Whole exome sequencing identification of novel candidate genes in patients with proliferative diabetic retinopathy
Rare or novel gene variants in patients with proliferative diabetic retinopathy may contribute to disease development. We performed whole exome sequencing (WES) on patients at the phenotypic extremes of diabetic retinal complications: 57 patients diagnosed with proliferative diabetic retinopathy (PD...
Gespeichert in:
Veröffentlicht in: | Vision research (Oxford) 2017-10, Vol.139, p.168-176 |
---|---|
Hauptverfasser: | , , , , , , , , , , , , , , , , |
Format: | Artikel |
Sprache: | eng |
Schlagworte: | |
Online-Zugang: | Volltext |
Tags: |
Tag hinzufügen
Keine Tags, Fügen Sie den ersten Tag hinzu!
|
container_end_page | 176 |
---|---|
container_issue | |
container_start_page | 168 |
container_title | Vision research (Oxford) |
container_volume | 139 |
creator | Ung, Cindy Sanchez, Angie V. Shen, Lishuang Davoudi, Samaneh Ahmadi, Tina Navarro-Gomez, Daniel Chen, Ching J. Hancock, Heather Penman, Alan Hoadley, Suzanne Consugar, Mark Restrepo, Carlos Shah, Vinay A. Arboleda-Velasquez, Joseph F. Sobrin, Lucia Gai, Xiaowu Kim, Leo A. |
description | Rare or novel gene variants in patients with proliferative diabetic retinopathy may contribute to disease development. We performed whole exome sequencing (WES) on patients at the phenotypic extremes of diabetic retinal complications: 57 patients diagnosed with proliferative diabetic retinopathy (PDR) as cases and 13 patients with no diabetic retinopathy despite at least 10years of type 2 diabetes as controls. Thirty-one out of the 57 cases and all 13 controls were from the African American Proliferative Diabetic Retinopathy Study (AA). The rest of the cases were of mixed ethnicities (ME). WES identified 721 candidate genes with rare or novel non-synonymous variants found in at least one case with PDR and not present in any controls. After filtering for genes with null alleles in greater than two cases, 28 candidate genes were identified in our ME cases and 16 genes were identified in our AA cases. Our analysis showed rare and novel variants within these genes that could contribute to the development of PDR, including rare non-synonymous variants in FAM132A, SLC5A9, ZNF600, and TMEM217. We also found previously unidentified variants in VEGFB and APOB. We found that VEGFB, VPS13B, PHF21A, NAT1, ZNF600, PKHD1L1 expression was reduced in human retinal endothelial cells (HRECs) cultured under high glucose conditions. In an exome sequence analysis of patients with PDR, we identified variants in genes that could contribute to pathogenesis. Six of these genes were further validated and found to have reduced expression in HRECs under high glucose conditions, suggestive of an important role in the development of PDR. |
doi_str_mv | 10.1016/j.visres.2017.03.007 |
format | Article |
fullrecord | <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_5912887</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><els_id>S0042698917300548</els_id><sourcerecordid>1891145958</sourcerecordid><originalsourceid>FETCH-LOGICAL-c463t-b6c997e00f6ae5aaf74282ba4928e5db4b1782f022fd433df5857be83488d8a73</originalsourceid><addsrcrecordid>eNp9kUFv1DAQhS0EokvhHyDkI5cE23ES54KEKmiRKvVC1aPl2OPdWWXtxc6m9N_j1ZYCl15syfPNm_F7hLznrOaMd5-29YI5Qa4F433Nmpqx_gVZcdWrqu1k95KsGJOi6gY1nJE3OW9ZIVoxvCZnQsmGq65fkXS3iRNQ-BV3QDP8PECwGNYUHYQZPVozYww0ehriAhO1Jjh0Zga6hgCZYqD7ghQ403ucN3Sf4oQeUnlcgDo0I8xoaSpniAXdPLwlr7yZMrx7vM_J7bevPy6uquuby-8XX64rK7tmrsbODkMPjPnOQGuM76VQYjRyEApaN8qR90p4JoR3smmcb1Xbj6AaqZRTpm_OyeeT7v4w7sDZsmMyk94n3Jn0oKNB_X8l4Eav46LbgQuljgIfHwVSLMbkWe8wW5gmEyAesuZq4Fy2Q6sKKk-oTTGXVPzTGM70MS691ae49DEuzRpdwihtH_5d8anpTz5__wDFqAUh6WyL2RYcJrCzdhGfn_AbX3StEw</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>1891145958</pqid></control><display><type>article</type><title>Whole