Auditory complications among childhood cancer survivors and health-related quality of life: a PanCareLIFE study.

PURPOSE: Auditory complications are potential side effects from childhood cancer treatment. Yet, limited evidence exists about the impact of auditory complications-particularly tinnitus-on health-related quality of life (HRQoL) among childhood cancer survivors (CCS). We determined the prevalence of hearing loss and tinnitus in the European PanCareLIFE cohort of CCS and examined its effect on HRQoL. METHODS: We included CCS from four European countries who were diagnosed at age ≤ 18 years; survived ≥ 5 years; and aged 25-44 years at study. We assessed HRQoL (Short Form 36), hearing loss, and tinnitus using questionnaires. We used multivariable linear regression to examine associations between these two auditory complications and HRQoL adjusting for socio-demographic and clinical factors. RESULTS: Our study population consisted of 6,318 CCS (53% female; median age at cancer diagnosis 9 years interquartile range [IQR] 5-13 years) with median age at survey of 31 years (IQR 28-35 years). Prevalence was 7.5% (476/6,318; confidence interval [CI]: 6.9-8.2) for hearing loss and 7.6% (127/1,668; CI: 6.4-9.0) for tinnitus. CCS with hearing loss had impaired physical (coefficient [coef.] -4.3, CI: -7.0 to -1.6) and mental (coef. -3.2, CI: -5.5 to -0.8) HRQoL when compared with CCS with normal hearing. Tinnitus was associated with impaired physical (coef. -8.2, CI: -11.8 to -4.7) and mental (coef. -5.9, CI: -8.8 to -3.1) HRQoL. CONCLUSION: We observed reduced HRQoL among CCS with hearing loss and tinnitus. Our findings indicate timely treatment of hearing loss and tinnitus may contribute to quality of life of survivors. IMPLICATIONS FOR CANCER SURVIVORS: CCS who experience auditory complications should be counseled about possible therapeutic and supportive measures during follow-up care.

Severity of hearing loss after platinum chemotherapy in childhood cancer survivors.

BACKGROUND: Hearing loss is a potential side effect from childhood cancer treatment. We described the severity of hearing loss assessed by audiometry in a representative national cohort of childhood cancer survivors (CCS) and identified clinical risk factors. PROCEDURE: We included all CCS from the Swiss Childhood Cancer Registry who were diagnosed ≤18 age and treated with platinum-based chemotherapy between 1990 and 2014. We extracted audiograms, treatment-related information, and demographic data from medical records. Two reviewers independently assessed the severity of hearing loss at latest follow-up using the Münster Ototoxicity Scale. We used ordered logistic regression to identify clinical risk factors for severity of hearing loss. RESULTS: We analyzed data from 270 CCS. Median time from cancer diagnosis to last audiogram was 5 years (interquartile range 2.5-8.1 years). We found 53 (20%) CCS with mild, 78 (29%) with moderate, and 75 (28%) with severe hearing loss. Higher severity grades were associated with (a) younger age at cancer diagnosis (odds ratio [OR] 5.4, 95% confidence interval [CI]: 2.5-12.0 for <5 years); (b) treatment in earlier years (OR 4.8, 95% CI: 2.1-11.0 for 1990-1995); (c) higher cumulative cisplatin doses (OR 13.5, 95% CI: 4.7-38.8 for >450 mg/m2 ); (d) concomitant cranial radiation therapy (CRT) (OR 4.4, 95% CI: 2.5-7.8); and (e) hematopoietic stem cell transplantation (HSCT) (OR 2.7, 95% CI: 1.0-7.2). CONCLUSION: Three of four CCS treated with platinum-based chemotherapy experienced some degree of hearing loss. We recommend closely monitoring patient's hearing function if treated at a young age with high cumulative cisplatin doses, and concomitant CRT as part of long-term care.

[The Bavarian Midwives Bonus - A Contribution to Monitoring Freelance Midwifery Care in Bavaria]. / Der Bayerische Hebammenbonus ­ Beitrag zum Monitoring der freiberuflichen Hebammenversorgung in der Geburtshilfe in Bayern.

