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      The effect of MRI-based screening and selection on the prevalence of syringomyelia in the Dutch and Danish Cavalier King Charles Spaniels

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          Abstract

          Introduction

          Syringomyelia (SM) is a heritable disorder causing a fluid filled cavity (FFC) in the spinal cord with a reported overall prevalence of 39 to 46% in the Cavalier King Charles Spaniels (CKCS). Breeders started screening their CKCS with MRI in the Netherlands since 2004 and in Denmark since 2015. The goal of this study was to evaluate the effect of MRI-based selection in breeding on the prevalence of SM.

          Method

          MRI scans of 2,125 purebred CKCS were available. SM was defined as having a visible FFC in the spinal cord. The prevalence of SM per year of birth was calculated, and a logistic regression was used to evaluate the affected status of offspring from affected versus unaffected parents and age category of the parent and study the combined effect of parental status and age-category to evaluate the effect on the affected status of the offspring.

          Results

          The mean FFC in affected CKCS was 2.03 ± 1.47 mm and ranged from 0.5 to 9 mm (median of 1.5 mm). An age effect exists as older CKCS, which has a higher frequency of being affected compared with younger CKCS. There was no significant sex predilection for SM in this dataset. The mean prevalence of SM decreased slightly from 38% (2010–2014; 2.8 ± 1.3 years of age (mean ± sd); median 2.6 years) to 27% (2015–2019; 2.4 ± 1.2 years of age; median 2.1 years) in the screened population of CKCS ( p = 4.3e-07). Breeding with two affected parents increased the odds ratio with 3.08 for producing affected offspring (95% CI 1.58–6.04) compared with breeding with unaffected parents.

          Discussion

          MRI-based screening and selection against SM led to a minimal decrease in the prevalence of SM in the Dutch and Danish CKCS population. Breeding with dogs with SM significantly increases the risk of affected offspring. As the disorder is progressive with age, and based on the results of this study, MRI-based screening for all CKCS is recommended at an age of 3 years or older, and to reduce SM more effectively, CKCS affected with SM should not be used for breeding.

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          Most cited references39

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          OsiriX: an open-source software for navigating in multidimensional DICOM images.

          A multidimensional image navigation and display software was designed for display and interpretation of large sets of multidimensional and multimodality images such as combined PET-CT studies. The software is developed in Objective-C on a Macintosh platform under the MacOS X operating system using the GNUstep development environment. It also benefits from the extremely fast and optimized 3D graphic capabilities of the OpenGL graphic standard widely used for computer games optimized for taking advantage of any hardware graphic accelerator boards available. In the design of the software special attention was given to adapt the user interface to the specific and complex tasks of navigating through large sets of image data. An interactive jog-wheel device widely used in the video and movie industry was implemented to allow users to navigate in the different dimensions of an image set much faster than with a traditional mouse or on-screen cursors and sliders. The program can easily be adapted for very specific tasks that require a limited number of functions, by adding and removing tools from the program's toolbar and avoiding an overwhelming number of unnecessary tools and functions. The processing and image rendering tools of the software are based on the open-source libraries ITK and VTK. This ensures that all new developments in image processing that could emerge from other academic institutions using these libraries can be directly ported to the OsiriX program. OsiriX is provided free of charge under the GNU open-source licensing agreement at http://homepage.mac.com/rossetantoine/osirix.
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            Syringomyelia: current concepts in pathogenesis, diagnosis, and treatment.

            Syringomyelia is a condition that results in fluid-containing cavities within the parenchyma of the spinal cord as a consequence of altered cerebrospinal fluid dynamics. This review discusses the history and the classification of the disorder, the current theories of pathogenesis, and the advanced imaging modalities used in the diagnosis. The intramedullary pulse pressure theory (a new pathophysiologic concept of syringomyelia) also is presented. In addition, the current understanding of the painful nature of this condition is discussed and the current trends in medical and surgical management are reviewed.
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              Prevalence of asymptomatic syringomyelia in Cavalier King Charles spaniels.

              The prevalence of syringomyelia was investigated in a sample population of 555 Cavalier King Charles spaniels. All dogs, which were declared by their owners to be showing no clinical signs of syringomyelia, underwent MRI to determine the presence or absence of the condition. Data were analysed by logistic regression to determine the effects of sex and age on the prevalence of syringomyelia. Only increased age was found to have a significant effect. The prevalence of syringomyelia was 25 per cent in dogs aged 12 months, increasing to a peak of 70 per cent in dogs aged 72 months or more.
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                Author and article information

                Contributors
                Role: Role: Role: Role: Role:
                Role: Role:
                URI : https://loop.frontiersin.org/people/2613292/overviewRole: Role: Role: Role: Role:
                URI : https://loop.frontiersin.org/people/711531/overviewRole: Role: Role: Role: Role: Role: Role: Role: Role: Role: Role: Role:
                Journal
                Front Vet Sci
                Front Vet Sci
                Front. Vet. Sci.
                Frontiers in Veterinary Science
                Frontiers Media S.A.
                2297-1769
                29 January 2024
                2024
                : 11
                : 1326621
                Affiliations
                [1] 1Expertise Centre of Genetics, Department of Clinical Sciences, Faculty of Veterinary Medicine, University of Utrecht , Utrecht, Netherlands
                [2] 2Evidensia Referral Hospital Arnhem , Arnhem, Netherlands
                Author notes

                Edited by: Andrea Tipold, University of Veterinary Medicine Hannover, Germany

                Reviewed by: Clare Rusbridge, University of Surrey, United Kingdom

                Shinji Tamura, Tamura Animal Clinic, Japan

                *Correspondence: Paul J. J. Mandigers, p.j.j.mandigers@ 123456uu.nl
                Article
                10.3389/fvets.2024.1326621
                10859423
                38348108
                c59945eb-8826-4226-b86e-ed3f37768155
                Copyright © 2024 Limpens, Smits, Fieten and Mandigers.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                : 23 October 2023
                : 09 January 2024
                Page count
                Figures: 1, Tables: 8, Equations: 0, References: 39, Pages: 9, Words: 7234
                Categories
                Veterinary Science
                Original Research
                Custom metadata
                Veterinary Neurology and Neurosurgery

                breeding,welfare,chiari,central canal dilatation,magnetic resonance imaging

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