Gicquel C, Cabrol S, Schneid H, Girard F, Le Bouc Y
Laboratoire d'Explorations Fonctionnelles Endocriniennes, Hôpital Trousseau, Paris, France.
J Med Genet. 1992 Aug;29(8):547-51. doi: 10.1136/jmg.29.8.547.
Turner's syndrome is a common disorder which occurs in around 1/3000 live births in girls. Diagnostic use of polymorphic DNA markers for the X chromosome could help to reduce the number of time consuming karyotype analyses needed. The M27 beta probe maps on the X chromosome to Xcen-Xp11-22 and in 83% of female subjects detects heterozygosity with multiallelic polymorphism. In Southern blotting, a single X chromosome yields a single hybridisation band. In this study, genomic DNA was extracted from leucocytes of 49 patients with Turner's syndrome (karyotypes: 45,XO, n = 29; 45,XO/46,XX, n = 4; 46,Xi(Xq), n = 1; 45,XO/46,Xi(Xq), n = 4; 45,XO/46,Xr(X), n = 4; 45,XO/46,XY, n = 4; 46,XXp-, n = 3), digested with EcoRI or HindIII, and analysed by Southern blotting. The molecular data for each patient were compared with DNA controls (homozygous 46,XX, heterozygous 46,XX and 46,XY DNA). A single band of reduced intensity compared to homozygous 46,XX control DNA was seen in 41 cases. Two hybridisation bands of different intensities were seen in four patients, in one of whom mosaicism was suspected on the basis of molecular analysis, despite a 45,XO karyotype. In four cases, Turner's syndrome failed to be detected: one 45,XO/46,XX mosaicism with only 4% of 45,XO cells and three distal Xp deletions. DNA analysis appears to be a useful and rapid tool in screening for Turner's syndrome and could be an alternative to cytogenetic analysis in diagnosing the disorder when severe growth retardation or delayed puberty are not accompanied by a Turner phenotype.
特纳综合征是一种常见疾病,在女孩活产中的发生率约为1/3000。使用多态性DNA标记对X染色体进行诊断有助于减少所需的耗时核型分析次数。M27β探针定位于X染色体的Xcen - Xp11 - 22区域,在83%的女性受试者中可检测到多等位基因多态性的杂合性。在Southern印迹法中,单个X染色体产生一条杂交带。在本研究中,从49例特纳综合征患者的白细胞中提取基因组DNA(核型:45,XO,n = 29;45,XO/46,XX,n = 4;46,Xi(Xq),n = 1;45,XO/46,Xi(Xq),n = 4;45,XO/46,Xr(X),n = 4;45,XO/46,XY,n = 4;46,XXp - ,n = 3),用EcoRI或HindIII消化,然后进行Southern印迹分析。将每位患者的分子数据与DNA对照(纯合46,XX、杂合46,XX和46,XY DNA)进行比较。与纯合46,XX对照DNA相比,41例患者出现一条强度降低的条带。4例患者出现两条不同强度的杂交带,其中1例尽管核型为45,XO,但根据分子分析怀疑存在嵌合体。4例未检测到特纳综合征:1例为45,XO/46,XX嵌合体,其中45,XO细胞仅占4%,3例为Xp远端缺失。DNA分析似乎是筛查特纳综合征的一种有用且快速的工具,在诊断该疾病时,当严重生长发育迟缓或青春期延迟未伴有特纳表型时,可作为细胞遗传学分析的替代方法。