Earlier decline in sniff nasal inspiratory pressure than peak expiratory flow in children with Duchenne muscular dystrophy

Nève, Véronique; Edmé, Jean-Louis; Matran, Régis

doi:10.1183/09031936.00043214

Cited by 8 publications

(3 citation statements)

References 13 publications

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“…Studies on PFT and respiratory involvement have been conducted in DMD [16,20,24]. Furthermore, SNIP and pulmonary function tests have been shown to be strongly correlated with DMD, demonstrating that SNIP declines earlier than vital capacity [3,25]. Our most noteworthy result was that a low SNIP predicted respiratory muscle involvement in these patients, despite the normal FVC, indicating that FVC was insu cient to predict respiratory muscle involvement.…”

Section: Discussionmentioning

confidence: 66%

The Crucial Role of Polysomnography and Capnography in Children With Duchenne Muscular Dystrophy

Kalyoncu,

Gökdemir,

Yeğit

et al. 2024

Preprint

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Purpose Duchenne muscular dystrophy (DMD) is a severe, progressive condition characterized by muscle degeneration and weakness, significantly affecting respiratory function. This study aimed to evaluate the presence of sleep-disordered breathing (SDB) in children with DMD and investigate the relationships between sleep and respiratory function using spirometry, sniff nasal inspiratory pressure (SNIP), and polysomnography (PSG) along with capnography.Research Question: Can low SNIP be a guide for detecting respiratory muscle involvement early and determining the right time to perform early PSG and capnography in DMD?Study Design: Prospective, observational, cross-sectional study.Methods This study included DMD patients aged < 18 years. Pulmonary function tests were conducted using spirometry and SNIP, and maximum inspiratory and expiratory pressure were measured. PSG and capnography were performed within two weeks after the pulmonary function tests, and their relationships with each other were investigated.Results The study included 44 children. Obstructive sleep apnea syndrome (OSAS) was present in 70.5% of patients, while nocturnal hypoventilation was observed in 4.5%. SNIP values were significantly lower in patients with moderate-to-severe OSAS than in those without OSAS. An SNIP value below 40 cm H2O was associated with a 92.8% prevalence of OSAS.Conclusion SNIP is a valuable, noninvasive marker for the early detection of respiratory muscle involvement and SDB in patients with DMD. This study highlights the need for early and regular respiratory monitoring in children with DMD to enhance care and quality of life.

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Section: Discussionmentioning

confidence: 66%

The Crucial Role of Polysomnography and Capnography in Children With Duchenne Muscular Dystrophy

Kalyoncu,

Gökdemir,

Yeğit

et al. 2024

Preprint

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“…In a group of 51 children with Duchenne and Becker muscular dystrophy, aged 5-20 years old, SNIP measurement was found highly reproducible (coefficient of variability 8%) [93]. Nève et al also reported that SNIP decreases earlier than vital capacity and peak expiratory flow in subjects with DMD, demonstrating its value as an early marker of inspiratory strength decline [93,94]. In another study of 55 DMD patients aged from 6 to 19 years old, SNIP values decline by about 6% predicted per year [92].…”

Section: Respiratory Muscles 441 Pressurementioning

confidence: 95%

Non-Invasive Respiratory Assessment in Duchenne Muscular Dystrophy: From Clinical Research to Outcome Measures

Pennati

LoMauro

D’Angelo

et al. 2021

Life

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Ventilatory failure, due to the progressive wasting of respiratory muscles, is the main cause of death in patients with Duchenne muscular dystrophy (DMD). Reliable measures of lung function and respiratory muscle action are important to monitor disease progression, to identify early signs of ventilatory insufficiency and to plan individual respiratory management. Moreover, the current development of novel gene-modifying and pharmacological therapies highlighted the urgent need of respiratory outcomes to quantify the effects of these therapies. Pulmonary function tests represent the standard of care for lung function evaluation in DMD, but provide a global evaluation of respiratory involvement, which results from the interaction between different respiratory muscles. Currently, research studies have focused on finding novel outcome measures able to describe the behavior of individual respiratory muscles. This review overviews the measures currently identified in clinical research to follow the progressive respiratory decline in patients with DMD, from a global assessment to an individual structure–function muscle characterization. We aim to discuss their strengths and limitations, in relation to their current development and suitability as outcome measures for use in a clinical setting and as in upcoming drug trials in DMD.

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“…Linear mixed model procedure was used to analyze the change of CMI and HR max versus a broken function of IC dyn that took 0 before the knot and subject’s IC dyn after the knot; the knot position was fixed by the smallest Akaike information criterion (AIC) using Gauss–Newton method. 31 , 32 Thus, mean and 95% confidence interval of optimal knot, intercept (no change), and slope (increase) were estimated by nonlinear mixed model. As these two variables are used interchangeably in the literature to define CI, we took the middle of the overlap area of the IC dyn knot from these two relationships.…”

Section: Methodsmentioning

confidence: 99%

Chronotropic incompetence can limit exercise tolerance in COPD patients with lung hyperinflation

Hulo

Inamo

Dehon

et al. 2016

COPD

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PurposeMetabolic-chronotropic relationship is the only concept that assesses the entire chronotropic function during exercise, as it takes into account individual fitness. To better understand interrelationships between chronotropic incompetence (CI), dynamic hyperinflation (DH) and exercise limitation among Global initiative for chronic Obstructive Lung Disease (GOLD) stages of chronic obstructive pulmonary disease (COPD) disease severity, we evaluated cardiopulmonary responses to symptom-limited cycle exercise in stable patients.Patients and methodsWe prospectively studied 47 COPD patients classified by GOLD stage severity. Pulmonary function tests and cardiopulmonary responses to symptom-limited incremental exercise were studied. CI was defined by regression line between percent heart rate (HR) reserve and percent oxygen uptake (V’O2) reserve, ie, chronotropic-metabolic index (CMI). DH was defined from the knot resulting from the nonlinear regressions of inspiratory capacity changes from rest to peak (dynamic inspiratory capacity (ICdyn)) with percentage of maximal HR and CMI.ResultsAerobic capacity (median interquartile ranges) peak V’O2, 24.3 (23.6; 25.2), 18.5 (15.5; 21.8), 17.5 (15.4; 19.1) mL·kg−1·min−1 and CMI worsened according to GOLD severity. The optimal knot of ICdyn was equal to −0.34 L. The multivariate logistic regression showed a strong relationship between CI (outcome) and DH (odds ratio [confidence interval 95]) 25 (3.5; 191.6).ConclusionCOPD patients with DH have a poor cardiovascular response to exercise, which may be attributed to CI.

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Earlier decline in sniff nasal inspiratory pressure than peak expiratory flow in children with Duchenne muscular dystrophy

Cited by 8 publications

References 13 publications

The Crucial Role of Polysomnography and Capnography in Children With Duchenne Muscular Dystrophy

The Crucial Role of Polysomnography and Capnography in Children With Duchenne Muscular Dystrophy

Non-Invasive Respiratory Assessment in Duchenne Muscular Dystrophy: From Clinical Research to Outcome Measures

Chronotropic incompetence can limit exercise tolerance in COPD patients with lung hyperinflation

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