Bogatcheva Natalia V, Truong Anne, Feng Shu, Engel Wolfgang, Adham Ibrahim M, Agoulnik Alexander I
Department of Obstetrics and Gynecology, 6550 Fannin Street, Baylor College of Medicine, Houston, Texas 77030, USA.
Mol Endocrinol. 2003 Dec;17(12):2639-46. doi: 10.1210/me.2003-0096. Epub 2003 Aug 21.
During male development testes descend from their embryonic intraabdominal position into the scrotum. Two genes, encoding the insulin-like 3 peptide (INSL3) and the GREAT/LGR8 G protein-coupled receptor, control the differentiation of gubernaculum, the caudal genitoinguinal ligament critical for testicular descent. It was established that the INSL3 peptide activates GREAT/LGR8 receptor in vitro. Mutations of Insl3 or Great cause cryptorchidism (undescended testes) in mice. Overexpression of the transgenic Insl3 causes male-like gubernaculum differentiation, ovarian descent into lower abdominal position, and reduced fertility in females. To address the question whether Great deletion complements the mutant female phenotype caused by the Insl3 overexpression, we have produced Insl3 transgenic mice deficient for Great. Such females had a wild-type phenotype, demonstrating that Great was the only cognate receptor for Insl3 in vivo. We have established that pancreatic HIT cells, transfected with the INSL3 cDNA, produce functionally active peptide. Analysis of five INSL3 mutant variants detected in cryptorchid patients showed that P49S substitution renders functionally compromised peptide. Therefore, mutations in INSL3 might contribute to the etiology of cryptorchidism. We have also showed that synthetic insulin-like peptides (INSL4 and INSL6) were unable to activate LGR7 or GREAT/LGR8.
在雄性发育过程中,睾丸从胚胎期的腹腔内位置降至阴囊。两个基因,即编码胰岛素样3肽(INSL3)和GREAT/LGR8 G蛋白偶联受体的基因,控制着引带的分化,引带是对睾丸下降至关重要的尾侧生殖腹股沟韧带。已证实INSL3肽在体外可激活GREAT/LGR8受体。Insl3或Great的突变会导致小鼠隐睾(睾丸未降)。转基因Insl3的过表达会导致雄性样引带分化、卵巢降至下腹部位置以及雌性生育力降低。为了解决Great缺失是否能弥补由Insl3过表达引起的突变雌性表型这一问题,我们培育了缺乏Great的Insl3转基因小鼠。此类雌性具有野生型表型,表明Great是Insl3在体内唯一的同源受体。我们已证实,用INSL3 cDNA转染的胰腺HIT细胞可产生具有功能活性的肽。对在隐睾患者中检测到的5种INSL3突变变体的分析表明,P49S替代使肽的功能受损。因此,INSL3中的突变可能导致隐睾症的病因。我们还表明,合成胰岛素样肽(INSL4和INSL6)无法激活LGR7或GREAT/LGR8。