Laney Dawn A, Clarke Virginia, Foley Allison, Hall Eric W, Gillespie Scott E, Holida Myrl, Simmons Morgan, Wadley Alexandrea
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.
JIMD Rep. 2017;37:85-97. doi: 10.1007/8904_2017_17. Epub 2017 Mar 22.
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.
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。