Buckley Jason R, Kavarana Minoo N, Chowdhury Shahryar M, Scheurer Mark A
Department of Pediatrics, Division of Cardiology, Medical University of South Carolina, Charleston, SC, USA.
Congenit Heart Dis. 2015 May-Jun;10(3):E131-8. doi: 10.1111/chd.12241. Epub 2014 Dec 14.
Traditionally, karyotype and fluorescence in situ hybridization (FISH) were used for cytogenetic testing of infants with congenital heart disease (CHD) who underwent cardiac surgery at our institution. Recently, chromosome microarray analysis (CMA) has been performed in lieu of the traditional tests. A standardized approach to cytogenetic testing does not exist in this population. The purpose of this study was to assess the utility of CMA based on our current ordering practice.
We reviewed the records of all infants (<1 year old) who underwent cardiac surgery at our institution from January 2010 to June 2013. Data included results of all cytogenetic testing performed. Diagnostic yield was calculated as the percentage of significant abnormal results obtained by each test modality. Patients were grouped by classification of CHD.
Two hundred seventy-five (51%) of 535 infants who underwent cardiac surgery had cytogenetic testing. Of those tested, 154 (56%) had multiple tests performed and at least 18% were redundant or overlapping. The utilization of CMA has increased each year since its implementation. The diagnostic yield for karyotype, FISH and CMA was 10%, 12%, and 14%, respectively. CMA yield was significantly higher in patients with septal defects (33%, P = .01) compared with all other CHD classes. CMA detected abnormalities of unknown clinical significance in 13% of infants tested.
In our center, redundant cytogenetic testing is frequently performed in infants undergoing cardiac surgery. The utilization of CMA has increased over time and abnormalities of unknown clinical significance are detected in an important subset of patients. A screening algorithm that risk-stratifies based on classification of CHD and clinical suspicion may provide a practical, data-driven approach to genetic testing in this population and limit unnecessary resource utilization.
传统上,我们机构对接受心脏手术的先天性心脏病(CHD)婴儿进行细胞遗传学检测时采用核型分析和荧光原位杂交(FISH)。最近,已采用染色体微阵列分析(CMA)取代传统检测方法。在这一人群中不存在标准化的细胞遗传学检测方法。本研究的目的是根据我们目前的检测订单实践评估CMA的效用。
我们回顾了2010年1月至2013年6月在我们机构接受心脏手术的所有婴儿(<1岁)的记录。数据包括所有进行的细胞遗传学检测结果。诊断率以每种检测方式获得的显著异常结果的百分比计算。患者按CHD分类进行分组。
535例接受心脏手术的婴儿中有275例(51%)进行了细胞遗传学检测。在这些接受检测的婴儿中,154例(56%)进行了多项检测,且至少18%的检测是多余或重叠的。自实施以来,CMA的使用量每年都在增加。核型分析、FISH和CMA的诊断率分别为10%、12%和14%。与所有其他CHD类别相比,CMA在房间隔缺损患者中的诊断率显著更高(33%,P = 0.01)。CMA在13%的受检婴儿中检测到了临床意义不明的异常。
在我们中心,接受心脏手术的婴儿经常进行多余的细胞遗传学检测。随着时间的推移,CMA的使用量有所增加,并且在一个重要的患者亚组中检测到了临床意义不明的异常。基于CHD分类和临床怀疑进行风险分层的筛查算法可能为该人群的基因检测提供一种实用的、数据驱动的方法,并限制不必要的资源利用。