Cullen Robert D, Zdanski Carlton, Roush Patricia, Brown Carolyn, Teagle Holly, Pillsbury Harold C, Buchman Craig
The W. Paul Biggers, MD, Carolina Children's Communication Disorders Program, Department of Otolaryngology-Head and Neck Surgery, University of North Carolina at Chapel Hill, Chapel Hill, North Carolina 27599-7070, USA.
Laryngoscope. 2006 Jul;116(7):1273-5. doi: 10.1097/01.mlg.0000221959.67801.9b.
Waardenburg syndrome is an autosomal-dominant syndrome characterized by dystopia canthorum, hyperplasia of the eyebrows, heterochromia irides, a white forelock, and sensorineural hearing loss in 20% to 55% of patients. This patient population accounts for approximately 2% of congenitally deaf children. The purpose of this retrospective case review was to describe the outcomes for those children with Waardenburg syndrome who have undergone cochlear implantation.
Pediatric cochlear implant recipients with documented evidence of Waardenburg syndrome underwent retrospective case review. All patients received their cochlear implants at the study institution followed by outpatient auditory habilitation. Charts were reviewed for etiology and duration of deafness, age at time of cochlear implantation, perioperative complications, duration of use, and performance outcomes. Results of standard tests batteries for speech perception and production administered as a part of the patients' auditory habilitation were reviewed.
Seven patients with Waardenburg syndrome and cochlear implants were identified. The average age at implantation was 37 months (range, 18-64 months) and the average duration of use was 69 months (range, 12-143 months). All of these patients are active users of their devices and perform very well after implantation. There were no major complications in this small group of patients.
Children with congenital sensorineural hearing loss without other comorbidities (e.g., developmental delay, inner ear malformations) perform well when they receive cochlear implantation and auditory habilitation. Patients with Waardenburg syndrome can be expected to have above-average performance after cochlear implantation.
瓦登伯革氏综合征是一种常染色体显性综合征,其特征为内眦异位、眉毛增生、虹膜异色、前额白发,以及20%至55%的患者出现感音神经性听力损失。这一患者群体约占先天性耳聋儿童的2%。本回顾性病例分析的目的是描述接受人工耳蜗植入的瓦登伯革氏综合征患儿的治疗效果。
对有记录证明患有瓦登伯革氏综合征的儿科人工耳蜗植入受者进行回顾性病例分析。所有患者均在研究机构接受人工耳蜗植入,随后进行门诊听觉康复训练。查阅病历以了解耳聋的病因和持续时间、人工耳蜗植入时的年龄、围手术期并发症、使用时间和治疗效果。回顾了作为患者听觉康复训练一部分进行的语音感知和发音标准测试电池的结果。
确定了7名患有瓦登伯革氏综合征并接受人工耳蜗植入的患者。植入时的平均年龄为37个月(范围18 - 64个月),平均使用时间为69个月(范围12 - 143个月)。所有这些患者均积极使用其设备,植入后表现良好。这一小群患者中没有出现重大并发症。
没有其他合并症(如发育迟缓、内耳畸形)的先天性感音神经性听力损失儿童在接受人工耳蜗植入和听觉康复训练后表现良好。预计患有瓦登伯革氏综合征的患者在人工耳蜗植入后会有高于平均水平的表现。