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dc.contributor.authorBallinger, ML
dc.contributor.authorBest, A
dc.contributor.authorMai, PL
dc.contributor.authorKhincha, PP
dc.contributor.authorLoud, JT
dc.contributor.authorPeters, JA
dc.contributor.authorAchatz, MI
dc.contributor.authorChojniak, R
dc.contributor.authorBalieiro da Costa, A
dc.contributor.authorSantiago, KM
dc.contributor.authorGarber, J
dc.contributor.authorO'Neill, AF
dc.contributor.authorEeles, RA
dc.contributor.authorEvans, DG
dc.contributor.authorBleiker, E
dc.contributor.authorSonke, GS
dc.contributor.authorRuijs, M
dc.contributor.authorLoo, C
dc.contributor.authorSchiffman, J
dc.contributor.authorNaumer, A
dc.contributor.authorKohlmann, W
dc.contributor.authorStrong, LC
dc.contributor.authorBojadzieva, J
dc.contributor.authorMalkin, D
dc.contributor.authorRednam, SP
dc.contributor.authorStoffel, EM
dc.contributor.authorKoeppe, E
dc.contributor.authorWeitzel, JN
dc.contributor.authorSlavin, TP
dc.contributor.authorNehoray, B
dc.contributor.authorRobson, M
dc.contributor.authorWalsh, M
dc.contributor.authorManelli, L
dc.contributor.authorVillani, A
dc.contributor.authorThomas, DM
dc.contributor.authorSavage, SA
dc.date.accessioned2017-11-17T09:43:58Z
dc.date.issued2017-12
dc.identifier.citationJAMA oncology, 2017, 3 (12), pp. 1634 - 1639
dc.identifier.issn2374-2437
dc.identifier.urihttps://repository.icr.ac.uk/handle/internal/909
dc.identifier.eissn2374-2445
dc.identifier.doi10.1001/jamaoncol.2017.1968
dc.description.abstractImportance Guidelines for clinical management in Li-Fraumeni syndrome, a multiple-organ cancer predisposition condition, are limited. Whole-body magnetic resonance imaging (WBMRI) may play a role in surveillance of this high-risk population.Objective To assess the clinical utility of WBMRI in germline TP53 mutation carriers at baseline.Data sources Clinical and research surveillance cohorts were identified through the Li-Fraumeni Exploration Research Consortium.Study selection Cohorts that incorporated WBMRI for individuals with germline TP53 mutations from January 1, 2004, through October 1, 2016, were included.Data extraction and synthesis Data were extracted by investigators from each cohort independently and synthesized by 2 investigators. Random-effects meta-analysis methods were used to estimate proportions.Main outcomes and measures The proportions of participants at baseline in whom a lesion was detected that required follow-up and in whom a new primary malignant neoplasm was detected.Results A total of 578 participants (376 female [65.1%] and 202 male [34.9%]; mean [SD] age, 33.2 [17.1] years) from 13 cohorts in 6 countries were included in the analysis. Two hundred twenty-five lesions requiring clinical follow-up were detected by WBMRI in 173 participants. Sixty-one lesions were diagnosed in 54 individuals as benign or malignant neoplasms. Overall, 42 cancers were identified in 39 individuals, with 35 new localized cancers treated with curative intent. The overall estimated detection rate for new, localized primary cancers was 7% (95% CI, 5%-9%).Conclusions and relevance These data suggest clinical utility of baseline WBMRI in TP53 germline mutation carriers and may form an integral part of baseline clinical risk management in this high-risk population.
dc.formatPrint
dc.format.extent1634 - 1639
dc.languageeng
dc.language.isoeng
dc.rights.urihttps://www.rioxx.net/licenses/under-embargo-all-rights-reserved
dc.subjectHumans
dc.subjectLi-Fraumeni Syndrome
dc.subjectDiagnosis, Differential
dc.subjectMagnetic Resonance Imaging
dc.subjectPopulation Surveillance
dc.subjectMutation
dc.subjectAdolescent
dc.subjectAdult
dc.subjectMiddle Aged
dc.subjectFemale
dc.subjectMale
dc.subjectTumor Suppressor Protein p53
dc.subjectWhole Body Imaging
dc.subjectPractice Guidelines as Topic
dc.subjectEarly Detection of Cancer
dc.subjectYoung Adult
dc.titleBaseline Surveillance in Li-Fraumeni Syndrome Using Whole-Body Magnetic Resonance Imaging: A Meta-analysis.
dc.typeJournal Article
dcterms.dateAccepted2017-05-15
rioxxterms.funderThe Institute of Cancer Research
rioxxterms.identifier.projectUnspecified
rioxxterms.versionofrecord10.1001/jamaoncol.2017.1968
rioxxterms.licenseref.urihttps://www.rioxx.net/licenses/under-embargo-all-rights-reserved
rioxxterms.licenseref.startdate2017-12
rioxxterms.typeJournal Article/Review
dc.relation.isPartOfJAMA oncology
pubs.declined2017-11-01T12:14:18.186+0000
pubs.issue12
pubs.notesNot known
pubs.organisational-group/ICR
pubs.organisational-group/ICR/Primary Group
pubs.organisational-group/ICR/Primary Group/ICR Divisions
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Genetics and Epidemiology
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Genetics and Epidemiology/Oncogenetics
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging/Oncogenetics
pubs.organisational-group/ICR/Primary Group/Royal Marsden Clinical Units
pubs.organisational-group/ICR
pubs.organisational-group/ICR/Primary Group
pubs.organisational-group/ICR/Primary Group/ICR Divisions
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Genetics and Epidemiology
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Genetics and Epidemiology/Oncogenetics
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging
pubs.organisational-group/ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging/Oncogenetics
pubs.organisational-group/ICR/Primary Group/Royal Marsden Clinical Units
pubs.publication-statusPublished
pubs.volume3
pubs.embargo.termsNot known
icr.researchteamOncogeneticsen_US
dc.contributor.icrauthorEeles, Rosalinden
dc.contributor.icrauthorMarsden,en


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