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The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine.

ABSTRACT: Cerebrospinal fluid (CSF) physiology is important for the development and homeostasis of the central nervous system, and its disruption has been linked to scoliosis in zebrafish [1, 2]. Suspended in the CSF is an extracellular structure called the Reissner fiber, which extends from the brain through the central canal of the spinal cord. Zebrafish scospondin-null mutants are unable to assemble a Reissner fiber and fail to form a straight body axis during embryonic development [3]. Here, we describe hypomorphic missense mutations of scospondin, which allow Reissner fiber assembly and extension of a straight axis. However, during larval development, these mutants display progressive Reissner fiber disassembly, which is concomitant with the emergence of axial curvatures and scoliosis in adult animals. Using a scospondin-GFP knockin zebrafish line, we demonstrate several dynamic properties of the Reissner fiber in vivo, including embryonic fiber assembly, the continuous rostral to caudal movement of the fiber within the brain and central canal, and subcommissural organ (SCO)-spondin-GFP protein secretion from the floor plate to merge with the fiber. Finally, we show that disassembly of the Reissner fiber is also associated with the progression of axial curvatures in distinct scoliosis mutant zebrafish models. Together, these data demonstrate a critical role for the Reissner fiber for the maintenance of a straight body axis and spine morphogenesis in adult zebrafish. Our study establishes a framework for future investigations to address the cellular effectors responsible for Reissner-fiber-dependent regulation of axial morphology. VIDEO ABSTRACT.

SUBMITTER: Troutwine BR

PROVIDER: S-EPMC7891109 | biostudies-literature | 2020 Jun

REPOSITORIES: biostudies-literature

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The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine.

Troutwine Benjamin R BR Gontarz Paul P Konjikusic Mia J MJ Minowa Ryoko R Monstad-Rios Adrian A Sepich Diane S DS Kwon Ronald Y RY Solnica-Krezel Lilianna L Gray Ryan S RS

Current biology : CB 20200507 12

Cerebrospinal fluid (CSF) physiology is important for the development and homeostasis of the central nervous system, and its disruption has been linked to scoliosis in zebrafish [1, 2]. Suspended in the CSF is an extracellular structure called the Reissner fiber, which extends from the brain through the central canal of the spinal cord. Zebrafish scospondin-null mutants are unable to assemble a Reissner fiber and fail to form a straight body axis during embryonic development [3]. Here, we descri ...[more]

PMID: 32386529

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Project description:A precise and comprehensive definition of "normal" in vivo cervical kinematics does not exist due to high intersubject variability and the absence of midrange kinematic data. In vitro test protocols and finite element models that are validated using only end range of motion data may not accurately reproduce continuous in vivo motion.The primary objective of this study was to precisely quantify cervical spine intervertebral kinematics during continuous, functional flexion-extension in asymptomatic subjects. The advantages of assessing continuous intervertebral kinematics were demonstrated by comparing asymptomatic controls with patients with single-level anterior arthrodesis.Cervical spine kinematics were determined during continuous in vivo flexion-extension in a clinically relevant age group of asymptomatic controls and a group of patients with C5-C6 arthrodesis.The patient sample consisted of 6 patients with single-level (C5-C6) anterior arthrodesis (average age: 48.8±6.9 years; 1 male, 5 female; 7.6±1.2 months postsurgery) and 18 asymptomatic control subjects of similar age (average age: 45.6±5.8 years; 5 male, 13 female).Outcome measures included the physiologic measure of continuous kinematic motion paths at each cervical motion segment (C2-C7) during flexion-extension.Participants performed flexion-extension while biplane radiographs were collected at 30 images per second. A previously validated tracking process determined three-dimensional vertebral positions with submillimeter accuracy. Continuous flexion-extension rotation and anterior-posterior translation motion paths were adjusted for disc height and static orientation of each corresponding motion segment.Intersubject variability in flexion-extension angle was decreased 15% to 46% and intersubject variability in anterior-posterior translation was reduced 14% to 33% after adjusting for disc height and static orientation angle. Average intersubject variability in continuous motion paths was 1.9° in flexion-extension and 0.6 mm in translation. Third-order polynomial equations were determined to precisely describe the continuous flexion-extension and anterior-posterior translation motion path at each motion segment (all R2>0.99).A significant portion of the intersubject variability in cervical kinematics can be explained by the disc height and the static orientation of each motion segment. Clinically relevant information may be gained by assessing intervertebral kinematics during continuous functional movement rather than at static, end range of motion positions. The fidelity of in vitro cervical spine mechanical testing protocols may be evaluated by comparing in vitro kinematics to the continuous motion paths presented.

Dataset Information

The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine.

Publications

The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine.

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