Τίτλος:
Behavioural effects of extracellular matrix protein Fras1 depletion in the mouse
Γλώσσες Τεκμηρίου:
Αγγλικά
Περίληψη:
Fras1 is an extracellular protein of the basement membranes that surround embryonic epithelia, choroid plexuses and meninges in foetal mouse brain. Depletion of Fras1 in knockout mice results in sub-epidermal blistering and fusion of eyelids and digits as well as malformation of lungs and kidneys. Mutations in the human counterpart FRAS1 are responsible for the Fraser Syndrome with clinical manifestations similar to the murine phenotype. In addition, brain deformities or mental impairments have occasionally been reported in patients with Fraser Syndrome. In the present study, we explored the possible involvement of Fras1 in brain function, analysing its expression pattern in mouse brain and investigating aspects of Fras1−/− mice behaviour, related to the function of brain regions expressing Fras1. Transcripts were detected in choroid plexuses and in certain brain regions including cortical, hippocampal and amygdalar areas in juvenile mice. Behavioural tests revealed that Fras1−/− mice exhibit impaired egocentric spatial memory, aberrant olfactory learning and memory, markedly reduced fear memory in an auditory fear conditioning task, as well as reduced anxiety expression in open field and elevated plus maze tests. Moreover, the extracellular matrix organization has been severely affected in cortical and subcortical areas as demonstrated by Wisteria floribunda agglutinin immunolabelling. The widespread detection of Fras1 transcripts in the brain of both pre- and postnatal mice, as well as the behavioural and cellular disturbances exhibited by Fras1−/− adult mice provide evidence for the involvement of Fras1 in brain organization and function. © 2020 Federation of European Neuroscience Societies and John Wiley & Sons Ltd.
Συγγραφείς:
Kalpachidou, T.
Makrygiannis, A.K.
Pavlakis, E.
Stylianopoulou, F.
Chalepakis, G.
Stamatakis, A.
Περιοδικό:
The European Journal of Neuroscience
Εκδότης:
John Wiley and Sons Inc
Λέξεις-κλειδιά:
agglutinin; Fras1; ketamine; poly(methyl methacrylate); scleroprotein; unclassified drug; xylazine; FRAS1 protein, human; scleroprotein, adult; animal experiment; animal model; animal tissue; antibody labeling; anxiety; Article; behavior; choroid plexus; cohort analysis; conditioning; egocentricity; elevated plus maze test; fear conditioning test; female; fetus; Fraser syndrome; histology; hot plate test; immunodetection; immunohistochemistry; immunolocalization; in situ hybridization; knockout mouse; Morris water maze test; mouse; mRNA expression level; nociception; nonhuman; novel object recognition test; olfactory discrimination; olfactory system; open field test; passive avoidance test; pH; protein depletion; protein expression level; rotarod test; social recognition test; spatial learning; spatial memory; wild type mouse; Wisteria; animal; basement membrane; epidermis; genetics; human; phenotype, Animals; Basement Membrane; Epidermis; Extracellular Matrix Proteins; Humans; Mice; Mice, Knockout; Phenotype
