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Aim: In a previous study we found that non-breastfed children of asthmatic mothers (CAM) had lower FVC and FEV1 compared with non-breastfed children of non-asthmatic mothers (CnAM), but breastfeeding (BF) compensated, improving these... more
Aim: In a previous study we found that non-breastfed children of asthmatic mothers (CAM) had lower FVC and FEV1 compared with non-breastfed children of non-asthmatic mothers (CnAM), but breastfeeding (BF) compensated, improving these values in CAM (Dogaru 2010 ERJ 678s). We extended this study, analyzing plethysmographic lung volumes (LV) and alveolar size in a different group. Method: In 132 UK-born subjects (7-21yrs) we measured FRC and TLC by plethysmography and Apparent Diffusion Coefficient (ADC, proxy for alveolar size) by He3-magnetic resonance. We used linear regressions adjusting for LV predictors and potential confounders and stratified by maternal asthma (MA). Results: 32 (24%) of children were not BF, 38 (29%) were BF ≤3mo and 53 (40%) BF>3mo; 24 (18%) had asthmatic mothers. We found no significant associations between BF and alveolar size, FRC or TLC in the whole group. However, stratification by MA showed a) that non-breastfed CAM have lower FRC and TLC but similar ADC means compared with non-breastfed CnAM and b) a trend for higher FRC and TLC in CAM if breastfed (Table). ![Figure][1] Conclusion: These results support previous findings that reduced LV in non-BF CAM compared with non-BF CnAM are compensated by BF. Since alveolar size was similar in CAM and CnAM, we suggest that alveolar number is reduced in CAM without BF, but can be partially increased with BF. Confirmation will require larger samples. Funding: Asthma UK 07/048; SNF 3200B0-122341. [1]: pending:yes
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Knowledge about determinants of quality of life (QoL) in eosinophilic oesophagitis (EoO) patients helps to identify patients at risk of experiencing poor QoL and to tailor therapeutic interventions accordingly. To evaluate the impact of... more
Knowledge about determinants of quality of life (QoL) in eosinophilic oesophagitis (EoO) patients helps to identify patients at risk of experiencing poor QoL and to tailor therapeutic interventions accordingly. To evaluate the impact of symptom severity, endoscopic and histological activity on EoE-specific QoL in adult EoE patients. Ninety-eight adult EoE patients were prospectively included (64% male, median age 39 years). Patients completed two validated instruments to assess EoE-specific QoL (EoO-QoL-A) and symptom severity (adult EoE activity index patient-reported outcome) and then underwent esophagogastroduodenoscopy with biopsy sampling. Physicians reported standardised information on EoE-associated endoscopic and histological alterations. The Spearman's rank correlation coefficient was calculated to determine the relationship between QoL and symptom severity. Linear regression and analysis of variance was used to quantify the extent to which variations in severity of EoE symptoms, endoscopic and histological findings explain variations in QoL. Quality of life strongly correlated with symptom severity (r = 0.610, P < 0.001). While the variation in severity of symptoms, endoscopic and histological findings alone explained 38%, 35% and 22% of the variability in EoE-related QoL, respectively, these together explained 60% of variation. Symptom severity explained 18-35% of the variation in each of the five QoL subscale scores. Eosinophilic oesophagitis symptom severity and biological disease activity determine QoL in adult patients with eosinophilic oesophagitis. Therefore, reduction in both eosinophilic oesophagitis symptoms as well as biological disease activity is essential for improvement of QoL in adult patients. Clinicaltrials.gov number, NCT00939263.
