Maher E R, Bentley E, Yates J R, Latif F, Lerman M, Zbar B, Affara N A, Ferguson-Smith M A
Department of Pathology, University of Cambridge, United Kingdom.
Genomics. 1991 Aug;10(4):957-60. doi: 10.1016/0888-7543(91)90185-h.
Genetic linkage studies were performed in 22 families with von Hippel-Lindau (VHL) disease by using polymorphic DNA markers from distal chromosome 3p. Linkage was detected between VHL disease and the markers D3S18 (Zmax = 6.6 at theta = 0.0, confidence interval (CI) 0.00-0.06), RAF1 (Zmax = 5.9 at theta = 0.06, CI 0.01-0.16), and THRB (Zmax 3.4 at theta = 0.11). Multipoint linkage analysis localized the VHL disease gene within a small region (approximately 8 cM) of 3p25-p26 between RAF1 and (D3S191, D3S225) and close to the D3S18 locus. There was no evidence of locus heterogeneity, and families with and without pheochromocytoma showed linkage to D3S18. The identification of DNA markers flanking the VHL disease gene allows reliable presymptomatic and prenatal diagnosis to be offered to informative families.
利用来自3号染色体远端的多态性DNA标记,对22个患有希佩尔-林道(VHL)病的家族进行了遗传连锁研究。在VHL病与标记D3S18(在θ=0.0时Zmax = 6.6,置信区间(CI)0.00 - 0.06)、RAF1(在θ=0.06时Zmax = 5.9,CI 0.01 - 0.16)和THRB(在θ=0.11时Zmax = 3.4)之间检测到连锁关系。多点连锁分析将VHL病基因定位在3p25 - p26的一个小区域(约8厘摩)内,该区域位于RAF1与(D3S191、D3S225)之间且靠近D3S18位点。没有证据表明存在位点异质性,有和没有嗜铬细胞瘤的家族均显示与D3S18连锁。确定VHL病基因两侧的DNA标记,使得可以向有信息价值的家族提供可靠的症状前和产前诊断。