Evidence map›Paper›PMID 32097629›Full record

ArticleNeuron2020

Posterior Neocortex-Specific Regulation of Neuronal Migration by CEP85L Identifies Maternal Centriole-Dependent Activation of CDK5.

Andrew Kodani, Connor Kenny, Abbe Lai, Dilenny M Gonzalez, Edward Stronge, Gabrielle M Sejourne, Laura Isacco, Jennifer N Partlow, Anne O'Donnell, Kirsty McWalter and 13 more

Open access · bronzeAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Neuron, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.

0numbers the graph read from it
0cells of the map it votes in
10citing papers in PubMed
2.0field-weighted citation impact, top 15% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

10 citing papers in PubMed, 25 citations in OpenAlex.

  1. Article
  2. Review
  3. The Control of Cortical Folding: Multiple Mechanisms, Multiple Models.The Neuroscientist : a review journal bringing neurobiology, neurology and psychiatry · 2024
    Review
  4. Review
  5. Article
  6. MiR-582 Down-Regulates Lissencephaly-1 (Iranian journal of biotechnology · 2022
    Article
  7. Review
  8. The role of Cdk5 in neurological disorders.Frontiers in cellular neuroscience · 2022
    Review
  9. Article
  10. Cyclin-dependent kinases and rare developmental disorders.Orphanet journal of rare diseases · 2020
    Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

23 authors at 6 institutions in 2 countries.

Andrew KodaniDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA. Electronic address: andrew.kodani@childrens.harvard.edu.
Connor KennyDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA.
Abbe LaiDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA; Department of Neurology, Boston Children's Hospital, Boston, MA, USA.
Dilenny M GonzalezDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA.
Edward StrongeDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA.
Gabrielle M SejourneDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA.
Laura IsaccoDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA.
Jennifer N PartlowDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA.
Anne O'DonnellDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA; Center for Mendelian Genomics, The Broad Institute of MIT and Harvard, Cambridge, MA, USA.
Kirsty McWalterGeneDX, Gaithersburg, MD, USA.
Alicia B ByrneCenter for Mendelian Genomics, The Broad Institute of MIT and Harvard, Cambridge, MA, USA; Department of Genetics and Molecular Pathology, Centre for Cancer Biology, SA Pathology, Adelaide, SA, Australia; School of Pharmacy and Medical Sciences, University of South Australia, Adelaide, SA, Australia.
A James BarkovichDepartment of Radiology, University of California, San Francisco, San Francisco, CA, USA.
Edward YangDepartment of Radiology, Boston Children's Hospital, Boston, MA, USA.
R Sean HillDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA.
Pawel GawlinskiDepartment of Medical Genetics, Institute of Mother and Child, Warsaw, Poland.
Wojciech WiszniewskiDepartment of Medical Genetics, Institute of Mother and Child, Warsaw, Poland; Department of Molecular and Medical Genetics, Oregon Health and Science University, Portland, OR, USA.
Julie S CohenDivision of Neurogenetics, Kennedy Krieger Institute, Baltimore, MD, USA; Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
S Ali FatemiDivision of Neurogenetics, Kennedy Krieger Institute, Baltimore, MD, USA; Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD, USA; Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
Kristin W BarananoDivision of Neurogenetics, Kennedy Krieger Institute, Baltimore, MD, USA; Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
Mustafa SahinDepartment of Neurology, Boston Children's Hospital, Boston, MA, USA.
David G VosslerDepartment of Neurology, University of Washington, Seattle, WA, USA.
Christopher J YuskaitisDepartment of Neurology, Boston Children's Hospital, Boston, MA, USA; Division of Epilepsy and Neurophysiology, Department of Neurology, Boston Children's Hospital, Boston, MA, USA.
Christopher A WalshDivision of Genetics and Genomics and Howard Hughes Medical Institute, Boston Children's Hospital, Boston, MA, USA; Department of Pediatrics and Neurology, Harvard Medical School, Boston, MA, USA. Electronic address: christopher.walsh@childrens.harvard.edu.
Boston Children's Hospital · USKennedy Krieger Institute · USBroad Institute · USOregon Health & Science University · USUniversity of California, San Francisco · USUniversity of Washington · US

Funding

Medical Scientist Training ProgramT32GM007753 · NIGMS · HARVARD UNIVERSITY (MEDICAL SCHOOL) · PI WALENSKY, LOREN DAVID · 1985 to 2021
$50.0M
Joint Center for Mendelian GenomicsUM1HG008900 · NHGRI · BROAD INSTITUTE, INC. · PI O'DONNELL-LURIA, ANNE, REHM, HEIDI L · 2016 to 2020
$16.5M
Cell Identity Determination In Human Brain: Somatic Mutation and Cell LineageR01NS032457 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI Alice Eunjung Lee, Christopher A. Walsh · 1995 to 2026
$12.7M
HUMAN EPILEPSY GENETICS--NEURONAL MIGRATION DISORDERSR01NS035129 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI WALSH, CHRISTOPHER A. · 1997 to 2023
$9.5M
The Child Heath Research CenterK12HD052896 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI Wendy K Chung · 2007 to 2026
$7.9M
How Zika virus disrupts neuronal cellular architectureR21NS104633 · NINDS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI KODANI, ANDREW TADASHI, REITER, JEREMY F · 2018 to 2019
$479k
Howard Hughes Medical InstituteNHGRI NIH HHS UM1 HG008900NICHD NIH HHS K12 HD052896NIGMS NIH HHS T32 GM007753NINDS NIH HHS R01 NS032457NINDS NIH HHS R01 NS035129NINDS NIH HHS R21 NS104633
6 · The paper itself

Abstract

Genes mutated in human neuronal migration disorders encode tubulin proteins and a variety of tubulin-binding and -regulating proteins, but it is very poorly understood how these proteins function together to coordinate migration. Additionally, the way in which regional differences in neocortical migration are controlled is completely unknown. Here we describe a new syndrome with remarkably region-specific effects on neuronal migration in the posterior cortex, reflecting de novo variants in CEP85L. We show that CEP85L is required cell autonomously in vivo and in vitro for migration, that it localizes to the maternal centriole, and that it forms a complex with many other proteins required for migration, including CDK5, LIS1, NDE1, KIF2A, and DYNC1H1. Loss of CEP85L disrupts CDK5 localization and activation, leading to centrosome disorganization and disrupted microtubule cytoskeleton organization. Together, our findings suggest that CEP85L highlights a complex that controls CDK5 activity to promote neuronal migration.

Indexed as

Cell MovementCentriolesChildChild, PreschoolCyclin-Dependent Kinase 5Cytoskeletal ProteinsFemaleHumansLissencephalyMaleMicrotubulesNeocortexNerve Tissue ProteinsNeuronsOncogene Proteins, FusionYoung AdultCDK5 protein, humanCEP85L protein, humanCyclin-Dependent Kinase 5Cytoskeletal ProteinsNerve Tissue ProteinsOncogene Proteins, FusionCDK5CentrosomeCEP85LDe novogeneticsLissencephalyneurodevelopmentPachygyria

Identifiers

PMID32097629
PMCPMC7255387
OpenAlexW3007873711

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.