000294425 001__ 294425 000294425 005__ 20241202094939.0 000294425 0247_ $$2doi$$a10.1158/1538-7445.AM2023-234 000294425 0247_ $$2ISSN$$a0099-7013 000294425 0247_ $$2ISSN$$a0099-7374 000294425 0247_ $$2ISSN$$a0008-5472 000294425 0247_ $$2ISSN$$a1538-7445 000294425 037__ $$aDKFZ-2024-02254 000294425 082__ $$a610 000294425 1001_ $$aGopisetty, Apurva$$b0 000294425 245__ $$aAbstract 234: ITCC-P4: Genomic profiling and analyses of pediatric patient tumor and patient-derived xenograft (PDX) models for high throughput in vivo testing 000294425 260__ $$c2023 000294425 3367_ $$0PUB:(DE-HGF)1$$2PUB:(DE-HGF)$$aAbstract$$babstract$$mabstract$$s1730992046_25079 000294425 3367_ $$033$$2EndNote$$aConference Paper 000294425 3367_ $$2BibTeX$$aINPROCEEDINGS 000294425 3367_ $$2DRIVER$$aconferenceObject 000294425 3367_ $$2DataCite$$aOutput Types/Conference Abstract 000294425 3367_ $$2ORCID$$aOTHER 000294425 520__ $$aAdvancements in state-of-the-art molecular profiling techniques have resulted in better understanding of pediatric cancers and driver events. It has become apparent that pediatric cancers are significantly more heterogeneous than previously thought as evidenced by the number of novel entities and subtypes that have been identified with distinct molecular and clinical characteristics. For most of these newly recognized entities there are extremely limited treatment options available. The ITCC-P4 consortium is an international collaboration between several European academic centers and pharmaceutical companies, with the overall aim to establish a sustainable platform of >400 molecularly well-characterized PDX models of high-risk pediatric cancers, their tumors and matching controls and to use the PDX models for in vivo testing of novel mechanism-of-action based treatments. Currently, 251 models are fully characterized, including 182 brain and 69 non-brain PDX models, representing 112 primary models, 92 relapse, 42 metastasis and 4 progressions under treatment models. Using low coverage whole-genome and whole exome sequencing, somatic mutation calling, DNA copy number and methylation analysis we aim to define genetic features in our PDX models and estimate the molecular fidelity of PDX models compared to their patient tumor. Based on DNA methylation profiling we identified 43 different tumor subgroups within 18 cancer entities. Mutational landscape analysis identified key somatic and germline oncogenic drivers. Ependymoma PDX models displayed the C11orf95-RELA fusion event, YAP1, C11orf95 and RELA structural variants. Medulloblastoma models were driven by MYCN, TP53, GLI2, SUFU and PTEN. High-grade glioma samples showed TP53, ATRX, MYCN and PIK3CA somatic SNVs, along with focal deletions in CDKN2A in chromosome 9. Neuroblastoma models were enriched for ALK SNVs and/or MYCN focal amplification, ATRX SNVs and CDKN2A/B deletions. Tumor mutational burden across entities and copy number analysis was performed to identify allele-specific copy number detection in tumor-normal pairs. Large chromosomal aberrations (deletions, duplications) detected in the PDX models were concurrent with molecular alterations frequently observed in each tumor type -isochromosome 17 was detected in 5 medulloblastoma models, while deletion of chromosome arm 1p or gain of parts of 17q in neuroblastomas which correlate with tumor progression. We observe clonal evolution of somatic variants not only in certain PDX-tumor pairs but also between disease states. The multi-omics approach in this study provides insight into the mutational landscape and patterns of the PDX models thus providing an overview of molecular mechanisms facilitating the identification and prioritization of oncogenic drivers and potential biomarkers for optimal treatment therapies. 