Mulley J C, Gedeon A K, Thorn K A, Bates L J, Sutherland G R
Am J Med Genet. 1987 Jun;27(2):435-48. doi: 10.1002/ajmg.1320270222.
Linkage data using the markers DXS51, F9, DXS15, and DXS52 are presented from 14 pedigrees segregating with the fragile X. Cytogenetic and DNA data were combined by two- or three-point linkage analysis for estimation of lod scores and carrier probabilities in potential carriers. Recombination frequencies (theta) corresponding to maximum z scores (zeta) were obtained for DXS51 (zeta = 3.45, theta = 0.0), DXS15 (zeta = 0.40, theta = 0.06), F9 (zeta = 3.15, theta = 0.09), and DXS52 (zeta = 3.60, theta = 0.11) with the fragile X. Considerable alterations to carrier probabilities occurred in some cases, especially when flanking markers were informative. The chance of mentally impaired offspring was reduced to 1% for five of eight women with prior carrier probabilities of 32%. Three pedigrees were identified in which mutation had possibly occurred. An alternative explanation for two of these was inheritance of the fragile X from normal males and for the other inheritance from a clinically normal woman. Probabilities were computed for each of these alternatives.
本文展示了利用DXS51、F9、DXS15和DXS52标记对14个与脆性X综合征相关的家系进行的连锁分析数据。通过两点或三点连锁分析将细胞遗传学和DNA数据相结合,以估计潜在携带者的lod分数和携带概率。获得了与脆性X综合征相关的DXS51(ζ = 3.45,θ = 0.0)、DXS15(ζ = 0.40,θ = 0.06)、F9(ζ = 3.15,θ = 0.09)和DXS52(ζ = 3.60,θ = 0.11)的重组频率(θ)对应于最大z分数(ζ)。在某些情况下,携带概率发生了相当大的变化,特别是当侧翼标记具有信息性时。对于八位先前携带概率为32%的女性中的五位,其智力受损后代的几率降至1%。鉴定出三个可能发生突变的家系。其中两个家系的另一种解释是脆性X从正常男性遗传而来,另一个家系的解释是从临床正常的女性遗传而来。计算了每种替代情况的概率。