exome sequencing identification of novel candidate genes in patients with proliferative diabetic retinopathy</title><source>Elsevier ScienceDirect Journals Complete</source><source>EZB-FREE-00999 freely available EZB journals</source><creator>Ung, Cindy ; Sanchez, Angie V. ; Shen, Lishuang ; Davoudi, Samaneh ; Ahmadi, Tina ; Navarro-Gomez, Daniel ; Chen, Ching J. ; Hancock, Heather ; Penman, Alan ; Hoadley, Suzanne ; Consugar, Mark ; Restrepo, Carlos ; Shah, Vinay A. ; Arboleda-Velasquez, Joseph F. ; Sobrin, Lucia ; Gai, Xiaowu ; Kim, Leo A.</creator><creatorcontrib>Ung, Cindy ; Sanchez, Angie V. ; Shen, Lishuang ; Davoudi, Samaneh ; Ahmadi, Tina ; Navarro-Gomez, Daniel ; Chen, Ching J. ; Hancock, Heather ; Penman, Alan ; Hoadley, Suzanne ; Consugar, Mark ; Restrepo, Carlos ; Shah, Vinay A. ; Arboleda-Velasquez, Joseph F. ; Sobrin, Lucia ; Gai, Xiaowu ; Kim, Leo A.</creatorcontrib><description>Rare or novel gene variants in patients with proliferative diabetic retinopathy may contribute to disease development. We performed whole exome sequencing (WES) on patients at the phenotypic extremes of diabetic retinal complications: 57 patients diagnosed with proliferative diabetic retinopathy (PDR) as cases and 13 patients with no diabetic retinopathy despite at least 10years of type 2 diabetes as controls. Thirty-one out of the 57 cases and all 13 controls were from the African American Proliferative Diabetic Retinopathy Study (AA). The rest of the cases were of mixed ethnicities (ME). WES identified 721 candidate genes with rare or novel non-synonymous variants found in at least one case with PDR and not present in any controls. After filtering for genes with null alleles in greater than two cases, 28 candidate genes were identified in our ME cases and 16 genes were identified in our AA cases. Our analysis showed rare and novel variants within these genes that could contribute to the development of PDR, including rare non-synonymous variants in FAM132A, SLC5A9, ZNF600, and TMEM217. We also found previously unidentified variants in VEGFB and APOB. We found that VEGFB, VPS13B, PHF21A, NAT1, ZNF600, PKHD1L1 expression was reduced in human retinal endothelial cells (HRECs) cultured under high glucose conditions. In an exome sequence analysis of patients with PDR, we identified variants in genes that could contribute to pathogenesis. Six of these genes were further validated and found to have reduced expression in HRECs under high glucose conditions, suggestive of an important role in the development of PDR.</description><identifier>ISSN: 0042-6989</identifier><identifier>EISSN: 1878-5646</identifier><identifier>DOI: 10.1016/j.visres.2017.03.007</identifier><identifier>PMID: 28431867</identifier><language>eng</language><publisher>England: Elsevier Ltd</publisher><subject>Aberrant angiogenesis ; Candidate genes ; Diabetic retinopathy ; Whole exome sequencing</subject><ispartof>Vision research (Oxford), 2017-10, Vol.139, p.168-176</ispartof><rights>2017 Elsevier Ltd</rights><rights>Copyright © 2017 Elsevier Ltd. All rights reserved.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c463t-b6c997e00f6ae5aaf74282ba4928e5db4b1782f022fd433df5857be83488d8a73</citedby><cites>FETCH-LOGICAL-c463t-b6c997e00f6ae5aaf74282ba4928e5db4b1782f022fd433df5857be83488d8a73</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://dx.doi.org/10.1016/j.visres.2017.03.007$$EHTML$$P50$$Gelsevier$$Hfree_for_read</linktohtml><link.rule.ids>230,314,780,784,885,3550,27924,27925,45995</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/28431867$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Ung, Cindy</creatorcontrib><creatorcontrib>Sanchez, Angie V.