Thoracoabdominal asynchrony and paradoxical motion in Duchenne muscular dystrophy and healthy subjects during cough: A case control study

Marques, Layana; Fregonezi, Guilherme; Marcelino, Ana Aline; Dourado-Júnior, Mario Emílio Teixeira; Sarmento, António; Pennati, Francesca; Aliverti, Andréa; Resqueti, Vanessa

doi:10.1002/ppul.25412

Cited by 7 publications

(6 citation statements)

References 37 publications

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“…The symmetry of the breathing in mdx mice is of particular interest since DMD affected patients are known to exhibit signs of paradoxical breathing [ 44 , 45 , 46 ], which is defined by the changes in phase between the chest and the abdomen. Thus, simultaneity of the local maxima of the inspiration was analysed by means of ORDT.…”

Section: Resultsmentioning

confidence: 99%

Non-Invasive Optical Motion Tracking Allows Monitoring of Respiratory Dynamics in Dystrophin-Deficient Mice

Svetlove¹,

Albers²,

Hülsmann³

et al. 2022

Cells

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Duchenne muscular dystrophy (DMD) is the most common x-chromosomal inherited dystrophinopathy which leads to progressive muscle weakness and a premature death due to cardiorespiratory dysfunction. The mdx mouse lacks functional dystrophin protein and has a comparatively human-like diaphragm phenotype. To date, diaphragm function can only be inadequately mapped in preclinical studies and a simple reliable translatable method of tracking the severity of the disease still lacks. We aimed to establish a sensitive, reliable, harmless and easy way to assess the effects of respiratory muscle weakness and subsequent irregularity in breathing pattern. Optical respiratory dynamics tracking (ORDT) was developed utilising a camera to track the movement of paper markers placed on the thoracic-abdominal region of the mouse. ORDT successfully distinguished diseased mdx phenotype from healthy controls by measuring significantly higher expiration constants (k) in mdx mice compared to wildtype (wt), which were also observed in the established X-ray based lung function (XLF). In contrast to XLF, with ORDT we were able to distinguish distinct fast and slow expiratory phases. In mdx mice, a larger part of the expiratory marker displacement was achieved in this initial fast phase as compared to wt mice. This phenomenon could not be observed in the XLF measurements. We further validated the simplicity and reliability of our approach by demonstrating that it can be performed using free-hand smartphone acquisition. We conclude that ORDT has a great preclinical potential to monitor DMD and other neuromuscular diseases based on changes in the breathing patterns with the future possibility to track therapy response.

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Section: Resultsmentioning

confidence: 99%

Non-Invasive Optical Motion Tracking Allows Monitoring of Respiratory Dynamics in Dystrophin-Deficient Mice

Svetlove¹,

Albers²,

Hülsmann³

et al. 2022

Cells

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“…26 Thoracoabdominal asynchrony (TAA) results in an increased PA and can be observed in patients with obstructive airway disease, children with NMD, and preterm infants. 26,[34][35][36] To study the effect of cough and thorax positioning on TAA in patients with Duchenne muscular dystrophy (DMD), Marques et al 37 performed a cross-sectional study with 14 DMD patients and 12 agematched controls. All patients performed OEP, spirometry, respiratory muscle strength, and cough peak flow (CPF) measurement.…”

Section: Tidal Breathing Studiesmentioning

confidence: 99%

“…To study the effect of cough and thorax positioning on TAA in patients with Duchenne muscular dystrophy (DMD), Marques et al 37 performed a cross‐sectional study with 14 DMD patients and 12 age‐matched controls. All patients performed OEP, spirometry, respiratory muscle strength, and cough peak flow (CPF) measurement.…”

Section: Introductionmentioning

confidence: 99%

Pediatric pulmonology year in review 2021 and 2022: Physiology

Feld

Dobkin

Maniaci

et al. 2023

Pediatric Pulmonology

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“…Finally, Marques et al 13 assessed thoracoabdominal asynchrony and inspiratory paradoxical motion in DMD. In this cross‐sectional study with matched pair design the authors examine respiratory mechanics at different positionings during spontaneous breathing and cough.…”

Section: Upper Airway Controlmentioning

confidence: 99%