Research Interests: Quality of life, Medicine, Endoscopy, Humans, Internal Medicine, and 15 moreClinical research, Female, Male, Young Adult, Clinical Sciences, Aged, Middle Aged, Digestive System, Adult, Medicine and Health Sciences, Eosinophilic Esophagitis, Severity of Illness Index, Surveys and Questionnaires, Esophagogastroduodenoscopy, and Pharmacology and pharmaceutical sciences
Background: In a cross-sectional collaborative analysis within the EU project BESTCILIA, we had found significantly lower height and slightly lower BMI in PCD patients younger than 20 years of age compared to national reference data. We... more
Background: In a cross-sectional collaborative analysis within the EU project BESTCILIA, we had found significantly lower height and slightly lower BMI in PCD patients younger than 20 years of age compared to national reference data. We now assessed changes in height and BMI over time during the growth period. Methods: We used longitudinal measurements of height and BMI from PCD patients of the iPCD Cohort from 10 countries. We calculated height and BMI z-scores using national references and used generalized estimating equations (GEE) to identify changes over time, as a function of sex. Results: We analysed 1833 repeated measurements of height and BMI from 343 patients aged 0-20 years. We found a significantly lower height (z-score: -0.16, 95%CI: -0.30 to -0.02, p=0.03) and BMI (-0.15, -0.31 to -0.03, p=0.05) in PCD patients compared to reference. We found no significant change in height z-scores (p=0.93) but a small increase in BMI z-scores (0.02 per year, 0.01 to 0.03, p Conclusions: Height z-scores were significantly reduced in children with PCD compared to reference values, but they did not change over time. This might suggest that PCD affects growth already at an early stage of life but there is no catch-up until the age of 20. In the ongoing study, we will look at birth weights, stratify by levels of diagnostic certainty and determine patient characteristics associated with growth, such as lung function, microbiology and clinical characteristics. Funding: FP7 grant 305404,SNF 32003B-162820,Lungenliga Bern,Milena Carvanal–Pro Kartagener.
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Background: In the framework of the EU project BESTCILIA we had found significantly lower lung function (FEV 1 ) in a multinational cross-sectional dataset of patients with PCD compared to the Global lung function initiative (GLI) 2012... more
Background: In the framework of the EU project BESTCILIA we had found significantly lower lung function (FEV 1 ) in a multinational cross-sectional dataset of patients with PCD compared to the Global lung function initiative (GLI) 2012 reference values. We now assessed changes in FEV 1 over time in children and adolescents with PCD. Methods: In our analysis we included longitudinal measurements of FEV 1 from PCD patients from 11 countries using the iPCD cohort. We calculated FEV 1 z-scores using the GLI reference and used generalised estimating equation (GEE) models to identify changes of lung function over time, as a function of sex. Results: We analysed 1431 repeated measurements of FEV 1 from 276 patients aged 6-20 years. We found a significant decline in FEV 1 z-scores over time (-0.07 per year, 95% CI: -0.08 to -0.06, p 1 z-scores between sexes (p=0.79). Conclusions: FEV 1 z-scores of PCD children and adolescents declined significantly over time compared to normal reference, which suggests that lung growth of PCD patients is already impaired from early childhood. In the ongoing study, we will determine patient characteristics associated with changes in lung function, for example BMI, level of diagnostic certainty, microbiology and available clinical characteristics. Funding: FP7 grant 305404, SNF 32003B-162820, Milena Carvanal –Pro Kartagener, Lungenliga Bern.
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PURPOSE Radiation to the bone and exposure to alkylating agents increases the risk of bone cancer among survivors of childhood cancer, but there is uncertainty regarding the risks of bone tissue radiation doses below 10 Gy and the... more
PURPOSE Radiation to the bone and exposure to alkylating agents increases the risk of bone cancer among survivors of childhood cancer, but there is uncertainty regarding the risks of bone tissue radiation doses below 10 Gy and the dose-response relationship for specific types of chemotherapy. METHODS Twelve European countries contributed 228 cases and 228 matched controls to a nested case-control study within a cohort of 69,460 5-year survivors of childhood cancer. Odds ratios (ORs) of developing bone cancer for different levels of cumulative radiation exposure and cumulative doses of specific types of chemotherapy were calculated. Excess ORs were calculated to investigate the shape and extent of any dose-response relationship. RESULTS The OR associated with bone tissue exposed to 1-4 Gy was 4.8-fold (95% CI, 1.2 to 19.6) and to 5-9 Gy was 9.6-fold (95% CI, 2.4 to 37.4) compared with unexposed bone tissue. The OR increased linearly with increasing dose of radiation ( Ptrend < .00...
