Investigation of the pathogenesis of ADAR1 gene in dyschromatosis symmetrica hereditaria

The pathogenesis of dyschromatosis symmetrica hereditaria (DSH) has not been well defined. In this study, we sought to investigate the influence of the ADAR1 gene on DSH both in vitro and in vivo. Morpholino knockdown of adar1 in zebrafish produced phenotypes characterized by polarity changes, and a...

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Bibliographic Details
Published in:Experimental dermatology 2024-02, Vol.33 (2), p.e15031-n/a
Main Authors: Zhang, Jia, Jiang, Chengyi, Wang, Zhen, Fang, Biqing, He, Jin, Li, Ming
Format: Article
Language:English
Subjects:
ADAR1
ADAR1 gene
Adenosine Deaminase - genetics
Animals
Apoptosis
cell polarity
Danio rerio
dyschromatosis symmetrica hereditaria
Humans
Melanocytes
Mutation
Pathogenesis
Pedigree
Phenotypes
Pigmentation Disorders - congenital
RNA-Binding Proteins - genetics
zebrafish
Zebrafish - genetics
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Summary:The pathogenesis of dyschromatosis symmetrica hereditaria (DSH) has not been well defined. In this study, we sought to investigate the influence of the ADAR1 gene on DSH both in vitro and in vivo. Morpholino knockdown of adar1 in zebrafish produced phenotypes characterized by polarity changes, and abnormal migration and distribution of melanocytes. Differential expression of C‐KIT and distinct patterns of apoptosis between hyperpigmented and hypopigmented areas in DSH patient were detected by means of immunohistochemical methods and TUNEL assays, respectively. This study revealed that adar1 knockdown in a zebrafish model resulted in abnormal migration and changes in the cell polarity of melanocytes, and provided novel insight into the mechanism of DSH pathogenesis.
ISSN:0906-6705
1600-0625
DOI:10.1111/exd.15031