Cryo-EM of retinoschisin branched networks suggests an intercellular adhesive scaffold in the retina
Identifieur interne : 000D89 ( Pmc/Curation ); précédent : 000D88; suivant : 000D90Cryo-EM of retinoschisin branched networks suggests an intercellular adhesive scaffold in the retina
Auteurs : J. Bernard Heymann ; Camasamudram Vijayasarathy ; Rick K. Huang ; Altaira D. Dearborn ; Paul A. Sieving ; Alasdair C. StevenSource :
- The Journal of Cell Biology [ 0021-9525 ] ; 2019.
Abstract
Mutations in the essential retinal protein retinoschisin (RS1) cause a form of macular degeneration. Heymann et al. use cryo-EM to show that RS1 assembles into branched networks that may play a stabilizing role in maintaining the integrity of the retina.
Url:
DOI: 10.1083/jcb.201806148
PubMed: 30630865
PubMed Central: 6400569
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Cryo-EM of retinoschisin branched networks suggests an intercellular adhesive scaffold in the retina</title><author><name sortKey="Heymann, J Bernard" sort="Heymann, J Bernard" uniqKey="Heymann J" first="J. Bernard" last="Heymann">J. Bernard Heymann</name><affiliation><nlm:aff id="aff1"><institution>Laboratory for Structural Biology Research, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Vijayasarathy, Camasamudram" sort="Vijayasarathy, Camasamudram" uniqKey="Vijayasarathy C" first="Camasamudram" last="Vijayasarathy">Camasamudram Vijayasarathy</name><affiliation><nlm:aff id="aff2"><institution>Section on Translational Research for Retinal and Macular Degeneration, National Institute on Deafness and Other Communication Disorders, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Huang, Rick K" sort="Huang, Rick K" uniqKey="Huang R" first="Rick K." last="Huang">Rick K. Huang</name><affiliation><nlm:aff id="aff3"><institution>Cryo-Electron Microscopy Facility, Janelia Research Campus, Howard Hughes Medical Institute, Ashburn, VA</institution></nlm:aff></affiliation></author><author><name sortKey="Dearborn, Altaira D" sort="Dearborn, Altaira D" uniqKey="Dearborn A" first="Altaira D." last="Dearborn">Altaira D. Dearborn</name><affiliation><nlm:aff id="aff1"><institution>Laboratory for Structural Biology Research, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Sieving, Paul A" sort="Sieving, Paul A" uniqKey="Sieving P" first="Paul A." last="Sieving">Paul A. Sieving</name><affiliation><nlm:aff id="aff2"><institution>Section on Translational Research for Retinal and Macular Degeneration, National Institute on Deafness and Other Communication Disorders, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation><affiliation><nlm:aff id="aff4"><institution>National Eye Institute, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Steven, Alasdair C" sort="Steven, Alasdair C" uniqKey="Steven A" first="Alasdair C." last="Steven">Alasdair C. Steven</name><affiliation><nlm:aff id="aff1"><institution>Laboratory for Structural Biology Research, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">30630865</idno><idno type="pmc">6400569</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6400569</idno><idno type="RBID">PMC:6400569</idno><idno type="doi">10.1083/jcb.201806148</idno><date when="2019">2019</date><idno type="wicri:Area/Pmc/Corpus">000D89</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000D89</idno><idno type="wicri:Area/Pmc/Curation">000D89</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">000D89</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Cryo-EM of retinoschisin branched networks suggests an intercellular adhesive scaffold in the retina</title><author><name sortKey="Heymann, J Bernard" sort="Heymann, J Bernard" uniqKey="Heymann J" first="J. Bernard" last="Heymann">J. Bernard Heymann</name><affiliation><nlm:aff id="aff1"><institution>Laboratory for Structural Biology Research, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Vijayasarathy, Camasamudram" sort="Vijayasarathy, Camasamudram" uniqKey="Vijayasarathy C" first="Camasamudram" last="Vijayasarathy">Camasamudram Vijayasarathy</name><affiliation><nlm:aff id="aff2"><institution>Section on Translational Research for Retinal and Macular Degeneration, National Institute on Deafness and Other Communication Disorders, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Huang, Rick K" sort="Huang, Rick K" uniqKey="Huang R" first="Rick K." last="Huang">Rick K. Huang</name><affiliation><nlm:aff id="aff3"><institution>Cryo-Electron Microscopy Facility, Janelia Research Campus, Howard Hughes Medical Institute, Ashburn, VA</institution></nlm:aff></affiliation></author><author><name sortKey="Dearborn, Altaira D" sort="Dearborn, Altaira