Abstract：
:Mouse models that replicate facets of human neurological diseases are often used at the pre-clinical stage to better understand the underlying mechanisms of a disease and test the target engagement of potential therapeutic interventions. We recently characterized a mouse model of childhood-onset parkinsonism-dystonia, a disease caused by a homozygous loss-of-function mutation in the SLC39A14 gene. The disease manifests itself phenotypically by impairments in locomotor behaviour and postural abnormalities. Our initial characterization of the model revealed that the Slc39a14-/- mice showed altered Mn homeostasis and compromised locomotor performance in vertical pole-descending, horizontal beam-traversing, and rotarod tests (Jenkitkasemwong et al., 2018). However, some of the mice also displayed torticollis and Straub tail. In this study, we investigated whether these postural abnormalities affected the performance in the above motility tests and consequently, biased and compromised the external validity of reported abnormal locomotor profiles. Our analyses showed that the Slc39a14-/- mice displaying torticollis and/or Straub tail had tests scores comparable to scores of their counterparts that never displayed these postural abnormalities. The z-score general index of performance revealed that the Slc39a14-/- model presents a complex pathological motor phenotype relevant to the complexity of phenotypes identified in childhood-onset parkinsonism-dystonia.
journal_name
Brain Res Bulljournal_title
Brain research bulletinauthors
Giraldo G,Janus Cdoi
10.1016/j.brainresbull.2020.10.018subject
Has Abstractpub_date
2021-01-01 00:00:00pages
54-63eissn
0361-9230issn
1873-2747pii
S0361-9230(20)30676-6journal_volume
166pub_type
杂志文章abstract：:The chronic mild stress (CMS) model has been used as an animal model of depression which induces anhedonic behavior in rodents. The present study was aimed to evaluate the behavioral and physiological effects of administration of beta-carboline harmine in rats exposed to CMS procedure. To this aim, after 40 days of ex...
journal_title：Brain research bulletin
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更新日期：1994-01-01 00:00:00
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pub_type： 杂志文章
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pub_type： 杂志文章
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journal_title：Brain research bulletin
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journal_title：Brain research bulletin
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journal_title：Brain research bulletin
pub_type： 杂志文章
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journal_title：Brain research bulletin
pub_type： 杂志文章,评审
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