Identification of novel cerebellar developmental transcriptional regulators with motif activity analysis.


Journal

BMC genomics
ISSN: 1471-2164
Titre abrégé: BMC Genomics
Pays: England
ID NLM: 100965258

Informations de publication

Date de publication:
18 Sep 2019
Historique:
received: 02 10 2018
accepted: 26 08 2019
entrez: 20 9 2019
pubmed: 20 9 2019
medline: 28 1 2020
Statut: epublish

Résumé

The work of the FANTOM5 Consortium has brought forth a new level of understanding of the regulation of gene transcription and the cellular processes involved in creating diversity of cell types. In this study, we extended the analysis of the FANTOM5 Cap Analysis of Gene Expression (CAGE) transcriptome data to focus on understanding the genetic regulators involved in mouse cerebellar development. We used the HeliScopeCAGE library sequencing on cerebellar samples over 8 embryonic and 4 early postnatal times. This study showcases temporal expression pattern changes during cerebellar development. Through a bioinformatics analysis that focused on transcription factors, their promoters and binding sites, we identified genes that appear as strong candidates for involvement in cerebellar development. We selected several candidate transcriptional regulators for validation experiments including qRT-PCR and shRNA transcript knockdown. We observed marked and reproducible developmental defects in Atf4, Rfx3, and Scrt2 knockdown embryos, which support the role of these genes in cerebellar development. The successful identification of these novel gene regulators in cerebellar development demonstrates that the FANTOM5 cerebellum time series is a high-quality transcriptome database for functional investigation of gene regulatory networks in cerebellar development.

Sections du résumé

BACKGROUND BACKGROUND
The work of the FANTOM5 Consortium has brought forth a new level of understanding of the regulation of gene transcription and the cellular processes involved in creating diversity of cell types. In this study, we extended the analysis of the FANTOM5 Cap Analysis of Gene Expression (CAGE) transcriptome data to focus on understanding the genetic regulators involved in mouse cerebellar development.
RESULTS RESULTS
We used the HeliScopeCAGE library sequencing on cerebellar samples over 8 embryonic and 4 early postnatal times. This study showcases temporal expression pattern changes during cerebellar development. Through a bioinformatics analysis that focused on transcription factors, their promoters and binding sites, we identified genes that appear as strong candidates for involvement in cerebellar development. We selected several candidate transcriptional regulators for validation experiments including qRT-PCR and shRNA transcript knockdown. We observed marked and reproducible developmental defects in Atf4, Rfx3, and Scrt2 knockdown embryos, which support the role of these genes in cerebellar development.
CONCLUSIONS CONCLUSIONS
The successful identification of these novel gene regulators in cerebellar development demonstrates that the FANTOM5 cerebellum time series is a high-quality transcriptome database for functional investigation of gene regulatory networks in cerebellar development.

Identifiants

pubmed: 31533632
doi: 10.1186/s12864-019-6063-9
pii: 10.1186/s12864-019-6063-9
pmc: PMC6751898
doi:

Substances chimiques

Atf4 protein, mouse 0
Regulatory Factor X Transcription Factors 0
Rfx3 protein, mouse 0
SCRATCH2 protein, mouse 0
Transcription Factors 0
Activating Transcription Factor 4 145891-90-3

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

718

Subventions

Organisme : Genome Canada
ID : 174CDE

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Auteurs

Thomas J Ha (TJ)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.
Division of Neurology, Department of Pediatrics, University of British Columbia and BC Children's Hospital, Vancouver, BC, Canada.

Peter G Y Zhang (PGY)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.

Remi Robert (R)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.

Joanna Yeung (J)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.

Douglas J Swanson (DJ)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.

Anthony Mathelier (A)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.
Centre for Molecular Medicine Norway (NCMM), Nordic EMBL Partnership, University of Oslo, 0318, Oslo, Norway.
Department of Cancer Genetics, Institute for Cancer Research, Oslo University Hospital, Radiumhospitalet, 0372, Oslo, Norway.

Wyeth W Wasserman (WW)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.

Sujin Im (S)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada.

Masayoshi Itoh (M)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Center for Life Science Technologies, Yokohama, Japan.

Hideya Kawaji (H)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Center for Life Science Technologies, Yokohama, Japan.

Timo Lassmann (T)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Center for Life Science Technologies, Yokohama, Japan.
Telethon Kids Institute, The University of Western Australia, 100 Roberts Road, Subiaco, Subiaco, Western Australia, 6008, Australia.

Carsten O Daub (CO)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Center for Life Science Technologies, Yokohama, Japan.

Erik Arner (E)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Center for Life Science Technologies, Yokohama, Japan.

Piero Carninci (P)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Center for Life Science Technologies, Yokohama, Japan.

Yoshihide Hayashizaki (Y)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Preventive Medicine and Diagnosis Innovation Program, Wako, Japan.

Alistair R R Forrest (ARR)

RIKEN Omics Science Center (OSC), 1-7-22 Suehiro-cho, Tsurumi-ku, Yokohama, 230-0045, Japan.
RIKEN Center for Life Science Technologies, Yokohama, Japan.

Daniel Goldowitz (D)

Centre for Molecular Medicine and Therapeutics at the BC Children's Hospital Research Institute, Department of Medical Genetics, University of British Columbia, Vancouver, BC, Canada. dang@cmmt.ubc.ca.

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Classifications MeSH