Swiss Childhood Cancer Survivor Study Research Articles

Incidence and prevalence of musculoskeletal health conditions in survivors of childhood and adolescent cancers: A report from the Swiss childhood cancer survivor study.

Childhood cancer and its treatment can cause damage to the musculoskeletal system. We aimed to determine the incidence and prevalence of musculoskeletal health conditions (MSHC) in survivors, and to investigate differences by cancer-related characteristics. We used data from the Childhood Cancer Registry and the Swiss Childhood Cancer Survivor Study, including survivors (≥5 years since diagnosis; diagnosed 1976-2015 at <20 years of age) aged ≥15 years at study. Cumulative incidence and prevalence of MSHCs (osteoporosis, limb length discrepancy, limited joint mobility, bone/joint pain, scoliosis, changes to chest/ribs and amputation) were calculated from self-reported data. We included 2645 survivors (53% men; median age 24 years, range 15-59 years). Prevalence and cumulative incidence of any MSHC was 21% and 26%, respectively. Incidence rate for any MSHC was 15.6/1000 person-years. Scoliosis (8%), bone/joint pain (7%) and limited joint mobility (7%) were the most prevalent MSHC. MSHC co-occurred with other health conditions in 87% of survivors. We found increased rates of MSHC in women (RR = 1.4, 95%CI: 1.2-1.7), bone tumour survivors (RR = 6.0, 95%CI: 4.5-7.9), survivors older at diagnosis (11-15 years: RR = 1.8, 95%CI: 1.5-2.3), after a relapse (RR = 1.5, 95%CI: 1.3-1.9), treatment with surgery (RR = 1.2, 95%CI: 1.0-1.5), chemotherapy (RR = 1.4, 95%CI: 1.1-1.8) or stem cell transplantation (RR = 1.6, 95%CI: 1.0-2.5), and more recent year of diagnosis (2011-2015: RR = 4.3, 95%CI: 2.8-6.8). MSHCs are prevalent in survivors, the risk is increasing in younger survivor cohorts, and MSHCs usually occur in multimorbid survivors. Strengthening of rehabilitation services and appropriate referrals are needed to mitigate the effects of the cancer and cancer treatment.

Validation of self-reported cardiovascular problems in childhood cancer survivors by contacting general practitioners: feasibility and results

BackgroundEpidemiological studies often rely on self-reported health problems and validation greatly improves study quality. In a study of late effects after childhood cancer, we validated self-reported cardiovascular problems by contacting general practitioners (GPs). This paper describes: (a) the feasibility of this approach; and (b) the agreement between survivor-reports and reports from their GP.MethodsThe Swiss Childhood Cancer Survivor Study (SCCSS) contacts all childhood cancer survivors registered in the Swiss Childhood Cancer Registry since 1976 who survived at least 5 years from cancer diagnosis. We validated answers of all survivors who reported a cardiovascular problem in the questionnaire. Reported cardiovascular problems were hypertension, arrhythmia, congestive heart failure, myocardial infarction, angina pectoris, stroke, thrombosis, and valvular problems. In the questionnaire, we further asked survivors to provide a valid address of their GP and a consent for contact. We sent case-report forms to survivors’ GPs and requested information on cardiovascular diagnoses of their patients. To determine agreement between information reported by survivors and GPs, we calculated Cohen’s kappa (κ) coefficients for each category of cardiovascular problems.ResultsWe used questionnaires from 2172 respondents of the SCCSS. Of 290 survivors (13% of 2172) who reported cardiovascular problems, 166 gave consent to contact their GP and provided a valid address. Of those, 135 GPs (81%) replied, and 128 returned the completed case-report form. Survivor-reports were confirmed by 54/128 GPs (42%). Of the 54 GPs, 36 (28% of 128) confirmed the problems as reported by the survivors; 11 (9% of 128) confirmed the reported problem(s) and gave additional information on more cardiovascular outcomes; and seven GPs (5% of 128) confirmed some, but not all cardiovascular problems. Agreement between GPs and survivors was good for stroke (κ = 0.79), moderate for hypertension (κ = 0.51), arrhythmias (κ = 0.41), valvular problems (κ = 0.41) and thrombosis (κ = 0.56), and poor for coronary heart disease (κ = 0.15) and heart failure (κ = 0.32).ConclusionsDespite excellent GP compliance, it was found unfeasible to validate self-reported cardiovascular problems via GPs because they do not serve as gatekeepers in the Swiss health care system. It is thus necessary to develop other validation methods to improve the quality of patient-reported outcomes.

