Prospective longitudinal association between repeated multiple breath washout measurements and computed tomography scores in children with cystic fibrosis

Sandvik, Rikke Mulvad; Kongstad, Thomas; Green, Kent; Voldby, Christian; Buchvald, Frederik; Skov, Marianne; Pressler, Tacjana; Nielsen, Kim G.

doi:10.1016/j.jcf.2020.09.010

Cited by 19 publications

(23 citation statements)

References 31 publications

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“…[23] Therefore, while LCI was stable during early school age years, we cannot assume that there was no disease progression; even more sensitive measures might be needed in the modern era of CF care to track early lung disease. [12] We have reported clinimetric properties of LCI values for school-age children. The range of values observed in healthy controls were consistent with recently published normative ranges using similar equipment and protocols [24].…”

Section: Discussionmentioning

confidence: 99%

“…Overall, between the two sites, there were 582 MBW measurements in HC and 845 measurements in CF subjects available from both preschool and school-age visits. On average the healthy control group had 13 visits (IQR 11; 15) during the two study periods, whereas the CF group had 14 visits on average (IQR [12][13][14][15][16]. As this was an observational study, not all subjects routinely attended quarterly visits, and MBW testing was not always logistically feasible to complete as clinical care always took priority.…”

Section: Summary Of the Study Populationmentioning

confidence: 99%

“…[6] Although spirometry may be normal, lung imaging studies have consistently demonstrated the presence of significant and progressive disease, including air trapping and bronchiectasis. [7][8][9][10][11][12] Consequently, more sensitive measures of lung function are needed to guide the clinical management of young children with CF.…”

Section: Introductionmentioning

confidence: 99%

“…The lung clearance index (LCI) measured by the multiple breath washout (MBW) test is a sensitive physiological marker of ventilation inhomogeneity and can detect early signs of lung disease in children with CF. The LCI can differentiate between health and disease [5,[13][14][15], track disease progression [12,16,17] and identify clinically relevant changes [18] in preschool age children with CF. In a cohort of children with CF, diagnosed based on clinical symptomatology, Aurora et al reported that LCI during preschool years was predictive of LCI and FEV 1 during the school age years; [17] these findings were supported in a more recent Australian study.…”

Section: Introductionmentioning

confidence: 99%

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Determinants of lung disease progression measured by lung clearance index in children with cystic fibrosis

et al. 2021

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BackgroundThe lung clearance index (LCI) measured by the multiple breath washout (MBW) test is sensitive to early lung disease in children with cystic fibrosis (CF). While LCI worsens during the preschool years in CF, there is limited evidence to clarify whether this continues during the early school age years, and whether the trajectory of disease progression as measured by LCI is modifiable.MethodsA cohort of children (healthy (HC) and CF) previously studied for 12 months as preschoolers were followed during school age (5–10 years). LCI was measured every 3 months for a period of 24 months using the Exhalyzer® D MBW nitrogen washout device. Linear mixed effects regression was used to model changes in LCI over time.ResultsA total of 582 MBW measurements in 48 healthy subjects and 845 measurements in 64 CF subjects were available. The majority of children with CF had elevated LCI at the first preschool and first school age visits (57.8% (37/64)), whereas all but six had normal forced expiratory volume in 1 s (FEV1) values at the first school age visit. During school age years, the course of disease was stable (−0.02 units·year−1 (95% CI −0.14; 0.10). LCI measured during preschool years, as well as the rate of LCI change during this time period, were important determinants of LCI and FEV1, at school age.ConclusionPreschool LCI was a major determinant of school age LCI; these findings further support that the preschool years are critical for early intervention strategies.

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Section: Discussionmentioning

confidence: 99%

Section: Summary Of the Study Populationmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Determinants of lung disease progression measured by lung clearance index in children with cystic fibrosis

et al. 2021

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“…As a result, an array of functional and image-testing modalities have been and continue to be actively investigated on CF for their ability to provide an accurate assessment of airway disease ( 10 – 14 ). Thanks to technological advances, parameters obtained from the multiple-breath washout technique have emerged as providing an alternative, sensitive assessment of airway function.…”

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confidence: 99%

Defining the Clinical Utility of the Lung Clearance Index. Are We There Yet?

Spano

Milla

2021

Am J Respir Crit Care Med

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Contemporary N₂ and SF₆ multiple breath washout in infants and toddlers with cystic fibrosis

Sandvik

Gustafsson

Lindblad

et al. 2022

Pediatric Pulmonology

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Introduction: Multiple breath washout (MBW) is used for early detection of cystic fibrosis (CF) lung disease, with SF 6 MBW commonly viewed as the reference method.The use of N 2 MBW in infants and toddlers has been questioned for technical and physiological reasons, but a new correction of the N 2 signal has minimized the technical part. The present study aimed to assess the remaining differences and the contributing mechanisms for the differences between SF 6 and N 2 MBW,correctedsuch as tidal volume reduction during N 2 washout with pure O 2 .Method: This was a longitudinal multicenter cohort study. SF 6 MBW and N 2 MBW were performed prospectively at three CF centers in the same visits on 154 test occasions across 62 children with CF (mean age: 22.7 months). Offline analysis using identical algorithms to the commercially available program provided outcomes of N 2,original and N 2,corrected for comparison with SF 6 MBW.Results: Mean functional residual capacity, FRC N2,corrected was 14.3% lower than FRC N2, original , and 1.0% different from FRC SF6 . Lung clearance index, LCI N2,corrected was 25.2% lower than LCI N2,original , and 7.3% higher than LCI SF6 . Mean (SD) tidal volume decreased significantly during N 2 MBW corrected , compared to SF 6 MBW (−13.1 ml [−30.7; 4.6], p < 0.0001, equal to −12.0% [−25.7; 1.73]), but this tidal

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Prospective longitudinal association between repeated multiple breath washout measurements and computed tomography scores in children with cystic fibrosis

Cited by 19 publications

References 31 publications

Determinants of lung disease progression measured by lung clearance index in children with cystic fibrosis

Determinants of lung disease progression measured by lung clearance index in children with cystic fibrosis

Defining the Clinical Utility of the Lung Clearance Index. Are We There Yet?

Contemporary N₂ and SF₆ multiple breath washout in infants and toddlers with cystic fibrosis

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Prospective longitudinal association between repeated multiple breath washout measurements and computed tomography scores in children with cystic fibrosis

Cited by 19 publications

References 31 publications

Determinants of lung disease progression measured by lung clearance index in children with cystic fibrosis

Determinants of lung disease progression measured by lung clearance index in children with cystic fibrosis

Defining the Clinical Utility of the Lung Clearance Index. Are We There Yet?

Contemporary N2 and SF6 multiple breath washout in infants and toddlers with cystic fibrosis

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Contemporary N₂ and SF₆ multiple breath washout in infants and toddlers with cystic fibrosis