Cancer predisposition syndromes as risk factor for early second primary neoplasms after childhood cancer – a national cohort study
Copyright policy )Cancer predisposition syndromes as risk factor for early second primary neoplasms after childhood cancer – a national cohort study
ABSTRACTBackgroundChildhood cancer patients are at increased risk of second primary neoplasms (SPNs). We assessed incidence and risk factors for early SPNs with a focus on cancer predisposition syndromes (CPSs).Patients and methodsThis cohort study used data from the Swiss Childhood Cancer Registry. We included patients with first primary neoplasms (FPN) until age 21 years from 1986 to 2015 and identified SPNs occurring before age 21. We calculated standardized incidence ratios (SIR) and absolute excess risks (AER) using Swiss population cancer incidence data and cumulative incidence of SPNs. We calculated hazard ratios (HR) of risk factors for SPNs using Fine and Gray competing risk regression.ResultsAmong 8,074 childhood cancer patients, 304 (4%) were diagnosed with a CPS and 94 (1%) developed early SPNs. The incidence of SPNs was more than 10-fold increased in childhood cancer patients compared to neoplasms in the general population (SIR 10.6, 95%-confidence interval [CI] 8.7-13.1) and the AER was 179/100,000 person-years (CI 139-219). Cumulative incidence of SPNs 20 years after FPN diagnosis was 23% in patients with CPSs and 3% in those without. Risk factors for SPNs were CPSs (HR 7.8, CI 4.8-12.7), chemotherapy (HR 2.2, CI 1.1-4.6), radiotherapy (HR 1.9, CI 1.2-2.9), hematopoietic stem cell transplantation (HR 1.8, CI 1-3.3), and older age (15-20 years) at FPN diagnosis (HR 1.9, CI 1.1-3.2).ConclusionCPSs are associated with a high risk of SPNs before age 21 years. Identification of CPSs is important for appropriate cancer surveillance and targeted screening.
- University of Geneva Switzerland
- University of Bern Switzerland
- University of Zurich Switzerland
- University of Basel Switzerland
- University of Lausanne Switzerland
Male, Genetic testing, Swiss childhood cancer registry, Cancer survivors, Second primary neoplasms, Pediatrics, Neoplasms, Second Primary / diagnosis, Cancer Survivors, Risk Factors, Adolescent medicine, Neoplasms, 1306 Cancer Research, Registries, Child, Neoplastic Syndromes, Hereditary / epidemiology, Incidence, Age Factors, Hematopoietic Stem Cell Transplantation, Neoplasms, Second Primary, Neoplasms, Second Primary / epidemiology, Early detection of cancer, Neoplastic Syndromes, Hereditary / diagnosis, Child, Preschool, 2730 Oncology, Female, Switzerland, Adolescent, 610 Medicine & health, Antineoplastic Agents, Risk Assessment, Switzerland / epidemiology, 618, Young Adult, 360 Social problems & social services, Neoplastic Syndromes, Hereditary, Humans, Radiotherapy / adverse effects, Genetic counselling, Adolescent; Age Factors; Antineoplastic Agents/adverse effects; Cancer Survivors; Child; Child, Preschool; Female; Hematopoietic Stem Cell Transplantation/adverse effects; Humans; Incidence; Infant; Infant, Newborn; Male; Neoplasms, Second Primary/diagnosis; Neoplasms, Second Primary/epidemiology; Neoplastic Syndromes, Hereditary/diagnosis; Neoplastic Syndromes, Hereditary/epidemiology; Radiotherapy/adverse effects; Registries; Risk Assessment; Risk Factors; Switzerland/epidemiology; Young Adult; Adolescent medicine; Cancer survivors; Early detection of cancer; Genetic counselling; Genetic testing; Hereditary neoplastic syndromes; Neoplasms; Pediatrics; Second primary neoplasms; Swiss childhood cancer registry, Radiotherapy, Hereditary neoplastic syndromes, Antineoplastic Agents / adverse effects, Hematopoietic Stem Cell Transplantation / adverse effects, Infant, Newborn, Infant, 10036 Medical Clinic, ddc: ddc:618
Male, Genetic testing, Swiss childhood cancer registry, Cancer survivors, Second primary neoplasms, Pediatrics, Neoplasms, Second Primary / diagnosis, Cancer Survivors, Risk Factors, Adolescent medicine, Neoplasms, 1306 Cancer Research, Registries, Child, Neoplastic Syndromes, Hereditary / epidemiology, Incidence, Age Factors, Hematopoietic Stem Cell Transplantation, Neoplasms, Second Primary, Neoplasms, Second Primary / epidemiology, Early detection of cancer, Neoplastic Syndromes, Hereditary / diagnosis, Child, Preschool, 2730 Oncology, Female, Switzerland, Adolescent, 610 Medicine & health, Antineoplastic Agents, Risk Assessment, Switzerland / epidemiology, 618, Young Adult, 360 Social problems & social services, Neoplastic Syndromes, Hereditary, Humans, Radiotherapy / adverse effects, Genetic counselling, Adolescent; Age Factors; Antineoplastic Agents/adverse effects; Cancer Survivors; Child; Child, Preschool; Female; Hematopoietic Stem Cell Transplantation/adverse effects; Humans; Incidence; Infant; Infant, Newborn; Male; Neoplasms, Second Primary/diagnosis; Neoplasms, Second Primary/epidemiology; Neoplastic Syndromes, Hereditary/diagnosis; Neoplastic Syndromes, Hereditary/epidemiology; Radiotherapy/adverse effects; Registries; Risk Assessment; Risk Factors; Switzerland/epidemiology; Young Adult; Adolescent medicine; Cancer survivors; Early detection of cancer; Genetic counselling; Genetic testing; Hereditary neoplastic syndromes; Neoplasms; Pediatrics; Second primary neoplasms; Swiss childhood cancer registry, Radiotherapy, Hereditary neoplastic syndromes, Antineoplastic Agents / adverse effects, Hematopoietic Stem Cell Transplantation / adverse effects, Infant, Newborn, Infant, 10036 Medical Clinic, ddc: ddc:618
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