Goellner G M, Tester D, Thibodeau S, Almqvist E, Goldberg Y P, Hayden M R, McMurray C T
Molecular Neuroscience Program, Mayo Foundation, Rochester, MN 55905, USA.
Am J Hum Genet. 1997 Apr;60(4):879-90.
Two recent lines of evidence raise the possibility that instability in germ-line or somatic cells arises by a common mechanism that involves defective mismatch repair. Mutations in mismatch-repair proteins are known to cause instability in hereditary nonpolyposis colorectal cancer, instability that is physically similar to germ-line instability observed in Huntington disease (HD). Furthermore, both germ-line and somatic-cell instability are likely to be mitotic defects, the former occurring early in embryogenesis. To test the hypothesis that defective repair is a common prerequisite for instability, we have utilized two disease groups that represent different instability "conditions." Germ-line instability within simple tandem repeats (STR) at 10 loci in 29 HD families were compared with somatic instability at the same loci in 26 colon cancer (CC) patients with identified or suspected defects in mismatch-repair enzymes. HD is known to be caused by expansion within the CAG repeat of the locus, but the extent or pattern of STR instability outside this region has not been examined systematically. We find a distinctly different pattern of STR mutation in the two disease groups, suggesting different mechanisms. Instability in HD is generally confined to a single locus, whereas instability is widespread for the same loci in CC. Our data do not support a causative role for defective mismatch-repair enzymes in instability associated with HD; rather, our data are consistent with a model in which DNA structure may inhibit normal mismatch repair at the expansion site.
最近的两条证据线索表明,生殖细胞或体细胞中的不稳定性可能是由一种涉及缺陷错配修复的共同机制引起的。已知错配修复蛋白的突变会导致遗传性非息肉病性结直肠癌中的不稳定性,这种不稳定性在物理上类似于在亨廷顿舞蹈症(HD)中观察到的生殖系不稳定性。此外,生殖系和体细胞的不稳定性都可能是有丝分裂缺陷,前者发生在胚胎发育早期。为了检验缺陷修复是不稳定性的共同先决条件这一假设,我们利用了代表不同不稳定性“情况”的两个疾病组。将29个HD家族中10个位点的简单串联重复序列(STR)内的生殖系不稳定性与26名错配修复酶存在已确定或疑似缺陷的结肠癌(CC)患者相同位点的体细胞不稳定性进行了比较。已知HD是由该位点的CAG重复序列内的扩增引起的,但该区域外STR不稳定性的程度或模式尚未得到系统研究。我们在这两个疾病组中发现了明显不同的STR突变模式,提示存在不同机制。HD中的不稳定性通常局限于单个位点,而CC中相同位点的不稳定性则很普遍。我们的数据不支持缺陷错配修复酶在与HD相关的不稳定性中起致病作用;相反,我们的数据与一种模型一致,即DNA结构可能在扩增位点抑制正常的错配修复。