External Validation of the Discriminative Validity of the ReSVinet Score and Development of Simplified ReSVinet Scores in Secondary Care.

BACKGROUND: There is no consensus on how to best quantify disease severity in infants with respiratory syncytial virus (RSV) and/or bronchiolitis; this lack of a sufficiently validated score complicates the provision of clinical care and, the evaluation of trials of therapeutics and vaccines. The ReSVinet score appears to be one of the most promising; however, it is too time consuming to be incorporated into routine clinical care. We aimed to develop and externally validate simplified versions of this score. METHODS: Data from a multinational (the Netherlands, Spain, and United Kingdom) multicenter case-control study of infants with RSV were used to develop simplified versions of the ReSVinet score by conducting a grid search to determine the best combination of equally weighted parameters to maximize for the discriminative ability (measured by area under the receiver operating characteristic curve [AUROC]) across a range of outcomes (hospitalization, intensive care unit admission, ventilation requirement). Subsequently discriminative validity of the score for a range of secondary care outcomes was externally validated by secondary analysis of datasets from Rwanda and Colombia. RESULTS: Three candidate simplified scores were identified using the development dataset; they were excellent (AUROC >0.9) at discriminating for a range of outcomes, and their performance was not significantly different from the original ReSVinet score despite having fewer parameters. In the external validation datasets, the simplified scores were moderate to excellent (AUROC, 0.7-1) across a range of outcomes. In all outcomes, except in a single dataset for predicting admission to the high-dependency unit, they performed at least as well as the original ReSVinet score. CONCLUSIONS: The candidate simplified scores developed require further external validation in larger datasets, ideally from resource-limited settings before any recommendation regarding their use.

Corrigendum: Case Report: Two Monochorionic Twins With a Critically Different Course of Progressive Osseous Heteroplasia.

[This corrects the article DOI: 10.3389/fped.2021.662669.].

Case Report: Two Monochorionic Twins With a Critically Different Course of Progressive Osseus Heteroplasia.

Progressive osseous heteroplasia (POH; OMIM 166350) is a rare autosomal-dominant genetic disorder in which extra-skeletal bone forms within skin and muscle tissue. POH is one of the clinical manifestations of an inactivating mutation in the GNAS gene. GNAS gene alterations are difficult matter to address, as GNAS alleles show genetic imprinting and produce several transcript products, and the same mutation may lead to strikingly different phenotypes. Also, most of the publications concerning POH patients are either clinical depictions of a case (or a case series), descriptions of their genetic background, or a tentative correlation of both clinical and molecular findings. Treatment for POH is rarely addressed, and POH still lacks therapeutic options. We describe a unique case of POH in two monochorionic twins, who presented an almost asymptomatic vs. the severe clinical course, despite sharing the same mutation and genetic background. We also report the results of the therapeutic interventions currently available for heterotopic ossification in the patient with the severe course. This article not only critically supports the assumption that the POH course is strongly influenced by factors beyond genetic background but also remarks the lack of options for patients suffering an orphan disease, even after testing drugs with promising in vitro results.

The ReSVinet Score for Bronchiolitis: A Scale for All Seasons.

The ReSVinet Scale is a clinical score for the assessment of respiratory difficulty in infancy. Our aim was to provide an update on the characteristics that make this tool different to any other. We descriptively compared the characteristics of the ReSVinet Scale with other options that can be found in the current literature or mobile apps stores. The ReSVinet Scale has been devised for its use regardless of the age of the child, entity causing respiratory symptoms, and setting. To this date, it is the only pediatric respiratory scale for acute respiratory distress that provides a parental version, and it is also the first one to have undergone validation in primary care. The tool can be found either in paper format or can be downloaded via App stores for devices running on Android or iOS systems. It has also been adopted recently as the assessment instrument for several clinical trials and for an IMI2 initiative (RESCEU project). All the aforementioned characteristics of the ReSVinet Scale should help toward its dissemination, as currently no other clinical tool for the assessment of respiratory difficulty can offer those advantages.

