Publications by authors named "Claudia E Kuehni"

246 Publications

Longitudinal lung function in childhood cancer survivors after hematopoietic stem cell transplantation.

Bone Marrow Transplant 2021 Nov 8. Epub 2021 Nov 8.

Childhood Cancer Research Group, Institute of Social and Preventive Medicine (ISPM), University of Bern, Bern, Switzerland.

Longitudinal data on pulmonary function after pediatric allogeneic or autologous hematopoietic stem cell transplantation (HSCT) are rare. We examined pulmonary function and associated risk factors in 5-year childhood cancer survivors (CCSs) longitudinally. We included 74 CCSs diagnosed between 1976 and 2010, treated with HSCT, and with at least two pulmonary function tests performed during follow-up. Median follow-up was 9 years (range 6-13). We described pulmonary function as z-scores for lung volumes (forced vital capacity [FVC], residual volume [RV], total lung capacity [TLC]), flows (forced expiratory volume in 1 s [FEV1], maximal mid-expiratory flow [MMEF]), and diffusion capacity for carbon monoxide (DLCO) and assessed associations with potential risk factors using multivariable regression analysis. The median z-scores for FEV1, FVC, and TLC were below the expected throughout the follow-up period. This was not the case for RV, MMEF and DLCO. Female gender, radiotherapy to the chest, and relapse were associated with lower z-scores of FEV1, FVC, MMEF, RV or DLCO. Childhood cancer survivors after HSCT are at risk of pulmonary dysfunction. The complex and multifactorial etiology of pulmonary dysfunction emphasizes the need for longitudinal prospective studies to better characterize the course and causes of pulmonary function impairment in CCSs.
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http://dx.doi.org/10.1038/s41409-021-01509-1DOI Listing
November 2021

Diagnosis of primary ciliary dyskinesia: discrepancy according to different algorithms.

ERJ Open Res 2021 Oct 1;7(4). Epub 2021 Nov 1.

Division of Paediatric Respiratory Medicine and Allergology, Dept of Paediatrics, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.

Background: Diagnosis of primary ciliary dyskinesia (PCD) is challenging since there is no gold standard test. The European Respiratory (ERS) and American Thoracic (ATS) Societies developed evidence-based diagnostic guidelines with considerable differences.

Objective: We aimed to compare the algorithms published by the ERS and the ATS with each other and with our own PCD-UNIBE algorithm in a clinical setting. Our algorithm is similar to the ERS algorithm with additional immunofluorescence staining. Agreement (Cohen's κ) and concordance between the three algorithms were assessed in patients with suspicion of PCD referred to our diagnostic centre.

Results: In 46 out of 54 patients (85%) the final diagnosis was concordant between all three algorithms (30 PCD negative, 16 PCD positive). In eight patients (15%) PCD diagnosis differed between the algorithms. Five patients (9%) were diagnosed as PCD only by the ATS, one (2%) only by the ERS and PCD-UNIBE, one (2%) only by the ATS and PCD-UNIBE, and one (2%) only by the PCD-UNIBE algorithm. Agreement was substantial between the ERS and the ATS (κ=0.72, 95% CI 0.53-0.92) and the ATS and the PCD-UNIBE (κ=0.73, 95% CI 0.53-0.92) and almost perfect between the ERS and the PCD-UNIBE algorithms (κ=0.92, 95% CI 0.80-1.00).

Conclusion: The different diagnostic algorithms lead to a contradictory diagnosis in a considerable proportion of patients. Thus, an updated, internationally harmonised and standardised PCD diagnostic algorithm is needed to improve diagnostics for these discordant cases.
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http://dx.doi.org/10.1183/23120541.00353-2021DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8558472PMC
October 2021

Predictors for participation in DNA self-sampling of childhood cancer survivors in Switzerland.

BMC Med Res Methodol 2021 10 30;21(1):236. Epub 2021 Oct 30.

CANSEARCH research platform for paediatric oncology and haematology, Faculty of Medicine, Department of Pediatrics, Gynecology, and Obstetrics, University of Geneva, Geneva, Switzerland.

Background: Research on germline genetic variants relies on enough eligible participants which is difficult to achieve for rare diseases such as childhood cancer. With self-collection kits, participants can contribute genetic samples conveniently from their home. Demographic and clinical factors were identified previously that influenced participation in mailed self-collection. People with pre-existing heritable diagnoses might participate differently in germline DNA collection which might render sampling biased in this group. In this nationwide cross-sectional study, we analysed predictive factors of participation in DNA self-collection including heritable diagnoses.

Methods: We identified childhood cancer survivors from the Swiss Childhood Cancer Registry for invitation to germline DNA self-sampling in September 2019. Participants received saliva sampling kits by postal mail at their home, were asked to fill them, sign an informed consent, and send them back by mail. Two reminders were sent to non-participants by mail. We compared demographic, clinical, and treatment information of participants with non-participants using univariable and multivariable logistic regression models.

Results: We invited 928 childhood cancer survivors in Switzerland with a median age of 26.5 years (interquartile range 19-37), of which 463 (50%) participated. After the initial send out of the sampling kit, 291 (63%) had participated, while reminder letters led to 172 additional participants (37%). Foreign nationality (odds ratio [OR] 0.5; 95%-confidence interval [CI] 0.4-0.7), survivors aged 30-39 years at study versus other age groups (OR 0.5; CI 0.4-0.8), and survivors with a known cancer predisposition syndrome (OR 0.5; CI 0.3-1.0) were less likely to participate in germline DNA collection. Survivors with a second primary neoplasm (OR 1.9; CI 1.0-3.8) or those living in a French or Italian speaking region (OR 1.3; CI 1.0-1.8) tended to participate more.

Conclusions: We showed that half of childhood cancer survivors participated in germline DNA self-sampling relying completely on mailing of sample kits. Written reminders increased the response by about one third. More targeted recruitment strategies may be advocated for people of foreign nationality, aged 30-39 years, and those with cancer predisposition syndromes. Perceptions of genetic research and potential barriers to participation of survivors need to be better understood.

