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

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  • Jenna R. Sternberg

    (Institut du Cerveau et de la Moelle épinière (ICM) - Hôpital Pitié-Salpêtrière
    Harvard University)

  • 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
    ESPCI Paris)

  • 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
    Massachusetts General Hospital)

  • Shusaku Kurisu

    (Riken Brain Science Institute
    Tokushima University Graduate School of Medical Science)

  • 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)

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.

Suggested Citation

  • 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. McD, 2018. "Pkd2l1 is required for mechanoception in cerebrospinal fluid-contacting neurons and maintenance of spine curvature," Nature Communications, Nature, vol. 9(1), pages 1-10, December.
    • Handle: RePEc:nat:natcom:v:9:y:2018:i:1:d:10.1038_s41467-018-06225-x
    DOI: 10.1038/s41467-018-06225-x
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    Cited by:

    1. Paul W. Chrystal & Nils J. Lambacher & Lance P. Doucette & James Bellingham & Elena R. Schiff & Nicole C. L. Noel & Chunmei Li & Sofia Tsiropoulou & Geoffrey A. Casey & Yi Zhai & Nathan J. Nadolski & , 2022. "The inner junction protein CFAP20 functions in motile and non-motile cilia and is critical for vision," Nature Communications, Nature, vol. 13(1), pages 1-22, December.

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