Chamberlain S, Shaw J, Wallis J, Rowland A, Chow L, Farrall M, Keats B, Richter A, Roy M, Melancon S
Department of Biochemistry and Molecular Genetics, St. Mary's Hospital, University of London, England.
Am J Hum Genet. 1989 Apr;44(4):518-21.
Classical Friedreich ataxia, a progressive, neurodegenerative disorder involving both the central and peripheral nervous systems, has been subclassified according to the observed clinical heterogeneity. The variations in the age at onset and in the spectrum and severity of symptoms have previously been interpreted as evidence of genetic heterogeneity. We have studied the linkage between the disorder and closely linked DNA markers in families of distinct ethnic origins, including the "typical" French-Canadians and the Acadian population of Louisiana. The disease in these two populations, both of continental French origin, has a very similar initial clinical picture. However, a marked difference in the rate of progression of the obligatory symptoms after 10 years of apparent disease is observed. A total of 553 individuals from 80 families with 202 affected members have been typed with the chromosome 9 marker MCT112, which we have previously shown to be closely linked to the disease locus. Evidence for linkage was observed in all families with the generation of a combined total lod score of 25.09 at a recombination fraction of theta = .00, providing strong evidence for genetic homogeneity at this locus for the classical form of this disease.
典型的弗里德赖希共济失调是一种累及中枢和周围神经系统的进行性神经退行性疾病,根据观察到的临床异质性已被进一步分类。发病年龄以及症状谱和严重程度的差异以前被解释为遗传异质性的证据。我们研究了不同种族起源家族中该疾病与紧密连锁的DNA标记之间的连锁关系,包括“典型的”法裔加拿大人和路易斯安那州的阿卡迪亚人群。这两个人口群体都起源于法国大陆,该疾病具有非常相似的初始临床症状。然而,在明显患病10年后,强制性症状的进展速度存在显著差异。我们用9号染色体标记MCT112对来自80个家庭的553人进行了分型,这些家庭中有202名受影响成员,我们之前已证明该标记与疾病位点紧密连锁。在所有家庭中均观察到连锁证据,在重组率θ = 0.00时,联合总lod分数为25.09,为该疾病经典形式在该位点的遗传同质性提供了有力证据。