D" uniqKey="Dearborn A" first="Altaira D." last="Dearborn">Altaira D. Dearborn</name><affiliation><nlm:aff id="aff1"><institution>Laboratory for Structural Biology Research, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Sieving, Paul A" sort="Sieving, Paul A" uniqKey="Sieving P" first="Paul A." last="Sieving">Paul A. Sieving</name><affiliation><nlm:aff id="aff2"><institution>Section on Translational Research for Retinal and Macular Degeneration, National Institute on Deafness and Other Communication Disorders, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation><affiliation><nlm:aff id="aff4"><institution>National Eye Institute, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author><author><name sortKey="Steven, Alasdair C" sort="Steven, Alasdair C" uniqKey="Steven A" first="Alasdair C." last="Steven">Alasdair C. Steven</name><affiliation><nlm:aff id="aff1"><institution>Laboratory for Structural Biology Research, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD</institution></nlm:aff></affiliation></author></analytic><series><title level="j">The Journal of Cell Biology</title><idno type="ISSN">0021-9525</idno><idno type="eISSN">1540-8140</idno><imprint><date when="2019">2019</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Mutations in the essential retinal protein retinoschisin (RS1) cause a form of macular degeneration. 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<front><journal-meta><journal-id journal-id-type="nlm-ta">J Cell Biol</journal-id><journal-id journal-id-type="iso-abbrev">J. Cell Biol</journal-id><journal-id journal-id-type="publisher-id">jcb</journal-id><journal-id journal-id-type="hwp">jcb</journal-id><journal-title-group><journal-title>The Journal of Cell Biology</journal-title></journal-title-group><issn pub-type="ppub">0021-9525</issn><issn pub-type="epub">1540-8140</issn><publisher><publisher-name>Rockefeller University Press</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">30630865</article-id><article-id pub-id-type="pmc">6400569</article-id><article-id pub-id-type="publisher-id">201806148</article-id><article-id pub-id-type="doi">10.1083/jcb.201806148</article-id><article-categories><subj-group subj-group-type="heading"><subject>Research Articles</subject><subj-group><subject>Article</subject></subj-group></subj-group><subj-group subj-group-type="hwp-journal-coll"><subject>1</subject><subject>20</subject><subject>46</subject></subj-group></article-categories><title-group><article-title>Cryo-EM of retinoschisin branched networks suggests an intercellular adhesive scaffold in the retina</article-title><alt-title alt-title-type="short">Retinoschisin forms an adhesive scaffold</alt-title></title-group><contrib-group><contrib contrib-type="author"><contrib-id contrib-id-type="orcid" authenticated="true">http://orcid.org/0000-0002-8872-5326</contrib-id><name><surname>Heymann</surname><given-names>J. Bernard</given-names></name><xref ref-type="aff" rid="aff1">1</xref><xref ref-type="corresp" rid="cor5"></xref></contrib><contrib contrib-type="author"><name><surname>Vijayasarathy</surname><given-names>Camasamudram</given-names></name><xref ref-type="aff" rid="aff2">2</xref></contrib><contrib contrib-type="author"><name><surname>Huang</surname><given-names>Rick K.</given-names></name><xref ref-type="aff" rid="aff3">3</xref></contrib><contrib contrib-type="author"><contrib-id contrib-id-type="orcid" authenticated="true">http://orcid.org/0000-0003-0463-5233</contrib-id><name><surname>Dearborn</surname><given-names>Altaira D.</given-names></name><xref ref-type="aff" rid="aff1">1</xref></contrib><contrib contrib-type="author"><name><surname>Sieving</surname><given-names>Paul A.</given-names></name><xref ref-type="aff" rid="aff2">2</xref><xref ref-type="aff" rid="aff4">4</xref></contrib><contrib contrib-type="author"><contrib-id contrib-id-type="orcid" authenticated="true">http://orcid.org/0000-0003-4104-6497</contrib-id><name><surname>Steven</surname><given-names>Alasdair C.