Haines J L, Short M P, Kwiatkowski D J, Jewell A, Andermann E, Bejjani B, Yang C H, Gusella J F, Amos J A
Molecular Neurogenetics Laboratory, Massachusetts General Hospital, Charlestown 02129.
Am J Hum Genet. 1991 Oct;49(4):764-72.
Tuberous sclerosis (TSC) is an autosomal dominant disorder with both neurological and cutaneous manifestations often resulting in significant disability. Although it has been studied clinically and biochemically for many years, the underlying pathophysiology remains unknown. Genetic linkage analysis provides an alternative strategy for understanding the genetic etiology of this disease. Genetic linkage of a gene for TSC to loci in 9q32-9q34 has been reported but has not been a universal finding, since absence of linkage to 9q loci, as well as linkage to loci on 11q, have also been reported. We present here data on 22 families (21 previously unreported) segregating TSC. Our results strongly support a TSC locus in the 9q32-34 region for approximately one-third of families and provide significant evidence for genetic heterogeneity. Application of newly described highly polymorphic dinucleotide repeat marker loci in TSC greatly enhanced the informativeness of our pedigrees and was vital for detecting the heterogeneity. No clear evidence of linkage to chromosome 11q22 markers was found, suggesting that a still unidentified TSC locus elsewhere in the genome may account for the majority of TSC families.
结节性硬化症(TSC)是一种常染色体显性疾病,具有神经和皮肤表现,常导致严重残疾。尽管对其进行临床和生化研究已有多年,但潜在的病理生理学仍不清楚。基因连锁分析为理解该疾病的遗传病因提供了一种替代策略。已报道TSC的一个基因与9q32 - 9q34位点存在基因连锁,但这并非普遍现象,因为也有报道称不存在与9q位点的连锁以及与11q位点的连锁。我们在此展示了22个(21个此前未报道)分离TSC的家系数据。我们的结果有力地支持了约三分之一的家系中TSC位点位于9q32 - 34区域,并为遗传异质性提供了重要证据。新描述的高度多态性二核苷酸重复标记位点在TSC中的应用极大地增强了我们家系图谱的信息量,对于检测异质性至关重要。未发现与染色体11q22标记存在明确连锁的证据,这表明基因组中其他尚未确定的TSC位点可能占大多数TSC家系。