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The Impact of Fabry Disease on Reproductive Fitness.法布里病对生殖健康的影响。
JIMD Rep. 2017;37:85-97. doi: 10.1007/8904_2017_17. Epub 2017 Mar 22.
2
Diagnosing lysosomal storage disorders: Fabry disease.溶酶体贮积症的诊断:法布里病
Curr Protoc Hum Genet. 2013;Chapter 17:Unit17.13. doi: 10.1002/0471142905.hg1713s77.
3
Fabry Disease: prevalence of affected males and heterozygotes with pathogenic mutations identified by screening renal, cardiac and stroke clinics, 1995-2017.法布里病:1995 年至 2017 年通过对肾脏、心脏和中风诊所进行筛查,确定了受影响男性和携带致病性突变的杂合子的患病率。
J Med Genet. 2018 Apr;55(4):261-268. doi: 10.1136/jmedgenet-2017-105080. Epub 2018 Jan 12.
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In Patients with an α-Galactosidase A Variant, Small Nerve Fibre Assessment Cannot Confirm a Diagnosis of Fabry Disease.在患有α-半乳糖苷酶A变异体的患者中,小神经纤维评估无法确诊法布里病。
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Genetic Screening of Mutations Associated with Fabry Disease in a Nationwide Cohort of Juvenile Idiopathic Arthritis Patients.在全国范围内的青少年特发性关节炎患者队列中对与法布里病相关的突变进行基因筛查。
Front Med (Lausanne). 2017 Mar 1;4:12. doi: 10.3389/fmed.2017.00012. eCollection 2017.
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Absence of α-galactosidase cross-correction in Fabry heterozygote cultured skin fibroblasts.法布里病杂合子培养的皮肤成纤维细胞中缺乏α-半乳糖苷酶交叉校正。
Mol Genet Metab. 2015 Feb;114(2):268-73. doi: 10.1016/j.ymgme.2014.11.005. Epub 2014 Nov 12.
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Quality of life of patients with Fabry disease.法布里病患者的生活质量。
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Fabry disease: polymorphic haplotypes and a novel missense mutation in the GLA gene.法布里病:GLA 基因的多态单体型和一种新的错义突变。
Clin Genet. 2012 Mar;81(3):224-33. doi: 10.1111/j.1399-0004.2011.01689.x. Epub 2011 May 25.
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Impact of Nontreatment Duration and Keratopathy on Major Adverse Cardiovascular Events in Fabry Disease: A Nationwide Cohort Study.非治疗持续时间和角膜病变对法布里病主要不良心血管事件的影响:一项全国性队列研究
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引用本文的文献

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Fabry disease in females: organ involvement and clinical outcomes compared with the general population (103/150 characters).女性法布里病:与普通人群相比的器官受累情况及临床结局(103/150字符)
Orphanet J Rare Dis. 2025 Aug 13;20(1):433. doi: 10.1186/s13023-025-03922-x.
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Infertility in Fabry's Disease: role of hypoxia and inflammation in determining testicular damage.法布里病的不孕:缺氧和炎症在决定睾丸损伤中的作用。
Front Endocrinol (Lausanne). 2024 Feb 22;15:1340188. doi: 10.3389/fendo.2024.1340188. eCollection 2024.
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Fabry Disease in Women: Genetic Basis, Available Biomarkers, and Clinical Manifestations.女性法布里病:遗传基础、可用生物标志物和临床表现。
Genes (Basel). 2023 Dec 26;15(1):37. doi: 10.3390/genes15010037.
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X Chromosome Inactivation in Carriers of Fabry Disease: Review and Meta-Analysis.X 染色体失活在法布里病携带者中的作用:综述和荟萃分析。
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Semen and male genital tract characteristics of patients with Fabry disease: the FERTIFABRY multicentre observational study.法布里病患者的精液及男性生殖道特征:FERTIFABRY多中心观察性研究
Basic Clin Androl. 2019 May 15;29:7. doi: 10.1186/s12610-019-0088-4. eCollection 2019.

