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We began analyzing https://www.jneurosci.org/content/27/52/14515, but it redirected us to https://www.jneurosci.org/content/27/52/14515. The analysis below is for the second page.

Title[redir]:
Proprioceptive Sensory Neuropathy in Mice with a Mutation in the Cytoplasmic Dynein Heavy Chain 1 Gene | Journal of Neuroscience
Description:
Mice heterozygous for the radiation-induced Sprawling ( Swl ) mutation display an early-onset sensory neuropathy with muscle spindle deficiency. The lack of an H reflex despite normal motor nerve function in the hindlimbs of these mutants strongly suggests defective proprioception. Immunohistochemical analyses reveal that proprioceptive sensory neurons are severely compromised in the lumbar dorsal root ganglia of newborn Swl /+ mice, whereas motor neuron numbers remain unaltered even in aged animals. We have used positional cloning to identify a nine base-pair deletion in the cytoplasmic dynein heavy chain 1 gene ( Dync1h1 ) in this mutant. Furthermore, we demonstrate that Loa /+ mice, which have previously been shown to carry a missense point mutation in Dync1h1 that results in late-onset motor neuron loss, also present with a severe, early-onset proprioceptive sensory neuropathy. Interestingly, in contrast to the Loa mutation, the Swl mutation does not delay disease progression in a motor neuron disease mouse model overexpressing a human mutant superoxide dismutase (SOD1G93A) transgene. Together, we provide in vivo evidence that distinct mutations in cytoplasmic dynein can either result in a pure sensory neuropathy or in a sensory neuropathy with motor neuron involvement.

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Keywords {๐Ÿ”}

mice, swl, sensory, motor, neurons, dynch, neuron, scholar, dynein, mutation, proprioceptive, muscle, loa, test, fig, openurlcrossrefpubmedgoogle, cytoplasmic, lumbar, drg, spindles, number, gene, sodga, mutant, survival, cell, grip, strength, antibody, mutations, reduced, disease, function, chain, nerve, loss, mouse, degeneration, transport, analysis, hindlimbs, mutants, students, hafezparast, duchen, drgs, compared, reflex, neuropathy, spindle,

Topics {โœ’๏ธ}

charcot-marie-tooth disease minus-end-directed motor protein independent n-ethyl-n-nitrosourea charcot-marie-tooth' mannโ€“whitney rank-sum test rabbit polyclonal anti-parvalbumin calcitonin gene-related peptide brain-derived neurotrophic factor disrupted neurotrophin/trk signaling standard dye-terminator chemistry activated ngf/trka complex early-onset sensory neuropathy kaplan-meier survival analyses mammalian genetic unit/harwell efferent ฮฑ-motor fibers multi-subunit complex essential dynein-dependent neuronal degeneration multiple neuron-specific processes c57bl/6j genomic dna fitc-conjugated secondary antibodies trka/ngf receptor gene high-resolution linkage analysis constant-current stimulator dynein-dependent axonal transport viable swl/swl mutants swl/swl mutants die radiation-induced mutation sprawling target-dependent neurons main content log references โ†ต ahmad-annuar early-onset hsn swl/sod1g93a mice showed afferent ia fibers dorsal root ganglia authors editorial board collagen iv antibody male wild-type motor neuron disease motor neuron degeneration egr3-positive muscle spindles c-terminal end issue journal limb proprioceptive afferents ฮฑ-motor neuron proprioceptive sensory defect trk neurotrophin receptors sensory neuron degeneration n-terminal portion cargo-binding domain jneurosci editorial board

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