BACKGROUND: Supply bottlenecks in midwifery care could intensify due to increasing birth rates in Bavaria. Midwives are already fully booked and can only take care of pregnant women by increasing their working hours. The situation of self-employed midwives is particularly precarious. Bavaria has been supporting self-employed midwives by granting the Bavarian midwife bonus of up to € 1,000 per year since September 2018. This analysis aims to describe the current status of the unique Bavarian midwife bonus scheme and to describe current programmes to improve care in obstetrics in Bavaria. METHODS: All midwives who were self-employed, had their primary residence in Bavaria and had cared for at least four births in the application year were eligible for the Bavarian midwife bonus. We used data from application years 2017 and 2018 to describe the number of applications per year and the socio-demographic characteristics of the applicants and analyzed the regional distribution of the midwives and the regional supply density. RESULTS: In 2017, a total of 723 midwives received the bonus, in 2018 already 852 midwives. The average age of the applicants, who were exclusively female, was 44 years in the application year 2018. In 2018, 37 midwives per 100,000 women aged 15-44 were self-employed in obstetrics in Bavaria. A regional comparison showed an above-average supply density in the Upper Palatinate (52) and Lower Bavaria (54) and a below-average supply density in the Upper Franconia (13). CONCLUSION: The analysis of the Bavarian midwifery bonus enables a glance, albeit limited, into the supply situation of self-employed midwives in Bavaria. Whether the political instruments can improve the supply situation in Bavaria have to be evaluated using newly generated representative data. Central registration of midwives in Bavaria could be useful.

6-year course of sleep homeostasis in a case with epilepsy-aphasia spectrum disorder.

The epilepsy-aphasia spectrum consists of epilepsies with a strong activation of epileptic discharges during non-rapid-eye-movement (NREM) sleep, variable seizure burden and language problems. The homeostatic decrease of slow waves (SW) during NREM sleep (i.e. their amplitude/slope and power) has been related to brain recovery and cognitive function. Epileptic discharges during NREM-sleep were related to an impairment of the decrease of the slope of SW and to cognitive deficits. In this longitudinal case study, we aim to relate this electrophysiological marker, i.e. overnight change of slope of SW, to imaging and behavior. We report a young girl with a fluctuating course in the epilepsy-aphasia spectrum, ranging from the benign end with self-limited childhood epilepsy with centrotemporal spikes (SLECTS) to the severe end with epileptic encephalopathy with continuous spike waves during sleep (CSWS) with two phases of cognitive regression. She was documented over a period of six years including 12 PSGs, six language fMRIs and seven neuropsychological assessments. We longitudinally studied focal and total spike wave index (SWI), detected SW during NREM sleep, calculated their slopes (first and last hour of NREM sleep and overnight change). Deterioration of overnight decrease of the slope of SW was paralleled by the occurrence of the EEG picture of bilateral synchronous electrical status epilepticus during sleep (ESES) and neuropsychological deficits, and this impairment was reversible with resolution of ESES and was accompanied by cognitive improvement. A laterality switch from left to right sided language dominance occurred during recovery from the second regression phase. This might reflect a compensating process. Later, the laterality switched back to the left, possibly facilitated by a low SWI on the left hemisphere. The qualitative analysis of this case supports the view that the longitudinal course of the overnight change of the slope of SW, as an objective, quantitative EEG measure, is related to the course of cognitive function and functional language MR analysis.

Experiences, opinions and expectations of health care providers towards an intensive care unit follow-up clinic: Qualitative study and online survey.

OBJECTIVES: Independent of the underlying disease, intensive care unit survivors often suffer from cognitive, physical and mental impairments, also known as post-intensive care syndrome (PICS). Specific follow-up services are recommended for these patients. This study aims to capture the perspectives of health care providers on the development of the first intensive care unit follow-up-clinic in Germany. RESEARCH METHODOLOGY: A qualitative study with six focus groups (n = 41) and six expert interviews, followed by a quantitative survey was conducted, involving nine different professions. Qualitative and quantitative data were analysed using thematic analysis and descriptive statistics, respectively. FINDINGS: Participants described aftercare of former intensive care unit patients as complex and appreciated the idea of an intensive care unit follow-up clinic to improve continuity of care and multidisciplinary collaboration. The favoured model combined diagnostics and targeted referral of patients to specialists and therapists with the provision of information. In the survey, participants disagreed on how to implement this referral system but agreed that assessments should be multidimensional. CONCLUSION: The necessity of and important criteria for the design of an intensive care unit follow-up clinic were identified. We will integrate these data with further evidence to develop a concept for a complex intervention.

Incidence Estimation in Post-ICU Populations: Challenges and Possible Solutions When Using Claims Data.