Chronic health conditions after childhood Langerhans cell histiocytosis: Results from the Swiss Childhood Cancer Survivor Study.

Langerhans cell histiocytosis (LCH) is a rare disease characterized by dysregulated proliferation of myeloid marrow progenitors and subsequent organ infiltration. While LCH is associated with a favorable prognosis, some survivors may develop chronic health conditions (CHC) because of the disease. In this study, we aimed to assess the spectrum and prevalence of CHC among LCH survivors compared with siblings and identify factors associated with the development of CHC. The Swiss Childhood Cancer Survivor Study sent questionnaires to all ≥ 5-year LCH survivors registered in the Swiss Childhood Cancer Registry and diagnosed between 1976 and 2015. Siblings also received similar questionnaires. We compared CHC prevalence between LCH survivors and siblings and used logistic regression to identify determinants of CHC. A total of 123 LCH survivors participated in the study, with a response rate of 69%. Median time since diagnosis was 13years (interquartile range 9-20). Among LCH survivors, 59% had at least one CHC. Cardiovascular (13% vs. 6%), endocrine (15% vs. 2%), musculoskeletal (22% vs. 13%), and digestive (15% vs. 8%) CHC were more common among LCH survivors compared to siblings (all p < 0.05). Factors most strongly associated with the occurrence of CHC were multisystem LCH, multifocal bone involvement, and involvement of the pituitary gland. More than half of long-term LCH survivors suffered from one or more CHC and were affected considerably more than siblings. LCH survivors in follow-up care should be screened especially for cardiovascular, endocrine, musculoskeletal, and digestive conditions.

Risk factors for overweight and obesity after childhood acute lymphoblastic leukemia in North America and Switzerland: A comparison of two cohort studies.

After childhood acute lymphoblastic leukemia (ALL), sequelae include overweight and obesity, yet with conflicting evidence. We compared the prevalence of overweight and obesity between ≥5-year ALL survivors from the North American Childhood Cancer Survivor Study (CCSS) and the Swiss Childhood Cancer Survivor Study (SCCSS) and described risk factors. We included adult childhood ALL survivors diagnosed between 1976 and 1999. We matched CCSS participants (3:1) to SCCSS participants by sex and attained age. We calculated body mass index (BMI) from self-reported height and weight for 1287 CCSS and 429 SCCSS participants; we then compared those with siblings (2034) in North America and Switzerland (678) siblings. We assessed risk factors for overweight (BMI 25-29.9 kg/m2 ) and obesity (≥30 kg/m2 ) using multinomial regression. We found overweight and obesity significantly more common among survivors in North America when compared with survivors in Switzerland [overweight: 30%, 95% confidence interval (CI): 27-32 vs. 24%, 21-29; obesity: 29%, 27-32 vs. 7%, 5-10] and siblings (overweight: 30%, 27-32 vs. 25%, 22-29; obesity: 24%, 22-26 vs. 6%, 4-8). Survivors in North America [odds ratio (OR) = 1.24, 1.01-1.53] and Switzerland (1.27, 0.74-2.21) were slightly more often obese than siblings. Among survivors, risk factors for obesity included residency in North America (5.8, 3.7-9.0); male (1.7, 1.3-2.3); attained age (≥45 years: 5.1, 2.4-10.8); Non-Hispanic Black (3.4, 1.6-7.0); low household income (2.3, 1.4-3.5); young age at diagnosis (1.6, 1.1-2.2). Cranial radiotherapy ≥18 Gray was only a risk factor for overweight (1.4, 1.0-1.8); steroids were not associated with overweight or obesity. Interaction tests found no evidence of difference in risk factors between cohorts. Although treatment-related risk for overweight and obesity were similar between regions, higher prevalence among survivors in North America identifies important sociodemographic drivers for informing health policy and targeted intervention trials.