本文引用的文献

1
The management and treatment of children with Fabry disease: A United States-based perspective.法布里病患儿的管理与治疗:基于美国的视角
Mol Genet Metab. 2016 Feb;117(2):104-13. doi: 10.1016/j.ymgme.2015.10.007. Epub 2015 Oct 23.
2
Quality of life in patients with Fabry disease: a systematic review of the literature.法布里病患者的生活质量:文献系统评价
Orphanet J Rare Dis. 2015 Jun 16;10:77. doi: 10.1186/s13023-015-0296-8.
3
Ten-year outcome of enzyme replacement therapy with agalsidase beta in patients with Fabry disease.法布里病患者使用β-半乳糖苷酶进行酶替代治疗的十年结果。
J Med Genet. 2015 May;52(5):353-8. doi: 10.1136/jmedgenet-2014-102797. Epub 2015 Mar 20.
4
Plasma globotriaosylsphingosine in relation to phenotypes of Fabry disease.与法布里病表型相关的血浆球三糖神经酰胺
J Med Genet. 2015 Apr;52(4):262-8. doi: 10.1136/jmedgenet-2014-102872. Epub 2015 Jan 16.
5
Lysosomal storage disorder screening implementation: findings from the first six months of full population pilot testing in Missouri.溶酶体贮积症筛查实施情况:密苏里州全面人群试点测试头六个月的发现。
J Pediatr. 2015 Jan;166(1):172-7. doi: 10.1016/j.jpeds.2014.09.023. Epub 2014 Oct 18.
6
Kidney function as an underestimated factor for reduced health related quality of life in patients with Fabry disease.肾功能是法布里病患者健康相关生活质量下降的一个被低估的因素。
BMC Nephrol. 2014 Nov 29;15:188. doi: 10.1186/1471-2369-15-188.
7
Fabry disease in infancy and early childhood: a systematic literature review.婴儿期和幼儿期的法布里病:系统文献回顾。
Genet Med. 2015 May;17(5):323-30. doi: 10.1038/gim.2014.120. Epub 2014 Sep 18.
8
Infertility and impaired fecundity in the United States, 1982-2010: data from the National Survey of Family Growth.1982 - 2010年美国的不孕不育与生育力受损:来自全国家庭成长调查的数据
Natl Health Stat Report. 2013 Aug 14(67):1-18, 1 p following 19.
9
Abortion incidence and service availability in the United States, 2011.美国 2011 年的堕胎发生率和服务提供情况。
Perspect Sex Reprod Health. 2014 Mar;46(1):3-14. doi: 10.1363/46e0414. Epub 2014 Feb 3.
10
Infertility service use in the United States: data from the National Survey of Family Growth, 1982-2010.美国的不孕不育服务利用情况:来自1982 - 2010年全国家庭成长调查的数据
Natl Health Stat Report. 2014 Jan 22(73):1-21.

法布里病对生殖健康的影响。

The Impact of Fabry Disease on Reproductive Fitness.

作者信息

Laney Dawn A, Clarke Virginia, Foley Allison, Hall Eric W, Gillespie Scott E, Holida Myrl, Simmons Morgan, Wadley Alexandrea

机构信息

Division of Medical Genetics, Department of Human Genetics, Emory University School of Medicine, 2165 North Decatur Road, Decatur, GA, 30033, USA.

Neurology Department, Massachusetts General Hospital, Boston, MA, USA.

出版信息

JIMD Rep. 2017;37:85-97. doi: 10.1007/8904_2017_17. Epub 2017 Mar 22.

DOI:10.1007/8904_2017_17
PMID:28324326
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5740039/
Abstract

Fabry disease (FD) is a pan-ethnic, X-linked, progressive lysosomal storage disorder caused by pathogenic mutations in the GLA gene. Published case reports and abstracts suggest that decreased reproductive fitness may occur in males with FD. In order to understand the impact of FD on reproductive fitness and increase the accuracy of reproductive genetic counseling, this study examines a large, multi-centered population of individuals with FD to determine if males have reduced reproductive fitness. Study data were collected on 376 patients through two, gender-specific surveys distributed across the United States and Canada. The number of biological live-born children among individuals with FD was compared to statistics from the general population. Information was also collected on reduced sperm count, depression, pain, use of assisted reproductive technology, and reproductive choice. On average, females affected by FD had more biological live-born children (1.8) than males affected by FD (1.1). However, males affected by FD had an increased mean number of biological children (1.1) compared to the mean number of biological children fathered by men in the United States (0.9). Sixteen of the 134 males with FD reported oligospermia, which suggests that an infertility work up may be indicated for males having difficulty impregnating their partners. In our large multicenter sample, males and females with FD do not exhibit reduced reproductive fitness; on average they have more biological children than the general population in the United States. This information should assist clinicians in providing accurate reproductive genetic counseling and treatment for individuals with FD.

摘要

法布里病(FD)是一种由GLA基因的致病性突变引起的全民族、X连锁、进行性溶酶体贮积症。已发表的病例报告和摘要表明,患有FD的男性可能出现生殖适应性下降。为了了解FD对生殖适应性的影响并提高生殖遗传咨询的准确性,本研究调查了一大群来自多个中心的FD患者,以确定男性的生殖适应性是否降低。通过在美国和加拿大各地开展的两项针对性别的调查,收集了376名患者的研究数据。将FD患者的亲生存活子女数量与普通人群的统计数据进行了比较。还收集了关于精子数量减少、抑郁、疼痛、辅助生殖技术的使用以及生殖选择等方面的信息。平均而言,受FD影响的女性所生的亲生存活子女(1.8个)比受FD影响的男性(1.1个)更多。然而,与美国男性亲生子女的平均数量(0.9个)相比,受FD影响的男性亲生子女的平均数量有所增加(1.1个)。134名患有FD的男性中有16名报告了少精子症,这表明对于难以使伴侣受孕的男性,可能需要进行不育检查。在我们这个大型多中心样本中,患有FD的男性和女性并未表现出生殖适应性降低;平均而言,他们的亲生子女数量比美国普通人群更多。这些信息应有助于临床医生为FD患者提供准确的生殖遗传咨询和治疗。