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Measuring childhood cancer late effects : evidence of a healthy survivor effect

Asdahl, Peter Haubjerg ; Ojha, Rohit Priyadarshi ; Winther, Jeanette Falck ; Holmqvist, Anna Sällfors LU ; de Fine Licht, Sofie ; Gudmundsdottir, Thorgerdur ; Madanat-Harjuoja, Laura ; Tryggvadottir, Laufey ; Andersen, Klaus Kaae and Hasle, Henrik (2017) In European Journal of Epidemiology 32(12). p.1089-1096
Abstract

Introduction: Given considerable focus on health outcomes among childhood cancer survivors, we aimed to explore whether survivor bias is apparent during long-term follow-up of childhood cancer survivors. Methods: We identified all 1-year survivors of cancer diagnosed before 20 years of age in Denmark, Finland, Iceland, and Sweden. From the general population, we randomly sampled a comparison cohort. Study individuals were followed for hospitalizations for diseases of the gastroenterological tract, endocrine system, cardiovascular system, or urinary tract from the start of the cancer registries to 2010. We estimated cumulative incidence with death as competing risk and used threshold regression to compare the hazards of the diseases of... (More)

Introduction: Given considerable focus on health outcomes among childhood cancer survivors, we aimed to explore whether survivor bias is apparent during long-term follow-up of childhood cancer survivors. Methods: We identified all 1-year survivors of cancer diagnosed before 20 years of age in Denmark, Finland, Iceland, and Sweden. From the general population, we randomly sampled a comparison cohort. Study individuals were followed for hospitalizations for diseases of the gastroenterological tract, endocrine system, cardiovascular system, or urinary tract from the start of the cancer registries to 2010. We estimated cumulative incidence with death as competing risk and used threshold regression to compare the hazards of the diseases of interest at ages 20, 40, 60, and 75 years. Results: Our study included 27,007 one-year survivors of childhood cancer and 165,620 individuals from the general population. The cumulative incidence of all four outcomes was higher for childhood cancer survivors during early adulthood, but for three outcomes, the cumulative incidence was higher for the general population after age 55 years. The hazard ratios (HRs) decreased for all outcomes with increasing age, and for two of the outcomes, the hazards were higher for the general population at older ages (endocrine diseases: age-specific HRs = 3.0, 1.4, 1.0, 0.87; Cardiovascular diseases: age-specific HRs = 4.1, 1.4, 0.97, 0.84). Conclusions: Our findings provide empirical evidence that survivor bias attenuates measures of association when comparing survivors with the general population. The design and analysis of studies among childhood cancer survivors, particularly as this population attains older ages, should account for survivor bias to avoid misinterpreting estimates of disease burden.

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author collaboration
publishing date
type
Contribution to journal
publication status
published
subject
keywords
Cancer survivorship, Childhood cancer, Competing risk, Healthy survivor effect, Late effects, Survivor bias
in
European Journal of Epidemiology
volume
32
issue
12
pages
1089 - 1096
publisher
Springer
external identifiers
  • scopus:85035095084
  • pmid:29185125
ISSN
0393-2990
DOI
10.1007/s10654-017-0305-4
language
English
LU publication?
no
id
3c4e2657-4526-4e24-99ff-e9c19ca5339f
date added to LUP
2017-12-14 14:34:47
date last changed
2024-10-14 20:09:23
@article{3c4e2657-4526-4e24-99ff-e9c19ca5339f,
  abstract     = {{<p>Introduction: Given considerable focus on health outcomes among childhood cancer survivors, we aimed to explore whether survivor bias is apparent during long-term follow-up of childhood cancer survivors. Methods: We identified all 1-year survivors of cancer diagnosed before 20 years of age in Denmark, Finland, Iceland, and Sweden. From the general population, we randomly sampled a comparison cohort. Study individuals were followed for hospitalizations for diseases of the gastroenterological tract, endocrine system, cardiovascular system, or urinary tract from the start of the cancer registries to 2010. We estimated cumulative incidence with death as competing risk and used threshold regression to compare the hazards of the diseases of interest at ages 20, 40, 60, and 75 years. Results: Our study included 27,007 one-year survivors of childhood cancer and 165,620 individuals from the general population. The cumulative incidence of all four outcomes was higher for childhood cancer survivors during early adulthood, but for three outcomes, the cumulative incidence was higher for the general population after age 55 years. The hazard ratios (HRs) decreased for all outcomes with increasing age, and for two of the outcomes, the hazards were higher for the general population at older ages (endocrine diseases: age-specific HRs = 3.0, 1.4, 1.0, 0.87; Cardiovascular diseases: age-specific HRs = 4.1, 1.4, 0.97, 0.84). Conclusions: Our findings provide empirical evidence that survivor bias attenuates measures of association when comparing survivors with the general population. The design and analysis of studies among childhood cancer survivors, particularly as this population attains older ages, should account for survivor bias to avoid misinterpreting estimates of disease burden.</p>}},
  author       = {{Asdahl, Peter Haubjerg and Ojha, Rohit Priyadarshi and Winther, Jeanette Falck and Holmqvist, Anna Sällfors and de Fine Licht, Sofie and Gudmundsdottir, Thorgerdur and Madanat-Harjuoja, Laura and Tryggvadottir, Laufey and Andersen, Klaus Kaae and Hasle, Henrik}},
  issn         = {{0393-2990}},
  keywords     = {{Cancer survivorship; Childhood cancer; Competing risk; Healthy survivor effect; Late effects; Survivor bias}},
  language     = {{eng}},
  month        = {{11}},
  number       = {{12}},
  pages        = {{1089--1096}},
  publisher    = {{Springer}},
  series       = {{European Journal of Epidemiology}},
  title        = {{Measuring childhood cancer late effects : evidence of a healthy survivor effect}},
  url          = {{http://dx.doi.org/10.1007/s10654-017-0305-4}},
  doi          = {{10.1007/s10654-017-0305-4}},
  volume       = {{32}},
  year         = {{2017}},
}