Trial Registration: Biobank: https://directory.bbmri-eric.eu/#/collection/bbmri-eric:ID:CH_HopitauxUniversitairesGeneve:collection:CH_BaHOP Research project : Clinicaltrials.gov: NCT04702321 .
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http://dx.doi.org/10.1186/s12874-021-01428-1DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8557596PMC
October 2021

Treatment Decisions in Children with Asthma in a Real-Life Clinical Setting: The Swiss Paediatric Airway Cohort.

J Allergy Clin Immunol Pract 2021 Oct 22. Epub 2021 Oct 22.

Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland; Division of Respiratory Medicine, Department of Paediatrics, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland. Electronic address:

Background: Asthma treatment should be modified according to symptom control and future risk, but there are scarce data on what drives treatment adjustments in routine tertiary care.

Objective: We studied factors that drive asthma treatment adjustment in pediatric outpatient clinics.

Methods: We performed a cross-sectional analysis of the Swiss Paediatric Airway Cohort, a clinical cohort of 0- to 16-year-old children seen by pediatric pulmonologists. We collected information on diagnosis, treatment, lung function, and FeNO from hospital records; and on symptoms, sociodemographic, and environmental factors from a parental questionnaire. We used reported symptoms to classify asthma control and categorized treatment according to the 2020 Global Initiative for Asthma guidelines. We used multivariable logistic regression to study factors associated with treatment adjustment (step-up or down vs no change).

Results: We included 551 children diagnosed with asthma (mean age, 10 years; 37% female). At the clinical visit, most children were prescribed Global Initiative for Asthma step 3 (35%). Compared with previsit treatment, 252 children remained on the same step (47%), 227 were stepped up (42%), and 58 were stepped down (11%). Female sex (adjusted odds ratio [aOR] = 1.61; 95% confidence interval [CI], 1.05-2.47), poor asthma control (aOR = 3.08; 95% CI, 1.72-5.54), and lower FEV Z-score (aOR = 0.70; 95% CI, 0.56-0.86 per one Z-score increase) were independently associated with treatment step-up, and low FeNO (aOR = 2.34; 95% CI, 1.23-4.45) was associated with treatment step-down, with marked heterogeneity between clinics.

Conclusions: In this tertiary care real-life study, we identified main drivers for asthma treatment adjustment. These findings may help improve both asthma management guidelines and clinical practice.
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http://dx.doi.org/10.1016/j.jaip.2021.10.026DOI Listing
October 2021

Agreement of parent- and child-reported wheeze and its association with measurable asthma traits.

Pediatr Pulmonol 2021 Dec 1;56(12):3813-3821. Epub 2021 Oct 1.

Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland.

Objectives: In epidemiological studies, childhood asthma is usually assessed with questionnaires directed at parents or children, and these may give different answers. We studied how well parents and children agreed when asked to report symptoms of wheeze and investigated whose answers were closer to measurable traits of asthma.

Methods: LuftiBus in the school is a cross-sectional survey of respiratory health among Swiss schoolchildren aged 6-17 years. We applied questionnaires to parents and children asking about wheeze and exertional wheeze in the past year. We assessed agreement between parent-child answers with Cohen's kappa (k), and associations of answers from children and parents with fractional exhaled nitric oxide (FeNO) and forced expiratory volume in 1 s over forced vital capacity (FEV /FVC), using quantile regression.

Results: We received questionnaires from 3079 children and their parents. Agreement was poor for reported wheeze (k = 0.37) and exertional wheeze (k = 0.36). Median FeNO varied when wheeze was reported by children (19 ppb, interquartile range [IQR]: 9-44), parents (22 ppb, IQR: 12-46), both (31 ppb, IQR: 16-55), or neither (11 ppb, IQR: 7-19). Median absolute FEV /FVC was the same when wheeze was reported by children (84%, IQR: 78-89) and by parents (84%, IQR: 78-89), lower when reported by both (82%, IQR: 78-87), and higher when reported by neither (87%, IQR: 82-91). For exertional wheeze findings were similar. Results did not differ by age or sex.

Conclusion: Our findings suggest that surveying both parents and children and combining their responses can help us to better identify children with measurable asthma traits.
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http://dx.doi.org/10.1002/ppul.25690DOI Listing
December 2021

A Comprehensive Approach for the Diagnosis of Primary Ciliary Dyskinesia-Experiences from the First 100 Patients of the PCD-UNIBE Diagnostic Center.

Diagnostics (Basel) 2021 Aug 25;11(9). Epub 2021 Aug 25.

Division of Paediatric Respiratory Medicine and Allergology, Department of Paediatrics, Inselspital, Bern University Hospital, University of Bern, 3010 Bern, Switzerland.

Primary ciliary dyskinesia (PCD) is a rare genetic disease characterized by dyskinetic cilia. Respiratory symptoms usually start at birth. The lack of diagnostic gold standard tests is challenging, as PCD diagnostics requires different methods with high expertise. We founded PCD-UNIBE as the first comprehensive PCD diagnostic center in Switzerland. Our diagnostic approach includes nasal brushing and cell culture with analysis of ciliary motility via high-speed-videomicroscopy (HSVM) and immunofluorescence labeling (IF) of structural proteins. Selected patients undergo electron microscopy (TEM) of ciliary ultrastructure and genetics. We report here on the first 100 patients assessed by PCD-UNIBE. All patients received HSVM fresh, IF, and cell culture (success rate of 90%). We repeated the HSVM with cell cultures and conducted TEM in 30 patients and genetics in 31 patients. Results from cell cultures were much clearer compared to fresh samples. For 80 patients, we found no evidence of PCD, 17 were diagnosed with PCD, two remained inconclusive, and one case is ongoing. HSVM was diagnostic in 12, IF in 14, TEM in five and genetics in 11 cases. None of the methods was able to diagnose all 17 PCD cases, highlighting that a comprehensive approach is essential for an accurate diagnosis of PCD.
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http://dx.doi.org/10.3390/diagnostics11091540DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8466881PMC
August 2021

Impact of era of diagnosis on cause-specific late mortality among 77 423 five-year European survivors of childhood and adolescent cancer: The PanCareSurFup consortium.

