Quitmann Julia, Rohenkohl Anja, Bullinger Monika, Chaplin John E, Herdman Michael, Sanz Dolores, Mimoun Emmanuelle, Feigerlova Eva, DeBusk Kendra, Power Michael, Wollmann Hartmut, Pleil Andreas
University Medical Center Hamburg-Eppendorf, Department of Medical Psychology, Martinistr. 52, 20246 Hamburg, Germany.
Sahlgrenska Academy at University of Gothenburg, Department of Paediatrics, Växthuset, Queen Silvia's Childrens Hospital, 41685 Gothenburg, Sweden.
Pediatr Endocrinol Rev. 2013 Dec;11(2):147-60.
Health-related quality of life (HrQoL) of the child diagnosed with short stature is an important outcome to be assessed both from the patient as well as from the parental perspective. The objective of this study was to review the literature on parent-reported HrQoL and to subsequently develop and psychometrically test the parent-reported version of the Quality of Life in Short Stature Youth (QoLISSY) instrument for use in clinical and epidemiologic research.
A review of the literature on parental assessment of child HrQoL via PUBMED was followed by a psychometric analysis of data collected within the European QoLISSY study, in which 686 eligible parents of short statured children/adolescents (aged 4-18 years) meeting inclusion criteria participated. Patient inclusion criteria were a height below -2 SD, a diagnosis of growth hormone deficiency (GHD) or idiopathic short stature (ISS), and treatment status in terms of receiving or not receiving recombinant human growth hormone therapy. Focus groups eliciting parental HrQoL statements, pilot testing with cognitive debriefing, and a field test in 317 parents with a retest in 148 parents were conducted simultaneously in France, Germany, Spain, Sweden and the UK. The psychometric performance of the parent-reported instrument, developed in parallel to the child/ adolescent self-report version, was assessed using standard tests of reliability and validity.
Literature search failed to identify a cross-culturally developed height specific instrument available for both patient self-report and parental observer report. Analysis of the QoLISSY focus group phase conducted separately in children, adolescents and parents yielded 169 items generated from parent focus groups. A cognitive debriefing exercise followed by a pilot test of preliminary psychometric characteristics resulted in deleting poorly performing items. Field testing of the parent-reported version suggested a three-domain core HrQoL structure with 22 items, additional 44 items assessing three mediator domains and two parent specific domains. The parent report version demonstrated good criterion and construct validity as well as internal consistency and test retest reliability.
The QoLISSY parent report questionnaire closes a gap in the simultaneous assessment of parent and child perception of HrQoL in an international context. It is based on items generated from the experience of short statured children, adolescents and their parents and is validated for use in five European languages. It is feasible, relevant for this population, psychometrically sound and is easy to administer in research and clinical settings.
从患者及其父母的角度评估被诊断为身材矮小儿童的健康相关生活质量（HrQoL）都是一项重要成果。本研究的目的是回顾关于父母报告的HrQoL的文献，并随后开发和进行心理测量学测试用于临床和流行病学研究的父母报告版身材矮小青少年生活质量（QoLISSY）工具。
通过PUBMED对关于父母对儿童HrQoL评估的文献进行回顾，随后对欧洲QoLISSY研究中收集的数据进行心理测量学分析，该研究中有686名符合纳入标准的身材矮小儿童/青少年（4至18岁）的合格父母参与。患者纳入标准为身高低于-2标准差、生长激素缺乏症（GHD）或特发性身材矮小（ISS）诊断以及接受或未接受重组人生长激素治疗的治疗状态。在法国、德国、西班牙、瑞典和英国同时进行了焦点小组访谈以引出父母关于HrQoL的陈述、进行有认知反馈的预测试以及对317名父母进行现场测试并对148名父母进行重新测试。与儿童/青少年自我报告版本并行开发的父母报告工具的心理测量学性能使用可靠性和有效性的标准测试进行评估。
文献检索未能找到一种跨文化开发的、适用于患者自我报告和父母观察者报告的身高特定工具。对分别在儿童、青少年和父母中进行的QoLISSY焦点小组阶段的分析产生了从父母焦点小组中生成的169个项目。在对初步心理测量学特征进行预测试并进行认知反馈后，删除了表现不佳的项目。父母报告版本的现场测试表明，HrQoL核心结构有三个领域，包含22个项目，另外44个项目评估三个中介领域和两个父母特定领域。父母报告版本显示出良好的标准效度和结构效度以及内部一致性和重测信度。
QoLISSY父母报告问卷填补了在国际背景下同时评估父母和儿童对HrQoL认知的空白。它基于身材矮小儿童、青少年及其父母的经历所产生的项目，并经过验证可用于五种欧洲语言。它可行、与该人群相关且心理测量学合理，并且在研究和临床环境中易于实施。
