Melamud A, Shen G-Q, Chung D, Xi Q, Simpson E, Li L, Peachey N S, Zegarra H, Hagstrom S A, Wang Q K, Traboulsi E I
J Med Genet. 2006 Jun;43(6):e27. doi: 10.1136/jmg.2005.031518.
We have defined a new genetic locus for an X linked form of retinitis pigmentosa (RP) on chromosome Xq28. We examined 15 members of a family in which RP appeared to be transmitted in an X linked manner. Ocular examinations were performed, and fundus photographs and electroretinograms were obtained for selected patients. Blood samples were obtained from all patients and an additional seven family members who were not given examinations. Visual acuity in four affected individuals ranged from 20/40 to 20/80+. Patients described the onset of night blindness and colour vision defects in the second decade of life, with the earliest at 13 years of age. Examined affected individuals had constricted visual fields and retinal findings compatible with RP. Based on full field electroretinography, cone function was more severely reduced than rod function. Female carriers had no ocular signs or symptoms and slightly reduced cone electroretinographic responses. Affected and non-affected family members were genotyped for 20 polymorphic markers on the X-chromosome spaced at 10 cM intervals. Genotyping data were analysed using GeneMapper software. Genotyping and linkage analyses identified significant linkage to markers DXS8061, DXS1073, and DXS1108 with two point LOD scores of 2.06, 2.17, and 2.20, respectively. Haplotype analysis revealed segregation of the disease phenotype with markers at Xq28.
我们在X染色体的Xq28区域确定了一个与X连锁型视网膜色素变性(RP)相关的新基因座。我们研究了一个家族中的15名成员,该家族中RP似乎以X连锁的方式遗传。对患者进行了眼部检查,并为部分患者拍摄了眼底照片和记录了视网膜电图。采集了所有患者以及另外7名未接受检查的家庭成员的血样。4名受影响个体的视力范围为20/40至20/80+。患者描述在生命的第二个十年开始出现夜盲和色觉缺陷,最早为13岁。接受检查的受影响个体视野缩小,视网膜表现与RP相符。基于全视野视网膜电图,视锥细胞功能比视杆细胞功能受损更严重。女性携带者没有眼部体征或症状,视锥细胞视网膜电图反应略有降低。对受影响和未受影响的家庭成员进行了X染色体上20个多态性标记的基因分型,这些标记间隔为10厘摩(cM)。使用GeneMapper软件分析基因分型数据。基因分型和连锁分析确定与标记DXS8061、DXS1073和DXS1108存在显著连锁,两点连锁分析的对数优势(LOD)分数分别为2.06、2.17和2.20。单倍型分析显示疾病表型与Xq28处的标记存在分离。