Respiratory Syncytial Virus Seasonality: A Global Overview.

Respiratory syncytial virus (RSV) is the leading cause of acute lower respiratory infections in children. By the age of 1 year, 60%-70% of children have been infected by RSV. In addition, early-life RSV infection is associated with the development of recurrent wheezing and asthma in infancy and childhood. The need for precise epidemiologic data regarding RSV as a worldwide pathogen has been growing steadily as novel RSV therapeutics are reaching the final stages of development. To optimize the prevention, diagnosis, and treatment of RSV infection in a timely manner, knowledge about the differences in the timing of the RSV epidemics worldwide is needed. Previous analyses, based on literature reviews of individual reports obtained from medical databases, have failed to provide global country seasonality patterns. Until recently, only certain countries have been recording RSV incidence through their own surveillance systems. This analysis was based on national RSV surveillance reports and medical databases from 27 countries worldwide. This is the first study to use original-source, high-quality surveillance data to establish a global, robust, and homogeneous report on global country-specific RSV seasonality.

Clinical respiratory scales: which one should we use?

INTRODUCTION: There are countless clinical respiratory scales for acute dyspnoea. Most healthcare professionals choose one based on previous personal experience or following local practice, unaware of the implications of their choice. The lack of critical comparisons between those different tools has been a widespread problem that only recently has begun to be addressed via score validation studies. Here we try to assess and compare the quality criteria of measurement properties of acute dyspnoea scores. Areas covered: A literature review was conducted by searching the PubMed database. Forty-five documents were deemed eligible as they reported the use or building of clinical scales, using at least two parameters, and applied these to an acute episode of respiratory dyspnoea. Our primary focus was the description of the validity, reliability and utility of 41 suitable scoring instruments. Differences in sample selection, study design, rater profiles and potential methodological shortcomings were also addressed. Expert commentary: All acute dyspnoea scores lack complete validation. In particular, the areas of measurement error and interpretability have not been addressed correctly by any of the tools reviewed. Frequent modification of pre-existing scores (in items composition and/or name), differences in study design and discrepancies in reviewed sources also hinder the search for an adequate tool.

Development and Validation of a New Clinical Scale for Infants with Acute Respiratory Infection: The ReSVinet Scale.

BACKGROUND AND AIMS: A properly validated scoring system allowing objective categorization of infants with acute respiratory infections (ARIs), avoiding the need for in-person assessment and that could also be used by non-health professionals is currently not available. We aimed to develop a new clinical assessment scale meeting these specifications. METHODS: We designed a clinical scale (ReSVinet scale) based on seven parameters (feeding intolerance, medical intervention, respiratory difficulty, respiratory frequency, apnoea, general condition, fever) that were assigned different values (from 0 to 3) for a total of 20 points.170 children under two years of age with ARI were assessed independently by three pediatricians using this scale. Parents also evaluated their offspring with an adapted version of the scale in a subset of 61 cases. The scale was tested for internal consistency (Cronbach's alpha), Pearson correlation coefficient for the items in the scale, inter-observer reliability (kappa index) and floor-ceiling effect. RESULTS: Internal consistency was good for all the observers, with the lowest Cronbach's alpha being 0.72. There was a strong correlation between the investigators (r-value ranged 0.76-0.83) and also between the results obtained by the parents and the investigators(r = 0.73). Light's kappa for the observations of the three investigators was 0.74. Weighted kappa in the group evaluated by the parents was 0.73. The final score was correlated with length of hospital stay, PICU admission and Wood-Downes Score. CONCLUSIONS: The ReSVinet scale may be useful and reliable in the evaluation of infants with ARI, particularly acute bronchiolitis, even with data obtained from medical records and when employed by parents. Although further studies are necessary, ReSVinet scale already complies with more score validation criteria than the vast majority of the alternatives currently available and used in the clinical practice.