“治愈我们的方法有很多”：普拉德-威利综合征患者如何看待自己和未来的临床试验。
'The cure for us is a lot of things': How young people with Prader-Willi syndrome view themselves and future clinical trials.
机构信息
Department of Psychology and Human Development, Vanderbilt Kennedy Center, Vanderbilt University, Nashville, Tennessee, USA.
出版信息
J Appl Res Intellect Disabil. 2022 Mar;35(2):460-470. doi: 10.1111/jar.12950. Epub 2021 Dec 13.
BACKGROUND
Despite work on the self-identities of people with intellectual disabilities, research has yet to describe the self-perceptions of people with Prader-Willi syndrome (PWS). The perspectives of those with PWS are also important for rapidly evolving clinical trials aimed at treating symptoms of PWS.
METHOD
Twenty-one young people with PWS were administered a semi-structured interview that assessed how they perceive their syndrome and clinical trials. Transcribed interviews were reliably coded using content-driven, applied thematic analyses.
RESULTS
Five themes emerged: struggles with chronic hunger and food-seeking that impede goals and relationships; struggles with anxiety and outbursts, schedule changes and school; distancing from PWS; needs for clinical trials that cure PWS, reduce hunger or anxiety, and lead to improved outcomes; and needs for advocacy and awareness of PWS.
CONCLUSIONS
Findings shed new light on the self-perceptions of those with PWS and have important implications for current interventions and future clinical trials.
背景
尽管人们对智力障碍者的自我认同进行了研究，但仍未描述普拉德-威利综合征（PWS）患者的自我认知。对于旨在治疗 PWS 症状的快速发展的临床试验而言，了解 PWS 患者的观点也很重要。
方法
对 21 名患有 PWS 的年轻人进行了半结构化访谈，评估了他们对自己的综合征和临床试验的看法。通过内容驱动的应用主题分析，对转录的访谈进行了可靠的编码。
结果
出现了五个主题：与慢性饥饿和觅食行为的斗争，这些行为会阻碍目标和人际关系；与焦虑和发作、日程变化和学校的斗争；与 PWS 的距离；对治愈 PWS、减少饥饿或焦虑以及改善结果的临床试验的需求；以及对 PWS 的宣传和意识的需求。
结论
研究结果为 PWS 患者的自我认知提供了新的视角，并对当前的干预措施和未来的临床试验具有重要意义。
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