{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T20:00:13Z","timestamp":1787169613589,"version":"build-2736575974"},"reference-count":45,"publisher":"Wiley","issue":"22","license":[{"start":{"date-parts":[[2023,6,29]],"date-time":"2023-06-29T00:00:00Z","timestamp":1687996800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["acsjournals.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cancer"],"published-print":{"date-parts":[[2023,11,15]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Germline genome sequencing in childhood cancer precision medicine trials may reveal pathogenic or likely pathogenic variants in cancer predisposition genes in more than 10% of children. These findings can have implications for diagnosis, treatment, and the child\u2019s and family\u2019s future cancer risk. Understanding parents\u2019 perspectives of germline genome sequencing is critical to successful clinical implementation.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>A total of 182 parents of 144 children (&lt;18 years of age) with poor\u2010prognosis cancers enrolled in the Precision Medicine for Children with Cancer trial completed a questionnaire at enrollment and after the return of their child\u2019s results, including clinically relevant germline findings (received by 13% of parents). Parents\u2019 expectations of germline genome sequencing, return of results preferences, and recall of results received were assessed. Forty\u2010five parents (of 43 children) were interviewed in depth.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>At trial enrollment, most parents (63%) believed it was at least \u201csomewhat likely\u201d that their child would receive a clinically relevant germline finding. Almost all expressed a preference to receive a broad range of germline genomic findings, including variants of uncertain significance (88%). Some (29%) inaccurately recalled receiving a clinically relevant germline finding. Qualitatively, parents expressed confusion and uncertainty after the return of their child\u2019s genome sequencing results by their child\u2019s clinician.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Many parents of children with poor\u2010prognosis childhood cancer enrolled in a precision medicine trial expect their child may have an underlying cancer predisposition syndrome. They wish to receive a wide scope of information from germline genome sequencing but may feel confused by the reporting of trial results.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/cncr.34917","type":"journal-article","created":{"date-parts":[[2023,6,29]],"date-time":"2023-06-29T06:20:22Z","timestamp":1688019622000},"page":"3620-3632","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Parents\u2019 expectations, preferences, and recall of germline findings in a childhood cancer precision medicine trial"],"prefix":"10.1002","volume":"129","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2134-2988","authenticated-orcid":false,"given":"Brittany C.","family":"McGill","sequence":"first","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claire E.","family":"Wakefield","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katherine M.","family":"Tucker","sequence":"additional","affiliation":[{"name":"Hereditary Cancer Centre Prince of Wales Hospital  Sydney New South Wales Australia"},{"name":"Prince of Wales Clinical School UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rebecca A.","family":"Daly","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark W.","family":"Donoghoe","sequence":"additional","affiliation":[{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"},{"name":"Stats Central UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janine","family":"Vetsch","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meera","family":"Warby","sequence":"additional","affiliation":[{"name":"Hereditary Cancer Centre Prince of Wales Hospital  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Noemi A.","family":"Fuentes\u2010Bolanos","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristine","family":"Barlow\u2010Stewart","sequence":"additional","affiliation":[{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"},{"name":"Faculty of Medicine and Health Royal North Shore Hospital  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judy","family":"Kirk","sequence":"additional","affiliation":[{"name":"Familial Cancer Service Westmead Hospital  Sydney New South Wales Australia"},{"name":"Sydney Medical School Westmead Institute for Medical Research  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eliza","family":"Courtney","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tracey A.","family":"O\u2019Brien","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Glenn M.","family":"Marshall","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark","family":"Pinese","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark J.","family":"Cowley","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vanessa","family":"Tyrrell","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rebecca J.","family":"Deyell","sequence":"additional","affiliation":[{"name":"Division of Paediatric Hematology\/Oncology\/Bone Marrow Transplantation British Columbia Children\u2019s Hospital and Research Institute  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David S.","family":"Ziegler","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"},{"name":"Children\u2019s Cancer Institute UNSW Sydney  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kate","family":"Hetherington","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales (UNSW) Sydney  Sydney New South Wales Australia"},{"name":"Kids Cancer Centre Sydney Children\u2019s Hospital  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,6,29]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41591-020-1072-4"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1158\/1078-0432.ccr-15-2717"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1158\/1078-0432.ccr-17-0381"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamanetworkopen.2019.2906"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2016.06.009"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1200\/jco.2019.37.15_suppl.10011"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.3390\/jpm8030030"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1056\/nejmoa1508054"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature25480"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1038\/s43018-021-00172-1"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1158\/2159-8290.cd-20-1631"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1158\/1078-0432.ccr-17-0834"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoncol.2021.4536"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoncol.2015.5699"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.trecan.2019.11.006"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00431-017-2997-6"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/ecc.12877"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pec.2018.11.007"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.3390\/children8040259"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.25520"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.26137"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1200\/po.19.00235"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.29908"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.3390\/jpm10010009"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1093\/jnci\/djj005"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10552-007-9108-3"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.2015.10080"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2013.201"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1136\/medethics-2013-101648"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.1159\/000439244"},{"key":"e_1_2_8_32_1","doi-asserted-by":"publisher","DOI":"10.1093\/jlb\/lsx038"},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12687-019-00434-7"},{"key":"e_1_2_8_34_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.32071"},{"key":"e_1_2_8_35_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.30581"},{"key":"e_1_2_8_36_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2014.134"},{"key":"e_1_2_8_37_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13073-014-0069-3"},{"key":"e_1_2_8_38_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmjopen-2019-034522"},{"key":"e_1_2_8_39_1","doi-asserted-by":"publisher","DOI":"10.1177\/1558689806298224"},{"key":"e_1_2_8_40_1","doi-asserted-by":"publisher","DOI":"10.1191\/1478088706qp063oa"},{"key":"e_1_2_8_41_1","doi-asserted-by":"publisher","DOI":"10.1177\/1359105308095063"},{"key":"e_1_2_8_42_1","doi-asserted-by":"publisher","DOI":"10.1080\/23294515.2022.2063994"},{"key":"e_1_2_8_43_1","doi-asserted-by":"publisher","DOI":"10.3390\/jpm12081327"},{"key":"e_1_2_8_44_1","doi-asserted-by":"publisher","DOI":"10.1200\/po.17.00104"},{"key":"e_1_2_8_45_1","doi-asserted-by":"publisher","DOI":"10.1200\/po.19.00213"},{"key":"e_1_2_8_46_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10689-017-9988-7"}],"container-title":["Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cncr.34917","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,12]],"date-time":"2025-10-12T12:52:10Z","timestamp":1760273530000},"score":1,"resource":{"primary":{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/10.1002\/cncr.34917"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,6,29]]},"references-count":45,"journal-issue":{"issue":"22","published-print":{"date-parts":[[2023,11,15]]}},"alternative-id":["10.1002\/cncr.34917"],"URL":"https:\/\/doi.org\/10.1002\/cncr.34917","archive":["Portico"],"relation":{},"ISSN":["0008-543X","1097-0142"],"issn-type":[{"value":"0008-543X","type":"print"},{"value":"1097-0142","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,6,29]]},"assertion":[{"value":"2022-08-29","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-05-15","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-06-29","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}