</given-names></name><xref ref-type="aff" rid="aff1">1</xref></contrib></contrib-group><aff id="aff1"><label>1</label><institution>Laboratory for Structural Biology Research, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD</institution></aff><aff id="aff2"><label>2</label><institution>Section on Translational Research for Retinal and Macular Degeneration, National Institute on Deafness and Other Communication Disorders, National Institutes of Health, Bethesda, MD</institution></aff><aff id="aff3"><label>3</label><institution>Cryo-Electron Microscopy Facility, Janelia Research Campus, Howard Hughes Medical Institute, Ashburn, VA</institution></aff><aff id="aff4"><label>4</label><institution>National Eye Institute, National Institutes of Health, Bethesda, MD</institution></aff><author-notes><corresp id="cor5">Correspondence to J. Bernard Heymann: <email>heymannb@mail.nih.gov</email></corresp></author-notes><pub-date pub-type="ppub"><day>04</day><month>3</month><year>2019</year></pub-date><volume>218</volume><issue>3</issue><fpage>1027</fpage><lpage>1038</lpage><history><date date-type="received"><day>21</day><month>6</month><year>2018</year></date><date date-type="rev-recd"><day>06</day><month>11</month><year>2018</year></date><date date-type="accepted"><day>07</day><month>12</month><year>2018</year></date></history><permissions><copyright-statement>© 2019 Heymann et al.</copyright-statement><copyright-year>2019</copyright-year><ali:free_to_read start_date="2019-09-02"></ali:free_to_read><license><ali:license_ref specific-use="vor">http://www.rupress.org/terms/</ali:license_ref><ali:license_ref specific-use="vor" start_date="2019-09-02">https://creativecommons.org/licenses/by-nc-sa/4.0/</ali:license_ref><license-p>This article is distributed under the terms of an Attribution–Noncommercial–Share Alike–No Mirror Sites license for the first six months after the publication date (see <ext-link ext-link-type="uri" xlink:href="http://www.rupress.org/terms/">http://www.rupress.org/terms/</ext-link>). After six months it is available under a Creative Commons License (Attribution–Noncommercial–Share Alike 4.0 International license, as described at <ext-link ext-link-type="uri" xlink:href="https://creativecommons.org/licenses/by-nc-sa/4.0/">https://creativecommons.org/licenses/by-nc-sa/4.0/</ext-link>).</license-p></license></permissions><self-uri xlink:role="icon" xlink:href="JCB_201806148_thumb.gif"></self-uri><abstract abstract-type="precis"><p>Mutations in the essential retinal protein retinoschisin (RS1) cause a form of macular degeneration. Heymann et al. use cryo-EM to show that RS1 assembles into branched networks that may play a stabilizing role in maintaining the integrity of the retina.</p></abstract><abstract><p>Mutations in the retinal protein retinoschisin (RS1) cause progressive loss of vision in young males, a form of macular degeneration called X-linked retinoschisis (XLRS). We previously solved the structure of RS1, a 16-mer composed of paired back-to-back octameric rings. Here, we show by cryo–electron microscopy that RS1 16-mers can assemble into extensive branched networks. We classified the different configurations, finding four types of interaction between the RS1 molecules. The predominant configuration is a linear strand with a wavy appearance. Three less frequent types constitute the branch points of the network. In all cases, the “spikes” around the periphery of the double rings are involved in these interactions. In the linear strand, a loop (usually referred to as spike 1) occurs on both sides of the interface between neighboring molecules. Mutations in this loop suppress secretion, indicating the possibility of intracellular higher-order assembly. These observations suggest that branched networks of RS1 may play a stabilizing role in maintaining the integrity of the retina.</p></abstract><abstract abstract-type="graphical"><title>Graphical Abstract</title><p><fig id="GA" orientation="portrait" position="float"><graphic xlink:href="JCB_201806148_GA"></graphic></fig></p></abstract><funding-group><award-group id="sp1"><funding-source><institution-wrap><institution>National Institutes of Health</institution><institution-id institution-id-type="DOI">https://doi.org/10.13039/100000002,%22National%20Institutes%20of%20Health%22,</institution-id></institution-wrap></funding-source></award-group><award-group id="sp2"><funding-source><institution-wrap><institution>National Eye Institute</institution><institution-id institution-id-type="DOI">https://doi.org/10.13039/100000053,%22National%20Eye%20Institute%22,</institution-id></institution-wrap></funding-source></award-group><award-group id="sp3"><funding-source><institution-wrap><institution>National Institute on Deafness and Other Communication Disorders</institution><institution-id institution-id-type="DOI">https://doi.org/10.13039/100000055,%22National%20Institute%20on%20Deafness%20and%20Other%20Communication%20Disorders%22,</institution-id></institution-wrap></funding-source></award-group><award-group id="sp4"><funding-source>National Institute for Arthritis, Musculoskeletal and Skin Diseases</funding-source></award-group></funding-group></article-meta></front></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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