Int J Cancer 2021 Sep 22. Epub 2021 Sep 22.

German Childhood Cancer Registry (GCCR), Division of Childhood Cancer Epidemiology, Institute of Medical Biostatistics, Epidemiology and Informatics (IMBEI), Johannes Gutenberg University Mainz, Mainz, Germany.

Late mortality of European 5-year survivors of childhood or adolescent cancer has dropped over the last 60 years, but excess mortality persists. There is little information concerning secular trends in cause-specific mortality among older European survivors. PanCareSurFup pooled data from 12 cancer registries and clinics in 11 European countries from 77 423 five-year survivors of cancer diagnosed before age 21 between 1940 and 2008 followed for an average age of 21 years and a total of 1.27 million person-years to determine their risk of death using cumulative mortality, standardized mortality ratios (SMR), absolute excess risks (AER), and multivariable proportional hazards regression analyses. At the end of follow-up 9166 survivors (11.8%) had died compared to 927 expected (SMR 9.89, 95% confidence interval [95% CI] 9.69-10.09), AER 6.47 per 1000 person-years, (95% CI 6.32-6.62). At 60 to 68 years of attained age all-cause mortality was still higher than expected (SMR = 2.41, 95% CI 1.90-3.02). Overall cumulative mortality at 25 years from diagnosis dropped from 18.4% (95% CI 16.5-20.4) to 7.3% (95% CI 6.7-8.0) over the observation period. Compared to the diagnosis period 1960 to 1969, the mortality hazard ratio declined for first neoplasms (P for trend <.0001) and for infections (P < .0001); declines in relative mortality from second neoplasms and cardiovascular causes were less pronounced (P = .1105 and P = .0829, respectively). PanCareSurFup is the largest study with the longest follow-up of late mortality among European childhood and adolescent cancer 5-year survivors, and documents significant mortality declines among European survivors into modern eras. However, continuing excess mortality highlights survivors' long-term care needs.
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http://dx.doi.org/10.1002/ijc.33817DOI Listing
September 2021

Clinical data for paediatric research: the Swiss approach : Proceedings of the National Symposium in Bern, Switzerland, Dec 5-6, 2019.

BMC Proc 2021 Sep 20;15(Suppl 13):19. Epub 2021 Sep 20.

Institute of Social and Preventive Medicine (ISPM), University of Bern, Mittelstrasse 43, 3012, Bern, Switzerland.

Background And Purpose: Continuous improvement of health and healthcare system is hampered by inefficient processes of generating new evidence, particularly in the case of rare diseases and paediatrics. Currently, most evidence is generated through specific research projects, which typically require extra encounters with patients, are costly and entail long delays between the recognition of specific needs in healthcare and the generation of necessary evidence to address those needs. The Swiss Personalised Health Network (SPHN) aims to improve the use of data obtained during routine healthcare encounters by harmonizing data across Switzerland and facilitating accessibility for research. The project "Harmonising the collection of health-related data and biospecimens in paediatric hospitals throughout Switzerland (SwissPedData)" was an infrastructure development project funded by the SPHN, which aimed to identify and describe available data on child health in Switzerland and to agree on a standardised core dataset for electronic health records across all paediatric teaching hospitals. Here, we describe the results of a two-day symposium that aimed to summarise what had been achieved in the SwissPedData project, to put it in an international context, and to discuss the next steps for a sustainable future. The target audience included clinicians and researchers who produce and use health-related data on children in Switzerland.

Key Highlights: The symposium consisted of state-of-the-art lectures from national and international keynote speakers, workshops and plenary discussions. This manuscript summarises the talks and discussions in four sections: (I) a description of the Swiss Personalized Health Network and the results of the SwissPedData project; (II) examples of similar initiatives from other countries; (III) an overview of existing health-related datasets and projects in Switzerland; and (IV) a summary of the lessons learned and future prospective from workshops and plenary discussions.

Implications: Streamlined processes linking initial collection of information during routine healthcare encounters, standardised recording of this information in electronic health records and fast accessibility for research are essential to accelerate research in child health and make it affordable. Ongoing projects prove that this is feasible in Switzerland and elsewhere. International collaboration is vital to success. The next steps include the implementation of the SwissPedData core dataset in the clinical information systems of Swiss hospitals, the use of this data to address priority research questions, and the acquisition of sustainable funding to support a slim central infrastructure and local support in each hospital. This will lay the foundation for a national paediatric learning health system in Switzerland.
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http://dx.doi.org/10.1186/s12919-021-00226-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8450032PMC
September 2021

External background ionizing radiation and childhood cancer: Update of a nationwide cohort analysis.

J Environ Radioact 2021 Nov 11;238-239:106734. Epub 2021 Sep 11.

Institute of Social and Preventive Medicine (ISPM), University of Bern, Bern, Switzerland. Electronic address:

Background: Exposure to high doses of ionizing radiation is known to cause cancer. Exposure during childhood is associated with a greater excess relative risk for leukemia and tumors of the central nervous system (CNS) than exposure in later life. Cancer risks associated with low-dose exposure (<100 mSv) are uncertain. We previously investigated the association between the incidence of childhood cancer and levels of exposure to external background radiation from terrestrial gamma and cosmic rays in Switzerland using data from a nationwide census-based cohort study. Here, we provide an update of that study using an extended follow-up period and an improved exposure model.

Methods: We included all children 0-15 years of age registered in the Swiss national censuses 1990, 2000, and 2010-2015. We identified incident cancer cases during 1990-2016 using probabilistic record linkage with the Swiss Childhood Cancer Registry. Exposure to terrestrial and cosmic radiation at children's place of residence was estimated using geographic exposure models based on aerial spectrometric gamma-ray measurements. We estimated and included the contribution from Cs deposition after the Chernobyl accident. We created a nested case-control sample and fitted conditional logistic regression models adjusting for sex, year of birth, neighborhood socioeconomic position, and modelled outdoor NO concentration. We also estimated the population attributable fraction for childhood cancer due to external background radiation.

