Xu Zheng-feng, Yi Long, Mo Xu-ming, Hu Yali, Wang Dong-jin, Zhu Rui-fang, Jiang Yong-zhong, Wu Xing, Wu Zhong, Shen Li, Zhang Ying, Zhong Xiao-ling
Drum Tower Hospital, Nanjing University Medical School, Nanjing, Jiangsu, 210008 PR China.
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2006 Jun;23(3):250-5.
To ascertain 5 short tandem repeat (STR) markers as qualified tools for detecting chromosome 22q11 deletion and to understand the prevalence and clinical importance of the deletions in patients with congenital heart diseases (CHD) from Chinese Han population.
The authors selected 5 new tetranucleotide repeat markers, 22D_4_1, 22D_4_2, 22D_4_3, 22D_4_4 and D22S873 located in the proximal region of chromosome 22q11 deletion. One hundred and sixty-three unselected CHD patients and their unaffected parents were analyzed by genotyping of these new tetranucleotide STR markers to detect 22q11 deletion. With fluorescence in situ hybridization (FISH, LSI dual color DNA probe), the deletion status was confirmed in all patients with deletions and some patients without deletions.
The heterozygosity of these STR markers in normal population was more than 0.7, except for 22D_4_1 and 22D_4_2 that were 0.65 and 0.52 respectively. Twelve cases of 163 CHD patients (7.36%) had the deletions at chromosome 22q11. The deletions were confirmed in 9 of 12 patients by FISH, except for 2 cases who had unique nested deletion and 1 case who had nested distal deletion. One hundred and ten patients were associated with ventricular septal defect (VSD); and 9 (8.18%) of these cases had microdeletion. Twenty-one patients were associated with tetralogy of Fallot (TOF); and 3 (14.3%) of these cases had microdeletion.
This study demonstrated that genotyping of 5 STR markers was a useful mean of detecting 22q11 microdeletion in clinical diagnosis owing to its rapid experimental procedure, cost effectiveness and high resolution. 22q11 deletion was common in CHD patients, particularly in VSD and TOF patients, from Chinese Han population.
确定5个短串联重复序列(STR)标记作为检测22q11染色体缺失的合格工具,并了解中国汉族先天性心脏病(CHD)患者中该缺失的发生率及临床意义。
作者选择了位于22q11染色体缺失近端区域的5个新的四核苷酸重复标记,即22D_4_1、22D_4_2、22D_4_3、22D_4_4和D22S873。通过对这些新的四核苷酸STR标记进行基因分型,分析了163例未经选择的CHD患者及其未受影响的父母,以检测22q11缺失。采用荧光原位杂交(FISH,LSI双色DNA探针)技术,对所有缺失患者及部分无缺失患者的缺失状态进行了确认。
这些STR标记在正常人群中的杂合度均大于0.7,但22D_4_1和22D_4_2分别为0.65和0.52。163例CHD患者中有12例(7.36%)存在22q11染色体缺失。12例患者中有9例经FISH确认存在缺失,其中2例为独特的嵌套缺失,1例为嵌套远端缺失。110例患者合并室间隔缺损(VSD),其中9例(8.18%)存在微缺失。21例患者合并法洛四联症(TOF),其中3例(14.3%)存在微缺失。
本研究表明,5个STR标记的基因分型因其实验过程快速、成本效益高和分辨率高,是临床诊断中检测22q11微缺失的有用方法。22q11缺失在中国汉族CHD患者中较为常见,尤其是在VSD和TOF患者中。