Neuromuscular Junction Defects in Mice with Mutation of dynein heavy chain 1
Publication Date
February 04, 2011
Journal
PLOS ONE
Authors
Stephanie L. Courchesne, Maria F. Pazyra Murphy, Daniel J. Lee & Rosalind A. Segal
Volume
6
Issue
2
Pages
e16753
DOI
https://dx.plos.org/10.1371/journal.pone.0016753
Publisher URL
http://journals.plos.org/plosone/article?id=10.1371%2Fjournal.pone.0016753
PubMed
http://www.ncbi.nlm.nih.gov/pubmed/21346813
PubMed Central
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3035627
Europe PMC
http://europepmc.org/abstract/MED/21346813
Scopus
79951573512
Mendeley
http://www.mendeley.com/research/neuromuscular-junction-defects-mice-mutation-dynein-heavy-chain-1
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Mendeley | Further Information

{"title"=>"Neuromuscular junction defects in mice with mutation of dynein heavy chain 1", "type"=>"journal", "authors"=>[{"first_name"=>"Stephanie L.", "last_name"=>"Courchesne", "scopus_author_id"=>"15755410400"}, {"first_name"=>"Maria F.", "last_name"=>"Pazyra-Murphy", "scopus_author_id"=>"26029127700"}, {"first_name"=>"Daniel J.", "last_name"=>"Lee", "scopus_author_id"=>"55547132020"}, {"first_name"=>"Rosalind A.", "last_name"=>"Segal", "scopus_author_id"=>"7102229846"}], "year"=>2011, "source"=>"PLoS ONE", "identifiers"=>{"issn"=>"19326203", "isbn"=>"1932-6203 (Electronic)\\r1932-6203 (Linking)", "doi"=>"10.1371/journal.pone.0016753", "pui"=>"361260693", "sgr"=>"79951573512", "pmid"=>"21346813", "scopus"=>"2-s2.0-79951573512"}, "id"=>"7929e57e-b82c-3069-aab6-6aae988fb298", "abstract"=>"Disruptions in axonal transport have been implicated in a wide range of neurodegenerative diseases. Cramping 1 (Cra1/+) and Legs at odd angles (Loa/+) mice, with hypomorphic mutations in the dynein heavy chain 1 gene, which encodes the ATPase of the retrograde motor protein dynein, were originally reported to exhibit late onset motor neuron disease. Subsequent, conflicting reports suggested that sensory neuron disease without motor neuron loss underlies the phenotypes of Cra1/+ and Loa/+ mice. Here, we present behavioral and anatomical analyses of Cra1/+ mice. We demonstrate that Cra1/+ mice exhibit early onset, stable behavioral deficits, including abnormal hindlimb posturing and decreased grip strength. These deficits do not progress through 24 months of age. No significant loss of primary motor neurons or dorsal root ganglia sensory neurons was observed at ages where the mice exhibited clear symptomatology. Instead, there is a decrease in complexity of neuromuscular junctions. These results indicate that disruption of dynein function in Cra1/+ mice results in abnormal morphology of neuromuscular junctions. The time course of behavioral deficits, as well as the nature of the morphological defects in neuromuscular junctions, suggests that disruption of dynein function in Cra1/+ mice causes a developmental defect in synapse assembly or stabilization.", "link"=>"http://www.mendeley.com/research/neuromuscular-junction-defects-mice-mutation-dynein-heavy-chain-1", "reader_count"=>28, "reader_count_by_academic_status"=>{"Unspecified"=>2, "Professor > Associate Professor"=>4, "Researcher"=>8, "Student > Ph. D. Student"=>8, "Student > Postgraduate"=>1, "Student > Master"=>4, "Professor"=>1}, "reader_count_by_user_role"=>{"Unspecified"=>2, "Professor > Associate Professor"=>4, "Researcher"=>8, "Student > Ph. D. Student"=>8, "Student > Postgraduate"=>1, "Student > Master"=>4, "Professor"=>1}, "reader_count_by_subject_area"=>{"Unspecified"=>3, "Biochemistry, Genetics and Molecular Biology"=>2, "Agricultural and Biological Sciences"=>16, "Medicine and Dentistry"=>6, "Psychology"=>1}, "reader_count_by_subdiscipline"=>{"Medicine and Dentistry"=>{"Medicine and Dentistry"=>6}, "Psychology"=>{"Psychology"=>1}, "Agricultural and Biological Sciences"=>{"Agricultural and Biological Sciences"=>16}, "Biochemistry, Genetics and Molecular Biology"=>{"Biochemistry, Genetics and Molecular Biology"=>2}, "Unspecified"=>{"Unspecified"=>3}}, "reader_count_by_country"=>{"Japan"=>1, "United Kingdom"=>1, "Australia"=>1, "France"=>1, "Germany"=>1}, "group_count"=>0}

