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The Proprioceptive System Masterminds Spinal Alignment: Insight into the Mechanism of Scoliosis.

Abstract 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.
PMID
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Authors

Mayor MeshTerms
Keywords

Egr3

Golgi tendon organ

Runx3

dorsal root ganglia

idiopathic scoliosis

mouse

muscle spindle

proprioception

Journal Title developmental cell
Publication Year Start




PMID- 28829946
OWN - NLM
STAT- In-Process
DA  - 20170822
LR  - 20170822
IS  - 1878-1551 (Electronic)
IS  - 1534-5807 (Linking)
VI  - 42
IP  - 4
DP  - 2017 Aug 21
TI  - The Proprioceptive System Masterminds Spinal Alignment: Insight into the
      Mechanism of Scoliosis.
PG  - 388-399.e3
LID - S1534-5807(17)30598-1 [pii]
LID - 10.1016/j.devcel.2017.07.022 [doi]
AB  - 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.
CI  - Copyright (c) 2017 Elsevier Inc. All rights reserved.
FAU - Blecher, Ronen
AU  - Blecher R
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel; Department of Orthopedic Surgery, Assaf HaRofeh Medical Center, Sackler
      Faculty of Medicine, Tel Aviv University, Zerrifin 70300, Israel.
FAU - Krief, Sharon
AU  - Krief S
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel.
FAU - Galili, Tal
AU  - Galili T
AD  - Department of Statistics and Operations Research, Tel Aviv University, Tel Aviv
      69978, Israel.
FAU - Biton, Inbal E
AU  - Biton IE
AD  - Department of Veterinary Resources, Weizmann Institute of Science, Rehovot 76100,
      Israel.
FAU - Stern, Tomer
AU  - Stern T
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel.
FAU - Assaraf, Eran
AU  - Assaraf E
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel; Department of Orthopedic Surgery, Assaf HaRofeh Medical Center, Sackler
      Faculty of Medicine, Tel Aviv University, Zerrifin 70300, Israel.
FAU - Levanon, Ditsa
AU  - Levanon D
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel.
FAU - Appel, Elena
AU  - Appel E
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel.
FAU - Anekstein, Yoram
AU  - Anekstein Y
AD  - Department of Orthopedic Surgery, Assaf HaRofeh Medical Center, Sackler Faculty
      of Medicine, Tel Aviv University, Zerrifin 70300, Israel.
FAU - Agar, Gabriel
AU  - Agar G
AD  - Department of Orthopedic Surgery, Assaf HaRofeh Medical Center, Sackler Faculty
      of Medicine, Tel Aviv University, Zerrifin 70300, Israel.
FAU - Groner, Yoram
AU  - Groner Y
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel.
FAU - Zelzer, Elazar
AU  - Zelzer E
AD  - Department of Molecular Genetics, Weizmann Institute of Science, Rehovot 76100,
      Israel. Electronic address: [email protected]
LA  - eng
PT  - Journal Article
PL  - United States
TA  - Dev Cell
JT  - Developmental cell
JID - 101120028
OTO - NOTNLM
OT  - Egr3
OT  - Golgi tendon organ
OT  - Runx3
OT  - dorsal root ganglia
OT  - idiopathic scoliosis
OT  - mouse
OT  - muscle spindle
OT  - proprioception
EDAT- 2017/08/23 06:00
MHDA- 2017/08/23 06:00
CRDT- 2017/08/23 06:00
PHST- 2016/12/08 [received]
PHST- 2017/06/10 [revised]
PHST- 2017/07/24 [accepted]
AID - S1534-5807(17)30598-1 [pii]
AID - 10.1016/j.devcel.2017.07.022 [doi]
PST - ppublish
SO  - Dev Cell. 2017 Aug 21;42(4):388-399.e3. doi: 10.1016/j.devcel.2017.07.022.