Lai Jin-Shei, Jensen Sally E, Patel Zabin S, Listernick Robert, Charrow Joel
Department of Medical Social Sciences, Northwestern University Feinberg School of Medicine, Chicago, Illinios.
Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, Illinios.
Am J Med Genet A. 2017 Jan;173(1):79-87. doi: 10.1002/ajmg.a.37987. Epub 2016 Sep 26.
Neurofibromatosis Type 1 (NF1) plexiform neurofibromas (pNFs) are associated with a variety of symptoms and concerns that affect patients' quality of life (QOL), highlighting the value of incorporating the patients' perspective when evaluating treatment outcomes. To better conceptualize the experience of patients with pNFs, this qualitative study sought to identify the most important outcomes to assess from the perspective of patients, families, and clinicians. Clinicians, patients age 5 years old and above, and parents of patients aged 5-17 years participated in semi-structured interviews to elicit the pNF symptoms/concerns considered most important to assess. The data were analyzed using an iterative coding procedure and the frequency with which symptoms/concerns emerged was tabulated. Eight clinicians, 31 patients, and 17 parents of patients participated in semi-structured interviews. The most frequently reported concerns raised by patients across all age groups included pain, appearance/disfigurement, social activity/role participation, stigma, and anxiety. For parents, physical functioning was the primary concern, followed by pain, social activity/role participation, appearance/disfigurement, and social relationships. The resulting conceptual framework included five domains to represent the most important identified symptoms/concerns: pain, social functioning, physical function impact, stigma, and emotional distress. This conceptual framework describing the symptoms/concerns of patients with pNF can help investigators create a measurement system to improve assessment of aspects of QOL only patients can report on. It may also provide the ability to identify symptoms/concerns that might warrant referrals to various clinical disciplines. © 2016 Wiley Periodicals, Inc.
1型神经纤维瘤病(NF1)丛状神经纤维瘤(pNFs)与多种影响患者生活质量(QOL)的症状和问题相关,这凸显了在评估治疗效果时纳入患者观点的价值。为了更好地理解pNFs患者的经历,这项定性研究旨在从患者、家属和临床医生的角度确定最重要的评估结果。临床医生、5岁及以上的患者以及5 - 17岁患者的父母参与了半结构化访谈,以引出被认为对评估最重要的pNF症状/问题。使用迭代编码程序对数据进行分析,并将症状/问题出现频率制成表格。8名临床医生、31名患者和17名患者父母参与了半结构化访谈。所有年龄组患者最常报告的问题包括疼痛、外观/毁容、社交活动/角色参与、耻辱感和焦虑。对父母来说,身体功能是主要关注点,其次是疼痛、社交活动/角色参与、外观/毁容和社会关系。由此产生的概念框架包括五个领域,以代表确定的最重要症状/问题:疼痛、社会功能、身体功能影响、耻辱感和情绪困扰。这个描述pNF患者症状/问题的概念框架可以帮助研究人员创建一个测量系统,以改进对只有患者才能报告的生活质量方面的评估。它还可能提供识别可能需要转诊到各个临床学科的症状/问题的能力。© 2016威利期刊公司