O'Donnell D M, Zoghbi H Y
Baylor College of Medicine, Houston, Texas 77030, USA.
Curr Opin Pediatr. 1995 Dec;7(6):715-25. doi: 10.1097/00008480-199512000-00016.
The relationship between the expansion of trinucleotide repeat sequences and human disease hs been the subject of a significant volume of study since the identification of a CGG repeat sequence in the mutated gene responsible for the fragile X syndrome. Six other neurologic diseases are now known to result from a triplet repeat expansion of either CTG or CAG nucleotides. Of particular interest to the pediatrician or pediatric subspecialist is the phenomenon of "anticipation," now clarified by mechanisms inherent to trinucleotide repeat expansions. The progressive enlargement of repeat sequences in successive generations of affected kindreds correlates inversely with the age of onset and, in some cases, the severity of the disease. The presentation of a young patient with symptoms ranging from developmental delay to movement disorders and ataxia requires that the physician involved in the child's care be aware of these diseases, their phenotypic variability, and their effects in previous generations. In this manner, pertinent history, including family history, may be obtained, relevant diagnostic testing initiated, and appropriate referrals facilitated.
自从在导致脆性X综合征的突变基因中鉴定出CGG重复序列以来,三核苷酸重复序列的扩增与人类疾病之间的关系一直是大量研究的主题。现在已知另外六种神经系统疾病是由CTG或CAG核苷酸的三联体重复扩增引起的。儿科医生或儿科专科医生特别感兴趣的是“遗传早现”现象,现在已通过三核苷酸重复扩增所固有的机制得以阐明。在受影响的家族连续几代中,重复序列的逐渐扩大多发病年龄呈负相关,在某些情况下,还与疾病的严重程度呈负相关。一名年轻患者出现从发育迟缓到运动障碍和共济失调等症状,这就要求参与该儿童护理的医生了解这些疾病、它们的表型变异性以及它们在前几代中的影响。通过这种方式,可以获取相关病史,包括家族史,启动相关诊断测试,并促进适当的转诊。