迈向以患者为中心的遗传病严重程度分类。

IF 3.7 2区 生物学 Q2 BIOCHEMISTRY & MOLECULAR BIOLOGY
Mojca Cizek Sajko, Lovro Vidmar, Bernarda Prosenc, Brigita Grum, Gordana Njenjic, Borut Peterlin
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引用次数: 0

摘要

疾病严重程度的概念在许多临床情况下都会遇到,尤其是遗传性疾病。然而,仍然没有定义严重性的标准化方法。本研究旨在建立一种以患者为中心的方法,将遗传疾病特征划分为严重程度组,重点关注准父母的观点。此外,我们对基于准父母观点的分类和基于医疗专业人员的分类进行了比较分析。对参加产前课程的320名孕妇及其伴侣进行了问卷调查。这项调查收集了28种疾病特征的评分,反映了它们的严重程度。利用这些评级,我们构建了两个采用聚集层次聚类的统计模型。对我们的队列来说,最重要的疾病特征是寿命缩短,严重的智力残疾,以及从童年或青春期开始严重的行动能力受损。我们的三层准父母模型揭示了疾病特征的严重程度与准父母关于终止妊娠的决定之间的强烈关联。平均而言,89%的父母会因为Tier 1特征选择终止妊娠,55%的父母选择Tier 2特征,20%的父母选择Tier 3特征。我们发现我们的模型与基于医疗保健专业人员的分类相对较好地一致,主要区别在于,与专业人员相比,准父母认为寿命缩短比智力残疾更重要。通过这项研究,我们进一步了解了准父母在评估遗传疾病严重程度时认为必不可少的核心因素。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Towards a patient-centred classification of genetic disease severity.

The concept of disease severity is encountered in numerous clinical situations, especially with genetic disorders. However, there is still no standardized method for defining severity. This study aimed to establish a patient-centred approach for classifying genetic disease characteristics into severity groups, with a focus on the perspectives of expectant parents. Additionally, we conducted a comparative analysis between classifications based on expectant parents' perspectives and those based on healthcare professionals. A questionnaire was administered to 320 expectant women and their partners participating in prenatal classes. The survey gathered ratings for 28 disease characteristics, reflecting their perceived levels of severity. Using these ratings, we constructed two statistical models employing agglomerative hierarchical clustering. The disease characteristics that were most important to our cohort were a shortened lifespan, severe intellectual disability, and severe impaired mobility from childhood or adolescence onwards. Our three-tier expectant parent model revealed a strong association between the severity of disease characteristics and expectant parents' decisions regarding pregnancy termination. On average, 89% of parents would choose to terminate a pregnancy for Tier 1 characteristics, 55% for Tier 2, and 20% for Tier 3. We found a relatively good alignment of our model with the healthcare professional-based classification, with the primary difference being that expectant parents considered a shortened lifespan more important than intellectual disability, compared to professionals. Through this study, we have gained additional insight into the core factors that expectant parents consider essential when assessing the severity of a genetic disease.

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来源期刊
European Journal of Human Genetics
European Journal of Human Genetics 生物-生化与分子生物学
CiteScore
9.90
自引率
5.80%
发文量
216
审稿时长
2 months
期刊介绍: The European Journal of Human Genetics is the official journal of the European Society of Human Genetics, publishing high-quality, original research papers, short reports and reviews in the rapidly expanding field of human genetics and genomics. It covers molecular, clinical and cytogenetics, interfacing between advanced biomedical research and the clinician, and bridging the great diversity of facilities, resources and viewpoints in the genetics community. Key areas include: -Monogenic and multifactorial disorders -Development and malformation -Hereditary cancer -Medical Genomics -Gene mapping and functional studies -Genotype-phenotype correlations -Genetic variation and genome diversity -Statistical and computational genetics -Bioinformatics -Advances in diagnostics -Therapy and prevention -Animal models -Genetic services -Community genetics
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