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ObjectivesPhysical activity is widely recommended in the treatment and management of cystic fibrosis (CF). Despite the numerous physical and psychological benefits, many young people with CF are not achieving the recommended levels of physical activity. The aim of this systematic review was to identify and synthesise available qualitative investigations exploring the motives for, barriers to and facilitators of physical activity among young people with CF.MethodsThe following six electronic databases were systematically searched: Applied Social Sciences Index and Abstracts (ASSIA), CINAH, EMBASE, MEDLINE, MEDLINE-in-process, PsycINFO up to August 2019. Keywords were used to identify qualitative research that explored engagement in physical activity among young people with CF. Titles and abstracts were screened by two independent reviewers, and potentially relevant articles were retrieved in full. Articles were eligible for inclusion if they employed any qualitative method and recruited participants under the age of 24 years with CF. Risk of bias of included studies was assessed via the Critical Appraisal Skills Programme. Results were synthesised using a thematic approach.ResultsSeven studies met our inclusion criteria and were included in the review. Overall, studies were of moderate to high quality. Thematic synthesis identified nine main themes that encompass motives for, barriers to and facilitators of physical activity among young people with CF. These were (1) Perceptions of physical activity. (2) Value attributed to physical activity. (3) Social influences. (4) Competing priorities. (5) Fluctuating health. (6) Normality. (7) Control beliefs. (8) Coping strategies. (9) Availability of facilities. Previous reviews have been unable to identify intervention characteristics that influence physical activity behaviour.ConclusionsThis review provides detailed information on the physical (biological—clinical), psychological, social and environmental influences on physical activity behaviour, thus providing numerous targets for future interventions. This in turn could facilitate promotion of physical activity among young people with CF.

PurposeExposures early in life, beginning in utero, have long-term impacts on mental and physical health. The ECHO prenatal and early childhood pathways to health consortium (ECHO-PATHWAYS) was established to examine the independent and combined impact of pregnancy and childhood chemical exposures and psychosocial stressors on child neurodevelopment and airway health, as well as the placental mechanisms underlying these associations.ParticipantsThe ECHO-PATHWAYS consortium harmonises extant data from 2684 mother–child dyads in three pregnancy cohort studies (CANDLE [Conditions Affecting Neurocognitive Development and Learning in Early Childhood], TIDES [The Infant Development and Environment Study] and GAPPS [Global Alliance to Prevent Prematurity and Stillbirth]) and collects prospective data under a unified protocol. Study participants are socioeconomically diverse and include a large proportion of Black families (38% Black and 51% White), often under-represented in research. Children are currently 5–15 years old. New data collection includes multimodal assessments of primary outcomes (airway health and neurodevelopment) and exposures (air pollution, phthalates and psychosocial stress) as well as rich covariate characterisation. ECHO-PATHWAYS is compiling extant and new biospecimens in a central biorepository and generating the largest placental transcriptomics data set to date (N=1083).Findings to dateEarly analyses demonstrate adverse associations of prenatal exposure to air pollution, phthalates and maternal stress with early childhood airway outcomes and neurodevelopment. Placental transcriptomics work suggests that phthalate exposure alters placental gene expression, pointing to mechanistic pathways for the developmental toxicity of phthalates. We also observe associations between prenatal maternal stress and placental corticotropin releasing hormone, a marker of hormonal activation during pregnancy relevant for child health. Other publications describe novel methods for examining exposure mixtures and the development of a national spatiotemporal model of ambient outdoor air pollution.Future plansThe first wave of data from the unified protocol (child age 8–9) is nearly complete. Future work will leverage these data to examine the combined impact of early life social and chemical exposures on middle childhood health outcomes and underlying placental mechanisms.

