Stoilova D, Child A, Trifan O C, Crick R P, Coakes R L, Sarfarazi M
Surgical Research Center, University of Connecticut Health Center, Farmington, Connecticut, 06030-1110, USA.
Genomics. 1996 Aug 15;36(1):142-50. doi: 10.1006/geno.1996.0434.
Primary open angle glaucoma (GLC1) is a common ocular disorder with a characteristic degeneration of the optic nerve and visual field defects that is often associated with an elevated intraocular pressure. The severe but rare juvenile-onset type has previously been mapped to 1q21-q31, and its genetic heterogeneity has been established. Herein, we present a new locus (GLC1B) for one form of GLC1 on chromosome 2cen-q13 with a clinical presentation of low to moderate intraocular pressure, onset in late 40s, and a good response to medical treatment. Two-point and haplotype analyses of affected and unaffected meioses in six families provided maximum linkage information with D2S417, GATA112EO3, D2S113, D2S373, and D2S274 (lod scores ranging from 3.11 to 6.48) within a region of 8.5 cM that is flanked by D2S2161 and D2S2264. Analysis of affected meioses alone revealed no recombination with an additional two markers (D2S2264 and D2S135) in a region of 11.2 cM that is flanked by D2S2161 and D2S176. Analysis of unaffected meioses identified only one healthy 86-year-old male who has inherited the entire affected haplotype and, hence, is a gene carrier for this condition. Eight additional families with similar and/or different clinical presentation did not show any linkage to this region and, therefore, provided evidence for genetic heterogeneity of adult-onset primary open angle glaucoma.
原发性开角型青光眼(GLC1)是一种常见的眼部疾病,其特征为视神经变性和视野缺损,常伴有眼压升高。严重但罕见的青少年型青光眼之前已被定位到1q21 - q31,并且其遗传异质性已得到证实。在此,我们报告了位于2号染色体cen - q13上的一种GLC1新位点(GLC1B),其临床表现为眼压低至中度,发病于40多岁后期,对药物治疗反应良好。对六个家系中患病和未患病减数分裂进行两点和单倍型分析,在8.5厘摩区域内与D2S417、GATA112EO3、D2S113、D2S373和D2S274提供了最大连锁信息(lod分数范围为3.11至6.48),该区域两侧为D2S2161和D2S2264。仅对患病减数分裂进行分析显示,在11.2厘摩区域内与另外两个标记(D2S2264和D2S135)无重组,该区域两侧为D2S2161和D2S176。对未患病减数分裂进行分析仅发现一名86岁健康男性,他继承了整个患病单倍型,因此是这种疾病的基因携带者。另外八个具有相似和/或不同临床表现的家系与该区域无连锁关系,因此为成人型原发性开角型青光眼的遗传异质性提供了证据。