期刊
DEVELOPMENTAL CELL
卷 42, 期 4, 页码 388-+出版社
CELL PRESS
DOI: 10.1016/j.devcel.2017.07.022
关键词
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资金
- European Research Council (ERC) [310098]
- Jeanne and Joseph Nissim Foundation for Life Sciences Research
- Y. Leon Benoziyo Institute for Molecular Medicine
- Beth Rom-Rymer
- Estate of David Levinson
- Jaffe Bernard and Audrey Foundation
- Georges Lustgarten Cancer Research Fund
- Estate of Bernard Bishin
- David and Fela Shapell Family
- Israel Science Foundation (ISF)
- European Research Council (ERC) [310098] Funding Source: European Research Council (ERC)
Maintaining posture requires tight regulation of the position and orientation of numerous spinal components. Yet, surprisingly little is known about this regulatory mechanism, whose failure may result in spinal deformity as in adolescent idiopathic scoliosis. Here, we use genetic mouse models to demonstrate the involvement of proprioception in regulating spine alignment. Null mutants for Runx3 transcription factor, which lack TrkC neurons connecting between proprioceptive mechanoreceptors and spinal cord, developed peripubertal scoliosis not preceded by vertebral dysplasia or muscle asymmetry. Deletion of Runx3 in the peripheral nervous system or specifically in peripheral sensory neurons, or of enhancer elements driving Runx3 expression in proprioceptive neurons, induced a similar phenotype. Egr3 knockout mice, lacking muscle spindles, but not Golgi tendon organs, displayed a less severe phenotype, suggesting that both receptor types may be required for this regulatory mechanism. These findings uncover a central role for the proprioceptive system in maintaining spinal alignment.
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