IntroductionPrimary ciliary dyskinesia (PCD) is a rare, genetic, multiorgan disease with an estimated prevalence of 1 in 10 000. It affects mainly the upper and lower airways due to impaired mucociliary clearance. Almost all patients have sinonasal or otologic (ear–nose–throat, ENT) problems, although the ENT clinical phenotype may present great variability. Despite that, data on PCD ENT manifestations are scarce and based on small single-centre studies. To date, we know little about the spectrum and severity of PCD ENT disease, its association with lung disease, its course over life and its determinants of prognosis.This study protocol describes the aims and methods of the first prospective, observational, multinational cohort study focusing on ENT disease in patients with PCD.Methods and analysisThe ENT prospective international cohort of patients with PCD (EPIC-PCD) is a prospective standardised observational clinical cohort set up as a multinational multicentre study, embedded into routine patient care. It aims to longitudinally characterise ENT disease in patients with PCD and its association with lung disease, and to identify determinants of its prognosis. Patients of all ages, diagnosed with PCD who undergo an ENT clinical assessment at least once a year at one of the participating centres will be invited to participate. Collected data include diagnostic test results, results of ENT examinations, lung function measurements, information on management of ENT disease and patient-reported data on clinical symptoms and health-related quality of life (QoL). Data are collected using the standardised PCD-specific FOLLOW-PCD form and the validated QoL-PCD questionnaire.Ethics and disseminationThe study has been reviewed and approved by the Human Research Ethics Committees at all participating centres, based on local legislation. The results of the study will be published in scientific journals, presented at scientific conferences and disseminated to participants and national patient organisations.Trial registrationNCT04611516.

ObjectivesTo determine the costs directly or indirectly related to bronchopulmonary dysplasia (BPD) in preterm infants. The secondary objective was to stratify the costs based on gestational age and/or birth weight.DesignSystematic literature review.SettingPubMed and Scopus were searched on 3 February 2020. Studies were selected based on eligibility criteria by two independent reviewers. Included studies were further searched to identify eligible references and citations.Two independent reviewers extracted data with a prespecified data extraction sheet, including items from a published checklist for quality assessment. The costs in the included studies are reported descriptively.Primary outcome measureCosts of BPD.ResultsThe 13 included studies reported the total costs or marginal costs of BPD. Most studies reported costs during birth hospitalisation (cost range: Int$21 392–Int$1 094 509 per child, equivalent to €19 103–€977 397, in 2019) and/or during the first year of life. One study reported costs during the first 2 years; two other studies reported costs later, during the preschool period and one study included a long-term follow-up. The highest mean costs were associated with infants born at extremely low gestational ages. The quality assessment indicated a low risk of bias in the reported findings of included studies.ConclusionsThis study was the first systematic review of costs associated with BPD. We confirmed previous reports of high costs and described the long-term follow-up necessary for preterm infants with BPD, particularly infants of very low gestational age. Moreover, we identified a need for studies that estimate costs outside hospitals and after the first year of life.PROSPERO registration numberCRD42020173234.

IntroductionA considerable burden of the tuberculosis (TB) epidemic is found in adolescents. The reasons for increased susceptibility to TB infection and higher incidence of TB disease in adolescence, compared with the 5–10 years old age group, are incompletely understood. Despite the pressing clinical and public health need to better understand and address adolescent TB, research in this field remains limited.Methods and analysisTeen TB is an ongoing prospective observational cohort study that aims to better understand the biology, morbidity and social context of adolescent TB. The study plans to recruit 50 adolescents (10–19 years old) with newly diagnosed microbiologically confirmed pulmonary TB disease and 50 TB-exposed controls without evidence of TB disease in Cape Town, South Africa, which is highly endemic for TB. At baseline, cases and controls will undergo a detailed clinical evaluation, chest imaging, respiratory function assessments and blood collection for viral coinfections, inflammatory cytokines and pubertal hormone testing. At 2 weeks, 2 months and 12 months, TB disease cases will undergo further chest imaging and additional lung function testing to explore the patterns of respiratory abnormalities. At week 2, cases will complete a multicomponent quantitative questionnaire about psychological and social impacts on their experiences and longitudinal, in-depth qualitative data will be collected from a nested subsample of 20 cases and their families.Ethics and disseminationThe study protocol has received ethical approval from the Stellenbosch University Health Research Ethics Committee (N19/10/148). The study findings will be disseminated through peer-reviewed publications, academic conferences and formal presentations to health professionals. Results will also be made available to participants and caregivers.

