Jones A C, Yamamura Y, Almasy L, Bohlega S, Elibol B, Hubble J, Kuzuhara S, Uchida M, Yanagi T, Weeks D E, Nygaard T G
Department of Neurology, Columbia University College of Physicians and Surgeons, New York, NY 10032, USA.
Am J Hum Genet. 1998 Jul;63(1):80-7. doi: 10.1086/301937.
Parkinson disease (PD) is a common neurodegenerative condition associated with degeneration of dopaminergic neurons in the zona compacta of the substantia nigra. There is increasing evidence that genetic factors play a role in the etiology of PD, although genetic heterogeneity is likely. An autosomal dominant syndrome with many similarities to sporadic PD has been mapped to 4q21-22 in a large Italian pedigree and has been found to be due to mutation of the alpha-synuclein gene. However, this gene appears to account for only a minority of PD, and a susceptibility locus for autosomal dominant parkinsonism has recently been mapped, on 2p13. Autosomal recessive juvenile parkinsonism (JP), which shows marked clinical similarity to PD, maps to 6q25.2-q27. We found linkage to this region in a group of 15 families from four distinct ethnic backgrounds. A full genomic screen excluded other candidate regions. We have constructed a detailed genetic map of the linked region and have mapped the position of the manganese superoxide dismutase gene (SOD2). Recombination events restricted the JP locus to a 6.9-cM region and excluded SOD2. The apparent homozygosity for null alleles at D6S955 in one family suggested a deletion and finer localization of the JP locus.
帕金森病(PD)是一种常见的神经退行性疾病,与黑质致密部多巴胺能神经元的变性有关。越来越多的证据表明遗传因素在PD的病因中起作用,尽管可能存在遗传异质性。在一个大型意大利家系中,一种与散发性PD有许多相似之处的常染色体显性综合征已被定位到4q21 - 22,并且已发现是由于α-突触核蛋白基因突变所致。然而,该基因似乎仅占少数PD病例,并且常染色体显性帕金森病的一个易感基因座最近已被定位到2p13。常染色体隐性青少年帕金森病(JP)与PD表现出明显的临床相似性,定位于6q25.2 - q27。我们在来自四个不同种族背景的15个家系中发现了与该区域的连锁关系。全基因组筛选排除了其他候选区域。我们构建了连锁区域的详细遗传图谱,并确定了锰超氧化物歧化酶基因(SOD2)的位置。重组事件将JP基因座限制在一个6.9厘摩的区域,并排除了SOD2。一个家系中D6S955处无效等位基因的明显纯合性提示了JP基因座的缺失和更精细的定位。