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Background Survivors of childhood cancer are at risk of subsequent primary malignant neoplasms (SPNs), but the risk for rarer types of SPNs, such as oral cancer, is uncertain. Previous studies included few oral SPNs, hence large-scale... more
Background Survivors of childhood cancer are at risk of subsequent primary malignant neoplasms (SPNs), but the risk for rarer types of SPNs, such as oral cancer, is uncertain. Previous studies included few oral SPNs, hence large-scale cohorts are required to identify groups at risks. Methods The PanCareSurFup cohort includes 69,460 5-year survivors of childhood cancer across Europe. Risks of oral SPNs were defined by standardised incidence ratios (SIRs), absolute excess risks and cumulative incidence. Results One hundred and forty-five oral SPNs (64 salivary gland, 38 tongue, 20 pharynx, 2 lip, and 21 other) were ascertained among 143 survivors. Survivors were at 5-fold risk of an oral SPN (95% CI: 4.4–5.6). Survivors of leukaemia were at greatest risk (SIR = 19.2; 95% CI: 14.6–25.2) followed by bone sarcoma (SIR = 6.4, 95% CI: 3.7–11.0), Hodgkin lymphoma (SIR = 6.2, 95% CI: 3.9–9.9) and soft-tissue sarcoma (SIR = 5.0, 95% CI: 3.0–8.5). Survivors treated with radiotherapy were at 33...
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Background: Childhood cancer survivors are at high risk for late effects. Regular attendance to long-term follow-up care is crucial and helps monitoring survivors’ health. The Theory of Planned Behaviour (TPB) was developed to predict a... more
Background: Childhood cancer survivors are at high risk for late effects. Regular attendance to long-term follow-up care is crucial and helps monitoring survivors’ health. The Theory of Planned Behaviour (TPB) was developed to predict a diverse range of health behaviours. We investigated whether the TPB helps explain attendance to follow-up care in Swiss survivors. We aimed to i) identify TPB-related predictors for the intention to attend follow-up, and ii) examine associations between intention and actual attendance. Methods: We sent a questionnaire to 716 eligible Swiss childhood cancer survivors (diagnosed with cancer aged <16 years; ≥5 years since diagnosis; aged 18 - 40 years at study). We assessed TPB-related predictors (attitude, subjective norm, perceived control), intention to attend follow-up care, and actual attendance. We applied structural equation modelling to investigate predictors of intention and logistic regression models to study the association between the int...
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The survival rate of children and adolescents diagnosed with cancer has markedly improved in the last decades and now exceeds 85% in Switzerland [1]. This results in about 5700 childhood cancer survivors living in Switzerland, who have... more
The survival rate of children and adolescents diagnosed with cancer has markedly improved in the last decades and now exceeds 85% in Switzerland [1]. This results in about 5700 childhood cancer survivors living in Switzerland, who have been diagnosed before age 14 years [2]. Most childhood cancer patients are cured from cancer and have decades of life ahead of them. Therefore, effective strategies to promote long-term health and quality of life are important. In this paper, we will briefly present major late effects experienced by childhood cancer survivors, and describe ongoing initiatives in the field of childhood cancer survivorship and long-term follow-up care (LTFU) in Switzerland.
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Background Lung resection is a controversial and understudied therapeutic modality in Primary Ciliary Dyskinesia (PCD). We assessed the prevalence of lung resection in PCD across countries and compared disease course in lobectomised and... more
Background Lung resection is a controversial and understudied therapeutic modality in Primary Ciliary Dyskinesia (PCD). We assessed the prevalence of lung resection in PCD across countries and compared disease course in lobectomised and non-lobectomised patients. Methods In the international iPCD cohort, we identified lobectomised and non-lobectomised age and sex-matched PCD patients and compared their characteristics, lung function and BMI cross-sectionally and longitudinally. Results Among 2896 patients in the iPCD cohort, 163 from 20 centers (15 countries) underwent lung resection (5.6%). Among adult patients, prevalence of lung resection was 8.9%, demonstrating wide variation among countries. Compared to the rest of the iPCD cohort, lobectomised patients were more often females, older at diagnosis, and more often had situs solitus. In about half of the cases (45.6%) lung resection was performed before presentation to specialized PCD centers for diagnostic work-up. Compared to co...
