---
_id: '1107'
abstract:
- lang: eng
text: The generation, migration, and differentiation of neurons requires the functional
integrity of the microtubule cytoskeleton. Mutations in the tubulin gene family
are known to cause various neurological diseases including lissencephaly, ocular
motor disorders, polymicrogyria and amyotrophic lateral sclerosis. We have previously
reported that mutations in TUBB5 cause microcephaly that is accompanied by severe
intellectual impairment and motor delay. Here we present the characterization
of a Tubb5 mouse model that allows for the conditional expression of the pathogenic
E401K mutation. Homozygous knockin animals exhibit a severe reduction in brain
size and in body weight. These animals do not show any significant impairment
in general activity, anxiety, or in the acoustic startle response, however, present
with notable defects in motor coordination. When assessed on the static rod apparatus
mice took longer to orient and often lost their balance completely. Interestingly,
mutant animals also showed defects in prepulse inhibition, a phenotype associated
with sensorimotor gating and considered an endophenotype for schizophrenia. This
study provides insight into the behavioral consequences of tubulin gene mutations.
acknowledgement: Austrian Science Fund (FWF) for funding this research [I914,P21092]
article_processing_charge: No
author:
- first_name: Martin
full_name: Breuss, Martin
last_name: Breuss
- first_name: Andi H
full_name: Hansen, Andi H
id: 38853E16-F248-11E8-B48F-1D18A9856A87
last_name: Hansen
- first_name: Lukas
full_name: Landler, Lukas
last_name: Landler
- first_name: David
full_name: Keays, David
last_name: Keays
citation:
ama: Breuss M, Hansen AH, Landler L, Keays D. Brain specific knockin of the pathogenic
Tubb5 E401K allele causes defects in motor coordination and prepulse inhibition.
Behavioural Brain Research. 2017;323:47-55. doi:10.1016/j.bbr.2017.01.029
apa: Breuss, M., Hansen, A. H., Landler, L., & Keays, D. (2017). Brain specific
knockin of the pathogenic Tubb5 E401K allele causes defects in motor coordination
and prepulse inhibition. Behavioural Brain Research. Elsevier. https://doi.org/10.1016/j.bbr.2017.01.029
chicago: Breuss, Martin, Andi H Hansen, Lukas Landler, and David Keays. “Brain Specific
Knockin of the Pathogenic Tubb5 E401K Allele Causes Defects in Motor Coordination
and Prepulse Inhibition.” Behavioural Brain Research. Elsevier, 2017. https://doi.org/10.1016/j.bbr.2017.01.029.
ieee: M. Breuss, A. H. Hansen, L. Landler, and D. Keays, “Brain specific knockin
of the pathogenic Tubb5 E401K allele causes defects in motor coordination and
prepulse inhibition,” Behavioural Brain Research, vol. 323. Elsevier, pp.
47–55, 2017.
ista: Breuss M, Hansen AH, Landler L, Keays D. 2017. Brain specific knockin of the
pathogenic Tubb5 E401K allele causes defects in motor coordination and prepulse
inhibition. Behavioural Brain Research. 323, 47–55.
mla: Breuss, Martin, et al. “Brain Specific Knockin of the Pathogenic Tubb5 E401K
Allele Causes Defects in Motor Coordination and Prepulse Inhibition.” Behavioural
Brain Research, vol. 323, Elsevier, 2017, pp. 47–55, doi:10.1016/j.bbr.2017.01.029.
short: M. Breuss, A.H. Hansen, L. Landler, D. Keays, Behavioural Brain Research
323 (2017) 47–55.
date_created: 2018-12-11T11:50:11Z
date_published: 2017-04-14T00:00:00Z
date_updated: 2023-09-20T11:37:25Z
day: '14'
ddc:
- '570'
- '571'
doi: 10.1016/j.bbr.2017.01.029
extern: '1'
external_id:
isi:
- '000397369100007'
file:
- access_level: open_access
content_type: application/pdf
creator: system
date_created: 2018-12-12T10:12:03Z
date_updated: 2018-12-12T10:12:03Z
file_id: '4921'
file_name: IST-2017-868-v1+1_1-s2.0-S0166432816309160-main.pdf
file_size: 2291511
relation: main_file
file_date_updated: 2018-12-12T10:12:03Z
has_accepted_license: '1'
intvolume: ' 323'
isi: 1
language:
- iso: eng
month: '04'
oa: 1
oa_version: Published Version
page: 47 - 55
publication: Behavioural Brain Research
publication_identifier:
issn:
- '01664328'
publication_status: published
publisher: Elsevier
publist_id: '6262'
pubrep_id: '868'
quality_controlled: '1'
status: public
title: Brain specific knockin of the pathogenic Tubb5 E401K allele causes defects
in motor coordination and prepulse inhibition
tmp:
image: /images/cc_by_nc_nd.png
legal_code_url: https://creativecommons.org/licenses/by-nc-nd/4.0/legalcode
name: Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International
(CC BY-NC-ND 4.0)
short: CC BY-NC-ND (4.0)
type: journal_article
user_id: c635000d-4b10-11ee-a964-aac5a93f6ac1
volume: 323
year: '2017'
...