ArticleNeuron2014
Katanin p80 regulates human cortical development by limiting centriole and cilia number.
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Article in Neuron, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 60 papers.
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60citing papers in PubMed
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60 citing papers in PubMed, 100 citations in OpenAlex.
- KATNAL2 dysfunction impairs microtubule dynamics and contributes to syndromic neurodevelopmental disease.Human molecular genetics · 2026Article
- Molecular Insights Into the Differential Binding Affinity of Human Katanin Hexamer for C-Terminal Tails of β-Tubulin Isotypes.Cytoskeleton (Hoboken, N.J.) · 2026Article
- Missense variants in KATNA1 alter microtubule dynamics and underlie dominant macular dystrophy.Research square · 2026Article
- Microtubule-Associated Proteins (MAPs) Are Multifunctional Cytoskeletal Proteins in the Testis That Regulate Spermatogenesis.Advances in experimental medicine and biology · 2025Review
- Exploring purine analogues as inhibitors against Katanin, a microtubule severing enzyme using molecular modeling approach.Scientific reports · 2024Article
- KATANIN-mediated microtubule severing is required for MTOC organisation and function in Marchantia polymorpha.Development (Cambridge, England) · 2024Article
- Microtubule-binding domains in Katanin p80 subunit are essential for severing activity in C. elegans.The Journal of cell biology · 2024Article
- The "microcephalic hydrocephalus" paradox as a paradigm of altered neural stem cell biology.Cerebral cortex (New York, N.Y. : 1991) · 2024Article
- Spastin is an essential regulator of male meiosis, acrosome formation, manchette structure and nuclear integrity.Development (Cambridge, England) · 2023Article
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- Combinatorial and antagonistic effects of tubulin glutamylation and glycylation on katanin microtubule severing.Developmental cell · 2022Article
- Resolving primary pathomechanisms driving idiopathic-like spinal curvature using a newiScience · 2022Article
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- Zika virus alters centrosome organization to suppress the innate immune response.EMBO reports · 2022Article
- STRIPAK regulation of katanin microtubule severing in the Caenorhabditis elegans embryo.Genetics · 2022Article
- Endosomal trafficking defects alter neural progenitor proliferation and cause microcephaly.Nature communications · 2022Article
- The Role of Spastin in Axon Biology.Frontiers in cell and developmental biology · 2022Review
- The Symmetry of Neural Stem Cell and Progenitor Divisions in the Vertebrate Brain.Frontiers in cell and developmental biology · 2022Review
- The SON RNA splicing factor is required for intracellular trafficking structures that promote centriole assembly and ciliogenesis.Molecular biology of the cell · 2021Article
- Article
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Authors and funding
24 authors at 16 institutions in 7 countries.
Wen F HuDivision of Genetics and Genomics, Department of Medicine, Boston Children's Hospital, Boston, MA 02115, USA; Manton Center for Orphan Disease Research, Boston Children's Hospital, Boston, MA 02115, USA; Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA 02115, USA; Program in Neuroscience, Harvard Medical School, Boston, MA 02115, USA; Harvard MD-PhD MSTP Program, Harvard Medical School, Boston, MA 02115, USA.
Oz PompInstitute of Medical Biology, Human Genetics and Embryology Laboratory, A(∗)STAR, Singapore 138648, Singapore.
Tawfeg Ben-OmranClinical and Metabolic Genetics Division, Department of Pediatrics, Hamad Medical Corporation, Doha, 3050, Qatar.
Andrew KodaniDepartment of Biochemistry and Biophysics, Cardiovascular Research Institute, University of California, San Francisco, San Francisco, CA 94158, USA.
Katrin HenkeDepartment of Orthopedic Research, Boston Children's Hospital, Boston, MA 02115, USA; Department of Genetics, Harvard Medical School, Boston, MA 02115, USA.
Ganeshwaran H MochidaDivision of Genetics and Genomics, Department of Medicine, Boston Children's Hospital, Boston, MA 02115, USA; Manton Center for Orphan Disease Research, Boston Children's Hospital, Boston, MA 02115, USA; Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA; Pediatric Neurology Unit, Department of Neurology, Massachusetts General Hospital, Boston, MA 02114, USA.
Timothy W YuDivision of Genetics and Genomics, Department of Medicine, Boston Children's Hospital, Boston, MA 02115, USA; Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA; Program in Medical and Population Genetics, Broad Institute of MIT and Harvard University, Cambridge, MA 02142, USA.
