Pkd2l1 is required for mechanoception in cerebrospinal fluid-contacting neurons and maintenance of spine curvature

Jenna R. Sternberg, Andrew E. Prendergast (), Lucie Brosse, Yasmine Cantaut-Belarif, Olivier Thouvenin, Adeline Orts-Del’Immagine, Laura Castillo, Lydia Djenoune, Shusaku Kurisu, Jonathan R. McDearmid, Pierre-Luc Bardet, Claude Boccara, Hitoshi Okamoto, Patrick Delmas and Claire Wyart ()
Additional contact information
Jenna R. Sternberg: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
Andrew E. Prendergast: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
Lucie Brosse: Laboratoire de Neurosciences Cognitives-UMR7291
Yasmine Cantaut-Belarif: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
Olivier Thouvenin: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
Adeline Orts-Del’Immagine: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
Laura Castillo: Laboratoire de Neurosciences Cognitives-UMR7291
Lydia Djenoune: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
Shusaku Kurisu: Riken Brain Science Institute
Jonathan R. McDearmid: University of Leicester
Pierre-Luc Bardet: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
Claude Boccara: ESPCI Paris
Hitoshi Okamoto: Riken Brain Science Institute
Patrick Delmas: Laboratoire de Neurosciences Cognitives-UMR7291
Claire Wyart: Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière

Nature Communications, 2018, vol. 9, issue 1, 1-10

Abstract: 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.

Date: 2018
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DOI: 10.1038/s41467-018-06225-x

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