Body image in adolescent survivors of childhood cancer: The role of chronic health conditions.

Cancer and its treatment may impair the body image of childhood cancer survivors during adolescence. We compared the body image between adolescent cancer survivors and their siblings, and determined whether survivors' body image is associated with socio-demographic characteristics, clinical characteristics and chronic health conditions. As part of the nationwide Swiss Childhood Cancer Survivor Study, we sent questionnaires to adolescents (aged 16-19years), who survived &gt;5years after having been diagnosed with childhood cancer between 1989 and 2010. Siblings received the same questionnaire. We assessed the level of agreement with three body image statements referring to body satisfaction and preferences for changes. Chronic health conditions were classified into cardiovascular, pulmonary, endocrine, musculoskeletal, renal/digestive, neurological and hearing or vision impairment. We used ordered logistic regression models to identify determinants of a more negative body image. Our study included 504 survivors (48% female) with a median age at study of 17.7years (interquartile range: 16.8-18.6) and 136 siblings. Survivors and siblings reported overall comparable levels of agreement with body image statements (all p&gt;.05). Female survivors (all odds ratio [ORs] ≥1.7), survivors treated with haematopoietic stem cell transplantation (HSCT; all ORs ≥2.2), and survivors with ≥2 chronic health conditions (all ORs ≥1.4) reported a more negative body image. This was particularly pronounced for survivors suffering from musculoskeletal or endocrine conditions. Female survivors, survivors treated with HSCT or with chronic health conditions are at risk of body image concerns during adolescence. Increased awareness among clinicians and targeted psychosocial support could mitigate such concerns.

Social, emotional, and behavioral functioning in young childhood cancer survivors with chronic health conditions

The cancer diagnosis and its intensive treatment may affect the long-term psycho-social adjustment of childhood cancer survivors. We aimed to describe social, emotional, and behavioral functioning and their determinants in young childhood cancer survivors. The nationwide Swiss Childhood Cancer Survivor Study sends questionnaires to parents of survivors aged 5-15 years, who have survived at least 5 years after diagnosis. We assessed social, emotional, and behavioral functioning using the Strengths and Difficulties Questionnaire (SDQ). The SDQ includes four difficulties scales (emotional, conduct, hyperactivity, peer problems), a total difficulties indicator, and one strength scale (prosocial). We compared the proportion of survivors with borderline and abnormal scores to reference values and used multivariable logistic regression to identify determinants. Our study included 756 families (response rate of 72%). Thirteen percent of survivors had abnormal scores for the total difficulties indicator compared to 10% in the general population. The proportion of survivors with abnormal scores was highest for the emotional scale (15%vs. 8% in the general population), followed by the peer problems scale (14%vs. 7%), hyperactivity (8%vs. 10%), and conduct scale (6%vs. 7%). Few survivors (4%vs. 7%) had abnormal scores on the prosocial scale. Children with chronic health conditions had a higher risk of borderline and abnormal scores on all difficulties scales (all p<0.05). Most childhood cancer survivors do well in social, emotional, and behavioral life domains, but children with chronic health conditions experience difficulties. Therefore, healthcare professionals should offer specific psycho-social support to these survivors.

Educational Attainment and Employment Outcome of Survivors of Pediatric CNS Tumors in Switzerland-A Report from the Swiss Childhood Cancer Survivor Study.