BACKGROUND: New or worsening cognitive, physical and/or mental health impairments after acute care for critical illness are referred to as "post-intensive care syndrome" (PICS). Little is known about the incidence of its components, since it is challenging to recruit patients after intensive care unit (ICU) treatment for observational studies. Claims data are particularly suited to achieve incidence estimates in difficult-to-recruit groups. However, some limitations remain when using claims data for empirical research on the outcome of ICU treatment. The objective of this article is to describe three challenges and possible solutions for the estimation of the incidence of PICS based on claims data METHODOLOGICAL CHALLENGES: THE PRESENCE OF COMPETING RISK BY DEATH, INVESTIGATING A SYNDROME AND DEALING WITH INTERVAL CENSORING: First, in (post) ICU populations the assumption of independence between the event of interest (diagnosis of PICS component) and the competing event (death) is violated. Competing risk is an event whose occurrence precludes the event of interest to be observed, and in ICU populations, death is a frequent secondary event. Methods that estimate incidence in the presence of competing risks are well-established but have not been applied to the scenario described above. Second, PICS is a complex syndrome and represented by various ICD-10 (International Classification of Diseases, 10th Revision) disease codes. The operationalization of this syndrome (case identification) and the validation of cases are particularly challenging. Third, another major challenge is that the exact date of the event of interest is not available in claims data. It is only known that the event occurred within a certain interval. This feature is called interval censoring. Recently, methods have been developed that address informative censoring due to competing risks in the presence of interval censoring. We will discuss how these methods could be used to tackle the problem when estimating PICS components. Alternatively, it could be possible to assign an exact date for each diagnosis by combining the diagnosis with the exact date of prescriptions of the respective medicines and/or medical services. CONCLUSION: Estimating incidence in post-ICU populations entails various methodological issues when using claims data. Investigators need to be aware of the presence of competing risks. The application of internal validation criteria to operationalize the event of interest is crucial to achieve reliable incidence estimates. The problem of interval censoring can be solved either by statistical methods or by combining information from different sources.

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

BACKGROUND: 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. PROCEDURES: 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. RESULTS: 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. CONCLUSIONS: 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.

Participation in People Living With Spinal Cord Injury in Switzerland: Degree and Associated Factors.

OBJECTIVE: To describe different domains of participation such as productive, leisure and social activities and describe sociodemographic and spinal cord injury (SCI)-related characteristics that are associated with participation in these domains in a large sample of community-dwelling individuals with SCI in Switzerland. DESIGN: Cross-sectional population-based survey within the Swiss Spinal Cord Injury Cohort Study. Participation in major life domains was measured by the Utrecht Scale for Evaluation of Rehabilitation-Participation (USER-Participation). Univariable unconditional analysis and unbiased recursive partitioning were used to identify the predominant associations of sociodemographic and SCI-related characteristics with multiple dimensions of participation. SETTING: Community. PARTICIPANTS: Swiss residents aged 16 years or older and living with traumatic or nontraumatic SCI (N=1549). INTERVENTIONS: Not applicable. MAIN OUTCOME MEASURE: The USER-Participation, a 32-item self-report questionnaire with 3 scales (Frequency, Restrictions, and Satisfaction) to assess key domains of participation (productive, leisure, social). RESULTS: Frequency (median 34.5 out of 100) in productive, outdoor leisure, and social activities was reduced with distinctive perceived restrictions in work and education, sports, and partner relationships. Domestic leisure activities (65.4%) and maintaining social relationships (76.1%) were those activities most often performed and with least perceived restrictions. Participants were generally satisfied with their current daily life activities. Lower scores across all participation scales were associated with more severe SCI, higher age, being female, not having a partner, and lower level of education. CONCLUSIONS: This study provides a thorough analysis of participation in major life domains of individuals with SCI in Switzerland. Different risk groups for reduced levels in participation in productive, leisure, and social activities were identified. This population-based evidence is instrumental to the better targeting of rehabilitation and policy interventions that aim to improve community participation.

Recommendations for ototoxicity surveillance for childhood, adolescent, and young adult cancer survivors: a report from the International Late Effects of Childhood Cancer Guideline Harmonization Group in collaboration with the PanCare Consortium.

Childhood, adolescent, and young adult (CAYA) cancer survivors treated with platinum-based drugs, head or brain radiotherapy, or both have an increased risk of ototoxicity (hearing loss, tinnitus, or both). To ensure optimal care and reduce consequent problems-such as speech and language, social-emotional development, and learning difficulties-for these CAYA cancer survivors, clinical practice guidelines for monitoring ototoxicity are essential. The implementation of surveillance across clinical settings is hindered by differences in definitions of hearing loss, recommendations for surveillance modalities, and remediation. To address these deficiencies, the International Guideline Harmonization Group organised an international multidisciplinary panel, including 32 experts from ten countries, to evaluate the quality of evidence for ototoxicity following platinum-based chemotherapy and head or brain radiotherapy, and formulate and harmonise ototoxicity surveillance recommendations for CAYA cancer survivors.

