Literature DB >> 26893377

The B3 Subunit of the Cone Cyclic Nucleotide-gated Channel Regulates the Light Responses of Cones and Contributes to the Channel Structural Flexibility.

Xi-Qin Ding1, Arjun Thapa2, Hongwei Ma2, Jianhua Xu2, Michael H Elliott3, Karla K Rodgers4, Marci L Smith5, Jin-Shan Wang6, Steven J Pittler5, Vladimir J Kefalov6.   

Abstract

Cone photoreceptor cyclic nucleotide-gated (CNG) channels play a pivotal role in cone phototransduction, which is a process essential for daylight vision, color vision, and visual acuity. Mutations in the cone channel subunits CNGA3 and CNGB3 are associated with human cone diseases, including achromatopsia, cone dystrophies, and early onset macular degeneration. Mutations in CNGB3 alone account for 50% of reported cases of achromatopsia. This work investigated the role of CNGB3 in cone light response and cone channel structural stability. As cones comprise only 2-3% of the total photoreceptor population in the wild-type mouse retina, we used Cngb3(-/-)/Nrl(-/-) mice with CNGB3 deficiency on a cone-dominant background in our study. We found that, in the absence of CNGB3, CNGA3 was able to travel to the outer segments, co-localize with cone opsin, and form tetrameric complexes. Electroretinogram analyses revealed reduced cone light response amplitude/sensitivity and slower response recovery in Cngb3(-/-)/Nrl(-/-) mice compared with Nrl(-/-) mice. Absence of CNGB3 expression altered the adaptation capacity of cones and severely compromised function in bright light. Biochemical analysis demonstrated that CNGA3 channels lacking CNGB3 were more resilient to proteolysis than CNGA3/CNGB3 channels, suggesting a hindered structural flexibility. Thus, CNGB3 regulates cone light response kinetics and the channel structural flexibility. This work advances our understanding of the biochemical and functional role of CNGB3 in cone photoreceptors.
© 2016 by The American Society for Biochemistry and Molecular Biology, Inc.

Entities:  

Keywords:  achromatopsia; cng channel; cone; ion channel; photoreceptor; phototransduction; retina; vision

Mesh:

Substances:

Year:  2016        PMID: 26893377      PMCID: PMC4861441          DOI: 10.1074/jbc.M115.696138

Source DB:  PubMed          Journal:  J Biol Chem        ISSN: 0021-9258            Impact factor:   5.157


  66 in total

1.  Mechanism of calcium/calmodulin inhibition of rod cyclic nucleotide-gated channels.

Authors:  Matthew C Trudeau; William N Zagotta
Journal:  Proc Natl Acad Sci U S A       Date:  2002-06-04       Impact factor: 11.205

2.  Chromophore supply rate-limits mammalian photoreceptor dark adaptation.

Authors:  Jin-shan Wang; Soile Nymark; Rikard Frederiksen; Maureen E Estevez; Susan Q Shen; Joseph C Corbo; M Carter Cornwall; Vladimir J Kefalov
Journal:  J Neurosci       Date:  2014-08-20       Impact factor: 6.167

3.  Folding and subunit assembly of photoreceptor peripherin/rds is mediated by determinants within the extracellular/intradiskal EC2 domain: implications for heterogeneous molecular pathologies.

Authors:  A F Goldberg; L M Fales; J B Hurley; N Khattree
Journal:  J Biol Chem       Date:  2001-09-11       Impact factor: 5.157

4.  Nrl is required for rod photoreceptor development.

Authors:  A J Mears; M Kondo; P K Swain; Y Takada; R A Bush; T L Saunders; P A Sieving; A Swaroop
Journal:  Nat Genet       Date:  2001-12       Impact factor: 38.330

5.  Mutations in the CNGB3 gene encoding the beta-subunit of the cone photoreceptor cGMP-gated channel are responsible for achromatopsia (ACHM3) linked to chromosome 8q21.

Authors:  S Kohl; B Baumann; M Broghammer; H Jägle; P Sieving; U Kellner; R Spegal; M Anastasi; E Zrenner; L T Sharpe; B Wissinger
Journal:  Hum Mol Genet       Date:  2000-09-01       Impact factor: 6.150

6.  Primate photopic sine-wave flicker ERG: vector modeling analysis of component origins using glutamate analogs.

Authors:  M Kondo; P A Sieving
Journal:  Invest Ophthalmol Vis Sci       Date:  2001-01       Impact factor: 4.799

7.  cGMP/Protein Kinase G Signaling Suppresses Inositol 1,4,5-Trisphosphate Receptor Phosphorylation and Promotes Endoplasmic Reticulum Stress in Photoreceptors of Cyclic Nucleotide-gated Channel-deficient Mice.

Authors:  Hongwei Ma; Michael R Butler; Arjun Thapa; Josh Belcher; Fan Yang; Wolfgang Baehr; Martin Biel; Stylianos Michalakis; Xi-Qin Ding
Journal:  J Biol Chem       Date:  2015-06-29       Impact factor: 5.157

8.  Molecular cloning and functional characterization of a new modulatory cyclic nucleotide-gated channel subunit from mouse retina.

Authors:  A Gerstner; X Zong; F Hofmann; M Biel
Journal:  J Neurosci       Date:  2000-02-15       Impact factor: 6.167

9.  CNGA3 mutations in hereditary cone photoreceptor disorders.

Authors:  B Wissinger; D Gamer; H Jägle; R Giorda; T Marx; S Mayer; S Tippmann; M Broghammer; B Jurklies; T Rosenberg; S G Jacobson; E C Sener; S Tatlipinar; C B Hoyng; C Castellan; P Bitoun; S Andreasson; G Rudolph; U Kellner; B Lorenz; G Wolff; C Verellen-Dumoulin; M Schwartz; F P Cremers; E Apfelstedt-Sylla; E Zrenner; R Salati; L T Sharpe; S Kohl
Journal:  Am J Hum Genet       Date:  2001-08-30       Impact factor: 11.025

Review 10.  Cyclic nucleotide-gated ion channels.

Authors:  U Benjamin Kaupp; Reinhard Seifert
Journal:  Physiol Rev       Date:  2002-07       Impact factor: 37.312

View more
  1 in total

1.  Delineating the Molecular and Phenotypic Spectrum of the CNGA3-Related Cone Photoreceptor Disorder in Pakistani Families.

Authors:  Sairah Yousaf; Nabeela Tariq; Zureesha Sajid; Shakeel A Sheikh; Tasleem Kausar; Yar M Waryah; Rehan S Shaikh; Ali M Waryah; Saumil Sethna; Saima Riazuddin; Zubair M Ahmed
Journal:  Genes (Basel)       Date:  2022-03-29       Impact factor: 4.141

  1 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.