Yan Y, Wu Q, Zhang L, Wang X, Dan S, Deng D, Sun L, Yao L, Ma Y, Wang L
Department of Ultrasound, Beijing Obstetrics and Gynecology Hospital, Capital Medical University, Beijing, P.R. China.
Ultrasound Obstet Gynecol. 2014 Apr;43(4):404-12. doi: 10.1002/uog.13236.
To evaluate the usefulness of array-based comparative genomic hybridization (aCGH) for prenatal genetic diagnosis of congenital heart disease (CHD), with and without associated anomalies, and to explore the relationship between submicroscopic chromosomal aberrations and CHD.
In this prospective study we investigated 76 consecutive singleton fetuses with abnormal cardiac ultrasound findings, normal karyotype and negative or no fluorescence in-situ hybridization results for 22q11.2 deletion syndrome. All pregnancies underwent aCGH in a comprehensive search for chromosomal aberrations. The relationship between copy number variations (CNVs) and CHD was determined by comparing clinical findings to chromosomal databases.
CNVs that were benign or had no clinical significance were detected in 18/76 (23.7%) cases. CNVs of unknown clinical significance (i.e. VOUS) were detected in 4/76 (5.3%) cases. Pathogenic CNVs were detected in 5/76 (6.6%) cases. Fetuses with CHD and additional structural abnormalities demonstrated no difference in number of pathogenic CNVs when compared with fetuses with isolated CHD (7.4% (n = 2/27) vs 6.1% (n = 3/49), P > 0.05).
In this study cohort, aCGH analysis significantly improved the detection of submicroscopic chromosomal aberrations in pregnancies with CHD, as compared with conventional cytogenetics. Our results suggest that aCGH can provide additional genetic information in fetuses with abnormal heart findings.
评估基于芯片的比较基因组杂交技术(aCGH)在先天性心脏病(CHD)产前基因诊断中的应用价值,包括有无合并其他异常情况,并探讨亚显微染色体畸变与CHD之间的关系。
在这项前瞻性研究中,我们调查了76例连续的单胎胎儿,这些胎儿心脏超声检查结果异常、核型正常且22q11.2缺失综合征的荧光原位杂交结果为阴性或无异常。所有孕妇均接受aCGH检测,以全面筛查染色体畸变。通过将临床结果与染色体数据库进行比较,确定拷贝数变异(CNV)与CHD之间的关系。
在18/76(23.7%)例中检测到良性或无临床意义的CNV。在4/76(5.3%)例中检测到临床意义不明的CNV(即VOUS)。在5/76(6.6%)例中检测到致病性CNV。与单纯CHD胎儿相比,患有CHD且伴有其他结构异常的胎儿在致病性CNV数量上无差异(7.4%(n = 2/27)对6.1%(n = 3/49),P>0.05)。
在本研究队列中,与传统细胞遗传学相比,aCGH分析显著提高了CHD妊娠中亚显微染色体畸变的检测率。我们的结果表明,aCGH可为心脏检查异常的胎儿提供额外的遗传信息。