Results: We included 3,401,113 children and identified 3,137 incident cases of cancer, including 951 leukemia, 495 lymphoma, and 701 CNS tumor cases. Median follow-up in the cohort was 6.0 years (interquartile range: 4.3-10.1) and median cumulative exposure since birth was 8.2 mSv (range: 0-31.2). Hazard ratios per 1 mSv increase in cumulative dose of external background radiation were 1.04 (95% CI: 1.01-1.06) for all cancers combined, 1.06 (1.01-1.10) for leukemia, 1.03 (0.98-1.08) for lymphoma, and 1.06 (1.01-1.11) for CNS tumors. Adjustment for potential confounders had little effect on the results. Based on these results, the estimated population attributable fraction for leukemia and CNS tumors due to external background radiation was 32% (7-49%) and 34% (5-51%), respectively.

Conclusions: Our results suggest that background ionizing radiation contributes to the risk of leukemia and CNS tumors in children.
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http://dx.doi.org/10.1016/j.jenvrad.2021.106734DOI Listing
November 2021

LuftiBus in the school (LUIS): a population-based study on respiratory health in schoolchildren.

Swiss Med Wkly 2021 08 10;151:w20544. Epub 2021 Aug 10.

Division of Respiratory Medicine, University Children's Hospital Zurich and Children's Research Centre, University of Zurich, Switzerland.

Respiratory disease is common in children and strongly associated with lifestyle and environmental exposures. Thus, it is important to study the epidemiology locally. The LuftiBus in the School (LUIS) study was set up to assess the respiratory health of schoolchildren in the canton of Zurich, Switzerland. LUIS is a cross-sectional population-based study that was carried out 2013 to 2016. Children aged 6–17 years living in the canton of Zurich were eligible to participate. All schools in the canton were approached and the school head decided whether the school would participate and with which classes. Consenting parents answered a standardised questionnaire at home and assenting children completed a shorter questionnaire by interview at school. Trained technicians measured children’s lung function, including spirometry, double tracer gas single-breath washout (DTG-SBW) and fractional exhaled nitric oxide (FeNO). Address histories of participants were geocoded to be linked with area-based socioeconomic measures and environmental exposures such as spatiotemporal air pollution estimates for specific time periods and locations. A subgroup was seen again 12 months later using the same procedures to collect longitudinal data. The study included 3870 children at baseline and 655 at the 1-year follow-up. Median age was 12.7 years; 281 (8%) had wheezed in the past year. At baseline we collected 3457 (89%) parental and 3546 (92%) child questionnaires, and 3393 (88%) FeNO, 3446 (89%) spirometry, and 1795 (46%) DTG-SBW measurements. LUIS is a rich resource of health-related data, with information on lung function, environmental exposures and respiratory health on Swiss schoolchildren.
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http://dx.doi.org/10.4414/smw.2021.20544DOI Listing
August 2021

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

BMC Cancer 2021 Jul 8;21(1):787. Epub 2021 Jul 8.

Childhood Cancer Registry, Institute of Social and Preventive Medicine, University of Bern, Mittelstrasse 43, 3012, Bern, Switzerland.

Background: Chest 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.

Methods: We 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.

Results: Forty-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.

Conclusion: It 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.
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http://dx.doi.org/10.1186/s12885-021-08425-zDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8268220PMC
July 2021

Hospital Contacts for Psychiatric Disorders in Parents of Children With Cancer in Denmark.

JNCI Cancer Spectr 2021 Jun 8;5(3):pkab036. Epub 2021 Apr 8.

Childhood Cancer Research Group, Danish Cancer Society Research Center, Copenhagen, Denmark.

Background: Having a child diagnosed with cancer is a devastating experience that may affect parents' mental health. We aimed to assess the risk of hospital contacts for psychiatric disorders in parents of children with cancer.

Methods: We conducted a nationwide population-based cohort study using Danish registry data. Parents of children diagnosed with cancer between 1982 and 2014 (n = 6689 mothers, n = 5509 fathers) were matched with comparison parents of cancer-free children (n = 67 544 mothers, n = 55 756 fathers). We used Cox proportional hazards models to estimate the risk of hospital contacts for any psychiatric disorder and specific disorders. Cox models were also used to investigate sociodemographic and cancer-related risk factors for psychiatric disorders.

Results: Incidence rates of hospital contacts for any psychiatric disorder were 426 per 100 000 person-years in mothers of children with cancer and 345 per 100 000 person-years in comparison mothers. For fathers, the respective incidence rates were 260 and 262 cases per 100 000 person-years. Compared with parents of cancer-free children, mothers of children with cancer were at an increased risk of hospital contacts for any psychiatric disorder (hazard ratio = 1.23, 95% confidence interval = 1.12 to 1.36), whereas no elevated risk was seen in fathers (hazard ratio = 0.99, 95% confidence interval = 0.87 to 1.13). Among mothers, risks were particularly elevated for affective and stress-related disorders. Parents of deceased children and children diagnosed at a younger age were at particular risk of hospital contacts for psychiatric disorders.

Conclusion: Hospital contacts for psychiatric disorders were overall rare. Health-care professionals should draw attention to subgroups of vulnerable parents to meet their needs of support and adequate treatment.
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http://dx.doi.org/10.1093/jncics/pkab036DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8164824PMC
June 2021

Dietary Intake and Diet Quality of Adult Survivors of Childhood Cancer and the General Population: Results from the SCCSS-Nutrition Study.

Nutrients 2021 May 22;13(6). Epub 2021 May 22.

Center for Primary Care and Public Health (Unisanté), University of Lausanne, 1010 Lausanne, Switzerland.