Background: Childhood cancer survivors diagnosed with a central nervous system (CNS) tumor are at risk for educational and vocational challenges. This study compared educational attainment and employment outcome in survivors of CNS tumors to survivors of other malignancies. Methods: The questionnaire-based Swiss Childhood Cancer Survivor Study (SCCSS) included cancer patients diagnosed between 1976 and 2010, aged ≤20 years, who survived ≥5 years after diagnosis. We classified participants aged ≥16 years into three groups: CNS tumor and non-CNS malignancy with and without CNS-directed treatment. We analyzed educational attainment, employment outcome and special schooling. Subgroup analyses included survivors aged ≥25 years. Results: We analyzed 2154 survivors, including 329 (15%) CNS tumor survivors, 850 (40%) non-CNS tumor survivors with and 975 (45%) without CNS-directed treatment. Fewer CNS tumor survivors aged ≥25 years reached tertiary education (44%) compared to those without CNS-directed treatment (51%) but performed similar to survivors with CNS-directed treatment (42%). Among CNS tumor survivors, 36 (14%) received special schooling. Higher parental education was associated with higher levels in survivors. Employment outcome did not significantly differ between the three diagnostic groups. A higher proportion of CNS tumor survivors received disability pension or were unemployed. Conclusions: Our findings suggest that CNS tumor survivors need more time to achieve their highest educational level. This should influence clinical care of these survivors by offering vocational counseling.

Cohort-based association study of germline genetic variants with acute and chronic health complications of childhood cancer and its treatment: Genetic Risks for Childhood Cancer Complications Switzerland (GECCOS) study protocol

IntroductionChildhood cancer and its treatment may lead to various health complications. Related impairment in quality of life, excess in deaths and accumulated healthcare costs are relevant. Genetic variations are suggested...

Validation of questionnaire-reported chest wall abnormalities with a telephone interview in Swiss childhood cancer survivors

BackgroundChest wall abnormalities are a poorly studied complication after treatment for childhood cancer. Chest wall abnormalities are not well-described in the literature, and little is known on the impact on daily life of survivors.MethodsWe investigated prevalence and risk factors of chest wall abnormalities in childhood cancer survivors in a nationwide, population-based cohort study (Swiss Childhood Cancer Survivor Study) with a questionnaire survey. We then interviewed a nested sample of survivors to validate types of chest wall abnormalities and understand their impact on the daily life of survivors.ResultsForty-eight of 2382 (95%CI 2–3%) survivors reported a chest wall abnormality. Risk factors were older age at cancer diagnosis (16–20 years; OR 2.5, 95%CI 1.0–6.1), lymphoma (OR 3.8, 95%CI 1.2–11.4), and central nervous system tumors (OR 9.5, 95%CI 3.0–30.1) as underlying disease, and treatment with thoracic radiotherapy (OR 2.0, 95%CI 1.0–4.2), surgery to the chest (OR 4.5, 95%CI 1.8–11.5), or chemotherapy (OR 2.9, 95%CI 1.0–8.1). The nature of the chest wall abnormalities varied and included thoracic wall deformities (30%), deformations of the spine (5%) or both (55%), and scars (10%). Chest wall abnormalities affected daily life in two thirds (13/20) of those who reported these problems and necessitated medical attention for 15 (75%) survivors.ConclusionIt is important that, during follow-up care, physicians pay attention to chest wall abnormalities, which are rare late effects of cancer treatment, but can considerably affect the well-being of cancer survivors.

Transplant characteristics and self-reported pulmonary outcomes in Swiss childhood cancer survivors after hematopoietic stem cell transplantation\u2014a cohort study

Childhood cancer survivors treated with hematopoietic stem cell transplantation are at high risk for pulmonary morbidity and mortality. In this retrospective study we described transplant characteristics of pediatric patients who underwent hematopoietic stem cell transplantation in Switzerland and how these characteristics changed over time, compared self-reported pulmonary outcomes between transplanted and non-transplanted survivors, and investigated risk factors for the reported pulmonary outcomes. As part of the population-based Swiss Childhood Cancer Survivor Study, we sent questionnaires to all ≥5-year childhood cancer survivors diagnosed 1976–2010 at age ≤20 years. We included 132 transplanted survivors and 368 matched non-transplanted survivors. During the study period transplant characteristics changed, with decreasing use of total body irradiation and increased use of peripheral blood stem cells and mismatched and unrelated donors as transplant source. One-fifth of transplanted survivors (20%, 95%CI 13–27%) and 18% of non-transplanted survivors (95%CI 13–21%) reported at least one pulmonary outcome. None of the analyzed factors was significantly associated with an increased risk of pulmonary outcomes in multivariable analysis. We found that pulmonary outcomes were frequently reported in transplanted and non-transplanted childhood cancer survivors, indicating a strong need for long-term pulmonary follow-up care.

