Spata7 is required for maintenance of the retinal connecting cilium.


Journal

Scientific reports
ISSN: 2045-2322
Titre abrégé: Sci Rep
Pays: England
ID NLM: 101563288

Informations de publication

Date de publication:
02 04 2022
Historique:
received: 13 01 2022
accepted: 22 03 2022
entrez: 3 4 2022
pubmed: 4 4 2022
medline: 6 4 2022
Statut: epublish

Résumé

SPATA7, an early onset LCA3 retinal disease gene, encodes a putative scaffold protein that is essential for the proper assembly of the connecting cilium (CC) complex in photoreceptors. Previous studies have shown that SPATA7 interacts with other photoreceptor-specific ciliary proteins, such as RPGR and RPGRIP1, and maintains the integrity of CC integrity. However, although it is known that Spata7 is required for early formation of the CC, it is unclear if Spata7 is also required for the maintenance of the CC. To investigate Spata7 function in the retina at the adult stage, loss of function was induced in the adult retina upon tamoxifen induction of an inducible Spata7 knockout allele (Spata7

Identifiants

pubmed: 35368022
doi: 10.1038/s41598-022-09530-0
pii: 10.1038/s41598-022-09530-0
pmc: PMC8976851
doi:

Substances chimiques

DNA-Binding Proteins 0
SPATA7 protein, mouse 0
Rhodopsin 9009-81-8

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

5575

Subventions

Organisme : NEI NIH HHS
ID : R01 EY028970
Pays : United States
Organisme : NIH HHS
ID : R01EY020540
Pays : United States
Organisme : NIH HHS
ID : R01EY018571
Pays : United States
Organisme : NIH HHS
ID : R01EY022356
Pays : United States

Informations de copyright

© 2022. The Author(s).

Références

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Auteurs

Jiaxiong Lu (J)

Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX, USA.

Kaitlyn Xiong (K)

Department of Molecular and Human Genetics, Human Genome Sequencing Center, Baylor College of Medicine, Houston, TX, USA.

Xinye Qian (X)

Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX, USA.

Jongsu Choi (J)

Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX, USA.

Yoon-Kyung Shim (YK)

Department of Pathology, Baylor College of Medicine, Houston, TX, USA.

Jacob Burnett (J)

Department of Pathology, Baylor College of Medicine, Houston, TX, USA.

Graeme Mardon (G)

Department of Pathology, Baylor College of Medicine, Houston, TX, USA.

Rui Chen (R)

Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX, USA. ruichen@bcm.edu.
Department of Molecular and Human Genetics, Human Genome Sequencing Center, Baylor College of Medicine, Houston, TX, USA. ruichen@bcm.edu.

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Classifications MeSH