000294425 588__ $$aDataset connected to CrossRef, Journals: inrepo02.dkfz.de 000294425 7001_ $$aFederico, Aniello$$b1 000294425 7001_ $$aSurdez, Didier$$b2 000294425 7001_ $$aIddir, Yasmine$$b3 000294425 7001_ $$aZaidi, Sakina$$b4 000294425 7001_ $$aSaint-Charles, Alexandra$$b5 000294425 7001_ $$aWaterfall, Joshua$$b6 000294425 7001_ $$aSaberi-Ansari, Elnaz$$b7 000294425 7001_ $$aWierzbinska, Justyna$$b8 000294425 7001_ $$aSchlicker, Andreas$$b9 000294425 7001_ $$aMack, Norman$$b10 000294425 7001_ $$aSchwalm, Benjamin$$b11 000294425 7001_ $$aPreviti, Christopher$$b12 000294425 7001_ $$aWeiser, Lena$$b13 000294425 7001_ $$aBuchhalter, Ivo$$b14 000294425 7001_ $$aBöttcher, Anna-Lisa$$b15 000294425 7001_ $$aSill, Martin$$b16 000294425 7001_ $$aAutry, Robert$$b17 000294425 7001_ $$aEstermann, Frank$$b18 000294425 7001_ $$aJones, David$$b19 000294425 7001_ $$aVolckmann, Richard$$b20 000294425 7001_ $$aZwijnenburg, Danny$$b21 000294425 7001_ $$aEggert, Angelika$$b22 000294425 7001_ $$aHeidenreich, Olaf$$b23 000294425 7001_ $$aIradier, Fatima$$b24 000294425 7001_ $$aJeremias, Irmela$$b25 000294425 7001_ $$aKovar, Heinrich$$b26 000294425 7001_ $$aKlusmann, Jan-Henning$$b27 000294425 7001_ $$aDebatin, Klaus-Michael$$b28 000294425 7001_ $$aBomken, Simon$$b29 000294425 7001_ $$aHamerlik, Petra$$b30 000294425 7001_ $$aHattersley, Maureen$$b31 000294425 7001_ $$aWitt, Olaf$$b32 000294425 7001_ $$aChesler, Louis$$b33 000294425 7001_ $$aMackay, Alan$$b34 000294425 7001_ $$aGojo, Johannes$$b35 000294425 7001_ $$aCairo, Stefano$$b36 000294425 7001_ $$aSchueler, Julia$$b37 000294425 7001_ $$aSchulte, Johannes$$b38 000294425 7001_ $$aGeoerger, Birgit$$b39 000294425 7001_ $$aMolenaar, Jan J.$$b40 000294425 7001_ $$aShields, David J.$$b41 000294425 7001_ $$aCaron, Hubert N.$$b42 000294425 7001_ $$aVassal, Gilles$$b43 000294425 7001_ $$aStancato, Louis F.$$b44 000294425 7001_ $$aPfister, Stefan M.$$b45 000294425 7001_ $$aJaeger, Natalie$$b46 000294425 7001_ $$aKoster, Jan$$b47 000294425 7001_ $$aKool, Marcel$$b48 000294425 7001_ $$aSchleiermacher, Gudrun$$b49 000294425 773__ $$0PERI:(DE-600)2036785-5$$a10.1158/1538-7445.AM2023-234$$gVol. 83, no. 7_Supplement, p. 234 - 234$$x1538-7445$$y2023 000294425 8564_ $$uhttps://inrepo02.dkfz.de/record/294425/files/Abstract%20234_Apurva%20Gopisetty.pdf 000294425 8564_ $$uhttps://inrepo02.dkfz.de/record/294425/files/Abstract%20234_Apurva%20Gopisetty.pdf?subformat=pdfa$$xpdfa 000294425 915__ $$0StatID:(DE-HGF)0200$$2StatID$$aDBCoverage$$bSCOPUS$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0300$$2StatID$$aDBCoverage$$bMedline$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0199$$2StatID$$aDBCoverage$$bClarivate Analytics Master Journal List$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)1050$$2StatID$$aDBCoverage$$bBIOSIS Previews$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0113$$2StatID$$aWoS$$bScience Citation Index Expanded$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0150$$2StatID$$aDBCoverage$$bWeb of Science Core Collection$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)1030$$2StatID$$aDBCoverage$$bCurrent Contents - Life Sciences$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)1190$$2StatID$$aDBCoverage$$bBiological Abstracts$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0160$$2StatID$$aDBCoverage$$bEssential Science Indicators$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)1110$$2StatID$$aDBCoverage$$bCurrent Contents - Clinical Medicine$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0100$$2StatID$$aJCR$$bCANCER RES : 2022$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0600$$2StatID$$aDBCoverage$$bEbsco Academic Search$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)0030$$2StatID$$aPeer Review$$bASC$$d2023-10-24 000294425 915__ $$0StatID:(DE-HGF)9910$$2StatID$$aIF >= 10$$bCANCER RES : 2022$$d2023-10-24 000294425 9801_ $$aEXTERN4VITA 000294425 980__ $$aabstract 000294425 980__ $$aEDITORS 000294425 980__ $$aI:(DE-He78)W510-20160331 000294425 980__ $$aI:(DE-He78)W500-20160331