Preschool Multiple-Breath Washout Testing. An Official American Thoracic Society Technical Statement

Robinson, Paul D.; Latzin, Philipp; Ramsey, Kathryn; Stanojevic, Sanja; Aurora, Paul; Davis, Stephanie D.; Gappa, Monika; Hall, Graham L.; Horsley, Alex; Jensen, Renée; Lum, Sooky; Milla, Carlos; Nielsen, Kim G.; Pittman, Jessica E.; Rosenfeld, Margaret; Singer, Florian; Subbarao, Padmaja; Gustafsson, Per; Ratjen, Félix

doi:10.1164/rccm.201801-0074st

Cited by 102 publications

(122 citation statements)

References 73 publications

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“…A minimum of two (ideally three) technically acceptable trials were collected. Technically acceptable trials were determined using published quality control criteria [19], based on consensus guideline recommendations [20]. A coefficient of variance (CoV) of within 10% for three trials and 5% for two trials was seen as reassuring of good data quality.…”

Section: Spirometrymentioning

confidence: 99%

Clinical and lung function outcomes in a cohort of children with severe asthma

et al. 2020

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Background: Uncontrolled severe asthma in children is burdensome and challenging to manage. This study aims to describe outcomes in children with uncontrolled severe asthma managed in a nurse-led severe asthma clinic (SAC). Methods: This retrospective analysis uses data collected from children referred by a paediatric respiratory specialist to a nurse-led SAC for uncontrolled severe asthma between 2014 and 2019. The pre-clinical assessments included a home visit to assess modifiable factors that could be addressed to improve control. A comprehensive lung function analysis was conducted at each visit. Interventions were personalised and included biologic agents. Statistical analysis was performed using nonparametric, two-tailed Mann-Whitney U-test, the parametric Student's t-test, or analysis of variance (ANOVA) as appropriate. Results: Twenty-three children with a median age of 12 years were seen once, and 16 were followed up. Compared to a non-asthmatic (NA) and asthmatic (A) age-matched cohort, children with severe asthma (SA) had a lower FEV1, and FVC% predicted before and after bronchodilator inhalation, and a higher mean Lung Clearance Index [LCI] (10.5 [SA] versus 7.3 [NA] versus 7.6 [A], p = 0.003). Almost 80% of children with SA had an abnormal LCI, and 48% had a reduced FEV1% at the first SAC visit. Asthma control and FEV1% predicted significantly improved at a follow-up visit, while LCI remained abnormal in the majority of children (83%). Conclusion: Over time, many children with severe asthma showed improved clinical outcomes and lung function while lung ventilation inhomogeneities persisted. Future appropriately controlled studies are required to determine if a nurse-led multidisciplinary SAC is associated with better outcomes.

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Section: Spirometrymentioning

confidence: 99%

Clinical and lung function outcomes in a cohort of children with severe asthma

et al. 2020

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“…These measurements are then aligned and combined, a process that is especially challenging at rapid respiratory rates such as those seen in infants. Small errors in signal alignment or delays in signal response time can lead to significant inaccuracy in the calculation of functional residual capacity (FRC) and LCI . The Manchester method eliminates this source of inaccuracy, as the flow is not directly measured, and expired gas is collected instead for volume measurement at the end of the test.…”

Section: Introductionmentioning

confidence: 99%

“…The Manchester method eliminates this source of inaccuracy, as the flow is not directly measured, and expired gas is collected instead for volume measurement at the end of the test. By removing the need for a pneumotachometer this also significantly reduces dead space in the system, another important consideration for ensuring accuracy in infant MBW . Finally, this method measures SF 6 directly rather than calculating it indirectly from molar mass measurements, and uses a photoacoustic gas analyser which has been shown to be accurate at low concentrations .…”

Section: Introductionmentioning

confidence: 99%

“…Small errors in signal alignment or delays in signal response time can lead to significant inaccuracy in the calculation of functional residual capacity (FRC) and LCI. 9,10 The Manchester method eliminates this source of inaccuracy, as the flow is not directly measured, and expired gas is collected instead for volume measurement at the end of the test. By removing the need for a pneumotachometer this also significantly reduces dead space in the system, another important consideration for ensuring accuracy in infant MBW.…”