Physical activity and screen time in children who survived cancer: A report from the Swiss Childhood Cancer Survivor Study.

Physical activity (PA) can reduce the risk of chronic adverse health conditions in childhood cancer survivors. We examined PA and sedentary screen time behavior in a nationwide study in Switzerland. The Swiss Childhood Cancer Survivor Study sent questionnaires to parents of all Swiss resident ≥5-year survivors diagnosed between 1995 and 2010. We assessed PA including compulsory school sport, recreational sport, commuting to school, and time spent with screen media in those aged 5-15 years, and compared results with international recommendations. We included 766 survivors with a median age at diagnosis of 2.8 (interquartile range 1.4-5.0) years and a median age at study of 12.5 (10.0-14.3) years. Median PA time was 7.3 (4.8-10.0) h/week and median screen time was 82 (45-120) min/day. Compulsory school sport hours and walking or cycling to school contributed significantly to total PA. Note that 55% of survivors met PA and 68% screen time recommendations. PA was lower for children living in regions of Switzerland speaking French or Italian compared to German, and for those who had a relapse or musculoskeletal/neurological conditions. Screen time was higher in males, children with lower parental education, and a migration background. PA and sedentary screen watching were associated with social factors, and PA also with clinical risk factors. Structural preventions that afford active commuting to school and sufficient school sport are essential, as is counseling vulnerable survivor groups such as those with musculoskeletal and neurological problems, and those who have had a relapse.

Nutritional Assessment of Childhood Cancer Survivors (the Swiss Childhood Cancer Survivor Study-Nutrition): Protocol for a Multicenter Observational Study.

BackgroundChildhood cancer survivors are at high risk of developing adverse late health effects. Poor nutritional intake may contribute to this risk, but information about dietary intake is limited.ObjectiveThis study will assess childhood cancer survivors’ dietary intake and compare two dietary assessment tools: a self-reported food frequency questionnaire, and dietary measurements from urine spot samples.MethodsIn a substudy of the Swiss Childhood Cancer Survivor Study (SCCSS), SCCSS-Nutrition, we assessed childhood cancer survivors’ dietary intake via a validated food frequency questionnaire. We sent a urine spot collection kit to a subset of 212 childhood cancer survivors from the French-speaking region of Switzerland to analyze urinary sodium, potassium, urea, urate, creatinine, and phosphate content. We will compare the food frequency questionnaire results with the urine spot analyses to quantify childhood cancer survivors’ intake of various nutrients. We collected data between March 2016 and March 2018.ResultsWe contacted 1599 childhood cancer survivors, of whom 919 (57.47%) returned a food frequency questionnaire. We excluded 11 childhood cancer survivors who were pregnant or were breastfeeding, 35 with missing dietary data, and 71 who had unreliable food frequency questionnaire data, resulting in 802 childhood cancer survivors available for food frequency questionnaire analyses. To a subset of 212 childhood cancer survivors in French-speaking Switzerland we sent a urine spot collection kit, and 111 (52.4%) returned a urine sample. We expect to have the results from analyses of these samples in mid-2019.ConclusionsThe SCCSS-Nutrition study has collected in-depth dietary data that will allow us to assess dietary intake and quality and compare two dietary assessment tools. This study will contribute to the knowledge of nutrition among childhood cancer survivors and is a step toward surveillance guidelines and targeted nutritional recommendations for childhood cancer survivors in Switzerland.Trial RegistrationClinicalTrials.gov NCT03297034; https://clinicaltrials.gov/ct2/show/NCT03297034International Registered Report Identifier (IRRID)DERR1-10.2196/14427

Cardiovascular disease after childhood acute lymphoblastic leukaemia: a cohort study.