</creatorcontrib><creatorcontrib>Shen, Lishuang</creatorcontrib><creatorcontrib>Davoudi, Samaneh</creatorcontrib><creatorcontrib>Ahmadi, Tina</creatorcontrib><creatorcontrib>Navarro-Gomez, Daniel</creatorcontrib><creatorcontrib>Chen, Ching J.</creatorcontrib><creatorcontrib>Hancock, Heather</creatorcontrib><creatorcontrib>Penman, Alan</creatorcontrib><creatorcontrib>Hoadley, Suzanne</creatorcontrib><creatorcontrib>Consugar, Mark</creatorcontrib><creatorcontrib>Restrepo, Carlos</creatorcontrib><creatorcontrib>Shah, Vinay A.</creatorcontrib><creatorcontrib>Arboleda-Velasquez, Joseph F.</creatorcontrib><creatorcontrib>Sobrin, Lucia</creatorcontrib><creatorcontrib>Gai, Xiaowu</creatorcontrib><creatorcontrib>Kim, Leo A.</creatorcontrib><title>Whole exome sequencing identification of novel candidate genes in patients with proliferative diabetic retinopathy</title><title>Vision research (Oxford)</title><addtitle>Vision Res</addtitle><description>Rare or novel gene variants in patients with proliferative diabetic retinopathy may contribute to disease development. We performed whole exome sequencing (WES) on patients at the phenotypic extremes of diabetic retinal complications: 57 patients diagnosed with proliferative diabetic retinopathy (PDR) as cases and 13 patients with no diabetic retinopathy despite at least 10years of type 2 diabetes as controls. Thirty-one out of the 57 cases and all 13 controls were from the African American Proliferative Diabetic Retinopathy Study (AA). The rest of the cases were of mixed ethnicities (ME). WES identified 721 candidate genes with rare or novel non-synonymous variants found in at least one case with PDR and not present in any controls. After filtering for genes with null alleles in greater than two cases, 28 candidate genes were identified in our ME cases and 16 genes were identified in our AA cases. Our analysis showed rare and novel variants within these genes that could contribute to the development of PDR, including rare non-synonymous variants in FAM132A, SLC5A9, ZNF600, and TMEM217. We also found previously unidentified variants in VEGFB and APOB. We found that VEGFB, VPS13B, PHF21A, NAT1, ZNF600, PKHD1L1 expression was reduced in human retinal endothelial cells (HRECs) cultured under high glucose conditions. In an exome sequence analysis of patients with PDR, we identified variants in genes that could contribute to pathogenesis. Six of these genes were further validated and found to have reduced expression in HRECs under high glucose conditions, suggestive of an important role in the development of PDR.</description><subject>Aberrant angiogenesis</subject><subject>Candidate genes</subject><subject>Diabetic retinopathy</subject><subject>Whole exome sequencing</subject><issn>0042-6989</issn><issn>1878-5646</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><recordid>eNp9kUFv1DAQhS0EokvhHyDkI5cE23ES54KEKmiRKvVC1aPl2OPdWWXtxc6m9N_j1ZYCl15syfPNm_F7hLznrOaMd5-29YI5Qa4F433Nmpqx_gVZcdWrqu1k95KsGJOi6gY1nJE3OW9ZIVoxvCZnQsmGq65fkXS3iRNQ-BV3QDP8PECwGNYUHYQZPVozYww0ehriAhO1Jjh0Zga6hgCZYqD7ghQ403ucN3Sf4oQeUnlcgDo0I8xoaSpniAXdPLwlr7yZMrx7vM_J7bevPy6uquuby-8XX64rK7tmrsbODkMPjPnOQGuM76VQYjRyEApaN8qR90p4JoR3smmcb1Xbj6AaqZRTpm_OyeeT7v4w7sDZsmMyk94n3Jn0oKNB_X8l4Eav46LbgQuljgIfHwVSLMbkWe8wW5gmEyAesuZq4Fy2Q6sKKk-oTTGXVPzTGM70MS691ae49DEuzRpdwihtH_5d8anpTz5__wDFqAUh6WyL2RYcJrCzdhGfn_AbX3StEw</recordid><startdate>20171001</startdate><enddate>20171001</enddate><creator>Ung, Cindy</creator><creator>Sanchez, Angie V.</creator><creator>Shen, Lishuang</creator><creator>Davoudi, Samaneh</creator><creator>Ahmadi, Tina</creator><creator>Navarro-Gomez, Daniel</creator><creator>Chen, Ching J.</creator><creator>Hancock, Heather</creator><creator>Penman, Alan</creator><creator>Hoadley, Suzanne</creator><creator>Consugar, Mark</creator><creator>Restrepo, Carlos</creator><creator>Shah, Vinay A.</creator><creator>Arboleda-Velasquez, Joseph F.</creator><creator>Sobrin, