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author:

Chen, Wenfeng (Chen, Wenfeng.) [1] (Scholars:陈文锋) | Zhong, Wenmiao (Zhong, Wenmiao.) [2] | Yu, Lingqi (Yu, Lingqi.) [3] | Lin, Xiang (Lin, Xiang.) [4] (Scholars:林翔) | Xie, Jiayu (Xie, Jiayu.) [5] | Liu, Zhenxing (Liu, Zhenxing.) [6]

Indexed by:

Scopus SCIE

Abstract:

Congenital stationary night blindness (CSNB) is a genetically heterogeneous inherited retinal disorder, caused by over 300 mutations in 17 different genes. While there are numerous fly models available for simulating ocular diseases, most are focused on mimicking retinitis pigmentosa (RP), with animal models specifically addressing CSNB limited to mammals. Here, we present a CSNB fly model associated with the mtt gene, utilizing RNA interference (RNAi) to silence the mtt gene in fly eyes (homologous to the mammalian GRM6 gene) and construct a CSNB model. Through this approach, we observed significant defects in the eye structure and function upon reducing mtt expression in fly eyes. This manifested as disruptions in the compound eye lens structure and reduced sensitivity to light responses. These results suggest a critical role for mtt in the function of fly adult eyes. Interestingly, we found that the mtt gene is not expressed in the photoreceptor neurons of adult flies but is localized to the inner lamina neurons. In summary, these results underscore the crucial involvement of mtt in fly retinal function, providing a framework for understanding the pathogenic mechanisms of CSNB and facilitating research into potential therapeutic interventions.

Keyword:

CSNB Drosophila eye GRM6 mtt retina

Community:

  • [ 1 ] [Chen, Wenfeng]Fuzhou Univ, Inst Life Sci, Coll Biol Sci & Engn, Fuzhou 350108, Peoples R China
  • [ 2 ] [Zhong, Wenmiao]Fuzhou Univ, Inst Life Sci, Coll Biol Sci & Engn, Fuzhou 350108, Peoples R China
  • [ 3 ] [Yu, Lingqi]Fuzhou Univ, Inst Life Sci, Coll Biol Sci & Engn, Fuzhou 350108, Peoples R China
  • [ 4 ] [Lin, Xiang]Fuzhou Univ, Inst Life Sci, Coll Biol Sci & Engn, Fuzhou 350108, Peoples R China
  • [ 5 ] [Xie, Jiayu]Chongqing Univ, Sch Med, Chongqing 400044, Peoples R China
  • [ 6 ] [Xie, Jiayu]Chinese Acad Agr Sci, Inst Apicultural Res, State Key Lab Resource Insects, Beijing 100093, Peoples R China
  • [ 7 ] [Liu, Zhenxing]Chinese Acad Agr Sci, Inst Apicultural Res, State Key Lab Resource Insects, Beijing 100093, Peoples R China

Reprint 's Address:

  • [Chen, Wenfeng]Fuzhou Univ, Inst Life Sci, Coll Biol Sci & Engn, Fuzhou 350108, Peoples R China;;[Liu, Zhenxing]Chinese Acad Agr Sci, Inst Apicultural Res, State Key Lab Resource Insects, Beijing 100093, Peoples R China;;

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Source :

INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

ISSN: 1661-6596

Year: 2024

Issue: 2

Volume: 25

4 . 9 0 0

JCR@2023

CAS Journal Grade:3

Cited Count:

WoS CC Cited Count: 1

SCOPUS Cited Count:

ESI Highly Cited Papers on the List: 0 Unfold All

WanFang Cited Count:

Chinese Cited Count:

30 Days PV: 0

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