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

PURPOSE: 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. METHODS: Within the Swiss Childhood Cancer Survivor Study, we sent questionnaires about hearing loss and HRQoL (KIDSCREEN-27) to parents of survivors aged 8-15 years. We stratified the effect of hearing loss on HRQoL by cancer diagnosis, using multivariable logistic regression and adjusting for sociodemographic and clinical factors. RESULTS: 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). CONCLUSION: 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.

Monitoring pulmonary health in Swiss childhood cancer survivors.

BACKGROUND: Childhood cancer survivors are at increased risk for pulmonary morbidity and mortality. International guidelines recommend pulmonary function tests (PFT) during follow-up care. This nationwide study assessed how many children received PFT within 5 years after pulmotoxic treatment in Switzerland, types of tests, and predictors for testing. METHODS: We included all children from the Swiss Childhood Cancer Registry who were diagnosed with cancer from 1990 to 2013 at age 0-16 years, survived for ≥2 years from diagnosis, and had pulmotoxic chemotherapy with bleomycin, busulfan, nitrosoureas, and/or chest radiotherapy. We searched medical records in all Swiss pediatric oncology clinics for PFT (spirometry, plethysmography, diffusion capacity of carbon monoxide [DLCO]) and treatment details. RESULTS: We found medical records for 372 children, of whom 147 had pulmotoxic chemotherapy and 323 chest radiotherapy. Only 185 had plethysmography and/or spirometry (50%), 122 had DLCO (33%). Testing varied by cancer center from 3% to 79% (P = 0.001). Central nervous system tumor survivors and those not treated according to study protocols had less plethysmography and/or spirometry (odds ratio (OR) 0.3 and 0.3), lymphoma survivors and those who were symptomatic had more PFT (plethysmography and/or spirometry: OR 5.9 and 8.7; DLCO: OR 3.4 and 2.3). Cumulative incidence (CuI) of PFT was 52% in the first 5 years after pulmotoxic treatment; most of the tests were done in the first 2 years after treatment (CuI 44%). CONCLUSION: Only half of the survivors exposed to pulmotoxic treatment have been followed up with PFT in Switzerland. We need to optimize, update, and implement monitoring guidelines.

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

Background: An increased risk of becoming overweight has been reported for childhood cancer survivors (CCSs), in particular leukemia survivors, although the evidence is inconclusive. Objective: We assessed the prevalence of overweight in CCSs, with a focus on leukemia survivors, compared it with their peers, and determined potential risk factors. Design: 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. Results: 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 5-14 y: 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). Conclusions: 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.

Audiological monitoring in Swiss childhood cancer patients.

BACKGROUND: Full audiological monitoring is the best strategy to detect hearing loss early and to provide timely intervention in the absence of a clinical method of otoprotection. Full monitoring requires audiological evaluation before, and then during and after ototoxic cancer treatment. In a worldwide context of monitoring protocols that vary substantially, we analyzed the audiological monitoring of childhood cancer patients over the last decade across treatment centers in Switzerland. PROCEDURE: We retrospectively searched for audiological evaluations in all nine Swiss Pediatric Oncology Centers. We analyzed proportions of patients who had audiological monitoring and described type and timing of monitoring. We determined predictors of audiological monitoring using multivariable logistic regression and described time trends. RESULTS: We included 185 patients from the Swiss Childhood Cancer Registry diagnosed from 2005 to 2013 who had platinum chemotherapy and/or cranial radiation ≥30 Gray and who were alive at time of study. Less than half of children, 43%, had full audiological monitoring (before, during, and after treatment), while 72% were tested after cancer treatment. Nonstudy patients were less likely to have had monitoring in all phases of cancer treatment. Patients who received treatment with cisplatin or both platinum chemotherapy and cranial radiation were more likely to have had monitoring after treatment. Monitoring during and after treatment increased over the study period, but monitoring before treatment was insufficient in all time periods. CONCLUSIONS: Our population-based study indicates that audiological monitoring is insufficient in Switzerland, particularly for nonstudy patients. Clinicians must become more aware of the importance of full audiological monitoring.

Long-term pulmonary disease among Swiss childhood cancer survivors.