Cardiovascular diseases (CVD) increase late morbidity and mortality in survivors of acute lymphoblastic leukaemia (ALL). We compared the risk of CVD in ALL survivors to siblings, examined time trends, quantified treatment-related risks, and investigated whether risk extends beyond patients treated with anthracyclines and chest radiotherapy. The Swiss Childhood Cancer Survivor Study assessed CVD by patient questionnaire in 5-year ALL survivors diagnosed between 1976 and 2005 and their siblings. Participants were asked whether a physician had ever told them that they had hypertension, arrhythmia, heart failure, myocardial infarction, angina pectoris, stroke, thrombosis or valvular problems. We investigated treatment-related risk factors for CVD using multivariable logistic regression, adjusting for demographic and socioeconomic factors, BMI, smoking, diabetes mellitus, alcohol consumption and physical activity. We contacted 707 survivors and 1299 siblings, 511 (72%) and 709 (55%) of whom responded, respectively. Survivors had a higher risk of developing CVD than siblings (odds ratio [OR] 1.9, 95% confidence interval 1.3&ndash;2.8), in particular heart failure (OR 13.9, 1.8&ndash;107.4). Compared to patients treated 1976&ndash;85, the risk of CVD was 1.4 (0.7&ndash;2.8) for those treated 1985&ndash;1994 and 1.5 (0.6&ndash;3.7) for those treated 1995&ndash;2005. The overall CVD risks after anthracycline treatment (OR 3.1, 2.0&ndash;4.7), haematopoietic stem cell transplantation (OR 8.0, 2.4&ndash;26.9) or relapse (OR 4.1, 1.9&ndash;8.8) were increased compared to those of siblings, while the CVD risks of survivors treated without anthracycline or chest radiotherapy were similar (OR 1.0; 0.5&ndash;2.0). Despite attempts to reduce cardiotoxicity in childhood cancer treatment, CVD risks in ALL survivors treated more recently do not seem to have declined.

Relationship status and quality of the partner relationship in parents of long-term childhood cancer survivors: the Swiss Childhood Cancer Survivor Study - Parents

Relationship status and quality of the partner relationship in parents of long-term childhood cancer survivors: the Swiss Childhood Cancer Survivor Study - Parents

Hearing loss and quality of life in survivors of paediatric CNS tumours and other cancers.

Hearing loss, a complication of cancer treatment, may reduce health-related quality of life (HRQoL), especially in childhood cancer survivors of central nervous system (CNS) tumours who often have multiple late effects. We examined the effect of hearing loss on HRQoL in young survivors of CNS and other childhood cancers. Within the Swiss Childhood Cancer Survivor Study, we sent questionnaires about hearing loss and HRQoL (KIDSCREEN-27) to parents of survivors aged 8-15years. We stratified the effect of hearing loss on HRQoL by cancer diagnosis, using multivariable logistic regression and adjusting for sociodemographic and clinical factors. Hearing loss was associated with impaired physical well-being [unadjusted estimated differences - 4.6 (CI - 9.2, - 0.1); adjusted - 4.0 (CI - 7.6, - 0.3)] and peers and social support [unadjusted - 6.7 (CI - 13.0, - 0.3); adjusted - 5.0 (CI - 10.5, 0.9)] scores in survivors of CNS tumours (n = 123), but not in children diagnosed with other cancers (all p-values > 0.20, n = 577). Clinicians should be alert to signs of reduced physical well-being and impaired relationships with peers. Especially survivors of CNS tumours may benefit most from strict audiological monitoring and timely intervention to mitigate secondary consequences of hearing loss on HRQoL.

Prevalence and reasons for smoking in adolescent Swiss childhood cancer survivors.

