• 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

黏多糖贮积症 I 型患者未经治疗和治疗期间的生长情况:来自 MPS I 登记处的数据。

Growth in individuals with attenuated mucopolysaccharidosis type I during untreated and treated periods: Data from the MPS I registry.

机构信息

The Lundquist Institute at Harbor-UCLA Medical Center, Torrance, California, USA.

Hospital Nacional de Pediatría J. P. Garrahan, Ciudad Autónoma de Buenos Aires, Buenos Aires, Argentina.

出版信息

Am J Med Genet A. 2022 Oct;188(10):2941-2951. doi: 10.1002/ajmg.a.62910. Epub 2022 Jul 23.

DOI:10.1002/ajmg.a.62910
PMID:35869927
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9545955/
Abstract

Mucopolysaccharidosis Type I (MPS I) is caused by deficiency of α-L-iduronidase. Short stature and growth deceleration are common in individuals with the attenuated MPS I phenotype. Study objectives were to assess growth in individuals with attenuated MPS I enrolled in The MPS I Registry while untreated and after initiation of enzyme replacement therapy (ERT) with laronidase (recombinant human iduronidase). Individuals in the MPS I Registry with at least one observation for height and assigned attenuated MPS I phenotype as of September 2020 were included. The cohort included 142 males and 153 females 2-18 years of age. Age and sex adjusted standardized height-for-age z-scores during the natural history and ERT-treatment periods were assessed using linear mixed model repeated measures analyses. Growth curves were estimated during both periods and compared to standard growth charts from the Center for Disease Control (CDC). There was a significantly slower decline in height z-scores with age during the ERT-treated period compared to the natural history period. Estimated average height z-scores in the ERT-treatment versus the natural history period at age 10 were -2.4 versus -3.3 in females and -1.4 versus -2.9 in males (females first treated 3 year; males <4.1 year). While median height remained below CDC standards during both the natural history and ERT-treated periods for individuals with attenuated MPS I, laronidase ERT was associated with slower declines in height z-scores.

摘要

黏多糖贮积症 I 型(MPS I)是由α-L-艾杜糖苷酸酶缺乏引起的。在表型减弱的 MPS I 个体中,身材矮小和生长减速是常见的。研究目的是评估在未经治疗和开始使用拉罗尼酶(重组人艾杜糖苷酸酶)进行酶替代治疗(ERT)的情况下,入组 MPS I 登记处的表型减弱的 MPS I 个体的生长情况。截至 2020 年 9 月,MPS I 登记处至少有一次身高观察结果且被分配为表型减弱的 MPS I 的个体被纳入研究。该队列包括 142 名男性和 153 名女性,年龄为 2-18 岁。使用线性混合模型重复测量分析评估自然史和 ERT 治疗期间的年龄和性别调整标准化身高年龄 z 评分。在两个时期都估计了生长曲线,并与疾病控制与预防中心(CDC)的标准生长图表进行了比较。与自然史期间相比,ERT 治疗期间身高 z 评分随年龄的下降速度明显较慢。ERT 治疗与自然史期间 10 岁时女性的平均身高 z 评分分别为-2.4 和-3.3,男性分别为-1.4 和-2.9(女性首次治疗 3 岁;男性<4.1 岁)。虽然在自然史和 ERT 治疗期间,表型减弱的 MPS I 个体的中位数身高仍低于 CDC 标准,但拉罗尼酶 ERT 与身高 z 评分下降速度较慢相关。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b00/9545955/634501bb100c/AJMG-188-2941-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b00/9545955/7980ac5c2c6d/AJMG-188-2941-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b00/9545955/2262da28ca3d/AJMG-188-2941-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b00/9545955/634501bb100c/AJMG-188-2941-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b00/9545955/7980ac5c2c6d/AJMG-188-2941-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b00/9545955/2262da28ca3d/AJMG-188-2941-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6b00/9545955/634501bb100c/AJMG-188-2941-g003.jpg