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Pulmonary exacerbations are a cause of significant morbidity in patients with primary ciliary dyskinesia (PCD) and are frequently used as an outcome measure in clinical research into chronic lung diseases. So far, there has been no... more
Pulmonary exacerbations are a cause of significant morbidity in patients with primary ciliary dyskinesia (PCD) and are frequently used as an outcome measure in clinical research into chronic lung diseases. So far, there has been no consensus on the definition of pulmonary exacerbations in PCD.30 multidisciplinary experts and patients developed a consensus definition for children and adults with PCD. Following a systematic review, the panel used a modified Delphi process with a combination of face-to-face meetings and e-surveys to develop a definition that can be used in research settings for children and adults with PCD.A pulmonary exacerbation was defined by the presence of three or more of the following seven items: 1) increased cough, 2) change in sputum volume and/or colour, 3) increased shortness of breath perceived by the patient or parent, 4) decision to start or change antibiotic treatment because of perceived pulmonary symptoms, 5) malaise, tiredness, fatigue or lethargy, 6...
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BackgroundUncertainty about cure puts childhood cancer survivors at risk of mental distress. We asked survivors if they had been told they had been cured and investigated associated factors.ProcedureWe used nationwide registry data and a... more
BackgroundUncertainty about cure puts childhood cancer survivors at risk of mental distress. We asked survivors if they had been told they had been cured and investigated associated factors.ProcedureWe used nationwide registry data and a questionnaire survey for ≥five‐year survivors of childhood cancer (n = 301), followed by online focus groups with a purposive sample of Swiss pediatric oncologists (n = 17). Discussions were coded by investigators using thematic analysis.ResultsOverall, 235 among 301 survivors (78%; 95% confidence interval, 73%–83%) reported having been told they were cured. The proportion was 89% (81%–97%) among lymphoma and 84% (77%–91%) among leukemia survivors, but only 49% (33%–65%) among central nervous system tumor survivors. Pediatric oncologists acknowledged that telling survivors they are cured may reassure them that their cancer lies behind them. However, many refrained from telling all patients. Reasons included the possibility of late effects (cure disr...
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Follow-up care is important for childhood cancer survivors to facilitate early detection and treatment of late effects. We aimed to describe preferences for different organisational aspects and models of follow-up care among Swiss... more
Follow-up care is important for childhood cancer survivors to facilitate early detection and treatment of late effects. We aimed to describe preferences for different organisational aspects and models of follow-up care among Swiss childhood cancer survivors, and characteristics associated with preferences for different models. We contacted 720 survivors aged 18+ years, diagnosed with cancer after 1990 (age 0-16 years), registered in the Swiss Childhood Cancer Registry (SCCR), and Swiss resident, who previously participated in a baseline survey. They received questionnaires to assess attendance and preferences for follow-up (rated on 4-point scales, 0-3). Clinical information was available from the SCCR. Survivors (n = 314: response rate 43.6%; 47.8% still attended follow-up) rated clinical reasons for follow-up higher than supportive reasons (p < .001). They rated checking for cancer recurrence (mean = 2.78, SD = 0.53) and knowing about risks for my children most important (mean ...