Childhood cancer survivors (CCSs) are at increased risk of developing chronic health conditions. This may potentially be reduced by a balanced diet. We aimed to compare dietary intake and diet quality using the Alternative Healthy Eating Index (AHEI) of adult CCSs and the general Swiss population. A food frequency questionnaire (FFQ) was completed by CCSs with a median age of 34 (IQR: 29-40) years. We compared dietary intake of 775 CCSs to two population-based cohorts who completed the same FFQ: 1276 CoLaus and 2529 Bus Santé study participants. CCSs consumed particular inadequate amounts of fiber and excessive amounts of sodium and saturated fat. Dietary intake was similar in CCSs and the general population. The mean AHEI was low with 49.8 in CCSs (men: 47.7, women: 51.9), 52.3 in CoLaus (men: 50.2, women: 54.0), and 53.7 in Bus Santé (men: 51.8, women: 54.4) out of a maximum score of 110. The AHEI scores for fish, fruit, vegetables, and alcohol were worse in CCSs than in the general population, whereas the score for sugar-sweetened beverages was better (all < 0.001). Diet quality at follow-up did not differ between clinical characteristics of CCSs. Long-term CCSs and the general population have poor dietary intake and quality in Switzerland, which suggests similar population-based interventions for everyone.
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http://dx.doi.org/10.3390/nu13061767DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8224580PMC
May 2021

Rotavirus disease and health care utilisation among children under 5 years of age in highly developed countries: A systematic review and meta-analysis.

Vaccine 2021 05 29;39(22):2917-2928. Epub 2021 Apr 29.

Institute of Social and Preventive Medicine, University of Bern, Mittlestrasse 43, 3012 Bern, Switzerland; Paediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Freiburgstrasse, 3010 Bern, Switzerland. Electronic address:

Background: Rotavirus (RV) infection is the leading cause of diarrhoea-associated morbidity and mortality globally among children under 5 years of age. RV vaccination is available, but has not been implemented in many national immunisation plans, especially in highly developed countries. This systematic review aimed to estimate the prevalence and incidence of health care use for RV gastroenteritis (RVGE) among children aged under 5 years in highly developed countries without routine RV vaccination.

Methods: We searched MEDLINE and Embase databases from January 1 2000 to December 17 2018 for publications reporting on incidence or prevalence of RVGE-related health care use in children below 5 years of age: primary care and emergency department (ED) visits, hospitalisations, nosocomial infections and deaths. We included only studies with laboratory-confirmed RV infection, undertaken in highly developed countries with no RV routine vaccination plans. We used random effects meta-analysis to generate summary estimates with 95% confidence intervals (CI) and prediction intervals.

Results: We screened 4033 abstracts and included 74 studies from 21 countries. Average incidence rates of RVGE per 100 000 person-years were: 2484 (95% CI 697-5366) primary care visits, 1890 (1597-2207) ED visits, 500 (422-584) hospitalisations, 34 (20-51) nosocomial infections and 0.04 (0.02-0.07) deaths. Average proportions of cases of acute gastroenteritis caused by RV were: 21% (95% CI 16-26%) for primary care visits; 32% (25-38%) for ED visits; 41% (36-47%) for hospitalisations, 29% (25-34%) for nosocomial infections and 12% (8-18%) for deaths. Results varied widely between and within countries, and heterogeneity was high (I > 90%) in most models.

Conclusion: RV in children under 5 years causes many healthcare visits and hospitalisations, with low mortality, in highly developed countries without routine RV vaccination. The health care use estimates for RVGE obtained by this study can be used to model RV vaccine cost-effectiveness in highly developed countries.
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http://dx.doi.org/10.1016/j.vaccine.2021.04.039DOI Listing
May 2021

Birth characteristics and childhood leukemia in Switzerland: a register-based case-control study.

Cancer Causes Control 2021 Jul 20;32(7):713-723. Epub 2021 Apr 20.

Institute of Social and Preventive Medicine (ISPM), University of Bern, Mittelstrasse 43, 3012, Bern, Switzerland.

Purpose: Initial genetic alterations in the development of childhood leukemia occur in utero or before conception; both genetic and environmental factors are suspected to play a role. We aimed to investigate the associations between childhood leukemia and perinatal characteristics including birth order, birth interval to older siblings, parental age, birth weight, and multiple birth.

Methods: We identified cases diagnosed between 1981 and 2015 and born in Switzerland between 1969 and 2015 from the Swiss Childhood Cancer Registry and randomly sampled five controls per case from national birth records matched on date of birth, sex, and municipality of residence at birth. We used conditional logistic regression to investigate associations between perinatal characteristics and leukemia at ages 0-15 and 0-4 years, and the subtypes acute lymphoblastic leukemia (ALL) and acute myeloid leukemia (AML).

Results: The study included 1,403 cases of leukemia. We observed increased risks associated with high birth weight (adjusted OR 1.37, 95% CI 1.12-1.69) and multiple birth (1.89, 1.24-2.86). These associations were similar for ALL and stronger for leukemia at ages 0-4 years. For AML, we observed an increased risk for higher birth order (3.08, 0.43-22.03 for fourth or later born children). We found no associations with other perinatal characteristics.

Conclusion: This register-based case-control study adds to the existing evidence of a positive association between high birth weight and risk of childhood leukemia. Furthermore, it suggests children from multiple births are at an increased risk of leukemia.
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http://dx.doi.org/10.1007/s10552-021-01423-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8184536PMC
July 2021

Hearing loss in childhood cancer survivors.

Lancet Child Adolesc Health 2021 05;5(5):e17

Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland; Division of Pediatric Hematology/Oncology, Department of Pediatrics, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.

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http://dx.doi.org/10.1016/S2352-4642(21)00099-7DOI Listing
May 2021

European Respiratory Society clinical practice guidelines for the diagnosis of asthma in children aged 5-16 years.

Eur Respir J 2021 10 4;58(5). Epub 2021 Nov 4.

Division of Respiratory Medicine, University Children's Hospital Zurich and Childhood Research Center, Zurich, Switzerland.

Background: Diagnosing asthma in children represents an important clinical challenge. There is no single gold-standard test to confirm the diagnosis. Consequently, over- and under-diagnosis of asthma is frequent in children.