相似文献

1
Growth in individuals with attenuated mucopolysaccharidosis type I during untreated and treated periods: Data from the MPS I registry.黏多糖贮积症 I 型患者未经治疗和治疗期间的生长情况:来自 MPS I 登记处的数据。
Am J Med Genet A. 2022 Oct;188(10):2941-2951. doi: 10.1002/ajmg.a.62910. Epub 2022 Jul 23.
2
Anthropometric data of 14 patients with mucopolysaccharidosis I: retrospective analysis and efficacy of recombinant human alpha-L-iduronidase (laronidase).黏多糖贮积症 I 型 14 例患者的人体测量学数据:回顾性分析及重组人α-L-艾杜糖苷酸酶(拉罗尼酶)的疗效。
Mol Genet Metab. 2010 Jan;99(1):10-7. doi: 10.1016/j.ymgme.2009.08.008.
3
Growth patterns for untreated individuals with MPS I: Report from the international MPS I registry.未经治疗的 MPS I 个体的生长模式:来自国际 MPS I 注册中心的报告。
Am J Med Genet A. 2019 Dec;179(12):2425-2432. doi: 10.1002/ajmg.a.61378. Epub 2019 Oct 22.
4
Early treatment with laronidase improves clinical outcomes in patients with attenuated MPS I: a retrospective case series analysis of nine sibships.早期使用拉罗尼酶治疗可改善轻型黏多糖贮积症I型患者的临床结局:对9个同胞家族的回顾性病例系列分析
Orphanet J Rare Dis. 2015 Oct 7;10:131. doi: 10.1186/s13023-015-0344-4.
5
18-year follow-up of enzyme-replacement therapy in two siblings with attenuated mucopolysaccharidosis I.对两名患有轻度黏多糖贮积症 I 型的同胞进行酶替代疗法的 18 年随访。
Am J Med Genet A. 2023 Feb;191(2):564-569. doi: 10.1002/ajmg.a.63029. Epub 2022 Nov 5.
6
Effect of recombinant human growth hormone on changes in height, bone mineral density, and body composition over 1-2 years in children with Hurler or Hunter syndrome.重组人生长激素对 Hurler 或 Hunter 综合征患儿 1-2 年内身高、骨密度和身体成分变化的影响。
Mol Genet Metab. 2014 Feb;111(2):101-6. doi: 10.1016/j.ymgme.2013.11.013. Epub 2013 Dec 11.
7
Effects of neonatal enzyme replacement therapy and simvastatin treatment on cervical spine disease in mucopolysaccharidosis I dogs.新生犬酶替代疗法和辛伐他汀治疗对黏多糖贮积症I型犬颈椎疾病的影响。
J Bone Miner Res. 2014 Dec;29(12):2610-7. doi: 10.1002/jbmr.2290.
8
Progressive eye pathology in mucopolysaccharidosis type I mice and effects of enzyme replacement therapy.黏多糖贮积症Ⅰ型小鼠的进行性眼部病变及酶替代治疗的效果。
Clin Exp Ophthalmol. 2020 Apr;48(3):334-342. doi: 10.1111/ceo.13713. Epub 2020 Jan 30.
9
Laronidase.拉罗尼酶
BioDrugs. 2002;16(4):316-8. doi: 10.2165/00063030-200216040-00009.
10
Changes in hair morphology of mucopolysaccharidosis I patients treated with recombinant human alpha-L-iduronidase (laronidase, Aldurazyme).接受重组人α-L-艾杜糖醛酸酶(拉罗尼酶,Aldurazyme)治疗的黏多糖贮积症I型患者毛发形态的变化
Am J Med Genet A. 2005 Dec 15;139(3):199-203. doi: 10.1002/ajmg.a.31021.

引用本文的文献

1
Mucopolysaccharidosis: What Pediatric Rheumatologists and Orthopedics Need to Know.黏多糖贮积症:儿科风湿病学家和骨科医生需要了解的内容。
Diagnostics (Basel). 2022 Dec 27;13(1):75. doi: 10.3390/diagnostics13010075.

本文引用的文献

1
MPS I: Early diagnosis, bone disease and treatment, where are we now?MPS I:早期诊断、骨骼疾病和治疗,我们现在在哪里?
J Inherit Metab Dis. 2021 Nov;44(6):1289-1310. doi: 10.1002/jimd.12431. Epub 2021 Sep 15.
2
Failures of Endochondral Ossification in the Mucopolysaccharidoses.黏多糖贮积症中的软骨内骨化失败。
Curr Osteoporos Rep. 2020 Dec;18(6):759-773. doi: 10.1007/s11914-020-00626-y. Epub 2020 Oct 16.
3
Growth patterns for untreated individuals with MPS I: Report from the international MPS I registry.未经治疗的 MPS I 个体的生长模式:来自国际 MPS I 注册中心的报告。
Am J Med Genet A. 2019 Dec;179(12):2425-2432. doi: 10.1002/ajmg.a.61378. Epub 2019 Oct 22.
4
Growth impairment and limited range of joint motion in children should raise suspicion of an attenuated form of mucopolysaccharidosis: expert opinion.儿童生长障碍和关节运动范围受限应引起对黏多糖贮积症衰减形式的怀疑:专家意见。
Eur J Pediatr. 2019 Apr;178(4):593-603. doi: 10.1007/s00431-019-03330-x. Epub 2019 Feb 11.
5
Growth impairment in mucopolysaccharidoses.黏多糖贮积症中的生长障碍。
Mol Genet Metab. 2018 May;124(1):1-10. doi: 10.1016/j.ymgme.2018.03.004. Epub 2018 Mar 16.
6
The effect of galsulfase enzyme replacement therapy on the growth of patients with mucopolysaccharidosis VI (Maroteaux-Lamy syndrome).瓜尔硫酸酯酶替代治疗对黏多糖贮积症 VI 型(马罗托劳里综合征)患者生长的影响。
Mol Genet Metab. 2017 Sep;122(1-2):107-112. doi: 10.1016/j.ymgme.2017.03.008. Epub 2017 Mar 31.
7
Relative frequency and estimated minimal frequency of Lysosomal Storage Diseases in Brazil: Report from a Reference Laboratory.巴西溶酶体贮积症的相对频率和估计最小频率:来自一家参考实验室的报告。
Genet Mol Biol. 2017 Jan-Mar;40(1):31-39. doi: 10.1590/1678-4685-GMB-2016-0268. Epub 2017 Mar 16.
8
Outcomes of Long-Term Treatment with Laronidase in Patients with Mucopolysaccharidosis Type I.I 型黏多糖贮积症患者用拉罗尼酶进行长期治疗的结果。
J Pediatr. 2016 Nov;178:219-226.e1. doi: 10.1016/j.jpeds.2016.08.033.
9
12 year follow up of enzyme-replacement therapy in two siblings with attenuated mucopolysaccharidosis I: the important role of early treatment.对两名患有轻型黏多糖贮积症 I 型的同胞进行酶替代疗法的 12 年随访:早期治疗的重要作用。
BMC Med Genet. 2016 Mar 10;17:19. doi: 10.1186/s12881-016-0284-4.
10
Early treatment with laronidase improves clinical outcomes in patients with attenuated MPS I: a retrospective case series analysis of nine sibships.早期使用拉罗尼酶治疗可改善轻型黏多糖贮积症I型患者的临床结局:对9个同胞家族的回顾性病例系列分析
Orphanet J Rare Dis. 2015 Oct 7;10:131. doi: 10.1186/s13023-015-0344-4.