ObjectiveTo identify risk factors and develop an early prediction model for bronchopulmonary dysplasia (BPD) in preterm infants with a gestational age (GA) under 32 weeks.DesignA single-centre, retrospective case–control study.SettingA tertiary neonatal intensive care unit in Fujian Province, China.Participants762 infants (GA under 32 weeks, survived beyond the corrected GA of 36 weeks) who were hospitalised in our department between January 2018 and December 2023.InterventionNone.Main outcome measuresThe primary outcome was the diagnosis of BPD at 36 weeks of corrected GA, with diagnostic criteria based on the 2018 criteria established by the National Institute of Child Health and Human Development.ResultsA total of 762 infants was enrolled in the study, including 572 in the training cohort, among whom 138 (24.13%) had BPD. Multivariate logistic regression identified the following as independent predictors for BPD development: birth weight (OR=0.997, 95% CI 0.996 to 0.998, p<0.001), chest X-ray score within 24 hours of birth (OR=1.777, 95% CI 1.253 to 2.521, p=0.001), chest X-ray score at day 7 after birth (OR=1.756, 95% CI 1.264 to 2.440, p<0.001) and extended invasive mechanical ventilation to day 7 after birth (OR=4.280, 95% CI 2.149 to 8.526, p<0.001). In training and validation cohorts, the area under the curve was 0.873 (95% CI 0.839 to 0.907) and 0.848 (95% CI 0.778 to 0.917), respectively. In addition, the calibration curves showed good agreement between the column-line graph model and the actual observations. A decision curve analysis indicated a significantly better net benefit of the model.ConclusionsHigher chest X-ray scores within 7 days of life and extended invasive mechanical ventilation to day 7 after birth were significantly associated with BPD. The model had good predictive performance for predicting BPD using birth weight, chest X-ray scores within 24 hours of birth and at day 7 after life, and the state of invasive mechanical ventilation at day 7 after birth.

ObjectivesTo explore the experiences and perceptions of children with bronchiectasis and their parents regarding an 8-week play-based therapeutic exercise programme.DesignQualitative study with inductive content analysis.SettingIndividual semistructured interviews were conducted. Interview recordings were transcribed verbatim, and coding was guided by the content. Content categories were established via consensus moderation.Participants10 parents and 10 children with bronchiectasis aged 5–12 years.ResultsFrom the perspective of children, the most important components of the programme were fun with friends and being active at home as a family. Parents valued the community-based sessions, perceived the programme to be engaging and motivating. Parents perceived improvements in their child’s endurance, coordination and physical activity level. They described the home programme as fun but noted that finding time was difficult. Both parents and children thought that in-person exercise sessions would be better than exercise sessions delivered online.ConclusionsChildren who participated in the play-based exercise programme, found it fun, motivating and accessible. Parents perceived positive impacts on fitness, coordination and physical activity.Trial registration numberThe trial was registered with, Australian and New Zealand Clinical Trials Register (ACTRN12619001008112).

IntroductionIndividuals with Down syndrome (DS) are predisposed to obstructive sleep apnoea (OSA) due to craniofacial features (eg, midface hypoplasia, glossoptosis) and studies have shown that the prevalence of OSA in this population is markedly increased compared with that of typically developing children. Adenotonsillectomy is considered the first-line treatment for childhood OSA. However, persistent OSA is common, thus many children with DS are referred for positive airway pressure (PAP) therapy initiation; PAP appears to be an important aspect of living with DS. PAP has been shown to be highly effective in the general population for treating OSA and improving OSA-associated neurobehavioural symptoms, such as quality of life, behaviour, mood, daytime sleepiness and school performance. However, PAP as a treatment for OSA has not been well-studied in children with DS. Therefore, we designed a multicentre randomised controlled trial recruiting children with DS and OSA at three academic institutions, aged 6–18 years, referred for PAP initiation to treat OSA.Methods and analysis86 participants will be randomised to a 6-month intensive behavioural intervention (INT) to improve PAP adherence versus standard clinical care and underwent standardised evaluations of quality of life, behaviour, attention, PAP adherence and healthcare utilisation at baseline, 6 months and 12 months.Ethics and disseminationThis study has been approved by the institutional review board at Children’s Hospital of Philadelphia (IRB of record, IRB # 20–0 17 512). Cincinnati Children’s Medical Center and University of Miami delegated IRB review and approval responsibility to Children’s Hospital of Philadelphia through reliance agreements as mandated by National Institutes of Health (NIH). All participants will be minors; consent will be obtained from parents and assent from participants will be obtained when possible. The intervention tested in this trial is considered not greater than minimal risk, and no identifiable data will be reported. As required by the NIH, a data safety monitoring board (DSMB) has been formed, who will review and approve the protocol and any protocol changes prior to implementation. The study team will send biannual reports and hold a biannual meeting with the DSMB to review any safety and protocol concerns. Findings will be presented at national conferences pertinent to this topic and published in peer-reviewed medical journals. In addition, findings will be shared in the lay format with DS associations around the world and used for training of healthcare providers and trainees (R25HD118212). Further, data collected will be deposited in a repository (National Sleep Research Resource; sleepdata.org) after completion of the study to maximise use by scientific community.Trial registration numberNCT04132999.