Mollie B WoodworthDivision of Genetics and Genomics, Department of Medicine, Boston Children's Hospital, Boston, MA 02115, USA; Manton Center for Orphan Disease Research, Boston Children's Hospital, Boston, MA 02115, USA; Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA 02115, USA; Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA.
Carine BonnardInstitute of Medical Biology, Human Genetics and Embryology Laboratory, A(∗)STAR, Singapore 138648, Singapore.
Grace Selva RajInstitute of Medical Biology, Human Genetics and Embryology Laboratory, A(∗)STAR, Singapore 138648, Singapore.
Thong Teck TanInstitute of Medical Biology, Human Genetics and Embryology Laboratory, A(∗)STAR, Singapore 138648, Singapore.
Hanan HamamyDepartment of Genetic Medicine and Development, Geneva University, Geneva 1211, Switzerland.
Amira MasriDepartment of Paediatrics, Faculty of Medicine, University of Jordan, Amman, 11942, Jordan.
Mohammad ShboulInstitute of Medical Biology, Human Genetics and Embryology Laboratory, A(∗)STAR, Singapore 138648, Singapore.
Muna Al SaffarDivision of Genetics and Genomics, Department of Medicine, Boston Children's Hospital, Boston, MA 02115, USA; Manton Center for Orphan Disease Research, Boston Children's Hospital, Boston, MA 02115, USA; Department of Pediatrics, Faculty of Medicine and Health Sciences, United Arab Emirates University, Al-Ain, United Arab Emirates.
Jennifer N PartlowDivision of Genetics and Genomics, Department of Medicine, Boston Children's Hospital, Boston, MA 02115, USA; Manton Center for Orphan Disease Research, Boston Children's Hospital, Boston, MA 02115, USA; Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA 02115, USA.
Mohammed Al-DosariDepartment of Pharmacognosy, College of Pharmacy, King Saud University, Riyadh 11451, Saudi Arabia.
Anas AlazamiDepartment of Genetics, King Faisal Specialist Hospital and Research Centre, Riyadh 11211, Saudi Arabia.
Mohammed AlowainDepartment of Medical Genetics, King Faisal Specialist Hospital and Research Centre, Riyadh 11211, Saudi Arabia; Department of Anatomy and Cell Biology, College of Medicine, Alfaisal University, Riyadh 11533, Saudi Arabia.
Fowzan S AlkurayaDepartment of Genetics, King Faisal Specialist Hospital and Research Centre, Riyadh 11211, Saudi Arabia; Department of Anatomy and Cell Biology, College of Medicine, Alfaisal University, Riyadh 11533, Saudi Arabia.
Jeremy F ReiterDepartment of Biochemistry and Biophysics, Cardiovascular Research Institute, University of California, San Francisco, San Francisco, CA 94158, USA.
Matthew P HarrisDepartment of Orthopedic Research, Boston Children's Hospital, Boston, MA 02115, USA; Department of Genetics, Harvard Medical School, Boston, MA 02115, USA. Electronic address: harris@genetics.med.harvard.edu.
Bruno ReversadeInstitute of Medical Biology, Human Genetics and Embryology Laboratory, A(∗)STAR, Singapore 138648, Singapore; Institute of Molecular and Cellular Biology, A(∗)STAR, Singapore 138648, Singapore; Department of Paediatrics, National University of Singapore, Singapore 119260, Singapore. Electronic address: bruno@reversade.com.
Christopher A WalshDivision of Genetics and Genomics, Department of Medicine, Boston Children's Hospital, Boston, MA 02115, USA; Manton Center for Orphan Disease Research, Boston Children's Hospital, Boston, MA 02115, USA; Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA 02115, USA; Program in Neuroscience, Harvard Medical School, Boston, MA 02115, USA; Harvard MD-PhD MSTP Program, Harvard Medical School, Boston, MA 02115, USA; Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA; Department of Neurology, Harvard Medical School, Boston, MA 02115, USA. Electronic address: christopher.walsh@childrens.harvard.edu.