Methods: A task force supported by the European Respiratory Society has developed these evidence-based clinical practice guidelines for the diagnosis of asthma in children aged 5-16 years using nine Population, Intervention, Comparator and Outcome (PICO) questions. The task force conducted systematic literature searches for all PICO questions and screened the outputs from these, including relevant full-text articles. All task force members approved the final decision for inclusion of research papers. The task force assessed the quality of the evidence using the Grading of Recommendations, Assessment, Development and Evaluation (GRADE) approach.

Results: The task force then developed a diagnostic algorithm based on the critical appraisal of the PICO questions, preferences expressed by lay members and test availability. Proposed cut-offs were determined based on the best available evidence. The task force formulated recommendations using the GRADE Evidence to Decision framework.

Conclusion: Based on the critical appraisal of the evidence and the Evidence to Decision framework, the task force recommends spirometry, bronchodilator reversibility testing and exhaled nitric oxide fraction as first-line diagnostic tests in children under investigation for asthma. The task force recommends against diagnosing asthma in children based on clinical history alone or following a single abnormal objective test. Finally, this guideline also proposes a set of research priorities to improve asthma diagnosis in children in the future.
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http://dx.doi.org/10.1183/13993003.04173-2020DOI Listing
October 2021

COVID-PCD: a participatory research study on the impact of COVID-19 in people with primary ciliary dyskinesia.

ERJ Open Res 2021 Jan 22;7(1). Epub 2021 Mar 22.

Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland.

COVID-PCD is a participatory study initiated by people with primary ciliary dyskinesia (PCD) who have an essential vote in all stages of the research from the design of the study to the recruitment of participants, and interpretation and communication of the study results. COVID-PCD aims to collect epidemiological data in real-time from people with PCD throughout the pandemic to describe incidence of coronavirus disease 2019 (COVID-19), symptoms and course of disease; identify risk factors for prognosis; and assess experiences, wishes and needs. The study is advertised through patient support groups and participants register online on the study website (www.covid19pcd.ispm.ch). The study invites persons of any age from anywhere in the world with a suspected or confirmed PCD. A baseline questionnaire assesses details on PCD diagnosis, habitual symptoms and COVID-19 episodes that occurred before study entry. Afterwards, participants receive a weekly follow-up questionnaire with questions on incident severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infections, current symptoms, social contact behaviour and physical activity. Occasional thematic questionnaires are sent out focussing on emerging questions of interest chosen by people with PCD. In case of hospitalisation, patients or family members are asked to obtain a hospital report. Results are continuously analysed and summaries put online. The study started recruitment on April 30, 2020, and 556 people with PCD completed the baseline questionnaire by November 2, 2020. The COVID-PCD study is a participatory study that follows people with PCD during the COVID-19 pandemic, helps to empower affected persons, and serves as a platform for communication between patients, physicians and researchers.
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http://dx.doi.org/10.1183/23120541.00843-2020DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7983255PMC
January 2021

Continuous recording of vital signs with a wearable device in pediatric patients undergoing chemotherapy for cancer-an operational feasibility study.

Support Care Cancer 2021 Sep 3;29(9):5283-5292. Epub 2021 Mar 3.

Pediatric Hematology/Oncology, Department of Pediatrics, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.

Purpose: Pediatric patients with cancer are at high risk for severe infections. Infections can trigger changes of vital signs long before clinical symptoms arise. Continuous recording may detect such changes earlier than discrete measurements. We aimed to assess the feasibility of continuous recording of vital signs by a wearable device (WD) in pediatric patients undergoing chemotherapy for cancer.

Methods: In this prospective, observational single-center study, pediatric patients under chemotherapy wore the Everion® WD for 14 days. The predefined patient-specific goal was heart rate recorded in good quality during ≥18/24 h per day, on ≥7 consecutive days. The predefined criterion to claim feasibility was ≥15/20 patients fulfilling this patient-specific goal.

Results: Twenty patients were included (median age, 6 years; range, 2-16). Six patients aged 3-16 years fulfilled the patient-specific goal. Quality of heart rate recording was good during 3992 of 6576 (61%) hours studied and poor during 300 (5%) hours, and no data was recorded during 2284 (35%) hours. Eighteen of 20 participants indicated that this WD is acceptable to measure vital signs in children under chemotherapy.

Conclusion: The predefined feasibility criterion was not fulfilled. This was mainly due to important compliance problems and independent of the WD itself. However, continuous recording of vital signs was possible across a very wide age range in pediatric patients undergoing chemotherapy for cancer. We recommend to study feasibility in the Everion® again, plus in further WDs, applying measures to enhance compliance.

Trial Registration: ClinicalTrials.gov (NCT04134429) on October 22, 2019.
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http://dx.doi.org/10.1007/s00520-021-06099-8DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7925259PMC
September 2021

Health behaviour of women with Turner Syndrome.

Acta Paediatr 2021 08 2;110(8):2424-2429. Epub 2021 Mar 2.

Department of Pediatric Endocrinology, Diabetology and Metabolism, Inselspital, Bern University Children's Hospital, University of Bern, Bern, Switzerland.

Aim: This study assessed lifestyle-related risk factors for cardiovascular disease in young women with Turner syndrome.

Methods: In 2012, we sent a questionnaire to women with Turner syndrome aged ≥18 years and living in Switzerland with questions on socio-demographic and medical data as well as health behaviour. We compared the reported lifestyle with that of women from the Swiss Health Survey 2012, a representative survey of the general population.

Results: Fifty-seven per cent (45/79) of women with Turner syndrome answered the questionnaire (mean age: 24 years). Eighty per cent (36/45) had never smoked compared with 58% (1156/1972) of the general population (p < 0.01). Women with Turner syndrome engaged less often in binge drinking (34% vs. 71%) (p < 0.001), but consumed alcohol equally often as the general population (p = 0.327). They performed sports as often as the general population (p = 0.34), but only one quarter (11/45) of women with Turner syndrome adhered to official physical activity recommendations.

