2020 journal article
Expiratory dysfunction in young dogs with golden retriever muscular dystrophy
NEUROMUSCULAR DISORDERS, 30(11), 930–937.
author keywords: Duchenne muscular dystrophy (DMD); Golden retriever muscular dystrophy (GRMD); Spirometry; Respiratory inductance plethysmography; Pulmonary function test; Peak expiratory flow
MeSH headings : Animals; Diaphragm / physiopathology; Disease Models, Animal; Dog Diseases / physiopathology; Dogs; Exhalation; Muscular Dystrophy, Animal / physiopathology; Muscular Dystrophy, Duchenne / physiopathology; Peak Expiratory Flow Rate; Respiratory Function Tests
TL;DR:
Tidal breathing spirometry and respiratory inductance plethysmography were performed in GRMD dogs at approximately 3 and 6 months of age, corresponding to approximately 5-10 years in DMD, when clinical trials are often conducted.
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UN Sustainable Development Goal Categories
3. Good Health and Well-being
(Web of Science; OpenAlex)
Source: Web Of Science
Added: December 21, 2020
Respiratory disease is a leading cause of morbidity in people with Duchenne muscular dystrophy and also occurs in the golden retriever muscular dystrophy (GRMD) model. We have previously shown that adult GRMD dogs have elevated expiratory flow as measured non-invasively during tidal breathing. This abnormality likely results from increased chest and diaphragmatic recoil associated with fibrosis and remodeling. Treatments must reverse pathologic effects on the diaphragm and other respiratory muscles to maximally reduce disease morbidity and mortality. Here, we extended our work in adults to younger GRMD dogs to define parameters that would be helpful in preclinical trials. Tidal breathing spirometry and respiratory inductance plethysmography were performed in GRMD dogs at approximately 3 and 6 months of age, corresponding to approximately 5–10 years in DMD, when clinical trials are often conducted. Expiratory flows were markedly elevated in GRMD versus normal dogs at 6 months. Values increased in GRMD dogs between 3 and 6 months, providing a 3-month window to assess treatment efficacy. These changes in breathing mechanics have not been previously identified at such an early age. Expiratory flow measured during tidal breathing of unsedated young GRMD dogs could be a valuable marker of respiratory mechanics during preclinical trials.