Lumbroso S, Lobaccaro J M, Belon C, Amram S, Bachelard B, Garandeau P, Sultan C
Unité de Biochimie Endocrinienne du Développement et de la Reproduction, Hôpital Lapeyronie, Montpellier, France.
Eur J Endocrinol. 1994 Apr;130(4):327-32. doi: 10.1530/eje.0.1300327.
In a large family with Reifenstein syndrome, we previously performed molecular analysis of the androgen receptor gene. Direct sequencing showed a G-A point mutation at position 2818 of exon 7, which was responsible for an arginine-histidine substitution at position 840 of the androgen receptor. In this family, the proband's mother became pregnant and wished to know whether she was carrying an unaffected fetus. Polymerase chain reactions of the sex-determining region of the Y chromosome (the SRY gene) on trophoblastic DNA at week 14 revealed a 46,XY genotype. Sequencing analysis showed the canonical sequence (CGT, encoding an Arg residue), suggesting that the fetus was not affected. The expectation of normal male sexual development was confirmed by detection of normal male external genitalia through ultrasonography at week 24. These data confirm that sequence analysis of the androgen receptor gene on trophoblastic DNA is the most reliable method for prenatally diagnosing or excluding androgen insensitivity syndrome in high-risk families.
在一个患有赖芬斯坦综合征的大家庭中,我们之前对雄激素受体基因进行了分子分析。直接测序显示外显子7第2818位存在G-A点突变,该突变导致雄激素受体第840位的精氨酸被组氨酸取代。在这个家庭中,先证者的母亲怀孕了,她想知道自己怀的胎儿是否未受影响。在孕14周时,对滋养层DNA上的Y染色体性别决定区域(SRY基因)进行聚合酶链反应,结果显示基因型为46,XY。测序分析显示为典型序列(CGT,编码精氨酸残基),表明胎儿未受影响。在孕24周时通过超声检查发现胎儿有正常的男性外生殖器,这证实了对正常男性性发育的预期。这些数据证实,对滋养层DNA上的雄激素受体基因进行序列分析是在高危家庭中产前诊断或排除雄激素不敏感综合征最可靠的方法。