Scopus | Further Information

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Figshare

  • {"files"=>["https://ndownloader.figshare.com/files/801052"], "description"=>"<p>A) α-bungarotoxin labeled NMJs (Scale bar  = 20 µm) B) Quantification of α-bungarotoxin labeling demonstrates no differences in the number of NMJs per section (N = 3 animals per genotype). C) Quantification of α-bungarotoxin labeled NMJs demonstrates no difference in total area (as measured by delineating outer edge and measuring area within that boundary, area analyzed is within white trace of 4Ai) of NMJs in <i>Cra1</i>/+ mice (N = 3 animals per genotype). D) NMJ complexity, as measured by the synaptic area of α-bungarotoxin label (4Aii), is significantly decreased in 2 month and 6 month old <i>Cra1</i>/+ mice (*P<0.05; N = 3 mice per genotype).</p>", "links"=>[], "tags"=>["junctions", "mice", "abnormal", "symptomatic"], "article_id"=>471418, "categories"=>["Physiology", "Neuroscience"], "users"=>["Stephanie L. Courchesne", "Maria F. Pazyra-Murphy", "Daniel J. Lee", "Rosalind A. Segal"], "doi"=>"https://dx.doi.org/10.1371/journal.pone.0016753.g004", "stats"=>{"downloads"=>1, "page_views"=>3, "likes"=>0}, "figshare_url"=>"https://figshare.com/articles/_Neuromuscular_junctions_of_Cra1_mice_are_abnormal_at_symptomatic_ages_/471418", "title"=>"Neuromuscular junctions of <i>Cra1</i>/+ mice are abnormal at symptomatic ages.", "pos_in_sequence"=>0, "defined_type"=>1, "published_date"=>"2013-02-20 20:48:33"}
  • {"files"=>["https://ndownloader.figshare.com/files/800774"], "description"=>"<p>A) Kaplan-Meier survival curves show a significant difference in survival between +/+ and <i>Cra1</i>/+ mice (*P<0.05). Cause of death is unknown, but there was no evidence of nervous system dysfunction. B) Analysis of the “cramping” posture of +/+ and <i>Cra1</i>/+ mice. Quantification of average cramping scores for +/+ and <i>Cra1</i>/+ mice from 15 days old through 12 months old shows an early onset, stable phenotype of increased cramping posture in the <i>Cra1</i>/+ mice (N = 17–34 +/+; N = 19–36 <i>Cra1</i>/+). C) Grip strength measurements for both fore- and hindlimb show significant impairments in grip strength for <i>Cra1</i>/+ mice beginning at early ages. Grip strength in <i>Cra1</i>/+ animals does not decline over time (*P<0.05; comparisons between +/+ and <i>Cra1</i>/+ mice were calculated at 3 month intervals; N = 5–34 +/+; N = 5–31 <i>Cra1</i>/+).</p>", "links"=>[], "tags"=>["mice", "non-progressive", "cramping", "hindlimbs", "reduced", "fore-"], "article_id"=>471131, "categories"=>["Physiology", "Neuroscience"], "users"=>["Stephanie L. Courchesne", "Maria F. Pazyra-Murphy", "Daniel J. Lee", "Rosalind A. Segal"], "doi"=>"https://dx.doi.org/10.1371/journal.pone.0016753.g001", "stats"=>{"downloads"=>2, "page_views"=>26, "likes"=>0}, "figshare_url"=>"https://figshare.com/articles/_Cra1_mice_show_early_onset_non_progressive_cramping_of_hindlimbs_and_reduced_grip_strength_in_fore_and_hindlimbs_/471131", "title"=>"<i>Cra1</i>/+ mice show early-onset, non-progressive cramping of hindlimbs and reduced grip strength in fore- and hindlimbs.", "pos_in_sequence"=>0, "defined_type"=>1, "published_date"=>"2013-02-20 20:46:56"}
  • {"files"=>["https://ndownloader.figshare.com/files/801142"], "description"=>"<p>A) α-bungarotoxin and GFP labeled NMJs. (Scale bar  = 20 µm). B) Motor neuron axons reveal a decrease in the proportion of NMJs with complete overlap between pre and post-synaptic zones and a compensatory increase in the proportion of NMJs with partial overlap in <i>Cra1/+</i> mice (*P<0.05). C) Labeling of nuclei by DAPI stain demonstrates a significant increase in central nuclei in <i>Cra1/+</i> mice, a measure of muscle fiber degeneration and regeneration (central nuclei indicated with red arrow; *P<0.05; N = 3 mice per genotype).