Pulmonary ventilation and micro‐structural findings in congenital diaphragmatic hernia

Spoel, Marjolein; Marshall, Helen; IJsselstijn, Hanneke; Parra‐Robles, Juan; Wiel, Els van der; Swift, Andrew J.; Rajaram, Smitha; Tibboel, Dick; Tiddens, Harm A.W.M.; Wild, Jim M.

doi:10.1002/ppul.23325

Cited by 25 publications

(28 citation statements)

References 30 publications

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“…In a cohort born before ECMO was introduced in the Netherlands (1991), mild airflow obstruction had deteriorated from childhood to young adulthood . Microstructural pulmonary changes not only in the ipsilateral lung but also in the contralateral lung were found in nonsmoking young adults in this cohort . Taking into account the very short duration of mechanical ventilation in this older group and the change in mortality rates over time, we assume that children with CDH who survived in the past decade suffered from more severe lung hypoplasia, were ventilated longer, and have more airflow obstruction.…”

Section: Discussionmentioning

confidence: 94%

Lung function in school‐aged congenital diaphragmatic hernia patients; a longitudinal evaluation

Toussaint-Duyster

Zijp

Spoel

et al. 2019

Pediatric Pulmonology

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Objective Children with congenital diaphragmatic hernia (CDH) are at risk for pulmonary morbidity. Data on longitudinal evaluation of lung function in CDH are scarce. We hypothesized that CDH patients would have impaired lung function that worsens over time. We evaluated lung function and its determinants at ages 8 and 12 years. Methods Dynamic and static lung volumes, and diffusion capacity were measured. Extracorporeal membrane oxygenation (ECMO) treatment, the standardized European neonatal treatment protocol, patch repair, duration of ventilation, type of initial mechanical ventilation, and nitric oxide treatment were entered as covariates in linear mixed models with standard deviation score (SDS) lung function parameters (FEV1, FEF 25‐75, and K CO) as dependent variables. Results Seventy‐six children (27 ECMO‐treated) born between 1999 and 2009 performed 113 reliable lung function tests. Severity of airflow obstruction deteriorated significantly from age 8 to 12 years: estimated mean difference (95% confidence interval [CI]) SDS FEV1 was −0.57 (−0.79 to −0.36) and SDS FEF25‐75 was −0.63 (−0.89 to −0.37), both P < .001. Static lung volumes were within normal range and unchanged over time: estimated mean difference (95% CI) SDS TLC −0.27 (−0.58 to 0.04); P = .085. SDS KCO was below normal at 8 and 12 years and remained stable: −0.06 (−0.22 to 0.35); P = .648. These observations were irrespective of ECMO treatment. FEV1 and FEF25‐75 were negatively associated with duration of ventilation (P < .001). Baseline data were not related with TLC or KCO. Conclusions CDH patients should be followed into adulthood as they are at risk for worsening airflow obstruction and decreased diffusion capacity at school age, irrespective of ECMO treatment.

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Section: Discussionmentioning

confidence: 94%

Lung function in school‐aged congenital diaphragmatic hernia patients; a longitudinal evaluation

Toussaint-Duyster

Zijp

Spoel

et al. 2019

Pediatric Pulmonology

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“…In previous studies, we showed that children born with major anatomical foregut anomalies and/or treated with ECMO are at risk for long‐term respiratory morbidity, reduced exercise capacity, and even deterioration of exercise capacity . In this RCT, we aimed to evaluate whether these children's exercise capacity can be improved.…”

Section: Perspectivementioning

confidence: 99%

“…We previously showed that children with CDH and EA are at risk for long‐term persistent respiratory morbidity, reduced exercise capacity, and motor function problems . Moreover, exercise capacity in neonatal ECMO survivors deteriorated between 5 and 12 years of age .…”

Section: Introductionmentioning

confidence: 99%

Improvement of exercise capacity following neonatal respiratory failure: A randomized controlled trial

Toussaint-Duyster

Zijp

Takken

et al. 2019

Scandinavian Med Sci Sports

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Exercise capacity deteriorates in school‐aged children born with major anatomical foregut anomalies and/or treated with extracorporeal membrane oxygenation. The aim of the present study was to evaluate whether exercise capacity can be improved in the short term and long term in children born with anatomical foregut anomalies and/or treated with extracorporeal membrane oxygenation. Therefore, we evaluated two different interventions in this single‐blinded randomized controlled trial. Forty participants were randomly assigned to group A: standardized anaerobic high‐intensity interval training plus online lifestyle coaching program, B: online lifestyle coaching program only, or C: standard of care. Inclusion criteria were as follows: score ≤−1 standard deviation (SD) on the Bruce protocol. Exercise capacity was assessed at baseline (T0), after 3 months (T1), and after 12 months (T2). Exercise capacity improved over time: mean (SD) standard deviation score (SDS) endurance time: T0 −1.91 (0.73); T1 −1.35 (0.94); T2 −1.20 (1.03): both P < .001. No significant differences in maximal endurance time were found at T1 (group A‐C: estimated mean difference (SDS): 0.06 P = .802; group B‐C: −0.17 P = .733) or T2 (group A‐C: −0.13 P = .635; group B‐C: −0.18 P = .587). Exercise capacity improved significantly over time, irrespective of the study arm. Not only residual morbidities may be responsible for reduced exercise capacity. Parental awareness of reduced exercise capacity rather than specific interventions may have contributed. Monitoring of exercise tolerance and providing counseling on lifestyle factors that improve physical activity should be part of routine care, and aftercare should be offered on an individual basis.

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“…Since the respiratory system cellular maintenance and repair systems rely on a sufficiently numerous and well‐positioned set of progenitor cells, it would be expected that disruption of this repertoire would have long term effects on the respiratory system development. For example, Spoel et al imaged adult congenital diaphragmatic hernia survivors using hyper‐polarized 3 H‐MRI and demonstrated persistent micro‐structural alveolar enlargement.…”

Section: Neonatologymentioning

confidence: 99%

Pediatric pulmonology year in review 2016: Part 2

et al. 2017

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Our journal covers a broad range of research and scholarly topics related to children's respiratory disorders. For updated perspectives on the rapidly expanding knowledge in our field, we will summarize the past year's publications in our major topic areas, as well as selected publications in these areas from the core clinical journal literature outside our own pages. The current review covers articles on neonatal lung disease, pulmonary physiology, and respiratory infection.Word count: 71 3

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Pulmonary ventilation and micro‐structural findings in congenital diaphragmatic hernia

Abstract: Functional and micro-structural changes persist into adulthood in most CDH patients. Ipsilateral elevated (3) He ADC values are consistent with enlargement of mean dimensions of the confining lung micro-structure at the alveolar level.

Cited by 25 publications

References 30 publications

Lung function in school‐aged congenital diaphragmatic hernia patients; a longitudinal evaluation

Lung function in school‐aged congenital diaphragmatic hernia patients; a longitudinal evaluation

Improvement of exercise capacity following neonatal respiratory failure: A randomized controlled trial

Pediatric pulmonology year in review 2016: Part 2

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