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Introduction: Chronic non-specific cough (CNSC), i.e. recurrent cough with no identifiable underlying disease, is common in childhood. It is unclear if risk factors for CNSC are similar as for wheezing illness. We aimed to compare risk... more
Introduction: Chronic non-specific cough (CNSC), i.e. recurrent cough with no identifiable underlying disease, is common in childhood. It is unclear if risk factors for CNSC are similar as for wheezing illness. We aimed to compare risk factors for CNSC with risk factors for wheeze in 0-2 year olds. Methods: We analysed parent-completed questionnaires (response rate 79%) on 3406 children from the population-based Leicester Respiratory Cohorts. CNSC was defined as no current wheeze and a positive reply to at least 2 of the following: coughing more than others, night-time cough and cough without cold. We used multinomial logistic regression to compare potential risk factors (sex, ethnicity, family history, comorbidities, socioeconomic status (SES), exposure to infections, air pollutants, breastfeeding) between children with wheeze and those with CNSC. Results: 353 children (10%) had CNSC and 926 (27%) wheeze. Sex (male), rhinitis, hayfever, eczema and posseting were associated with both outcomes similarly. Parental history of atopic diseases, exposure to tobacco smoke, lower SES and lack of breastfeeding were associated with wheeze only (all p<0.01). Habitual snoring was more strongly associated with wheeze than with CNSC (test for difference p=0.03). South Asian ethnicity (RRR 1.3; 95%CI 1.0-1.7; p=0.06) and nursery care (1.8; 1.4-2.3; p<0.001) were positively associated only with CNSC. Conclusions: CNSC in 0-2 year olds has a different pattern of risk factors than wheezing illness. In particular, CNSC is less associated with personal and family history of atopy and more strongly with attendance to nursery care, suggesting infectious aetiology. Funding: SNF PDFMP3 137033; SNF 32003B-144068; Asthma UK 07/048.
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Preterm birth, low birth weight and high infant weight gain are associated with increased risks of childhood asthma. An important underlying pathway might be smaller airways and subsequent lower lung function. We examined the association... more
Preterm birth, low birth weight and high infant weight gain are associated with increased risks of childhood asthma. An important underlying pathway might be smaller airways and subsequent lower lung function. We examined the association of early growth characteristics with the risk of lower lung function, using data from 24,715 children of 24 European birth cohorts. Studies were eligible if they included children from 1989 onwards, had information on gestational age, birth weight or infant weight gain, and lung function (FEV1, FVC, FEF25-75, FEF75) at age 4-18 years. Random-effect meta-analyses were used with individual participant data and pooled effect estimates. Younger gestational age and lower birth weight were associated with lower lung function values (p-values Younger gestational age, lower weight at birth and higher infant weight gain are associated with lower lung function in childhood. Smaller airways might be partly responsible for associations of early growth characteristics with asthma.
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Research Interests: Demography, Pediatrics, Obesity, Asthma, Medicine, and 5 moreLogistic Regression, Cohort, Body Mass Index, confounding, and Wheeze
Objectives: There is little data on SARS-CoV-2 in people with rare chronic diseases. We studied incidence and severity of SARS-CoV-2 and its risk factors in people with primary ciliary dyskinesia (PCD) from May 2020 to May 2022.Methods:... more
Objectives: There is little data on SARS-CoV-2 in people with rare chronic diseases. We studied incidence and severity of SARS-CoV-2 and its risk factors in people with primary ciliary dyskinesia (PCD) from May 2020 to May 2022.Methods: We used self-reported questionnaire data from the COVID-PCD study at baseline or during weekly follow-ups. We studied factors associated with SARS-CoV-2 and symptoms using Poisson regression.Results: By May 2022, 728 people participated (40% male, median age 27 years; range 0–85). 87 (12%) reported SARS-CoV-2 at baseline or during follow-up and 62 people reported an incident SARS-CoV-2 infection during 716 person-years (incidence rate 9 per 100 person years). The strongest predictors for reporting SARS-CoV-2 were exposure during periods where Delta variant was dominant (IRR 4.52, 95% CI 1.92–10.6) and Omicron variants (IRR 13.3, 95% CI 5.2–33.8). Severity was mild; 12 (14%) were asymptomatic and 75 (86%) had symptoms among whom four were hospitalized...