Study design and objectiveCross-sectional, observational survey to describe the impact of allergic rhinitis (AR) on Australian children (2 to 15 years).MethodsParticipants (n=1541), parents of children aged 2 to 15 years, provided information on behalf of themselves and one eligible child in their household using a custom-built online questionnaire. Children were allocated to case (AR) or control (No AR) analysis groups based on a validated screening questionnaire.Statistical methodsThe study sample was stratified on age: primary analysis population (6 to 15 years, n=1111; AR=797, No AR=314); exploratory population (2 to 5 years). The primary endpoint, parent-perceived burden, was quantified using a validated measure of health status and analysed via comparison of means.ResultsThe majority of AR cases were treated (730/797; 90.3%) and classified as having moderate-severe, intermittent AR (549/797; 68.9%). Half reported adequate symptom control in the prior 2 weeks (389/797; 48.8%; OR=4.04; 95% CI (CI) 2.24 to 7.31). Having AR was associated with worse overall health status (7.4 vs 8.4, mean difference (least squares mean difference (LSMD))=−0.99; 95% CI −1.18 to −0.79), fewer days being happy (22.2 vs 25.9, LSMD=−3.68; 95% CI −4.82 to −2.54) and more days of poor physical (2.82 vs 0.78, LSMD=2.04; 95% CI 1.61 to 2.47) and emotional (2.14 vs 0.67, LSMD=1.47; 95% CI 1.02 to −1.92) health compared with not having AR. All of these outcomes were significantly (p<0.05) worse in children who reported inadequate symptom control. Having AR negatively impacted on schoolwork, sleep and other activities, and increased the likelihood of having comorbidities.ConclusionThe parent-perceived burden of AR in Australian children is high and it impacts many areas of day-to-day living. Inadequate symptom control is a key driver of the extent of this impact. Opportunities to optimise the management of AR in children include the adoption of self-assessment tools to gauge and monitor adequacy of symptom control.

ObjectiveTo identify reasons for poor asthma control in African children and adolescents.DesignSystematic reviewData sourcesPubMed, Scopus, CINHAL, PsycINFO, MEDLINE and Web of Science databases were systematically searched up to 31 May 2020. Hand searching was done on Sabinet, African Journal online and Google Scholar.Eligibility criteriaStudies identifying barriers to asthma control, where asthma control was assessed by the validated Asthma Control Test/Child Asthma Control Test and/or Asthma Control Questionnaire were included.Data extraction and synthesisTwo reviewers independently selected studies for inclusion with disagreements resolved by a research team discussion, including a third reviewer. Data were extracted using the Cochrane Effective Practice and Organization of Care data collection form. The quality of the included studies was assessed using the modified Newcastle-Ottawa quality assessment scale. Identified barriers were reported in a thematic narrative synthesis.Primary outcomesPoorly controlled asthma and associated factors.ResultsFrom 914 records, three studies conducted between 2014 and 2019 in Nigeria, Uganda and South Africa met the inclusion criteria. A total of 883 children aged 4–19 years were analysed. Older age, concurrent allergy and city-dwelling significantly impacted asthma control. Few children with asthma symptoms in the community had ever used inhaled corticosteroids (6.7%) and identified reasons included lack of asthma diagnosis (38.8%) and no prescribed treatment (47.6%).ConclusionAsthma control in African children is impacted by age, allergy, urbanisation and lack of access to asthma diagnosis and treatment. More studies focusing on identifying barriers to asthma control in Africa are needed.PROSPERORegistration no: CRD42020196755)