Deng Yuyuan, Sang Shushan, Wen Jie, Liu Yalan, Ling Jie, Chen Hongsheng, Cai Xinzhang, Mei Lingyun, Chen Xiaoya, Li Meng, Li Wu, Li Taoxi, He Chufeng, Feng Yong
Department of Otolaryngology, Xiangya Hospital, Central South University, Center for Medical Genetics, Central South University, Key Laboratory of Otolaryngology Major Disease Research of Hunan Province, Xiangya Hospital, Central South University, China.
Department of Otolaryngology, Xiangya Hospital, Central South University, China.
Int J Pediatr Otorhinolaryngol. 2018 Dec;115:114-119. doi: 10.1016/j.ijporl.2018.08.026. Epub 2018 Sep 12.
To evaluate the accuracy and validity of our protocol for prenatal diagnosis and genetic counseling in high-risk families at a clinic.
Fifteen unrelated families with recessive nonsyndromic hearing loss (NSHL) in their family history and a positive attitude towards prenatal diagnosis were recruited in the present study. According to genetic information for each family, Sanger sequencing, fluorescence polymerase chain reaction (PCR)-based congenital deafness gene detection kit and multiple PCR-based target gene capture and high-throughput sequencing were used. Genetic counseling was offered to all participating families by genetic counselors and otologists. Prenatal diagnosis was provided to families with detected pathogenic mutations and who were expected to participate in subsequent prenatal diagnosis.
In this study, confirmed pathogenic mutations were detected in eight families, who were defined as high-risk families. These families all participated in prenatal diagnosis with positive attitudes. One novel variant (c.1687dupA) in the SLC264 gene was detected in a family. Through genetic counseling, the recurrence probability of NSHL in fetuses was 25% in six families, 0% in one family, and 50% in one family. The results of fetal DNA detection showed that one fetal variant was wild type, three were heterozygous mutations in SLC26A4, and one was a compound heterozygous mutation in SLC26A4. Two variants were heterozygous mutations in GJB2, and one was a homozygous mutation in GJB2. According to the test results for fetal DNA, prenatal diagnosis found that six fetuses had normal hearing, whereas two fetuses suffered from NSHL. After birth, six infants predicted to have normal hearing passed a newborn hearing screening test and two infants predicted to have NSHL were diagnosed with NSHL and received cochlear implants.
Our protocol for prenatal diagnosis and genetic counseling provides detailed information that can assist couples in high-risk families in preparing for infant arrival and future family planning. For the affected neonates, prenatal diagnosis and genetic counseling achieve an "early screening, early diagnosis, early intervention" strategy.
评估我们在一家诊所为高危家庭进行产前诊断和遗传咨询方案的准确性和有效性。
本研究招募了15个有隐性非综合征性听力损失(NSHL)家族史且对产前诊断持积极态度的无血缘关系家庭。根据每个家庭的遗传信息,采用桑格测序、基于荧光聚合酶链反应(PCR)的先天性耳聋基因检测试剂盒以及基于多重PCR的目标基因捕获和高通量测序。遗传咨询师和耳科医生为所有参与家庭提供遗传咨询。为检测到致病突变且预期参与后续产前诊断的家庭提供产前诊断。
在本研究中,8个家庭检测到确诊的致病突变,这些家庭被定义为高危家庭。这些家庭均以积极态度参与了产前诊断。在一个家庭中检测到SLC264基因中的一个新变异(c.1687dupA)。通过遗传咨询,6个家庭中胎儿患NSHL的复发概率为25%,1个家庭为0%,1个家庭为50%。胎儿DNA检测结果显示,1个胎儿变异为野生型,3个为SLC26A4基因的杂合突变,1个为SLC26A4基因的复合杂合突变。2个变异为GJB2基因的杂合突变,1个为GJB2基因的纯合突变。根据胎儿DNA检测结果,产前诊断发现6例胎儿听力正常,而2例胎儿患有NSHL。出生后,6例预测听力正常的婴儿通过了新生儿听力筛查测试,2例预测患有NSHL的婴儿被诊断为NSHL并接受了人工耳蜗植入。
我们的产前诊断和遗传咨询方案提供了详细信息,可帮助高危家庭的夫妇为婴儿出生和未来计划生育做好准备。对于受影响的新生儿,产前诊断和遗传咨询实现了“早筛查、早诊断、早干预”策略。