Homozygous mutation in SPATA16 is associated with male infertility in human globozoospermia

被引:238
作者
Dam, Anika H. D. M.
Koscinski, Isabelle
Kremer, Jan A. M.
Moutou, Celine
Jaeger, Anne-Sophie
Oudakker, Astrid R.
Tournaye, Herman
Charlet, Nicolas
lagier-Tourenne, Clotilde
van Bokhoven, Hans
Viville, Stephane
机构
[1] Radboud Univ Nijmegen Med Ctr, Ctr Reprod, Dept Obstet & Gynecol, NL-6500 HB Nijmegen, Netherlands
[2] Radboud Univ Nijmegen Med Ctr, Dept Human Genet, NL-6500 HB Nijmegen, Netherlands
[3] Syndicat Inter Hosp Communaute Urbaine Strasbourg, Serv Biol Reprod, Ctr Med Chirurg & Obstet, Schiltigheim, France
[4] Inst Genet & Biol Mol Cellulaire, Dept Dev Biol, Illkirch Graffenstaden, France
[5] Univ Strasbourg, Fac Med, CHU Strasbourg, Strasbourg, France
[6] Dutch Speaking Brussels Free Univ, Ctr Reprod Med, Brussels, Belgium
关键词
D O I
10.1086/521314
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
Globozoospermia is a rare (incidence <0.1% in male infertile patients) form of teratozoospermia, mainly characterized by round-headed spermatozoa that lack an acrosome. It originates from a disturbed spermiogenesis, which is expected to be induced by a genetic factor. Several family cases and recessive mouse models with the same phenotype support this expectation. In this study, we present a consanguineous family with three affected brothers, in whom we have identified a homozygous mutation in the spermatogenesis-specific gene SPATA16. This is the first example of a nonsyndromic male infertility condition in humans caused by an autosomal gene defect, and it could also mean that the identification of other partners like SPATA16 could elucidate acrosome formation.
引用
收藏
页码:813 / 820
页数:8
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