Development of HRQOL Instruments for Children with CIs and their Parents

 描述(由申请人提供)：严重到严重的听力损失与健康相关生活质量(HRQOL)的可测量缺陷有关，反映出其对语言学习、社交和情感功能以及学习成绩的伴随影响。然而，人工耳蜗术的研究通常集中在与沟通发展(如听觉、言语、语言技能)相关的疗效的临床测量上，这并不能代表干预对日常功能的影响。HRQOL测量提供了对疾病及其治疗对身体、社会和情绪功能的影响的关键评估。到目前为止，还没有针对幼儿、青少年及其父母的针对CI的HRQOL测量(Morettin等人，2013年)。本研究的目的是为儿童人工耳蜗植入者及其父母开发首个HRQL量表。我们计划完成工具开发的前两个阶段：1)儿童出生到5岁的父母，2)6-12岁的学龄儿童，加上父母-代理测量和3)13-18岁顺式儿童的青少年。仪器开发将遵循FDA关于患者报告结果的指南(FDA，2009)。第一阶段包括综合和整合现有文献和以前针对这一人群的HRQOL工具，以及国家传播和信息中心的数据收集，以确保地理和种族/族裔多样性，并包括：1)整合以前的文献和工具，以 建立可测试的概念框架，2)与利益相关者(外科医生、听力专家、语言病理学家)组成焦点小组，3)对出生至18岁儿童的父母、6至18岁儿童的父母进行开放式访谈，以及4)对4个草案工具的认知测试。八个一小时的焦点小组将与利益相关者一起进行，如CI外科医生、儿科听力专家、听觉语言治疗师和社区提供者。接下来，将对110名家长和儿童进行开放式访谈并录制录音带，以确定耳聋和CI使用对几个功能领域的影响。将对所有焦点小组和不限成员名额访谈进行转录和内容分析，以确定关键主题并量化其获得认可的频率。然后，将根据特定内容的饱和矩阵生成项目，从而产生所有四种QOL-CI措施的草案版本。最后，将对96名家长和CI使用者(6至18岁)进行认知访谈，采用标准化的“大声思考”程序。认知访谈技术现在被广泛用于从受访者那里获得关于项目和回答量表的清晰度和全面性的信息。该项目的完成将产生最终的工具草案，将在国家心理测量学验证研究中进行实地测试，以评估可靠性、有效性和对变化的响应性。将使用电子格式开发文书，以提高其实用性和可解释性。这些工具将有助于评估CI OVER TIE的有效性，为其他干预措施(例如，听觉语言治疗、行为干预)提供以患者为中心的结果衡量标准，并为目标干预提供患者/家庭层面的信息。

 DESCRIPTION (provided by applicant): Severe to profound hearing loss is associated with measurable deficits in health-related quality of life (HRQoL), reflecting its concomitant effects o language learning, social and emotional functioning, and academic performance. However, studies of cochlear implantation (CI) typically focus on clinical measures of efficacy related to communication development (e.g., auditory, speech, language skills), which does not represent the intervention's effects on daily functioning. HRQoL measures provide a crucial assessment of the impact of the condition and its treatment on physical, social and emotional functioning. To date, there are no CI-specific HRQoL measures for young children, adolescents and their parents (Morettin et al., 2013). The purpose of this study is to develop the first HRQoL instruments for pediatric cochlear implant recipients and their parents. We plan to complete the first two phases of instrument development for: 1) parents of children birth to five years, 2) school-age children 6-12, plus a parent-proxy measure and 3) adolescents with CIs ages 13-18. Instrument development will adhere to the FDA Guidance on patient-reported outcomes (FDA, 2009). Phase I consists of a synthesis and integration of existing literature and prior HRQoL instruments for this population and data collection at national CI Centers to ensure geographic and racial/ethnic diversity, and includes: 1) an integration of prior literature and instruments to form a testable conceptual framework, 2) focus groups with stakeholders (surgeons, audiologists, speech pathologists), 3) open-ended interviews with parents of children birth to 18 years, children ages 6 to 18 years, and 4) cognitive testing of the 4 draft instruments. Eight one-hour focus groups will be conducted with stakeholders, such as CI surgeons, pediatric audiologists, auditory verbal therapists, and community providers. Next, open-ended interviews will be conducted and audiotaped with 110 parents and children to identify the effects of both deafness and CI use on several areas of functioning. All focus groups and open-ended interviews will be transcribed and content-analyzed to identify key themes and quantify the frequency of their endorsement. Items will then be generated based on saturation matrices of specific content, resulting in draft versions of all four QOL-CI measures. Lastly, cognitive interviews, using standardized "think aloud" procedures, will be performed with 96 parents and CI users (ages 6 to 18). Cognitive interviewing techniques are now widely used to gain information from respondents about the clarity and comprehensiveness of the items and response scales. Completion of this project will yield the final draft instruments to be field-testd in a national, psychometric validation study to assess reliability, validity, and responsivity to change. Instruments will be developed using an electronic format to enhance their utility and interpretability. These instruments will facilitate evaluation of the effectiveness of CI's over tie, provide patient-centered outcome measures for other interventions (e.g., auditory-verbal therapy, behavioral interventions), and provide information at the patient/family level for targete interventions.