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Hop Mice Display Sy...
Hop Mice Display Synchronous Hindlimb Locomotion and a Ventrally Fused Lumbar Spinal Cord Caused by a Point Mutation in Ttc26
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- Rabe Bernhardt, Nadine, 1978- (author)
- Uppsala universitet,Institutionen för neurovetenskap,Tech Univ Dresden, Univ Hosp Carl Gustav Carus, Dept Psychiat & Psychotherapy, D-01307 Dresden, Germany.
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- Memic, Fatima, 1981- (author)
- Karolinska Institutet,Uppsala universitet,Institutionen för neurovetenskap
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- Velica, Anna (author)
- Uppsala universitet,Funktionella neuronala nätverk
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- Tran, Michelle A. (author)
- Univ Calgary, Hotchkiss Brain Inst, Dept Comparat Biol & Expt Med, Calgary, AB T2N 4N1, Canada.
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- Vieillard, Jennifer (author)
- Uppsala universitet,Funktionella neuronala nätverk
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- Sayyab, Shumaila (author)
- Swedish University of Agricultural Sciences,Sveriges lantbruksuniversitet,Uppsala universitet,Institutionen för medicinsk biokemi och mikrobiologi,Institutionen för husdjursgenetik (HGEN),Department of Animal Breeding and Genetics
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- Chersa, Taha (author)
- Univ Calgary, Hotchkiss Brain Inst, Dept Comparat Biol & Expt Med, Calgary, AB T2N 4N1, Canada.
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- Andersson, Leif (author)
- Swedish University of Agricultural Sciences,Sveriges lantbruksuniversitet,Uppsala universitet,Institutionen för medicinsk biokemi och mikrobiologi,Swedish Univ Agr Sci, Dept Anim Breeding & Genet, S-75007 Uppsala, Sweden.;Texas A&M Univ, Dept Vet Integrat Biosci, College Stn, TX 77843 USA.,Institutionen för husdjursgenetik (HGEN),Department of Animal Breeding and Genetics,Uppsala University,Texas AandM University College Station
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- Whelan, Patrick J. (author)
- Univ Calgary, Hotchkiss Brain Inst, Dept Comparat Biol & Expt Med, Calgary, AB T2N 4N1, Canada.
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- Boije, Henrik (author)
- Uppsala universitet,Boije: Zebrafiskens neuronala nätverk
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- Kullander, Klas, 1966- (author)
- Uppsala universitet,Funktionella neuronala nätverk
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(creator_code:org_t)
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- Society for Neuroscience, 2022
- 2022
- English.
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In: eNeuro. - : Society for Neuroscience. - 2373-2822. ; 9:2
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Abstract
Subject headings
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- Identifying the spinal circuits controlling locomotion is critical for unravelling the mechanisms controlling the production of gaits. Development of the circuits governing left-right coordination relies on axon guidance molecules such as ephrins and netrins. To date, no other class of proteins have been shown to play a role during this process. Here, we have analyzed hop mice, which walk with a characteristic hopping gait using their hindlimbs in synchrony. Fictive locomotion experiments suggest that a local defect in the ventral spinal cord contributes to the aberrant locomotor phenotype. Hop mutant spinal cords had severe morphologic defects, including the absence of the ventral midline and a poorly defined border between white and gray matter. The hop mice represent the first model where, exclusively found in the lumbar domain, the left and right components of the central pattern generators (CPGs) are fused with a synchronous hindlimb gait as a functional consequence. These defects were associated with abnormal developmental processes, including a misplaced notochord and reduced induction of ventral progenitor domains. Whereas the underlying mutation in hop mice has been suggested to lie within the Ttc26 gene, other genes in close vicinity have been associated with gait defects. Mouse embryos carrying a CRISPR replicated point mutation within Ttc26 displayed an identical morphologic phenotype. Thus, our data suggest that the assembly of the lumbar CPG network is dependent on fully functional TTC26 protein.
Subject headings
- MEDICIN OCH HÄLSOVETENSKAP -- Medicinska och farmaceutiska grundvetenskaper -- Neurovetenskaper (hsv//swe)
- MEDICAL AND HEALTH SCIENCES -- Basic Medicine -- Neurosciences (hsv//eng)
- NATURVETENSKAP -- Biologi -- Utvecklingsbiologi (hsv//swe)
- NATURAL SCIENCES -- Biological Sciences -- Developmental Biology (hsv//eng)
Keyword
- central pattern generator
- midline fusion
- rabbit-like gait
- sonic hedgehog
- spinal cord
- synchrony
Publication and Content Type
- ref (subject category)
- art (subject category)
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Rabe Bernhardt, ...
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Memic, Fatima, 1 ...
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Velica, Anna
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Tran, Michelle A ...
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Vieillard, Jenni ...
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Sayyab, Shumaila
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Chersa, Taha
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Andersson, Leif
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Whelan, Patrick ...
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Boije, Henrik
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Kullander, Klas, ...
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- About the subject
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- MEDICAL AND HEALTH SCIENCES
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MEDICAL AND HEAL ...
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and Basic Medicine
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and Neurosciences
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- NATURAL SCIENCES
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NATURAL SCIENCES
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and Biological Scien ...
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and Developmental Bi ...
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eNeuro
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Uppsala University
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Karolinska Institutet
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Swedish University of Agricultural Sciences