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BACKGROUND Leukaemias comprise a heterogenous group of haematological malignancies. In CONCORD-3, we analysed data for children (aged 0-14 years) and adults (aged 15-99 years) diagnosed with a haematological malignancy during 2000-14 in... more
BACKGROUND Leukaemias comprise a heterogenous group of haematological malignancies. In CONCORD-3, we analysed data for children (aged 0-14 years) and adults (aged 15-99 years) diagnosed with a haematological malignancy during 2000-14 in 61 countries. Here, we aimed to examine worldwide trends in survival from leukaemia, by age and morphology, in young patients (aged 0-24 years). METHODS We analysed data from 258 population-based cancer registries in 61 countries participating in CONCORD-3 that submitted data on patients diagnosed with leukaemia. We grouped patients by age as children (0-14 years), adolescents (15-19 years), and young adults (20-24 years). We categorised leukaemia subtypes according to the International Classification of Childhood Cancer (ICCC-3), updated with International Classification of Diseases for Oncology, third edition (ICD-O-3) codes. We estimated 5-year net survival by age and morphology, with 95% CIs, using the non-parametric Pohar-Perme estimator. To control for background mortality, we used life tables by country or region, single year of age, single calendar year and sex, and, where possible, by race or ethnicity. All-age survival estimates were standardised to the marginal distribution of young people with leukaemia included in the analysis. FINDINGS 164 563 young people were included in this analysis: 121 328 (73·7%) children, 22 963 (14·0%) adolescents, and 20 272 (12·3%) young adults. In 2010-14, the most common subtypes were lymphoid leukaemia (28 205 [68·2%] patients) and acute myeloid leukaemia (7863 [19·0%] patients). Age-standardised 5-year net survival in children, adolescents, and young adults for all leukaemias combined during 2010-14 varied widely, ranging from 46% in Mexico to more than 85% in Canada, Cyprus, Belgium, Denmark, Finland, and Australia. Individuals with lymphoid leukaemia had better age-standardised survival (from 43% in Ecuador to ≥80% in parts of Europe, North America, Oceania, and Asia) than those with acute myeloid leukaemia (from 32% in Peru to ≥70% in most high-income countries in Europe, North America, and Oceania). Throughout 2000-14, survival from all leukaemias combined remained consistently higher for children than adolescents and young adults, and minimal improvement was seen for adolescents and young adults in most countries. INTERPRETATION This study offers the first worldwide picture of population-based survival from leukaemia in children, adolescents, and young adults. Adolescents and young adults diagnosed with leukaemia continue to have lower survival than children. Trends in survival from leukaemia for adolescents and young adults are important indicators of the quality of cancer management in this age group. FUNDING Children with Cancer UK, the Institut National du Cancer, La Ligue Contre le Cancer, Centers for Disease Control and Prevention, Swiss Re, Swiss Cancer Research foundation, Swiss Cancer League, Rossy Family Foundation, US National Cancer Institute, and the American Cancer Society.
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differences in perceived asthma control in childhood: guidelines and reality
Children with frequent respiratory symptoms in infancy have an increased risk for later wheezing, but the association with symptom dynamics is unknown. We developed an observer-independent method to characterise symptom dynamics and... more
Children with frequent respiratory symptoms in infancy have an increased risk for later wheezing, but the association with symptom dynamics is unknown. We developed an observer-independent method to characterise symptom dynamics and tested their association with subsequent respiratory morbidity.In this birth-cohort of healthy neonates, we prospectively assessed weekly respiratory symptoms during infancy, resulting in a time series of 52 symptom scores. For each infant, we calculated the transition probability between two consecutive symptom scores. We used these transition probabilities to construct a Markov matrix, which characterised symptom dynamics quantitatively using an entropy parameter. Using this parameter, we determined phenotypes by hierarchical clustering. We then studied the association between phenotypes and wheezing at 6 years.In 322 children with complete data for symptom scores during infancy (16 864 observations), we identified three dynamic phenotypes. Compared to...
