dc.contributor.author | Daltveit, Dagrun Slettebø | |
dc.date.accessioned | 2024-08-22T08:15:55Z | |
dc.date.available | 2024-08-22T08:15:55Z | |
dc.date.issued | 2024-09-02 | |
dc.date.submitted | 2024-08-05T12:19:52.860Z | |
dc.identifier | container/d5/f9/d8/94/d5f9d894-4dd8-438c-916b-5d72eb0dad64 | |
dc.identifier.isbn | 9788230853931 | |
dc.identifier.isbn | 9788230866832 | |
dc.identifier.uri | https://hdl.handle.net/11250/3147497 | |
dc.description.abstract | Bakgrunn: Medfødte misdannelser og kreft er begge blant de ti vanligste årsakene til tapte leveår grunnet død eller helsetap for barn mellom 0 og 19 år (ulykker og perinatale sykdommer ekskludert). Årsakene til begge sykdommene er i stor grad ukjente. Alvorlige misdannelser er imidlertid etablert som risikofaktorer for kreft blant barn, noe som kan tyde på en felles etiologi.
Hensikt: Vi undersøkte sammenhengen mellom det å ha en alvorlig misdannelse eller å ha et søsken med en alvorlig misdannelse og senere kreftutvikling; blant barn, ungdom og voksne, samt kjønnsforskjeller i denne sammenhengen blant barn.
Metode: Vi gjennomførte tre populasjonsbaserte nøstede kasus-kontrollstudier hvor vi kombinerte registerdata fra Danmark, Finland, Norge og Sverige. Personer registrert i fødselsregistrene mellom 1967 og 2014 som utviklet kreft ble definert som kasus. Kontrollene ble matchet på land og fødselsår. Eksponeringene vi undersøkte var alvorlige misdannelser blant individene eller deres søsken. Den relative risikoen for kreft assosiert med eksponeringen ble estimert som oddsratio fra logistiske regresjonsmodeller.
Resultat: Den relative risikoen for kreft blant personer med alvorlige misdannelser var 1,7 ganger høyere enn blant personer uten misdannelser. Den økte risikoen vedvarte inn i voksen alder (1,2 ganger høyere), spesielt gjaldt dette voksne med alvorlige misdannelser i hjerte, kjønnsorganer, nervesystemet, skjelettdysplasier og Down syndrom. Sammenhengene mellom alvorlige misdannelser og barnekreft var generelt sterkere blant jenter enn gutter. Blant personer som hadde søsken med alvorlige misdannelser, var risikoen for barnekreft (0 til 19 år) noe økt (1,09 ganger), mens den totale risikoen for kreft blant personer i alderen 0 til 46 år ikke var økt.
Konklusjon: Våre resultater stemmer overens med hypotesen om felles bakenforliggende årsaker til alvorlige misdannelser og kreft: genetiske, miljømessige eller en kombinasjon. Arbeidet danner grunnlaget for videre forskning på de biologiske mekanismene som ligger bak begge sykdommene. | en_US |
dc.description.abstract | Background: Globally, birth defects and childhood cancer are among the 10 most common causes of childhood disease burden (excluding perinatal diseases and injuries). There are few established risk factors for both diseases, but birth defects have consistently been associated with childhood cancer risk, suggesting a common aetiology. Given the large global public health impact of birth defects and childhood cancer, a broader understanding of the underlying causes is warranted.
Objectives: We aimed to explore the associations between having a major birth defect or having a sibling with a major birth defect and cancer among children, adolescents, and adults, and to evaluate if the associations among children differed by sex.
Methods: We performed three population-based nested case-control studies where we combined registry data from Denmark, Finland, Norway, and Sweden. Individuals registered in the birth registries between 1967 and 2014 who later developed cancer were defined as cases. Controls were frequency-matched on country and year of birth. The exposure of interest was major birth defects in the individuals or the siblings. The relative risk of cancer associated with the exposure was estimated as odds ratios from logistic regression models.
Results: The relative risk of overall cancer in individuals with birth defects compared to individuals without birth defects was 1.7. The increased risk persisted into adulthood (1.2-fold), in particular for individuals with congenital heart defects, genital organ defects, chromosomal anomalies, nervous system defects, and skeletal dysplasia. The birth defect-childhood cancer associations were generally stronger in girls than boys. The risk of childhood cancer (0 to 19 years) was slightly elevated (1.09-fold) in individuals whose siblings had birth defects, but the overall risk of cancer in individuals aged 0 to 46 years was not increased.
Conclusions: Our novel findings provide evidence consistent with common aetiologies of birth defects and cancer, such as shared genetic predisposition and environmental factors, and should motivate further research into possible biological mechanisms. | en_US |
dc.language.iso | eng | en_US |
dc.publisher | The University of Bergen | en_US |
dc.relation.haspart | Paper I: Daltveit, D. S., Klungsøyr, K., Engeland, A., Ekbom, A., Gissler, M., Glimelius, I., Grotmol, T., Madanat-Harjuoja, L., Ording, A. G., Sæther, S. M. M., Sørensen, H. T., Troisi, R., & Bjørge, T. (2020). Cancer risk in individuals with major birth defects: large Nordic population based case-control study among children, adolescents, and adults. BMJ, 371, m4060. The article is available at: <a href="https://hdl.handle.net/11250/2757151" target="blank">https://hdl.handle.net/11250/2757151</a>. | en_US |
dc.relation.haspart | Paper II: Daltveit, D. S., Klungsøyr, K., Engeland, A., Ekbom, A., Gissler, M., Glimelius, I., Grotmol, T., Madanat-Harjuoja, L., Ording, A. G., Sørensen, H. T., Troisi, R., & Bjørge, T. (2023). Sex differences in childhood cancer risk among children with major birth defects: a Nordic population-based nested case-control study. International journal of epidemiology, 52(2), 450–465. The article is available at: <a href="https://hdl.handle.net/11250/3037245" target="blank">https://hdl.handle.net/11250/3037245</a>. | en_US |
dc.relation.haspart | Paper III: Daltveit, D. S., Klungsøyr, K., Engeland, A., Ekbom, A., Gissler, M., Glimelius, I., Grotmol, T., Madanat-Harjuoja, L., Ording, A. G., Sørensen, H. T., Troisi, R., & Bjørge, T. (2023). Cancer risk in the siblings of individuals with major birth defects: a large Nordic population-based case-control study. International journal of epidemiology, 52(6), 1826–1835. The article is available at: <a href="https://hdl.handle.net/11250/3130406" target="blank">https://hdl.handle.net/11250/3130406</a>. | en_US |
dc.rights | Attribution-NoDerivs (CC BY-ND). This item's rights statement or license does not apply to the included articles in the thesis. | |
dc.rights.uri | https://creativecommons.org/licenses/by-nd/4.0/ | |
dc.title | Birth defects and cancer risk : Nordic population-based studies of cancer risk in children, adolescents, and adults with major birth defects and their siblings | en_US |
dc.type | Doctoral thesis | en_US |
dc.date.updated | 2024-08-05T12:19:52.860Z | |
dc.rights.holder | Copyright the Author. | en_US |
dc.contributor.orcid | 0000-0002-0903-1140 | |
dc.description.degree | Doktorgradsavhandling | |
fs.unitcode | 13-26-0 | |