Michaelides Katerina, Tuddenham Edward G D, Turner Cathy, Lavender Ben, Lavery Stuart A
Haemostasis and Thrombosis, MRC Clinical Sciences Centre, Faculty of Medicine, Imperial College, Hammersmith Hospital Campus, Du Cane Road, London W12 0NN, UK.
Thromb Haemost. 2006 Feb;95(2):373-9. doi: 10.1160/TH05-08-0574.
Haemophilia A is an X-linked, recessive, inherited bleeding disorder which affects 1 in 5000 males born worldwide. It is caused by mutations in the FactorVIII (F8) gene on chromosome Xq28. We describe for the first time two mutation specific, single cell protocols for pre-implantation genetic diagnosis (PGD) of haemophilia. A that enable the selection of both male and female unaffected embryos. This approach offers an alternative to sexing, frequently used for X-linked disorders, that results in the discarding of all male embryos including the 50% that would have been normal. Two families with a history of severe haemophilia. A requested carrier diagnosis and subsequently proceeded to PGD. The mutation in family 1 is a single nucleotide substitution c.5953C > T, R1966X in exon 18 and in family 2, c.5122C > T, R1689C in exon 14 of the F8 gene. Amplification efficiency was compared between distilled water and SDS/proteinase K cell lysis (98.0%, 96/98 and 80%, 112/140 respectively) using 238 single lymphocytes. Blastomeres from spare IVF cleavage-stage embryos donated for research showed amplification efficiencies of 83.3% (45/54) for the R1966X and 92.9% (13/14) for the R1689C mutations. The rate of allele dropout (ADO) on heterozygous lymphocytes was 1.1% (1/93) for R1966X and 5.94% (6/101) for R1689C mutations. A single PGD treatment cycle for family 1 resulted in two embryos for transfer but these failed to implant. However, with family 2, two embryos were transferred to the uterus on day 4 resulting in a successful singleton pregnancy and subsequent live birth of a normal non-carrier female.
甲型血友病是一种X连锁隐性遗传性出血性疾病,全球每5000名出生男性中就有1人受其影响。它由位于X染色体q28上的凝血因子VIII(F8)基因突变引起。我们首次描述了两种针对血友病的植入前基因诊断(PGD)的突变特异性单细胞方案。这两种方案能够筛选出未受影响的男性和女性胚胎。这种方法为常用于X连锁疾病的性别筛选提供了一种替代方案,性别筛选会导致所有男性胚胎被丢弃,其中包括50%本应正常的胚胎。两个有严重血友病家族史的家庭。一个家庭要求进行携带者诊断,随后进行了植入前基因诊断。家族1中的突变是外显子18中的单核苷酸替换c.5953C>T,R1966X;家族2中的突变是F8基因外显子14中的c.5122C>T,R1689C。使用238个单个淋巴细胞比较了蒸馏水和SDS/蛋白酶K细胞裂解液的扩增效率(分别为98.0%,96/98和80%,112/140)。用于研究的体外受精(IVF)分裂期备用胚胎的卵裂球对R1966X突变的扩增效率为83.3%(45/54),对R1689C突变的扩增效率为92.9%(13/14)。杂合淋巴细胞上等位基因脱失(ADO)率,R1966X突变为1.1%(1/93),R1689C突变为5.94%(6/101)。家族1的一个植入前基因诊断治疗周期产生了两个可供移植的胚胎,但这些胚胎未能着床。然而,对于家族2,在第4天将两个胚胎移植到子宫内,成功实现单胎妊娠,随后一名正常的非携带者女性顺利出生。