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Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome

22q11.2 deletion syndrome (22q11.2DS) is associated with a high risk of developing various psychiatric and developmental disorders, including schizophrenia and early-onset Parkinson’s disease. Recently, a mouse model of this disease, Del(3.0Mb)/+, mimicking the 3.0 Mb deletion which is most frequent...

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Autores principales: Tabata, Hidenori, Mori, Daisuke, Matsuki, Tohru, Yoshizaki, Kaichi, Asai, Masato, Nakayama, Atsuo, Ozaki, Norio, Nagata, Koh-ichi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10216591/
https://www.ncbi.nlm.nih.gov/pubmed/37238632
http://dx.doi.org/10.3390/biom13050763
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author Tabata, Hidenori
Mori, Daisuke
Matsuki, Tohru
Yoshizaki, Kaichi
Asai, Masato
Nakayama, Atsuo
Ozaki, Norio
Nagata, Koh-ichi
author_facet Tabata, Hidenori
Mori, Daisuke
Matsuki, Tohru
Yoshizaki, Kaichi
Asai, Masato
Nakayama, Atsuo
Ozaki, Norio
Nagata, Koh-ichi
author_sort Tabata, Hidenori
collection PubMed
description 22q11.2 deletion syndrome (22q11.2DS) is associated with a high risk of developing various psychiatric and developmental disorders, including schizophrenia and early-onset Parkinson’s disease. Recently, a mouse model of this disease, Del(3.0Mb)/+, mimicking the 3.0 Mb deletion which is most frequently found in patients with 22q11.2DS, was generated. The behavior of this mouse model was extensively studied and several abnormalities related to the symptoms of 22q11.2DS were found. However, the histological features of their brains have been little addressed. Here we describe the cytoarchitectures of the brains of Del(3.0Mb)/+ mice. First, we investigated the overall histology of the embryonic and adult cerebral cortices, but they were indistinguishable from the wild type. However, the morphologies of individual neurons were slightly but significantly changed from the wild type counterparts in a region-specific manner. The dendritic branches and/or dendritic spine densities of neurons in the medial prefrontal cortex, nucleus accumbens, and primary somatosensory cortex were reduced. We also observed reduced axon innervation of dopaminergic neurons into the prefrontal cortex. Given these affected neurons function together as the dopamine system to control animal behaviors, the impairment we observed may explain a part of the abnormal behaviors of Del(3.0Mb)/+ mice and the psychiatric symptoms of 22q11.2DS.
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spelling pubmed-102165912023-05-27 Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome Tabata, Hidenori Mori, Daisuke Matsuki, Tohru Yoshizaki, Kaichi Asai, Masato Nakayama, Atsuo Ozaki, Norio Nagata, Koh-ichi Biomolecules Article 22q11.2 deletion syndrome (22q11.2DS) is associated with a high risk of developing various psychiatric and developmental disorders, including schizophrenia and early-onset Parkinson’s disease. Recently, a mouse model of this disease, Del(3.0Mb)/+, mimicking the 3.0 Mb deletion which is most frequently found in patients with 22q11.2DS, was generated. The behavior of this mouse model was extensively studied and several abnormalities related to the symptoms of 22q11.2DS were found. However, the histological features of their brains have been little addressed. Here we describe the cytoarchitectures of the brains of Del(3.0Mb)/+ mice. First, we investigated the overall histology of the embryonic and adult cerebral cortices, but they were indistinguishable from the wild type. However, the morphologies of individual neurons were slightly but significantly changed from the wild type counterparts in a region-specific manner. The dendritic branches and/or dendritic spine densities of neurons in the medial prefrontal cortex, nucleus accumbens, and primary somatosensory cortex were reduced. We also observed reduced axon innervation of dopaminergic neurons into the prefrontal cortex. Given these affected neurons function together as the dopamine system to control animal behaviors, the impairment we observed may explain a part of the abnormal behaviors of Del(3.0Mb)/+ mice and the psychiatric symptoms of 22q11.2DS. MDPI 2023-04-27 /pmc/articles/PMC10216591/ /pubmed/37238632 http://dx.doi.org/10.3390/biom13050763 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Tabata, Hidenori
Mori, Daisuke
Matsuki, Tohru
Yoshizaki, Kaichi
Asai, Masato
Nakayama, Atsuo
Ozaki, Norio
Nagata, Koh-ichi
Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome
title Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome
title_full Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome
title_fullStr Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome
title_full_unstemmed Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome
title_short Histological Analysis of a Mouse Model of the 22q11.2 Microdeletion Syndrome
title_sort histological analysis of a mouse model of the 22q11.2 microdeletion syndrome
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10216591/
https://www.ncbi.nlm.nih.gov/pubmed/37238632
http://dx.doi.org/10.3390/biom13050763
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