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
{"title":"Interdisciplinary management and genetic evaluation of pediatric cancer predisposition syndromes: a retrospective cohort study.","authors":"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","doi":"10.1038/s41431-026-02244-1","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":12016,"journal":{"name":"European Journal of Human Genetics","volume":" ","pages":""},"PeriodicalIF":4.6000,"publicationDate":"2026-09-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"European Journal of Human Genetics","FirstCategoryId":"99","ListUrlMain":"https://doi.org/10.1038/s41431-026-02244-1","RegionNum":2,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"BIOCHEMISTRY & MOLECULAR BIOLOGY","Score":null,"Total":0}
引用次数: 0
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.
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