Conclusion: Although most women with Turner syndrome had a healthy lifestyle, only a minority had sufficient physical activity. Paediatricians should promote structured physical activity in girls with Turner syndrome from early childhood onwards to reduce their cardiovascular risk in adulthood and to increase long-term health-related quality of life.
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http://dx.doi.org/10.1111/apa.15814DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8359370PMC
August 2021

Cancer predisposition syndromes as a risk factor for early second primary neoplasms after childhood cancer - A national cohort study.

Eur J Cancer 2021 03 7;145:71-80. Epub 2021 Jan 7.

Institute of Social and Preventive Medicine, University of Bern, Switzerland; Division of Pediatric Hematology and Oncology, University Children`s Hospital Bern, Switzerland. Electronic address:

Background: Childhood 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 Methods: This cohort study used data from the Swiss Childhood Cancer Registry. We included patients with first primary neoplasms (FPNs) diagnosed before age 21 years from 1986 to 2015 and identified SPNs occurring before age 21. We calculated standardised incidence ratios (SIRs) and absolute excess risks (AERs) using Swiss population cancer incidence data, and cumulative incidence of SPNs. We calculated hazard ratios (HRs) of risk factors for SPNs using Fine and Gray competing risk regression.

Results: Among 8074 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 higher in childhood cancer patients than the incidence of 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 (CI: 12-41%) and 2.7% in those without (CI: 2.0-3.6%). 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), haematopoietic 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).

Conclusion: CPSs are associated with a high risk of SPNs before age 21 years. Identification of CPSs is important for appropriate cancer surveillance and targeted screening.
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http://dx.doi.org/10.1016/j.ejca.2020.11.042DOI Listing
March 2021

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

Bone Marrow Transplant 2021 05 25;56(5):1065-1076. Epub 2020 Nov 25.

Childhood Cancer Research Group, Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland.

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.
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http://dx.doi.org/10.1038/s41409-020-01137-1DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8113058PMC
May 2021

Risk of digestive cancers in a cohort of 69 460 five-year survivors of childhood cancer in Europe: the PanCareSurFup study.

Gut 2020 Nov 2. Epub 2020 Nov 2.

Princess Maxima Center for Pediatric Oncology, Utrecht, The Netherlands.

Background: Survivors of childhood cancer are at risk of subsequent primary neoplasms (SPNs), but the risk of developing specific digestive SPNs beyond age 40 years remains uncertain. We investigated risks of specific digestive SPNs within the largest available cohort worldwide.

Methods: The PanCareSurFup cohort includes 69 460 five-year survivors of childhood cancer from 12 countries in Europe. Risks of digestive SPNs were quantified using standardised incidence ratios (SIRs), absolute excess risks and cumulative incidence.

Results: 427 digestive SPNs (214 colorectal, 62 liver, 48 stomach, 44 pancreas, 59 other) were diagnosed in 413 survivors. Wilms tumour (WT) and Hodgkin lymphoma (HL) survivors were at greatest risk (SIR 12.1; 95% CI 9.6 to 15.1; SIR 7.3; 95% CI 5.9 to 9.0, respectively). The cumulative incidence increased the most steeply with increasing age for WT survivors, reaching 7.4% by age 55% and 9.6% by age 60 years (1.0% expected based on general population rates). Regarding colorectal SPNs, WT and HL survivors were at greatest risk; both seven times that expected. By age 55 years, 2.3% of both WT (95% CI 1.4 to 3.9) and HL (95% CI 1.6 to 3.2) survivors had developed a colorectal SPN-comparable to the risk among members of the general population with at least two first-degree relatives affected.

Conclusions: Colonoscopy surveillance before age 55 is recommended in many European countries for individuals with a family history of colorectal cancer, but not for WT and HL survivors despite a comparable risk profile. Clinically, serious consideration should be given to the implementation of colonoscopy surveillance while further evaluation of its benefits, harms and cost-effectiveness in WT and HL survivors is undertaken.
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http://dx.doi.org/10.1136/gutjnl-2020-322237DOI Listing
November 2020

Cardiovascular and Pulmonary Challenges After Treatment of Childhood Cancer.

Pediatr Clin North Am 2020 12;67(6):1155-1170

Childhood Cancer Research Platform, Institute of Social and Preventive Medicine, University of Bern, Mittelstrasse 43, Bern 3012, Switzerland; Department of Pediatrics, Inselspital, Bern University Hospital, University of Bern, Mittelstrasse 43, Bern 3012, Switzerland.

Childhood cancer survivors are at risk for developing cardiovascular disease and pulmonary disease related to cancer treatment. This might not become apparent until many years after treatment and varies from subclinical to life-threatening disease. Important causes are anthracyclines and radiotherapy involving heart, head, or neck for cardiovascular disease, and bleomycin, busulfan, nitrosoureas, radiation to the chest, and lung or chest surgery for pulmonary disease. Most effects are dose dependent, but genetic risk factors have been discovered. Treatment options are limited. Prevention and regular screening are crucial. Survivors should be encouraged to adopt a healthy lifestyle, and modifiable risk factors should be addressed.
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http://dx.doi.org/10.1016/j.pcl.2020.07.007DOI Listing
December 2020

Late Effects in Childhood Cancer Survivors: Early Studies, Survivor Cohorts, and Significant Contributions to the Field of Late Effects.

Pediatr Clin North Am 2020 12;67(6):1033-1049

Childhood Cancer Research Group, Danish Cancer Society Research Center, Strandboulevarden 49, 2100 Copenhagen, Denmark; Department of Clinical Medicine, Faculty of Health, Incuba Skejby, building 2, Palle Juul-Jensens Boulevard 82, 8200 Aarhus N, Denmark.

With improvement in cure of childhood cancer came the responsibility to investigate the long-term morbidity and mortality associated with the treatments accountable for this increase in survival. Several large cohorts of childhood cancer survivors have been established throughout Europe and North America to facilitate research on long-term complications of cancer treatment. The cohorts have made significant contributions to the understanding of early mortality, somatic late complications, and psychosocial outcomes among childhood cancer survivors, which has been translated into the design of new treatment protocols for pediatric cancers, with the goal to reduce the potential risk and severity of late effects.
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http://dx.doi.org/10.1016/j.pcl.2020.07.002DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8393933PMC
December 2020

Isolated night cough in children: how does it differ from wheeze?

