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Title: Pkd2l1 is required for mechanoception in cerebrospinal fluid-contacting neurons and maintenance of spine curvature.
Authors: Sternberg, JR
Prendergast, AE
Brosse, L
Cantaut-Belarif, Y
Thouvenin, O
Orts-Del'Immagine, A
Castillo, L
Djenoune, L
Kurisu, S
McDearmid, JR
Bardet, P-L
Boccara, C
Okamoto, H
Delmas, P
Wyart, C
First Published: 18-Sep-2018
Publisher: Nature Research (part of Springer Nature)
Citation: Nature Communications, 2018, volume 9, Article number: 3804
Abstract: Defects in cerebrospinal fluid (CSF) flow may contribute to idiopathic scoliosis. However, the mechanisms underlying detection of CSF flow in the central canal of the spinal cord are unknown. Here we demonstrate that CSF flows bidirectionally along the antero-posterior axis in the central canal of zebrafish embryos. In the cfap298tm304 mutant, reduction of cilia motility slows transport posteriorly down the central canal and abolishes spontaneous activity of CSF-contacting neurons (CSF-cNs). Loss of the sensory Pkd2l1 channel nearly abolishes CSF-cN calcium activity and single channel opening. Recording from isolated CSF-cNs in vitro, we show that CSF-cNs are mechanosensory and require Pkd2l1 to respond to pressure. Additionally, adult pkd2l1 mutant zebrafish develop an exaggerated spine curvature, reminiscent of kyphosis in humans. These results indicate that CSF-cNs are mechanosensory cells whose Pkd2l1-driven spontaneous activity reflects CSF flow in vivo. Furthermore, Pkd2l1 in CSF-cNs contributes to maintenance of natural curvature of the spine.
DOI Link: 10.1038/s41467-018-06225-x
eISSN: 2041-1723
Version: Publisher Version
Status: Peer-reviewed
Type: Journal Article
Rights: Copyright © the authors, 2018. This is an open-access article distributed under the terms of the Creative Commons Attribution License (, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Description: Data and MATLAB scripts used for analysis are available from the authors upon request. Supplementary Information accompanies this paper at
Appears in Collections:Published Articles, Dept. of Neuroscience, Psychology and Behaviour

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