Bateman J B, Kojis T L, Cantor R M, Heinzmann C, Ngo J T, Spence M A, Inana G, Kivlin J D, Curtis D, Sparkes R S
Vision Genetics Center, Jules Stein Eye Institute, UCLA School of Medicine.
Trans Am Ophthalmol Soc. 1993;91:299-307; discussion 307-8.
Norrie disease is a rare disease of newborn males caused by prenatal or perinatal retinal detachment, which may be associated with mental retardation, psychosis, and/or hearing loss. DXS7 (L1.28) and MAO A and B loci have been linked to the ND locus on the short arm of the X chromosome. Sequences homologous to OAT also have been mapped to the short arm of the X chromosome. We performed linkage analyses between the ND locus and one of the OAT-like clusters of sequences on the X chromosome (OATL1), using a ScaI RFLP in a ND family, and increased the previously calculated lod score (z) to over 3 (3.38; theta = 0.05). Similarly, we calculated a lod score of 4.06 (theta = 0.01) between the OATL1 and DXS7 loci. Alone, the OATL1 ScaI RFLP system is expected to be informative in 48% of females. If this system were used in combination with the DXS7 TaqI polymorphism, 71% of females would be informative for at least one of the markers and 21% would be informative for both. Because the OATL1 ScaI RFLP is a relatively common polymorphism, this system should be useful for the identification of ND carriers and affected male fetuses and newborns.
诺里病是一种罕见的新生儿男性疾病,由产前或围产期视网膜脱离引起,可能与智力迟钝、精神病和/或听力丧失有关。DXS7(L1.28)以及单胺氧化酶A和B基因座已与X染色体短臂上的诺里病基因座相关联。与鸟氨酸氨基转移酶(OAT)同源的序列也已定位到X染色体短臂上。我们在一个诺里病家族中使用ScaI限制性片段长度多态性(RFLP),对诺里病基因座与X染色体上一个类鸟氨酸氨基转移酶序列簇(OATL1)进行连锁分析,将先前计算的对数优势分数(z)提高到超过3(3.38;θ=0.05)。同样,我们计算出OATL1与DXS7基因座之间的对数优势分数为4.06(θ=0.01)。单独来看,预计OATL1 ScaI RFLP系统在48%的女性中具有信息价值。如果该系统与DXS7 TaqI多态性联合使用,71%的女性至少对其中一个标记具有信息价值,21%的女性对两个标记都具有信息价值。由于OATL1 ScaI RFLP是一种相对常见的多态性,该系统应有助于识别诺里病携带者以及受影响的男性胎儿和新生儿。