Interdisciplinary management and genetic evaluation of pediatric cancer predisposition syndromes: a retrospective cohort study.

IF 4.6 2区 生物学 Q2 BIOCHEMISTRY & MOLECULAR BIOLOGY
Nicola Dikow, Anna Lisa Nitschke, Steffen Hirsch, Eva Roth, Laura Orec, Tom Fischer, Daniela Choukair, Kerstin Grund, Peggy Lüttich, Christian Sutter, Katrin Hinderhofer, Heiko Brennenstuhl, Cornelis M van Tilburg, Olaf Witt, Markus Großmüller, Alexander Fichtner, Katrin Willig, David T W Jones, Maja Hempel, Andreas E Kulozik, Ute Bartels, Till Milde, Christian P Schaaf, Stefan M Pfister, Joachim Kunz, Kristian W Pajtler
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Abstract

Comprehensive knowledge of cancer predisposition syndromes (CPS) is essential for the implementation of surveillance programs with proven clinical benefit. An interdisciplinary expert panel was established at our center to facilitate early identification of children with suspected CPS. This retrospective cohort study assessed the diagnostic yield and clinical impact of this consultation model. A total of 144 pediatric patients were evaluated. Clinical characteristics, family histories, and molecular diagnostic results were systematically retrieved from medical records. Of the 144 individuals, 99 presented with clinical features suggestive of CPS, of whom 53 had cancer. Clinically asymptomatic children (45/144) were referred due to positive family history. Prior to consultation, 34 of 99 symptomatic patients had been diagnosed with CPS. Among those symptomatic and undiagnosed undergoing molecular genetic testing, a disease-causing variant was identified in CPS genes in 46% (23/50). Predictive testing in asymptomatic children revealed the familial pathogenic variant in 43.8% (14/32). Nearly all patients with confirmed CPS 93,4% (71/76) received surveillance recommendations. The interdisciplinary CPS consultation model contributes to identifying hereditary cancer predisposition in pediatric patients and families. This expert-led approach emphasizes the importance of personalized surveillance strategies for rare CPS entities and provides a scalable foundation for systematic CPS assessment and development of standardized, evidence-based surveillance protocols in pediatric oncology.

儿童癌症易感性综合征的跨学科管理和遗传评估:一项回顾性队列研究。
全面了解癌症易感综合征(CPS)是必要的监测方案的实施已证实的临床效益。我们中心成立了一个跨学科的专家小组,以促进早期识别疑似CPS的儿童。这项回顾性队列研究评估了这种咨询模式的诊断率和临床效果。共有144名儿科患者被评估。临床特征、家族史和分子诊断结果系统地从医疗记录中检索。在144个人中,99人表现出提示CPS的临床特征,其中53人患有癌症。临床无症状患儿(45/144)因家族史阳性而转诊。在会诊前,99例有症状的患者中有34例被诊断为CPS。在接受分子基因检测的有症状和未确诊的患者中,46%(23/50)在CPS基因中发现了致病变异。无症状儿童的预测检测显示家族性致病变异为43.8%(14/32)。几乎所有确诊CPS的患者93.4%(71/76)接受了监测建议。跨学科CPS会诊模式有助于确定儿科患者和家庭的遗传性癌症易感性。这种专家主导的方法强调了针对罕见CPS实体的个性化监测策略的重要性,并为儿科肿瘤学中系统的CPS评估和标准化、循证监测方案的发展提供了可扩展的基础。
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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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