Section: Introductionmentioning

confidence: 99%

“…By removing the need for a pneumotachometer this also significantly reduces dead space in the system, another important consideration for ensuring accuracy in infant MBW. 10,11 Finally, this method measures SF 6 directly rather than calculating it indirectly from molar mass measurements, and uses a photoacoustic gas analyser which has been shown to be accurate at low concentrations. 12 Recently, concerns have been raised that 4% SF 6 , which is used for infant MBW with a mass spectrometer or ultrasonic flowmeter, may alter infant breathing parameters thus rendering results inaccurate 13 ; the Manchester method uses 0.1% SF 6 with a similar viscosity to air, eliminating this concern.…”

Section: Introductionmentioning

confidence: 99%

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A novel method for infant multiple breath washout: First report in clinical practice

Murray

Pike

Horsley

2019

Pediatric Pulmonology

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Background Lung clearance index (LCI), measured using multiple breath inert gas washout (MBW) is a potentially useful test in infants with respiratory disease, particularly cystic fibrosis (CF). Clinical use is limited however by the need for specialist staff and equipment. We have previously described a novel method for infant MBW suitable for use outside of specialist laboratories. This study describes its performance in vivo in infants with CF and healthy controls, including a limited comparison with the respiratory mass spectrometer. Methods Children aged less than 2 years with CF and controls underwent MBW testing on a single occasion. The practical applicability of the system was determined by the number of successful duplicate tests and within‐subject repeatability. Results Twenty‐five children (seven with CF, 18 healthy controls, all sedated with chloral hydrate) attempted MBW. Twenty patients (seven with CF) successfully underwent duplicate testing (80% success rate). Mean within‐subject coefficient of variation for functional residual capacity (FRC) was 7.2% and for LCI 5.9%. Comparison of LCI with the mass spectrometer was limited but gave very similar values for LCI and FRC in those patients who underwent technically adequate tests with both methods. Conclusions We have described a new MBW method that is feasible and reproducible in sedated infants. Results fall within the expected range, and well within accuracy limits set by international guidelines. This could provide a more accessible alternative to previously described systems for infant MBW, and overcomes many of the technical challenges inherent in conventional MBW.

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Ex vivo model predicted in vivo efficacy of CFTR modulator therapy in a child with rare genotype

Terlizzi

Amato

Castellani

et al. 2021

Molec Gen & Gen Med

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Background New drugs that target the basic defect in cystic fibrosis (CF) patients may now be used in a large number of patients carrying responsive mutations. Nevertheless, further research is needed to extend the benefit of these treatments to patients with rare mutations that are still uncharacterized in vitro and that are not included in clinical trials. For this purpose, ex vivo models are necessary to preliminary assessing the effect of CFTR modulators in these cases. Method We report the clinical effectiveness of lumacaftor/ivacaftor therapy prescribed to a CF child with a rare genetic profile (p.Phe508del/p.Gly970Asp) after testing the drug on nasal epithelial cells. We observed a significant drop of the sweat chloride value, as of the lung clearance index. A longer follow‐up period is needed to define the effects of therapy on pancreatic status, although an increase of the fecal elastase values was found. Conclusion Drug response obtained on nasal epithelial cells correlates with changes in vivo therapeutic endpoints and can be a predictor of clinical efficacy of novel drugs especially in pediatric patients.

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Preschool Multiple-Breath Washout Testing. An Official American Thoracic Society Technical Statement

Abstract: Consensus recommendations are outlined to direct interested groups of manufacturers, researchers, and clinicians in preschool device design, test performance, and data analysis for the MBW technique.

Cited by 102 publications

References 73 publications

Clinical and lung function outcomes in a cohort of children with severe asthma

Clinical and lung function outcomes in a cohort of children with severe asthma

A novel method for infant multiple breath washout: First report in clinical practice

Ex vivo model predicted in vivo efficacy of CFTR modulator therapy in a child with rare genotype

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