</p>", "links"=>[], "tags"=>["post-synaptic", "defects", "neuromuscular", "junctions"], "article_id"=>471516, "categories"=>["Physiology", "Neuroscience"], "users"=>["Stephanie L. Courchesne", "Maria F. Pazyra-Murphy", "Daniel J. Lee", "Rosalind A. Segal"], "doi"=>"https://dx.doi.org/10.1371/journal.pone.0016753.g005", "stats"=>{"downloads"=>1, "page_views"=>8, "likes"=>0}, "figshare_url"=>"https://figshare.com/articles/_Synaptic_and_post_synaptic_defects_at_the_neuromuscular_junctions_in_Cra1_mice_/471516", "title"=>"Synaptic and post-synaptic defects at the neuromuscular junctions in <i>Cra1/+</i> mice.", "pos_in_sequence"=>0, "defined_type"=>1, "published_date"=>"2013-02-20 20:49:04"}
  • {"files"=>["https://ndownloader.figshare.com/files/800846"], "description"=>"<p>A) Motor neuron labeling with ChAT of +/+ and <i>Cra1</i>/+ mice at 6 months and 24 months of age (Scale bar  = 50 µm). B) Quantification of ChAT-labeled motor neurons reveals no difference in number between +/+ and <i>Cra1</i>/+ mice (N = 3 animals per genotype). C) Toluidine blue staining of the anterior horn of the spinal cord at 6 months and 24 months of age in +/+ and <i>Cra1/+</i> mice (white arrows indicate motor neurons, Scale bar  = 50 µm). D) Quantification of toluidine blue labeled motor neurons reveals no difference in number between +/+ and <i>Cra1</i>/+ mice (N = 3 animals per genotype).</p>", "links"=>[], "tags"=>["mice", "neuron", "symptomatic"], "article_id"=>471217, "categories"=>["Physiology", "Neuroscience"], "users"=>["Stephanie L. Courchesne", "Maria F. Pazyra-Murphy", "Daniel J. Lee", "Rosalind A. Segal"], "doi"=>"https://dx.doi.org/10.1371/journal.pone.0016753.g002", "stats"=>{"downloads"=>3, "page_views"=>55, "likes"=>0}, "figshare_url"=>"https://figshare.com/articles/_Cra1_mice_show_no_evidence_of_motor_neuron_loss_at_symptomatic_ages_/471217", "title"=>"<i>Cra1</i>/+ mice show no evidence of motor neuron loss at symptomatic ages.", "pos_in_sequence"=>0, "defined_type"=>1, "published_date"=>"2013-02-20 20:47:27"}
  • {"files"=>["https://ndownloader.figshare.com/files/800940"], "description"=>"<p>A) Proprioceptive sensory neuron labeling and quantification with parvalbumin, (B) ER81, and (C) TrkC shows no difference between of +/+ and <i>Cra1</i>/+ mice at 6 months of age (Scale bar  = 20 µm). D) Parvalbumin labeling of proprioceptive sensory neuron fibers within the spinal cord shows the central projection of these sensory neurons is intact in <i>Cra1</i>/+ mice (Scale bar  = 20 µm; N = 3 animals per genotype).</p>", "links"=>[], "tags"=>["mice", "proprioceptive", "sensory", "neuron", "symptomatic"], "article_id"=>471306, "categories"=>["Physiology", "Neuroscience"], "users"=>["Stephanie L. Courchesne", "Maria F. Pazyra-Murphy", "Daniel J. Lee", "Rosalind A. Segal"], "doi"=>"https://dx.doi.org/10.1371/journal.pone.0016753.g003", "stats"=>{"downloads"=>2, "page_views"=>8, "likes"=>0}, "figshare_url"=>"https://figshare.com/articles/_Cra1_mice_show_no_evidence_of_proprioceptive_sensory_neuron_loss_at_symptomatic_ages_/471306", "title"=>"<i>Cra1</i>/+ mice show no evidence of proprioceptive sensory neuron loss at symptomatic ages.", "pos_in_sequence"=>0, "defined_type"=>1, "published_date"=>"2013-02-20 20:47:58"}

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Relative Metric

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Net::HTTPInternalServerError

Source
Scopus
Time
2020-11-11 20:17:57 UTC
Target URL
https://api.elsevier.com/content/search/index:SCOPUS?query=DOI(10.1371%2Fjournal.pone.0016753)
Trace

/app/models/concerns/networkable.rb:21:in `get_result'
/app/models/source.rb:165:in `get_data'
/app/models/retrieval_status.rb:47:in `perform_get_data'
/app/jobs/source_job.rb:52:in `block (2 levels) in perform'
/app/jobs/source_job.rb:51:in `block in perform'
/app/jobs/source_job.rb:35:in `each'
/app/jobs/source_job.rb:35:in `perform'