age-related cataract: a comprehensive systematic review and meta-analysis.全球和地区与年龄相关的白内障患病率:全面的系统评价和荟萃分析。
Eye (Lond). 2020 Aug;34(8):1357-1370. doi: 10.1038/s41433-020-0806-3. Epub 2020 Feb 13.