Rule Payton D, Wolk Megan W, Hill Patrick L
Department of Psychological and Brain Sciences, Washington University in St. Louis, 1 Brookings Dr., St. Louis, MO 63130, United States.
Health Care Transit. 2024 Feb 28;2:100053. doi: 10.1016/j.hctj.2024.100053. eCollection 2024.
Wellbeing research among individuals with Charcot-Marie-Tooth disease (CMT) is limited. The goal of the current study is to characterize the challenges experienced by adults with CMT that researchers may miss by utilizing typical strategies to capture mental health and wellbeing.
In 2021, we recruited 288 US adults with CMT ( = 60 years, 59% Female, 93% White) to take part in an online survey. Participants were presented with validated wellbeing and health measures, in addition to newly created measures specific to CMT, and then asked to provide free-response feedback about aspects of their lived experiences missing from these measures. Thematic analysis were applied to the free-response data to identify CMT-related difficulties that may impact wellbeing among adults with CMT.
Results identified nine CMT-related challenges that are important to individuals with CMT and are often left out of wellbeing assessments in this population. The three most common themes identified were anxiety or worry, coping with loss of function or progression, and difficulty finding knowledgeable medical providers. Further, some aspects of the CMT experience may be comorbid.
These results can be used to inform future research and clinical practice in the CMT population.
关于夏科-马里-图思病（CMT）患者的健康状况研究有限。本研究的目的是描述成年CMT患者所经历的挑战，而这些挑战可能是研究人员采用典型策略来获取心理健康和幸福状况时所遗漏的。
2021年，我们招募了288名美国成年CMT患者（平均年龄 = 60岁，59%为女性，93%为白人）参与一项在线调查。除了针对CMT的新创建测量方法外，还向参与者展示了经过验证的健康和幸福测量方法，然后要求他们就这些测量方法中未涵盖的生活经历方面提供自由回答反馈。对自由回答数据进行主题分析，以确定可能影响成年CMT患者健康状况的与CMT相关的困难。
结果确定了九个与CMT相关的挑战，这些挑战对CMT患者很重要，并且在该人群的健康评估中经常被遗漏。确定的三个最常见主题是焦虑或担忧、应对功能丧失或病情进展以及难以找到知识渊博的医疗服务提供者。此外，CMT经历的某些方面可能存在共病情况。
这些结果可用于为CMT人群未来的研究和临床实践提供参考。