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<p><sup>a</sup>Diagnosis is classified according to the International Classification of Childhood Cancer – third edition <a... more
<p><sup>a</sup>Diagnosis is classified according to the International Classification of Childhood Cancer – third edition <a href="http://www.plosone.org/article/info:doi/10.1371/journal.pone.0047944#pone.0047944-SteliarovaFoucher1" target="_blank">[26]</a>. Abbreviations: CNS, Central Nervous System. <a href="http://www.plosone.org/article/info:doi/10.1371/journal.pone.0047944#pone-0047944-g001" target="_blank"><i>Figure 1A</i></a> shows the proportion of survivors reporting a limitation for sporting activities within each diagnostic group. The dotted line indicates the proportion in the sibling controls. <a href="http://www.plosone.org/article/info:doi/10.1371/journal.pone.0047944#pone-0047944-g001" target="_blank"><i>Figure 1B</i></a> shows limitations for daily activities (mean scores of the physical function score from the SF-36) stratified by type of diagnosis. Lower scores indicate increased limitations for daily tasks such as walking fast, carry heavy things, move a table, carry grocery bags, climbing stairs, bending down, walking a certain distance, bathing and clothing. The dotted line indicates the mean score of the sibling controls.</p
Research Interests: Physiology, Mathematics, Immunology, Biotechnology, Cancer, and 4 moreMedicine, Sports, Activities, and sf
<p>NOTE: Percentages are based upon available data for each variable.</p><p>Abbreviations: CNS, Central Nervous System; ICCC-3, International Classification of Childhood Cancer – Third Edition; n, number; n.a., not... more
<p>NOTE: Percentages are based upon available data for each variable.</p><p>Abbreviations: CNS, Central Nervous System; ICCC-3, International Classification of Childhood Cancer – Third Edition; n, number; n.a., not applicable.</p>a<p>Age- and sex-standardized numbers and percentages are given for siblings.</p>b<p>Non participants include: 107 without current address, 239 who did not respond, 85 who refused to participate, and 83 who answered an abridged questionnaire (<b><a href="http://www.plosone.org/article/info:doi/10.1371/journal.pone.0047944#pone.0047944.s001" target="_blank">Supplementary Figure S1</a></b>).</p>c<p>Column percentages are given.</p>d<p>P-value calculated from chi-square statistics comparing survivor participants and survivor non-participants.</p>e<p>Information derived from questionnaire survey is not available for non-responders.</p>f<p>Information on former cancer disease is not applicable for siblings.</p>g<p>Other malignant epithelial neoplasms, malignant melanomas and other or unspecified malignant neoplasms.</p>h<p>Each person can have had several treatments.</p
Research Interests: Physiology, Mathematics, Immunology, Biotechnology, Cancer, and 3 moreMedicine, Siblings, and Comparing
<p>Socio-demographic predictors of monthly income >4’500 CHF in survivors and siblings<a href="http://www.plosone.org/article/info:doi/10.1371/journal.pone.0155546#t003fn001"... more
<p>Socio-demographic predictors of monthly income >4’500 CHF in survivors and siblings<a href="http://www.plosone.org/article/info:doi/10.1371/journal.pone.0155546#t003fn001" target="_blank"><sup>a</sup></a>.</p
Research Interests: Marine Biology, Science Policy, Biotechnology, Cancer, Infectious Diseases, and 7 moreMedicine, Survivor, Survivors, CHF, CNS, SCCR, and Sibling
<p>Fig 2 shows the association of diagnostic group with having a monthly income of >4’500 CHF compared to siblings. <sup>a</sup>Multivariable analysis showing ORs adjusted for baseline and secondary socio-demographic... more
<p>Fig 2 shows the association of diagnostic group with having a monthly income of >4’500 CHF compared to siblings. <sup>a</sup>Multivariable analysis showing ORs adjusted for baseline and secondary socio-demographic variables that were significant in the univariable model (<a href="http://www.plosone.org/article/info:doi/10.1371/journal.pone.0155546#pone.0155546.t003" target="_blank">Table 3</a>); <sup>b</sup>Sibling population is standardized on age, gender, migration background and language region according to the survivor population.</p
Research Interests: Marine Biology, Science Policy, Biotechnology, Cancer, Infectious Diseases, and 7 moreMedicine, Survivor, Survivors, CHF, CNS, SCCR, and Sibling
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... more
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...
