date: 2022-03-08T05:51:28Z pdf:unmappedUnicodeCharsPerPage: 17 pdf:PDFVersion: 1.7 pdf:docinfo:title: Non-Invasive Optical Motion Tracking Allows Monitoring of Respiratory Dynamics in Dystrophin-Deficient Mice xmp:CreatorTool: LaTeX with hyperref Keywords: Duchenne muscular dystrophy; optical tracking; breathing dynamics; mdx mouse model; neuromuscular disorders access_permission:modify_annotations: true access_permission:can_print_degraded: true subject: 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. dc:creator: Angelika Svetlove, Jonas Albers, Swen Hülsmann, Marietta Andrea Markus, Jana Zschüntzsch, Frauke Alves and Christian Dullin dcterms:created: 2022-03-08T05:48:38Z Last-Modified: 2022-03-08T05:51:28Z dcterms:modified: 2022-03-08T05:51:28Z dc:format: application/pdf; version=1.7 title: Non-Invasive Optical Motion Tracking Allows Monitoring of Respiratory Dynamics in Dystrophin-Deficient Mice Last-Save-Date: 2022-03-08T05:51:28Z pdf:docinfo:creator_tool: LaTeX with hyperref access_permission:fill_in_form: true pdf:docinfo:keywords: Duchenne muscular dystrophy; optical tracking; breathing dynamics; mdx mouse model; neuromuscular disorders pdf:docinfo:modified: 2022-03-08T05:51:28Z meta:save-date: 2022-03-08T05:51:28Z pdf:encrypted: false dc:title: Non-Invasive Optical Motion Tracking Allows Monitoring of Respiratory Dynamics in Dystrophin-Deficient Mice modified: 2022-03-08T05:51:28Z cp:subject: 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. pdf:docinfo:subject: 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. Content-Type: application/pdf pdf:docinfo:creator: Angelika Svetlove, Jonas Albers, Swen Hülsmann, Marietta Andrea Markus, Jana Zschüntzsch, Frauke Alves and Christian Dullin X-Parsed-By: org.apache.tika.parser.DefaultParser creator: Angelika Svetlove, Jonas Albers, Swen Hülsmann, Marietta Andrea Markus, Jana Zschüntzsch, Frauke Alves and Christian Dullin meta:author: Angelika Svetlove, Jonas Albers, Swen Hülsmann, Marietta Andrea Markus, Jana Zschüntzsch, Frauke Alves and Christian Dullin dc:subject: Duchenne muscular dystrophy; optical tracking; breathing dynamics; mdx mouse model; neuromuscular disorders meta:creation-date: 2022-03-08T05:48:38Z created: 2022-03-08T05:48:38Z access_permission:extract_for_accessibility: true access_permission:assemble_document: true xmpTPg:NPages: 14 Creation-Date: 2022-03-08T05:48:38Z pdf:charsPerPage: 4002 access_permission:extract_content: true access_permission:can_print: true meta:keyword: Duchenne muscular dystrophy; optical tracking; breathing dynamics; mdx mouse model; neuromuscular disorders Author: Angelika Svetlove, Jonas Albers, Swen Hülsmann, Marietta Andrea Markus, Jana Zschüntzsch, Frauke Alves and Christian Dullin producer: pdfTeX-1.40.21 access_permission:can_modify: true pdf:docinfo:producer: pdfTeX-1.40.21 pdf:docinfo:created: 2022-03-08T05:48:38Z