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Fluids and Barriers of the CNS
Published in: Fluids and Barriers of the CNS 1/2023

Open Access 01-12-2023 | Research

SCO-spondin knockout mice exhibit small brain ventricles and mild spine deformation

Authors: Huixin Xu, Guillaume P. Dugué, Yasmine Cantaut-Belarif, François-Xavier Lejeune, Suhasini Gupta, Claire Wyart, Maria K. Lehtinen

Published in: Fluids and Barriers of the CNS | Issue 1/2023

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Abstract

Reissner’s fiber (RF) is an extracellular polymer comprising the large monomeric protein SCO-spondin (SSPO) secreted by the subcommissural organ (SCO) that extends through cerebrospinal fluid (CSF)-filled ventricles into the central canal of the spinal cord. In zebrafish, RF and CSF-contacting neurons (CSF-cNs) form an axial sensory system that detects spinal curvature, instructs morphogenesis of the body axis, and enables proper alignment of the spine. In mammalian models, RF has been implicated in CSF circulation. However, challenges in manipulating Sspo, an exceptionally large gene of 15,719 nucleotides, with traditional approaches has limited progress. Here, we generated a Sspo knockout mouse model using CRISPR/Cas9-mediated genome-editing. Sspo knockout mice lacked RF-positive material in the SCO and fibrillar condensates in the brain ventricles. Remarkably, Sspo knockout brain ventricle sizes were reduced compared to littermate controls. Minor defects in thoracic spine curvature were detected in Sspo knockouts, which did not alter basic motor behaviors tested. Altogether, our work in mouse demonstrates that SSPO and RF regulate ventricle size during development but only moderately impact spine geometry.
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Metadata
Title
SCO-spondin knockout mice exhibit small brain ventricles and mild spine deformation
Authors
Huixin Xu
Guillaume P. Dugué
Yasmine Cantaut-Belarif
François-Xavier Lejeune
Suhasini Gupta
Claire Wyart
Maria K. Lehtinen
Publication date
01-12-2023
Publisher
BioMed Central
Published in
Fluids and Barriers of the CNS / Issue 1/2023
Electronic ISSN: 2045-8118
DOI
https://doi.org/10.1186/s12987-023-00491-8

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