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Das Leben mit einer seltenen Krankheit ist oft eine jahrelange Suche nach der richtigen Diagnose. Hansruedi Silberschmidt schildert in seiner Autobiographie «husten verboten – Ein Leben mit einer seltenen Krankheit» seinen langen und... more
Das Leben mit einer seltenen Krankheit ist oft eine jahrelange Suche nach der richtigen Diagnose. Hansruedi Silberschmidt schildert in seiner Autobiographie «husten verboten – Ein Leben mit einer seltenen Krankheit» seinen langen und steinigen Weg vom kränklichen Buben mit Primärer Ziliärer Dyskinesie bis zum engagierten Betroffenen. «husten verboten» bietet auch einen kritischen Blick in die Geschichte der Schweizer Pädiatrie, beschreibt Institutionen und bewährte Praktiken, die uns bestens bekannt sind, die aus der Perspektive eines Patienten aber so absurd wirken, wie sie tatsächlich waren. Eine Einladung eines ehemaligen Patienten zu einem Diskurs in Augenhöhe. «husten verboten» ist spannend und liest sich in einem Zug.
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RationaleThe Predicting Asthma Risk in Children (PARC) tool uses questionnaire-based respiratory symptoms collected from preschool children to predict their risk of asthma 5 years later. The tool was originally developed and externally... more
RationaleThe Predicting Asthma Risk in Children (PARC) tool uses questionnaire-based respiratory symptoms collected from preschool children to predict their risk of asthma 5 years later. The tool was originally developed and externally validated in population-based settings and has not yet been validated in a clinical setting.ObjectiveTo externally validate the PARC tool in children seen in paediatric pulmonology clinics.MethodsThe Swiss Paediatric Airway Cohort (SPAC) is a prospective study of children seen in respiratory outpatient clinics across Switzerland. This analysis included children seen at ages 1-6 years for cough or wheeze at baseline and who completed the follow-up questionnaire 2 years later. The outcome was defined as current wheeze plus use of asthma medication. In sensitivity analyses, we explored effects of varied inclusion criteria and outcomes. We assessed performance by describing sensitivity, specificity, negative and positive predictive value (NPV, PPV), area ...
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Objectives: Primary ciliary dyskinesia (PCD) is a rare genetic disease that causes recurrent respiratory infections. People with PCD may be at high risk of severe COVID-19 and protection against SARS-CoV-2 is therefore important. We... more
Objectives: Primary ciliary dyskinesia (PCD) is a rare genetic disease that causes recurrent respiratory infections. People with PCD may be at high risk of severe COVID-19 and protection against SARS-CoV-2 is therefore important. We studied facemask usage and problems reported in relation with their use among people with PCD.Methods: We used data from COVID-PCD, an international observational cohort study. A questionnaire was e-mailed to participants in October 2020 that asked about facemask usage.Results: In total, 282 participants from 27 countries were included (Median age 32 years; 63% female). In total, 252 (89%) wore facemasks everywhere in public, 13 (5%) wore facemasks in most places, and 17 (6%) did not wear facemasks in public. Half of the participants reported that it was uncomfortable to wear facemasks because of runny nose, cough, or difficulty breathing. Participants less often wore facemasks when there was no national requirement.Conclusion: Most people with PCD wore ...
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RationaleChildhood cancer survivors (CCSs) are at increased risk for pulmonary morbidity due to exposure to lung-toxic treatments, including specific chemotherapies, radiotherapy, and surgery. Longitudinal data on lung function, with... more
RationaleChildhood cancer survivors (CCSs) are at increased risk for pulmonary morbidity due to exposure to lung-toxic treatments, including specific chemotherapies, radiotherapy, and surgery. Longitudinal data on lung function, with information on how outcomes change over time, in CCSs are scarce.ObjectivesTo investigate lung function trajectories in childhood cancer survivors over time and investigate the association with lung-toxic treatment.MethodsThis retrospective, multi-center cohort study included CCSs, who were diagnosed between 1990 and 2013 in Switzerland and had been exposed to lung-toxic chemotherapeutics or thoracic radiotherapy. Pulmonary function tests (PFTs) were obtained from hospital charts. We assessed quality of PFTs systematically and calculated z-scores and percentage predicted of forced expiratory volume in first second (FEV1), forced vital capacity (FVC), FEV1/FVC ratio, total lung capacity (TLC) and diffusion capacity for carbon-monoxide (DLCO) based on rec...
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European children: recommendations and clinical practice Full names, institutions and countries of all co‐authors