Smoking harms health, particularly that of childhood cancer survivors, who face risk of pulmonary and cardiovascular diseases because of chemotherapy and radiotherapy to the chest. This nationwide study assessed smoking habits and reasons for smoking in adolescent survivors and healthy peers. As part of the Swiss Childhood Cancer Survivor Study, we sent a questionnaire to all Swiss resident survivors, who were aged 16-19 years. We compared smoking status and reasons for smoking between 511 survivors, 141 of their siblings, and 1,727 adolescents in a representative population-based study, the Tobacco Monitoring Switzerland (TMS). Current smoking was less prevalent in survivors (17%) and their siblings (17%) compared with TMS (32%). Survivors and TMS adolescents gave similar reasons for smoking. Stress control, smoking being a habit, and good taste were the reasons for smoking cited most often in both groups. Peer smoking was more important in survivors (49%) than in TMS (34%, P=0.004). Most important reasons for not smoking in both groups were smoking being unhealthy and not wanting to be addicted. In Switzerland, survivors smoke as often as their siblings but less than the general population. Peer smoking was a more important reason for smoking in survivors than in the general population, suggesting that reducing smoking in peers could result in a reduction of smoking in survivors. Overall, reasons for smoking were very similar, thus interventions to reduce smoking in survivors could be the same as those used in the general population.

No evidence of overweight in long‐term survivors of childhood cancer after glucocorticoid treatment

Glucocorticoids can lead to weight gain during cancer treatment, but to the authors' knowledge, little is known regarding their long-term effects in childhood cancer survivors (CCS). As part of the Swiss Childhood Cancer Survivor Study, the authors sent a questionnaire to CCS aged <21 years at diagnosis who were residing in Switzerland, had survived ≥5 years, and were aged 15 to 45 years at the time of the survey. Cumulative doses of glucocorticoids were assessed from medical records and study protocols and body mass index was calculated from self-reported height and weight at the time of the survey. The authors compared the prevalence of overweight between CCS, their siblings, and the general population (Swiss Health Survey [SHS]) and investigated the association between overweight and treatment-related risk factors using multivariable logistic regression. The study included 1936 CCS, 546 siblings, and 9591 SHS participants. The median age of the CCS at the time of the survey was 24 years (interquartile range, 20-31 years) and the median time since diagnosis was 17 years (interquartile range, 12-22 years). At the time of the survey, approximately 26% of CCS were overweight, a percentage that was comparable to that among siblings (24%) and the SHS participants (25%). The prevalence of overweight was 24% in CCS treated with glucocorticoids only (686 CCS), 37% in those treated with cranial radiotherapy (CRT) (127 CCS), and 49% in those who received treatment with both glucocorticoids and CRT (101 CCS) (P < .001). The authors found no evidence of a dose-response relationship between cumulative glucocorticoid doses and overweight and no evidence that CRT modified the effect of the cumulative glucocorticoid dose on overweight. The results of the current study suggest that glucocorticoids used for the treatment of childhood cancer are not associated with long-term risk of overweight.

Overweight in childhood cancer survivors: the Swiss Childhood Cancer Survivor Study

An increased risk of becoming overweight has been reported for childhood cancer survivors (CCSs), in particular leukemia survivors, although the evidence is inconclusive. We assessed the prevalence of overweight in CCSs, with a focus on leukemia survivors, compared it with their peers, and determined potential risk factors. As part of the Swiss Childhood Cancer Survivor Study, we sent a questionnaire between 2007 and 2013 to all Swiss resident CCSs aged <21 y at diagnosis who had survived ≥5 y. We calculated body mass index (BMI) from medical records at diagnosis and self-reported heights and weights at survey. We calculated BMI z scores by using Swiss references for children and compared overweight prevalence in CCSs, their siblings, and the general population with the use of the Swiss Health Survey (SHS) and assessed risk factors for being overweight by using multivariable logistic regression. The study included 2365 CCSs, 819 siblings, and 9591 SHS participants. At survey, at an average of 15 y after diagnosis, the prevalence of overweight in CCSs overall (26%) and in leukemia survivors (26%) was similar to that in siblings (22%) and the general population (25%). Risk factors for being overweight in CCSs were male sex (OR: 1.8; 95% CI: 1.5, 2.1), both young (OR for ages 5-14y: 1.6; 95% CI: 1.2, 2.3) and older (range-OR for ages 25-29 y: 1.7; 95% CI: 1.2, 2.4; OR for ages 40-45 y: 4.0; 95% CI: 2.5, 6.5) age at study, lower education (OR: 1.4; 95% CI: 1.1, 1.8), migration background (OR: 1.3; 95% CI: 1.1, 1.7), and no sports participation (OR: 1.4; 95% CI: 1.1, 1.7). Risk factors for overweight were similar in peers. CCSs treated with cranial radiotherapy (≥20 Gy) were more likely to be overweight than their peers (OR: 1.6; 95% CI: 1.2, 2.2). The prevalence of and risk factors for being overweight are similar in long-term CCSs and their peers. This suggests that prevention methods can be the same as in the general population. An important exception is CCSs treated with cranial radiotherapy ≥20 Gy who may need extra attention during follow-up care. This study was registered at clinicaltrials.gov as NCT03297034.

