Details

Autor(en) / Beteiligte

• Troutwine, Benjamin R.
• Gontarz, Paul
• Konjikusic, Mia J.
• Minowa, Ryoko
• Monstad-Rios, Adrian
• Sepich, Diane S.
• Kwon, Ronald Y.
• Solnica-Krezel, Lilianna
• Gray, Ryan S.

Titel

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

Ist Teil von

• Current biology, 2020-06, Vol.30 (12), p.2353-2362.e3

Ort / Verlag

England: Elsevier Inc

Erscheinungsjahr

2020

Quelle

Free E-Journal (出版社公開部分のみ）

Beschreibungen/Notizen

• 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. [Display omitted] [Display omitted] •Hypomorphic scospondin mutants show disassembly of the Reissner fiber and scoliosis•SCO-spondin mislocalization in floor plate cells is associated with fiber loss•A scospondin-GFP knockin strain reveals dynamic properties of Reissner fiber in vivo•Loss of the Reissner fiber is a common feature of scoliosis in zebrafish Troutwine et al. report a new scospondin-GFP knockin zebrafish strain and demonstrate intriguing dynamic properties of the Reissner fiber in the brain and central canal in vivo. Using forward genetics and cell biological approaches, they demonstrate that Reissner fiber assembly is critical during spine morphogenesis in zebrafish.