de Sousa Andrade Susana Margarida, Monteiro Ana Rita Tomé, Martins Jorge Humberto Ferreira, Alves Marisa Costa, Santos Silva Luis Filipe, Quadros Jorge Manuel Cardoso, Ribeiro Carlos Alberto Reis
Cochlear Implant Unit, ENT Department, Centro Hospitalar de Coimbra, E.P.E. Quinta dos Vales - São Martinho do Bispo 3041-853 Coimbra, Portugal.
Int J Pediatr Otorhinolaryngol. 2012 Sep;76(9):1375-8. doi: 10.1016/j.ijporl.2012.06.010. Epub 2012 Jul 9.
The purpose of this study was to review the outcomes of children with documented Waardenburg syndrome implanted in the ENT Department of Centro Hospitalar de Coimbra, concerning postoperative speech perception and production, in comparison to the rest of non-syndromic implanted children.
A retrospective chart review was performed for children congenitally deaf who had undergone cochlear implantation with multichannel implants, diagnosed as having Waardenburg syndrome, between 1992 and 2011. Postoperative performance outcomes were assessed and confronted with results obtained by children with non-syndromic congenital deafness also implanted in our department. Open-set auditory perception skills were evaluated by using European Portuguese speech discrimination tests (vowels test, monosyllabic word test, number word test and words in sentence test). Meaningful auditory integration scales (MAIS) and categories of auditory performance (CAP) were also measured. Speech production was further assessed and included results on meaningful use of speech Scale (MUSS) and speech intelligibility rating (SIR).
To date, 6 implanted children were clinically identified as having WS type I, and one met the diagnosis of type II. All WS children received multichannel cochlear implants, with a mean age at implantation of 30.6±9.7months (ranging from 19 to 42months). Postoperative outcomes in WS children were similar to other nonsyndromic children. In addition, in number word and vowels discrimination test WS group showed slightly better performances, as well as in MUSS and MAIS assessment.
Our study has shown that cochlear implantation should be considered a rehabilitative option for Waardenburg syndrome children with profound deafness, enabling the development and improvement of speech perception and production abilities in this group of patients, reinforcing their candidacy for this audio-oral rehabilitation method.
本研究旨在回顾在科英布拉中心医院耳鼻喉科接受植入手术的Waardenburg综合征患儿的术后语音感知和发音结果,并与其他非综合征植入患儿进行比较。
对1992年至2011年间被诊断为Waardenburg综合征、接受多通道人工耳蜗植入的先天性耳聋患儿进行回顾性病历审查。评估术后表现结果,并与在我们科室接受植入的非综合征先天性耳聋患儿的结果进行对比。通过使用欧洲葡萄牙语语音辨别测试(元音测试、单音节词测试、数字词测试和句子中的词测试)来评估开放式听觉感知技能。还测量了有意义听觉整合量表(MAIS)和听觉表现类别(CAP)。进一步评估语音发音,并包括有意义语音使用量表(MUSS)和语音清晰度评级(SIR)的结果。
迄今为止,临床上确定有6名植入患儿患有I型Waardenburg综合征,1名符合II型诊断。所有Waardenburg综合征患儿均接受了多通道人工耳蜗植入,植入时的平均年龄为30.6±9.7个月(范围为19至42个月)。Waardenburg综合征患儿术后结果与其他非综合征患儿相似。此外,在数字词和元音辨别测试中,Waardenburg综合征组表现略好,在MUSS和MAIS评估中也是如此。
我们的研究表明,人工耳蜗植入应被视为重度耳聋的Waardenburg综合征患儿的一种康复选择,能够促进和改善该组患者的语音感知和发音能力,增强他们接受这种听觉-口语康复方法的适宜性。