Musarella M A, Burghes A, Anson-Cartwright L, Mahtani M M, Argonza R, Tsui L C, Worton R
Department of Ophthalmology, Hospital for Sick Children, Toronto, Ontario, Canada.
Am J Hum Genet. 1988 Oct;43(4):484-94.
The X-linked recessive type of retinitis pigmentosa (XLRP) causes progressive night blindness, visual field constriction, and eventual blindness in affected males by the third or fourth decade of life. The biochemical basis of the disease is unknown, and prenatal diagnosis and definitive carrier diagnosis remain elusive. Heterogeneity in XLRP has been suggested by linkage studies of families affected with XLRP and by phenotypic differences observed in female carriers. Localization of XLRP near Xp11.3 has been suggested by close linkage to an RFLP at the locus DXS7 (Xp11.3) detected by probe L1.28. In other studies a locus for XLRP with metallic sheen has been linked to the ornithine transcarbamylase (OTC) locus mapping to the Xp21 region. In this study, by linkage analysis using seven RFLP markers between Xp21 and Xcen, we examined four families with multiple affected individuals. Close linkage was found between XLRP and polymorphic sites OTC (theta = .06 with lod 5.69), DXS84 (theta = .05 with lod 4.08), and DXS206 (theta = .06 with lod 2.56), defined by probes OTC, 754, and XJ, respectively. The close linkage of OTC, 754, and XJ to XLRP localizes the XLRP locus to the Xp21 region. Data from recombinations in three of four families place the locus above L1.28 and below the Duchenne muscular dystrophy (DMD) gene, consistent with an Xp21 localization. In one family, however, one affected male revealed a crossover between XLRP and all DNA markers, except for the more distal DXS28 (C7), while his brother is recombined for this marker (C7) and not other, more proximal markers. This suggests that in this family the XLRP mutation maps near DXS28 and above the DMD locus.
X连锁隐性型视网膜色素变性(XLRP)可导致受影响男性在生命的第三个或第四个十年出现进行性夜盲、视野缩窄,并最终失明。该病的生化基础尚不清楚,产前诊断和明确的携带者诊断仍然难以实现。XLRP患者家系的连锁研究以及女性携带者中观察到的表型差异提示了XLRP的异质性。通过探针L1.28检测到与位于DXS7(Xp11.3)位点的RFLP紧密连锁,提示XLRP定位于Xp11.3附近。在其他研究中,一种伴有金属光泽的XLRP位点与定位于Xp21区域的鸟氨酸转氨甲酰酶(OTC)位点连锁。在本研究中,我们使用Xp21和Xcen之间的7个RFLP标记进行连锁分析,研究了4个有多个受累个体的家系。发现XLRP与分别由探针OTC、754和XJ定义的多态性位点OTC(θ = 0.06,lod值为5.69)、DXS84(θ = 0.05,lod值为4.08)和DXS206(θ = 0.06,lod值为2.56)紧密连锁。OTC、754和XJ与XLRP的紧密连锁将XLRP位点定位于Xp21区域。四个家系中有三个家系的重组数据表明该位点位于L1.28上方和杜兴肌营养不良(DMD)基因下方,与Xp21定位一致。然而,在一个家系中,一名受累男性显示XLRP与所有DNA标记之间发生了交换,但更远端的DXS28(C7)除外,而他的兄弟则在该标记(C7)处发生了重组,而在其他更近端的标记处未发生重组。这表明在这个家系中,XLRP突变位于DXS28附近且在DMD位点上方。