Lynch J R, Ogilvie D, Priestley L, Baigrie C, Smith R, Farndon P, Sykes B
University of Oxford, Institute of Molecular Medicine, John Radcliffe Hospital, Headington.
J Med Genet. 1991 Mar;28(3):145-50. doi: 10.1136/jmg.28.3.145.
Dominantly inherited osteogenesis imperfecta is consistently linked to the two loci encoding the alpha 1 and alpha 2 subunits of collagen 1, the predominant bone collagen. We have performed several prenatal diagnoses based on identification of the segregating allele at the concordant locus in chorionic villus samples both in families where the linkage can be independently shown and in those where it cannot. Especially in the latter category, calculation of the final risk must incorporate an estimate of genetic heterogeneity within the OI population to give a prior probability of linkage. This figure can then be modified for each family by additional information from concordant meioses.
显性遗传的成骨不全症始终与编码Ⅰ型胶原(主要的骨胶原)α1和α2亚基的两个基因座相关联。我们已经在绒毛膜绒毛样本中通过识别共分离等位基因进行了多次产前诊断,这些样本来自连锁关系可独立显示的家庭以及连锁关系无法独立显示的家庭。特别是在后者这类家庭中,最终风险的计算必须纳入对成骨不全症群体内遗传异质性的估计,以得出连锁的先验概率。然后,这个数字可以根据来自共分离减数分裂的额外信息针对每个家庭进行修正。