No evidence of response bias in a population-based childhood cancer survivor questionnaire survey - Results from the Swiss Childhood Cancer Survivor Study.

PurposeThis is the first study to quantify potential nonresponse bias in a childhood cancer survivor questionnaire survey. We describe early and late responders and nonresponders, and estimate nonresponse bias in a nationwide questionnaire survey of survivors.MethodsIn the Swiss Childhood Cancer Survivor Study, we compared characteristics of early responders (who answered an initial questionnaire), late responders (who answered after ≥1 reminder) and nonresponders. Sociodemographic and cancer-related information was available for the whole population from the Swiss Childhood Cancer Registry. We compared observed prevalence of typical outcomes in responders to the expected prevalence in a complete (100% response) representative population we constructed in order to estimate the effect of nonresponse bias. We constructed the complete population using inverse probability of participation weights.ResultsOf 2328 survivors, 930 returned the initial questionnaire (40%); 671 returned the questionnaire after ≥1reminder (29%). Compared to early and late responders, we found that the 727 nonresponders (31%) were more likely male, aged <20 years, French or Italian speaking, of foreign nationality, diagnosed with lymphoma or a CNS or germ cell tumor, and treated only with surgery. But observed prevalence of typical estimates (somatic health, medical care, mental health, health behaviors) was similar among the sample of early responders (40%), all responders (69%), and the complete representative population (100%). In this survey, nonresponse bias did not seem to influence observed prevalence estimates.ConclusionNonresponse bias may play only a minor role in childhood cancer survivor studies, suggesting that results can be generalized to the whole population of such cancer survivors and applied in clinical practice.

Validation of questionnaire-reported hearing with medical records: A report from the Swiss Childhood Cancer Survivor Study.

BackgroundHearing loss is a potential late effect after childhood cancer. Questionnaires are often used to assess hearing in large cohorts of childhood cancer survivors and it is important to know if they can provide valid measures of hearing loss. We therefore assessed agreement and validity of questionnaire-reported hearing in childhood cancer survivors using medical records as reference.ProcedureIn this validation study, we studied 361 survivors of childhood cancer from the Swiss Childhood Cancer Survivor Study (SCCSS) who had been diagnosed after 1989 and had been exposed to ototoxic cancer treatment. Questionnaire-reported hearing was compared to the information in medical records. Hearing loss was defined as ≥ grade 1 according to the SIOP Boston Ototoxicity Scale. We assessed agreement and validity of questionnaire-reported hearing overall and stratified by questionnaire respondents (survivor or parent), sociodemographic characteristics, time between follow-up and questionnaire and severity of hearing loss.ResultsQuestionnaire reports agreed with medical records in 85% of respondents (kappa 0.62), normal hearing was correctly assessed in 92% of those with normal hearing (n = 249), and hearing loss was correctly assessed in 69% of those with hearing loss (n = 112). Sensitivity of the questionnaires was 92%, 74%, and 39% for assessment of severe, moderate and mild bilateral hearing loss; and 50%, 33% and 10% for severe, moderate and mild unilateral hearing loss, respectively. Results did not differ by sociodemographic characteristics of the respondents, and survivor- and parent-reports were equally valid.ConclusionsQuestionnaires are a useful tool to assess hearing in large cohorts of childhood cancer survivors, but underestimate mild and unilateral hearing loss. Further research should investigate whether the addition of questions with higher sensitivity for mild degrees of hearing loss could improve the results.