ERJ Open Res 2020 Oct 13;6(4). Epub 2020 Oct 13.

Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland.

It has been postulated that some children with recurrent cough but no wheeze have a mild form of asthma (cough variant asthma), with similar risk factors and an increased risk of future wheeze. This longitudinal study compared risk factors for isolated night cough and for wheeze in the Leicester Respiratory Cohort in children aged 1, 4, 6 and 9 years and compared prognosis of children with isolated night cough, children with wheeze and asymptomatic children. We included 4101 children aged 1 year, 2854 aged 4 years, 2369 aged 6 years and 1688 aged 9 years. The prevalence of isolated night cough was 10% at age 1 year and 18% in older children. Prevalence of wheeze decreased from 35% at 1 year to 13% at 9 years. Although several risk factors were similar for cough and wheeze, day care, reflux and family history of bronchitis were more strongly associated with cough, and male sex and family history of asthma with wheeze. Over one-third of preschool children with cough continued to cough at school age, but their risk of developing wheeze was similar to that of children who were asymptomatic at earlier surveys. Wheeze tracked more strongly throughout childhood than cough. In conclusion, our study showed that only some risk factors for cough and wheeze were shared but many were not, and there was little evidence for an increased risk of future wheeze in children with isolated night cough. This provides little support for the hypothesis that recurrent cough without wheeze may indicate a variant form of asthma.
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http://dx.doi.org/10.1183/23120541.00217-2020DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7553117PMC
October 2020

Diagnosis in children with exercise-induced respiratory symptoms: A multi-center study.

Pediatr Pulmonol 2021 01 17;56(1):217-225. Epub 2020 Nov 17.

Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland.

Objective: Exercise-induced respiratory symptoms (EIS) are common in childhood and reflect different diseases that can be difficult to diagnose. In children referred to respiratory outpatient clinics for EIS, we compared the diagnosis proposed by the primary care physician with the final diagnosis from the outpatient clinic and described diagnostic tests and treatments.

Design: An observational study of respiratory outpatients aged 0-16 years nested in the Swiss Paediatric Airway Cohort (SPAC).

Patients: We included children with EIS as the main reason for referral. Information about diagnostic investigations, final diagnosis, and treatment prescribed came from outpatient records. We included 214 children (mean age 12 years, range 2-17, 54% males) referred for EIS.

Results: The final diagnosis was asthma in 115 (54%), extrathoracic dysfunctional breathing (DB) in 35 (16%), thoracic DB in 22 (10%), asthma plus DB in 23 (11%), insufficient fitness in 10 (5%), chronic cough in 6 (3%), and other diagnoses in 3 (1%). Final diagnosis differed from referral diagnosis in 115 (54%, 95%-CI 46%-60%). Spirometry, body plethysmography, and exhaled nitric oxide were performed in almost all, exercise-challenge tests in a third, and laryngoscopy in none. 91% of the children with a final diagnosis of asthma were prescribed inhaled medication and 50% of children with DB were referred to physiotherapy.

Conclusions: Diagnosis given at the outpatient clinic often differed from the diagnosis proposed by the referring physician. Diagnostic evaluations, management, and follow-up differed between clinics and diagnostic groups highlighting the need for evidence-based diagnostic guidelines and harmonized procedures for children seen for EIS.
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http://dx.doi.org/10.1002/ppul.25126DOI Listing
January 2021

Reported Symptoms Differentiate Diagnoses in Children with Exercise-Induced Respiratory Problems: Findings from the Swiss Paediatric Airway Cohort (SPAC).

J Allergy Clin Immunol Pract 2021 02 19;9(2):881-889.e3. Epub 2020 Sep 19.

Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland; Paediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Bern, Switzerland. Electronic address:

Background: Exercise-induced breathing problems with similar clinical presentations can have different etiologies. This makes distinguishing common diagnoses such as asthma, extrathoracic and thoracic dysfunctional breathing (DB), insufficient fitness, and chronic cough difficult.

Objective: We studied which parent-reported, exercise-induced symptoms can help distinguish diagnoses in children seen in respiratory outpatient clinics.

Methods: This study was nested in the Swiss Paediatric Airway Cohort, an observational study of children aged 0 to 17 years referred to pediatric respiratory outpatient clinics in Switzerland. We studied children aged 6 to 17 years and compared information on exercise-induced symptoms from parent-completed questionnaires between children with different diagnoses. We used multinomial regression to analyze whether parent-reported symptoms differed between diagnoses (asthma as base).

Results: Among 1109 children, exercise-induced symptoms were reported for 732 (66%) (mean age: 11 years, 318 of 732 [43%] female). Among the symptoms, dyspnea best distinguished thoracic DB (relative risk ratio [RRR]: 5.4, 95% confidence interval [CI]: 1.3-22) from asthma. Among exercise triggers, swimming best distinguished thoracic DB (RRR: 2.4, 95% CI: 1.3-6.2) and asthma plus DB (RRR: 1.8, 95% CI: 0.9-3.4) from asthma only. Late onset of symptoms was less common for extrathoracic DB (RRR: 0.1, 95% CI: 0.03-0.5) and thoracic DB (RRR: 0.4, 95% CI: 0.1-1.2) compared with asthma. Localization of dyspnea (throat vs chest) differed between extrathoracic DB (RRR: 2.3, 95% CI: 0.9-5.8) and asthma. Reported respiration phase (inspiration or expiration) did not help distinguish diagnoses.

Conclusion: Parent-reported symptoms help distinguish different diagnoses in children with exercise-induced symptoms. This highlights the importance of physicians obtaining detailed patient histories.
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http://dx.doi.org/10.1016/j.jaip.2020.09.012DOI Listing
February 2021
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