Abstract
BackgroundSurvivors of childhood cancer can develop adverse health events later in life. Infrequent occurrences and scarcity of structured information result in analytical and statistical challenges. Alternative statistical approaches are required to investigate the basis of late effects in smaller data sets.MethodsHere we describe sex-specific health care use, mortality and causal associations between primary diagnosis, treatment and outcomes in a small cohort (n = 2315) of 5-year survivors of childhood cancer (n = 2129) in southern Sweden and a control group (n = 11,882; age-, sex- and region-matched from the general population). We developed a constraint-based method for causal inference based on Bayesian estimation of distributions, and used it to investigate health care use and causal associations between diagnoses, treatments and outcomes. Mortality was analyzed by the Kaplan–Meier method.ResultsOur results confirm a significantly higher health care usage and premature mortality among childhood cancer survivors as compared to controls. The developed method for causal inference identifies 98 significant associations (p < 0.0001) where most are well known (n = 73; 74.5%). Hitherto undescribed associations are identified (n = 5; 5.1%). These were between use of alkylating agents and eye conditions, topoisomerase inhibitors and viral infections; pituitary surgery and intestinal infections; and cervical cancer and endometritis. We discuss study-related biases (n = 20; 20.4%) and limitations.ConclusionsThe findings contribute to a broader understanding of the consequences of cancer treatment. The study shows relevance for small data sets and causal inference, and presents the method as a complement to traditional statistical approaches.
Highlights
Survivors of childhood cancer can develop adverse health events later in life
We developed a method for causal inference and used it to investigate causal relations between details of the primary disease, its treatment history and health-related events that occur 5 years after the first childhood cancer diagnosis (CCD), using a population-based childhood cancer cohort with years of diagnoses ranging from 1970 to 201610
Most of the links were well known but some have not been described previously, to the best of our knowledge. These were between use of alkylating agents and eye conditions, topoisomerase inhibitors and viral infections; pituitary surgery and intestinal infections; and cervical cancer and endometritis
Summary
The purpose of the study was to (1) briefly describe the newly established cohort, (2) describe and discuss the causal inference method as suitable for smaller data sets and (3) report the links found between primary disease, treatment and outcomes
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