TY - JOUR
T1 - The rare disease neurofibromatosis 1 as a source of hereditary economic inequality
T2 - Evidence from Finland
AU - Johansson, Edvard
AU - Kallionpää, Roope A.
AU - Böckerman, Petri
AU - Peltonen, Sirkku
AU - Peltonen, Juha
N1 - Funding Information:
The study was funded by grants from the Turku University Hospital and the Cancer Foundation Finland. This work was generated within the European Reference Network on Genetic Tumour Risk Syndromes?Project ID Number 739547. European Reference Network on Genetic Tumour Risk Syndromes is partly cofunded by the European Union within the framework of the Third Health Programme, ERN-2016?Framework Partnership Agreement 2017?2021. Conceptualization: E.J. R.A.K. P.B. S.P. J.P.; Data Curation: E.J. R.A.K. S.P. J.P.; Formal Analysis: E.J.; Funding Acquisition: S.P. J.P.; Investigation: E.J. R.A.K. P.B. S.P. J.P.; Writing: E.J. R.A.K. P.B. S.P. J.P. The study was approved by the Ethics Committee of the Hospital District of Southwest Finland, and research permissions were secured from the Finnish Institute for Health and Welfare, Statistics Finland, and all participating hospitals. The study adhered to the principles set out in the Declaration of Helsinki. The study was register-based and retrospective and therefore exempted from obtaining informed consent from the participants.
Funding Information:
The study was funded by grants from the Turku University Hospital and the Cancer Foundation Finland. This work was generated within the European Reference Network on Genetic Tumour Risk Syndromes—Project ID Number 739547. European Reference Network on Genetic Tumour Risk Syndromes is partly cofunded by the European Union within the framework of the Third Health Programme, ERN-2016—Framework Partnership Agreement 2017–2021.
Publisher Copyright:
© 2021 American College of Medical Genetics and Genomics
PY - 2021
Y1 - 2021
N2 - Purpose: This study investigated whether individuals with neurofibromatosis 1 (NF1) fare worse than individuals without NF1 in terms of economic well-being. NF1 is relatively common in the population and provides an informative case of a rare hereditary disease. Methods: We examined a subset of 692 individuals with verified NF1 from the Finnish total population-based NF1 cohort and compared that with 7407 control individuals matched for age, sex, and municipality during 1997-2014. Economic well-being was operationalized with annual work earnings and total income, including social income transfers. Results: NF1 significantly worsened economic well-being. Low education, increased morbidity, and reduced labor market participation partly explained the effect of NF1. Yet, NF1 was independently associated with lower income even after adjusting for these factors. Furthermore, NF1 had a larger negative effect on income from work than it had on total income, which indicated that the Finnish social security system partly compensated the labor market losses suffered by individuals with NF1. NF1 had a larger impact on economic inequality for men than for women. Conclusion: NF1 contributes to economic inequality. A hereditary disease may convey worse economic well-being over several generations.
AB - Purpose: This study investigated whether individuals with neurofibromatosis 1 (NF1) fare worse than individuals without NF1 in terms of economic well-being. NF1 is relatively common in the population and provides an informative case of a rare hereditary disease. Methods: We examined a subset of 692 individuals with verified NF1 from the Finnish total population-based NF1 cohort and compared that with 7407 control individuals matched for age, sex, and municipality during 1997-2014. Economic well-being was operationalized with annual work earnings and total income, including social income transfers. Results: NF1 significantly worsened economic well-being. Low education, increased morbidity, and reduced labor market participation partly explained the effect of NF1. Yet, NF1 was independently associated with lower income even after adjusting for these factors. Furthermore, NF1 had a larger negative effect on income from work than it had on total income, which indicated that the Finnish social security system partly compensated the labor market losses suffered by individuals with NF1. NF1 had a larger impact on economic inequality for men than for women. Conclusion: NF1 contributes to economic inequality. A hereditary disease may convey worse economic well-being over several generations.
KW - Neurofibromatosis
KW - Nordic model
KW - Rare diseases
KW - Social income transfers
KW - Wages
UR - http://www.scopus.com/inward/record.url?scp=85121784490&partnerID=8YFLogxK
U2 - 10.1016/j.gim.2021.11.024
DO - 10.1016/j.gim.2021.11.024
M3 - Article
AN - SCOPUS:85121784490
SN - 1098-3600
JO - Genetics in Medicine
JF - Genetics in Medicine
ER -