Boston Children's Hospital · USInstitute of Medical Biology · SGAgency for Science, Technology and Research · SGHoward Hughes Medical Institute · USKing Faisal Specialist Hospital & Research Centre · SAUniversity of California, San Francisco · USAlfaisal University · SABroad Institute · USDana-Farber/Boston Children's Cancer and Blood Disorders Center · USHamad Medical Corporation · QAHarvard University · USKing Saud University · SAMassachusetts General Hospital · USNational University of Singapore · SGUniversity of Geneva · CHUniversity of Jordan · JO
Funding
Medical Scientist Training ProgramT32GM007753 · NIGMS · HARVARD UNIVERSITY (MEDICAL SCHOOL) · PI WALENSKY, LOREN DAVID · 1985 to 2021
$50.0MSYNAPTIC MECHANISMS IN HIPPOCAMPAL EPILEPTOGENESISP30HD018655 · NICHD · CHILDREN'S HOSPITAL BOSTON · PI POMEROY, SCOTT LOREN · 1985 to 2015
$26.2MMOLECULAR AND CELLULAR BASIS OF CARDIOVASCULAR DISEASET32HL007731 · NHLBI · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Brian L Black · 1992 to 2026
$12.4MHUMAN EPILEPSY GENETICS--NEURONAL MIGRATION DISORDERSR01NS035129 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI WALSH, CHRISTOPHER A. · 1997 to 2023
$9.5MHedgehog signaling at the cell's antenna: Smoothened and the primary ciliumR01AR054396 · NIAMS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Jeremy F Reiter · 2007 to 2026
$9.0MHuman autism genetics and activity dependent gene activationRC2MH089952 · NIMH · BOSTON CHILDREN'S HOSPITAL · PI WALSH, CHRISTOPHER A. · 2009 to 2010
$5.1MTransition zone control of ciliary signalingR01GM095941 · NIGMS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI REITER, JEREMY F · 2011 to 2018
$3.4MAnatomical atlas and transgenic toolkit for late skull formation in zebrafishU01DE024434 · NIDCR · UNIVERSITY OF PENNSYLVANIA · PI FISHER, SHANNON, HARRIS, MATTHEW P · 2014 to 2018
$3.3MKatanin p80: A Role for Regulation of Microtubule Severing in Cortical DevelopmenF31NS083111 · NINDS · HARVARD MEDICAL SCHOOL · PI HU, WEN FAN · 2013 to 2014
$38kHoward Hughes Medical InstituteNHLBI NIH HHS T32 HL007731NHLBI NIH HHS T32HL007731NIAMS NIH HHS R01 AR054396NIAMS NIH HHS R01AR054396NICHD NIH HHS P30 HD018655NIDCR NIH HHS 1U01DE024434-01NIDCR NIH HHS U01 DE024434NIGMS NIH HHS R01 GM095941NIGMS NIH HHS R01GM095941NIGMS NIH HHS T32 GM007753NIGMS NIH HHS T32GM007753NIMH NIH HHS RC2 MH089952NIMH NIH HHS RC2MH089952NINDS NIH HHS F31 NS083111NINDS NIH HHS F31NS083111NINDS NIH HHS R01 NS035129NINDS NIH HHS R01NS035129
Abstract
Katanin is a microtubule-severing complex whose catalytic activities are well characterized, but whose in vivo functions are incompletely understood. Human mutations in KATNB1, which encodes the noncatalytic regulatory p80 subunit of katanin, cause severe microlissencephaly. Loss of Katnb1 in mice confirms essential roles in neurogenesis and cell survival, while loss of zebrafish katnb1 reveals specific roles for katnin p80 in early and late developmental stages. Surprisingly, Katnb1 null mutant mouse embryos display hallmarks of aberrant Sonic hedgehog signaling, including holoprosencephaly. KATNB1-deficient human cells show defective proliferation and spindle structure, while Katnb1 null fibroblasts also demonstrate a remarkable excess of centrioles, with supernumerary cilia but deficient Hedgehog signaling. Our results reveal unexpected functions for KATNB1 in regulating overall centriole, mother centriole, and cilia number, and as an essential gene for normal Hedgehog signaling during neocortical development.
Indexed as
Adenosine TriphosphatasesAnimalsCase-Control StudiesCell ProliferationCentriolesCerebral CortexCiliaEmbryo, MammalianEmbryonic DevelopmentFibroblastsHedgehog ProteinsHumansKataninMiceMicrocephalyMutationAdenosine TriphosphatasesHedgehog ProteinsKataninShh protein, mouse
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