Ten-year survival of children with trisomy 13 or trisomy 18: A multi-registry European cohort study
dc.contributor.author | Glinianaia, Svetlana V. | |
dc.contributor.author | Rankin, Judith | |
dc.contributor.author | Tan, Joachim | |
dc.contributor.author | Loane, Maria | |
dc.contributor.author | Garne, Ester | |
dc.contributor.author | Cavero-Carbonell, Clara | |
dc.contributor.author | De Walle, Hermien E. K. | |
dc.contributor.author | Gatt, Miriam | |
dc.contributor.author | Gissler, Mika | |
dc.contributor.author | Klungsøyr, Kari | |
dc.contributor.author | Lelong, Natalie | |
dc.contributor.author | Neville, Amanda | |
dc.contributor.author | Pierini, Anna | |
dc.contributor.author | Tucker, David F. | |
dc.contributor.author | Urhoj, Stine Kjaer | |
dc.contributor.author | Wellesley, Diana Gay | |
dc.contributor.author | Morris, Joan K. | |
dc.date.accessioned | 2023-06-27T07:37:59Z | |
dc.date.available | 2023-06-27T07:37:59Z | |
dc.date.created | 2023-06-07T14:34:09Z | |
dc.date.issued | 2023 | |
dc.identifier.issn | 0003-9888 | |
dc.identifier.uri | https://hdl.handle.net/11250/3073344 | |
dc.description.abstract | Objective: To investigate the survival to 10 years of age of children with trisomy 13 (T13) and children with trisomy 18 (T18), born 1995–2014. Design: Population-based cohort study that linked mortality data to data on children born with T13 or T18, including translocations and mosaicisms, from 13 member registries of EUROCAT, a European network for the surveillance of congenital anomalies. Setting: 13 regions in nine Western European countries. Patients: 252 live births with T13 and 602 with T18. Main outcome measures: Survival at 1 week, 4 weeks and 1, 5 and 10 years of age estimated by random-effects meta-analyses of registry-specific Kaplan-Meier survival estimates. Results: Survival estimates of children with T13 were 34% (95% CI 26% to 46%), 17% (95% CI 11% to 29%) and 11% (95% CI 6% to 18%) at 4 weeks, 1 and 10 years, respectively. The corresponding survival estimates were 38% (95% CI 31% to 45%), 13% (95% CI 10% to 17%) and 8% (95% CI 5% to 13%) for children with T18. The 10-year survival conditional on surviving to 4 weeks was 32% (95% CI 23% to 41%) and 21% (95% CI 15% to 28%) for children with T13 and T18, respectively. Conclusions: This multi-registry European study found that despite extremely high neonatal mortality in children with T13 and T18, 32% and 21%, respectively, of those who survived to 4 weeks were likely to survive to age 10 years. These reliable survival estimates are useful to inform counselling of parents after prenatal diagnosis. | en_US |
dc.language.iso | eng | en_US |
dc.publisher | BMJ | en_US |
dc.title | Ten-year survival of children with trisomy 13 or trisomy 18: A multi-registry European cohort study | en_US |
dc.type | Journal article | en_US |
dc.type | Peer reviewed | en_US |
dc.description.version | acceptedVersion | en_US |
dc.rights.holder | Copyright 2023 the authors | en_US |
cristin.ispublished | true | |
cristin.fulltext | postprint | |
cristin.qualitycode | 2 | |
dc.identifier.doi | 10.1136/archdischild-2022-325068 | |
dc.identifier.cristin | 2152712 | |
dc.source.journal | Archives of Disease in Childhood | en_US |
dc.source.pagenumber | 461-467 | en_US |
dc.relation.project | EC/H2020/733001 | en_US |
dc.identifier.citation | Archives of Disease in Childhood. 2023, 108 (6), 461-467. | en_US |
dc.source.volume | 108 | en_US |
dc.source.issue | 6 | en_US |