Rodrigues Fidjy, Paneque Milena, Reis Cláudia, Venâncio Margarida, Sequeiros Jorge, Saraiva Jorge
ICBAS, Universidade do Porto, Porto, Portugal.
J Genet Couns. 2013 Aug;22(4):448-54. doi: 10.1007/s10897-012-9565-z. Epub 2013 Jan 26.
Recent advances in molecular genetics have allowed the determination of the genetic cause of some childhood non-syndromic deafness. In Portugal only a small proportion of families are referred to a clinical genetics service in order to clarify the etiology of the deafness and to provide genetic counseling. Consequently, there are no published studies of the prior beliefs of parents about the causes of hereditary deafness of their children and their genetic knowledge after receipt of genetic counseling. In order to evaluate the impact of genetic counseling, 44 parents of 24 children with the diagnosis of non-syndromic sensorineural prelingual deafness due to mutations in the GJB2 (connexin 26), completed surveys before and after genetic counseling. Before counseling 13.6 % of the parents knew the cause of deafness; at a post-counseling setting this percentage was significantly higher, with 84.1 % of the parents accurately identifying the etiology. No significant differences were found between the answers of mothers and fathers either before or after genetic counseling. Parents' level of education was a significant factor in pre-test knowledge. After genetic counseling 95.5 % of the parents stated that the consultation had met their expectations, 70.5 % remembered correctly the inheritance pattern, and 93.2 % correctly recalled the chance of risk of deafness. These results underline the importance of genetic counseling in demystifying parents' beliefs about the etiology of their children's deafness.
分子遗传学的最新进展使得确定某些儿童非综合征性耳聋的遗传病因成为可能。在葡萄牙,只有一小部分家庭会被转介到临床遗传学服务机构,以明确耳聋的病因并提供遗传咨询。因此,目前尚无关于父母对子女遗传性耳聋病因的先前认知以及他们在接受遗传咨询后的遗传知识的公开研究。为了评估遗传咨询的效果,对24名被诊断为由于GJB2(连接蛋白26)基因突变导致的非综合征性感音神经性语前聋儿童的44名家长在遗传咨询前后进行了问卷调查。咨询前,13.6%的家长知道耳聋的病因;在咨询后,这一比例显著提高,84.1%的家长准确识别了病因。在遗传咨询前后,父亲和母亲的回答均未发现显著差异。家长的教育水平是咨询前知识水平的一个重要因素。遗传咨询后,95.5%的家长表示咨询符合他们的期望,70.5%的家长正确记住了遗传模式,93.2%的家长正确回忆起耳聋的风险几率。这些结果强调了遗传咨询在消除父母对子女耳聋病因的误解方面的重要性。