BACKGROUND: Pulmonary diseases are potentially severe late complications of childhood cancer treatment that increase mortality risk among survivors. This nationwide study assesses the prevalence and incidence of pulmonary diseases in long-term childhood cancer survivors (CCS) and their siblings, and quantifies treatment-related risks. METHODS: As part of the Swiss Childhood Cancer Survivor Study, we studied CCS who were diagnosed between 1976 and 2005 and alive at least 5 years after diagnosis. We compared prevalence of self-reported pulmonary diseases (pneumonia, chest wall abnormalities, lung fibrosis, emphysema) between CCS and their siblings, calculated cumulative incidence of pulmonary diseases using the Kaplan-Meier method, and determined risk factors using multivariable logistic regression. RESULTS: CCS reported more pneumonias (10% vs. 7%, P = 0.020) and chest wall abnormalities (2% vs. 0.4%, P = 0.003) than siblings. Treatment with busulfan was associated with prevalence of pneumonia (odds ratio [OR] 4.0, 95% confidence interval [CI] 1.1-14.9), and thoracic surgery was associated with chest wall abnormalities and lung fibrosis (OR 4.1, 95% CI 1.6-10.7 and OR 6.3, 95% CI 1.7-26.6). Cumulative incidence of any pulmonary disease after 35 years of follow-up was 21%. For pneumonia, the highest cumulative incidence was seen in CCS treated with both pulmotoxic chemotherapy and radiotherapy to the thorax (23%). CONCLUSION: This nationwide study in CCS found an increased risk for pulmonary diseases, especially pneumonia, while still young, which indicates that CCS need long-term pulmonary follow-up.

Low adherence to dietary recommendations in adult childhood cancer survivors.

BACKGROUND & AIMS: Poor diet may increase the risk that childhood cancer survivors (CCS) will suffer from chronic disease. We compared adherence to national dietary recommendations between CCS, their siblings and the Swiss population, identified determinants of adherence, and assessed the association of adherence with cardiovascular disease (CVD) risk profiles. METHODS: As part of the Swiss Childhood Cancer Survivor Study (SCCSS), a questionnaire was sent to all Swiss resident CCS aged <21 years at diagnosis, who survived ≥5 years and were 16-45 years old at the time of the survey. We compared dietary adherence between CCS, their siblings and participants in the Swiss Health Survey (SHS), a representative survey of the general population. A multivariable logistic regression was used to assess characteristics associated with dietary adherence. We sorted CCS into four kinds of CVD risk groups based on type of treatment (anthracyclines, chest irradiation, a combination, or neither). RESULTS: We included 1864 CCS, 698 siblings and 8258 participants of the general population. Only 43% of the CCS met the recommended dietary intakes for meat, 34% for fruit, 30% for fish, 18% for dairy products, 11% for vegetables, and 7% for combined fruit and vegetables. Results were similar for both control groups. In all groups, dietary adherence was associated with gender, parental education, migration background, language region in Switzerland, smoking, alcohol consumption and sport participation. CCS with a higher CVD risk profile because of cardiotoxic treatment had no better adherence. CONCLUSIONS: CCS have similar food patterns as their siblings and the general population, and poorly adhere to current recommendations. Awareness of the importance of a healthy diet should be raised among CCS, to prevent chronic diseases like CVD.

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

BACKGROUND: Hearing 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. PROCEDURE: In 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. RESULTS: Questionnaire 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. CONCLUSIONS: Questionnaires 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.

Long-term auditory complications after childhood cancer: A report from the Swiss Childhood Cancer Survivor Study.

BACKGROUND: Auditory complications are an adverse event of childhood cancer treatment, especially common in children treated with platinum chemotherapy or cranial radiation. Variation between diagnostic childhood cancer groups has rarely been studied, and we do not know if the burden of auditory complications has changed over the last decades. PROCEDURE: Within the Swiss Childhood Cancer Survivor Study, we sent a questionnaire to all survivors who were diagnosed at age 16 years or less between 1976 and 2005. We compared prevalence of self-reported hearing loss and tinnitus between all diagnostic childhood cancer groups and siblings, used multivariable logistic regression to analyze the effect of treatment-related factors on hearing loss, and compared the cumulative incidence of hearing loss between different periods of cancer diagnosis. RESULTS: Prevalence of self-reported hearing loss was higher in survivors (10%) than in siblings (3%, P < 0.001), and highest in survivors of central nervous system tumors (25%). Significant risk factors were treatment with platinum compounds (carboplatin: odds ratio [OR] 2.4; cisplatin: OR 9.4), cranial radiation (>29 Gy: OR >1.7), or brain surgery (OR 2.2). Children diagnosed in 1986-1995, when platinum compounds came into widespread use, had a significantly higher cumulative incidence of hearing loss than those diagnosed in 1976-1985. In the most recent period, 1996-2005, the risk decreased again, both for patients treated with platinum compounds and with cranial radiation. CONCLUSIONS: Our data show that the burden of hearing loss has stabilized in recently treated survivors, suggesting that survivors have benefited from new treatment regimens that use less ototoxic radiation and more carefully dosed platinum compounds.