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澳大利亚遗传性视网膜疾病患者对眼部基因治疗的看法调查。

Survey of perspectives of people with inherited retinal diseases on ocular gene therapy in Australia.

机构信息

Ophthalmology, Department of Surgery, University of Melbourne, Melbourne, VIC, Australia.

Centre for Eye Research Australia, Royal Victorian Eye and Ear Hospital, Melbourne, VIC, Australia.

出版信息

Gene Ther. 2023 Apr;30(3-4):336-346. doi: 10.1038/s41434-022-00364-z. Epub 2022 Oct 2.

DOI:10.1038/s41434-022-00364-z
PMID:36183012
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10113139/
Abstract

Many gene therapies are in development for treating people with inherited retinal diseases (IRD). We hypothesized that potential recipients of gene therapy would have knowledge gaps regarding treatment. We aimed to assess knowledge, attitudes, and perceptions of genetic therapies among potential recipients with IRD, using a novel instrument we designed (Attitudes to Gene Therapy-Eye (AGT-Eye)) and their associations with demographic data, self-reported visual status, and tools assessing quality of life and attitudes toward clinical trials using a community-based cross-sectional survey of Australian adults with IRD. AGT-Eye, overall quality of life EQ-5D-5L, National Eye Institute Visual Functioning Questionnaire (NEI-VFQ-25) and Patient Attitudes to Clinical Trials (PACT-22) instruments were administered. Six hundred and eighty-one people completed the study, 51.7% women of mean age 53.5 years (SD ± 15.8). Most participants (91.6%) indicated they would likely accept gene therapy if it was available to them or family members. However, only 28.3% agreed that they had good knowledge of gene therapy. Most obtained information about gene therapy from the internet (49.3%). Respondents with post-graduate degrees scored highest compared to other educational levels on methods (p < 0.001) and outcomes (p = 0.003) and were more likely to see economic value of treatment (p = 0.043). Knowledge gaps were present regarding methods and outcomes of gene therapy. This survey has shown high level of interest in the IRD community for gene therapies, and highlights areas for improved clinician and patient education.

摘要

许多基因疗法正在开发中,用于治疗遗传性视网膜疾病(IRD)患者。我们假设基因治疗的潜在接受者在治疗方面存在知识差距。我们旨在使用我们设计的一种新工具(Attitudes to Gene Therapy-Eye(AGT-Eye))评估潜在的 IRD 患者对基因治疗的知识、态度和看法,并评估这些知识、态度和看法与人口统计学数据、自我报告的视觉状况以及评估生活质量和对临床试验态度的工具之间的关联,方法是对澳大利亚患有 IRD 的成年人进行基于社区的横断面调查。评估了 AGT-Eye、整体生活质量 EQ-5D-5L、美国国立眼科研究所视觉功能问卷(NEI-VFQ-25)和患者对临床试验的态度问卷(PACT-22)。681 人完成了这项研究,女性占 51.7%,平均年龄为 53.5 岁(标准差±15.8)。大多数参与者(91.6%)表示,如果基因治疗对他们或他们的家庭成员可用,他们很可能会接受。然而,只有 28.3%的人表示他们对基因治疗有很好的了解。大多数人通过互联网(49.3%)获取有关基因治疗的信息。与其他教育水平相比,拥有研究生学历的受访者在方法(p<0.001)和结果(p=0.003)方面得分最高,并且更有可能看到治疗的经济价值(p=0.043)。在基因治疗的方法和结果方面存在知识差距。这项调查表明,IRD 社区对基因治疗有很高的兴趣,并强调了需要改善临床医生和患者教育的领域。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/7ab3ce50acde/41434_2022_364_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/cad028e57f87/41434_2022_364_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/20b4acba55de/41434_2022_364_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/47764e935045/41434_2022_364_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/7ab3ce50acde/41434_2022_364_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/cad028e57f87/41434_2022_364_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/20b4acba55de/41434_2022_364_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/47764e935045/41434_2022_364_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3bfe/10113139/7ab3ce50acde/41434_2022_364_Fig4_HTML.jpg

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