RESUMO
AIM: To review the selection, measurement and reporting of outcomes in studies of interventions in Congenital Diaphragmatic Hernia (CDH). METHODS: We searched the Cochrane Central Register of Controlled Trials from 2000-2020 for randomised trials and observational studies. Outcomes reported were classified into seven key domains modelled on the patient journey. RESULTS: Our search yielded 118 papers; 27 were eligible. The most frequent domains measured were 'short-term markers of disease activity' (17/27), whereas long-term outcomes (3/27) and outcomes relating to functional health status (8/27) were reported infrequently. There was heterogeneity in the methods and timing of outcome reporting. Primary outcomes were varied and not always clearly stated. CONCLUSION: Long-term health and functional outcomes involving interventional studies in CDH are infrequently reported, which hinders the process of shared decision-making and evidence-based healthcare. A CDH core outcome set is needed to standardise outcome reporting that is relevant to both families and healthcare teams.
Assuntos
Hérnias Diafragmáticas Congênitas , Hérnias Diafragmáticas Congênitas/terapia , Humanos , Avaliação de Resultados em Cuidados de SaúdeRESUMO
BACKGROUND: The seasonality of respiratory syncytial virus (RSV) epidemics have been disrupted during the COVID-19 pandemic, possibly because of lockdowns and social restrictions reducing viral transmission. Given uncertainties around the severity of upcoming RSV bronchiolitis epidemics, debate exists whether palivizumab (RSV prophylaxis) should be administered to infants with Congenital Diaphragmatic Hernia (CDH), who may be vulnerable due to lung hypoplasia and pulmonary hypertension. AIM: To evaluate (1) if CDH infants have higher risk of admission with RSV bronchiolitis than infants in the general population; (2) if palivizumab prophylaxis may reduce this risk. METHODS: We included all eligible studies examining the risk(s) of RSV-positive bronchiolitis requiring hospital admission in (1) CDH infants without palivizumab prophylaxis versus infants in the general population and (2) CDH infants with prophylaxis versus CDH infants without prophylaxis. The primary outcome evaluated was the risk of admission with RSV bronchiolitis. Data are reported descriptively and meta-analysed when appropriate. RESULTS: Three eligible retrospective cohort studies were identified: one study found CDH to be an independent risk factor for RSV hospitalisation (odds ratio, 3.30; 95% confidence interval [CI], 2.01-4.4); two studies compared RSV hospitalisation rates in CDH patients who had palivizumab versus those that did not. The pooled risk ratio was 1.11 (95% CI, 0.29-4.23; p = .88). Overall, the quality of evidence was considered poor and one study was industry funded. CONCLUSION: Whether CDH infants are at particular risk of severe bronchiolitis remains unclear. There is no evidence from this current systematic review that CDH infants should routinely receive palivizumab vaccination prophylaxis.
Assuntos
Bronquiolite , COVID-19 , Hérnias Diafragmáticas Congênitas , Infecções por Vírus Respiratório Sincicial , Vírus Sincicial Respiratório Humano , Antivirais/uso terapêutico , Bronquiolite/tratamento farmacológico , Bronquiolite/epidemiologia , Bronquiolite/prevenção & controle , Controle de Doenças Transmissíveis , Hospitalização , Humanos , Lactente , Palivizumab/uso terapêutico , Pandemias , Prevalência , Infecções por Vírus Respiratório Sincicial/tratamento farmacológico , Infecções por Vírus Respiratório Sincicial/epidemiologia , Infecções por Vírus Respiratório Sincicial/prevenção & controle , Estudos Retrospectivos , SARS-CoV-2RESUMO
BACKGROUND: With improvements in clinical management and an increase in CDH survivorship there is a crucial need for better understanding of long-term health outcomes in CDH. AIM: To investigate the prevalence of cardiopulmonary health morbidity and health related quality of life (HRQoL) in CDH survivors. METHODS: We included all studies (n = 65) investigating long-term cardiopulmonary outcomes in CDH patients more than 2 years published in the last 30 years. The Newcastle-Ottawa Scale and the CASP checklist for cohort studies were utilized to assess study quality. Results were reported descriptively and collated by age group where possible. RESULTS: The incidence of pulmonary hypertension was highly variable (4.5-38%), though rates (%) appeared to diminish after 5 years of age. Lung function indices and radiological outcomes were frequently abnormal, and Health Related Quality of Life (HRQoL) reduced also. Long term diseases notably emphysema and COPD are not yet fully described in the contemporary literature. CONCLUSION: This study underscores cardiopulmonary health morbidity and a reduced HRQoL among CDH survivors. Where not already available dedicated multidisciplinary follow-up clinics should be established to support these vulnerable patients transition safely into adulthood. Future research is therefore needed to investigate the risk factors for cardiopulmonary ill health and morbidity in CDH survivors. TYPE OF STUDY: Systematic review of case control and cohort studies.