1159 - Characterization Of Golden Retriever Muscular Dystrophy Biomechanics With Motion Capture During A 6-month Longitudinal Study
Notice bibliographique
Résumé
INTRODUCTION: Muscular Dystrophies (MD) are incurable diseases that progressively weaken voluntary muscles [1]. The most severe type of MD is Duchenne Muscular Dystrophy (DMD) which affects 1 in 4000-6000 boys [2]. Patients are wheelchair bound in their teens and typically die from respiratory failure or cardiomyopathy with an average life expectancy of 26 years [3]. Golden Retriever Muscular Dystrophy (GRMD) is considered a good animal model for DMD due to its genetic homology and is used to study DMD and evaluate treatments [2]. The purpose of this study is to develop a non-invasive motion capture method based on gait analysis parameters to characterize GRMD biomechanics and ultimately assess treatments proposed to improve muscular control.METHODS: Twelve dogs were divided into a control group of 5 healthy dogs, a treatment group of 5 affected dogs treated with an adeno-associated virus gene therapy and a sham control of 2 untreated affected dogs. They were studied at 2, 4 and 6 months of age. Each dog was equipped with 18 reflective markers placed on anatomical landmarks on their hind limbs. Dogs were walked at a self selected speed within a volume covered by 8 near infrared motion capture cameras recording at 100Hz. Data was collected for several consecutive steps in the middle of the capture volume. Steps from the beginning and the end of the walk were excluded to avoid acceleration impact and to make sure collected data was representative of the dogu2019s gait. After gathering data according to walking speed, joint range of motion (ROM), stride length and duration, paw elevation, and movement correlation were calculated for each dog and compared at different time points. Studentu2019s T-tests were performed to assess the statistical significance of results between the three groups.. The analysis was blinded to minimize bias. The study was conducted according to institutional animal care guidelines and was approved by the Animal Use Protocol AUP 081065.RESULTS: GRMD dogs (treated and untreated) showed lower and decreasing ROM for the hock angle compared to healthy dogs. They had a similar ROM but with more extended limbs for the stifle angle and the pelvic tilt. Paw elevation and stride length ratios for all GRMD dogs decreased with age while it was stable for their counterparts. Step duration was on average 44u00b121% longer for healthy dogs and 19u00b114% longer for affected and treated dogs compared to affected and non-treated dogs. Other parameters did not show significant differences between the groups.DISCUSSION: This motion capture method has been able to identify significant differences between affected and non-affected subjects based on their ROM, paw elevation and stride length. The slight improvement trend seen on step duration for treated dogs could be attributed to gene therapy efficacy but further studies with more subjects have to be conducted to confirm this assessment.SIGNIFICANCE/CLINICAL RELEVANCE: Golden Retrievers are considered to be the most relevant model for Duchenne Muscular Dystrophy, and studying the first 6 months of life of GRMD dogs is critical as it is considered to correspond to the first 10 years of life of DMD children. The current biomechanical characterization of the disease evolution in the dog model suggests the possibility of a similar protocol adapted for humans. Our laboratory is currently going through IRB approval to start a DMD biomechanics research study at the Orthopedic Biomechanics Research Laboratory (Houston) in collaboration with Houston Methodist Research Institute.REFERENCES: [1] Dubowitz, V. (1977). Muscular Dystrophy (pp. 5-8). Karger Publishers.[2] Kornegay, J.N. (2017). Skeletal muscle, 7(1), 9.[3] Eagle, M. et al., (2002). Neuromuscular disorders, 12(10), 926-929.ACKNOWLEDGEMENTS: Texas A&M College of Veterinary Medicine provided the dogs and funding for this research project.
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Comment cette classification a été obtenuedéplier
Prédiction machine sur la base complète
Imitation des enseignantsNi prévalence calibrée, ni vérité terrain. Validation humaine à venir. Le volet Gemma est une étiquette directe du modèle pour chaque travail de la base, lue sur la notice réduite au titre. Le volet Codex est un classifieur appris des 10 348 étiquettes directes de Codex et calibré sur les taux pondérés de l'échantillon; les champs sans appui suffisant ne portent aucun appel Codex. Le mode candidate est l'union des deux volets; le consensus est leur intersection. Ces sorties portent le statut machine_predicted_unvalidated et ne sont pas des étiquettes humaines.
Scores du classifieur distillé par catégorie (deux têtes)
| Catégorie | Codex | Gemma |
|---|---|---|
| Métarecherche | 0,001 | 0,000 |
| Méta-épidémiologie (sens strict) | 0,000 | 0,000 |
| Méta-épidémiologie (sens large) | 0,000 | 0,000 |
| Bibliométrie | 0,001 | 0,000 |
| Études des sciences et des technologies | 0,000 | 0,000 |
| Communication savante | 0,000 | 0,000 |
| Science ouverte | 0,000 | 0,000 |
| Intégrité de la recherche | 0,000 | 0,000 |
| Charge utile insuffisante (le modèle a refusé de juger) | 0,001 | 0,000 |
Scores machine (provisoires)
Les deux têtes enseignantes du modèle étudiant, lues sur ce travail. Un score ordonne la base pour la relecture; il n'affirme jamais une catégorie, et le statut de validation accompagne chaque rangée tel quel.
Scores de référence d'un modèle non mature (critères de maturité non atteints, 7 itérations). Un score ordonne; il n'affirme jamais une catégorie.
score_only:v0-immature-baseline · tel quel depuis la passe de notation : score_only signifie que le nombre peut ordonner les travaux, et qu'aucune étiquette de catégorie n'en découleClassification
machine, non validéePrédiction automatique; un appel candidat d’une seule source (Gemma direct ou Codex distillé), pas un consensus.
Le détail, modèle par modèle et score par score, se trouve en fin de page sous « Comment cette classification a été obtenue ».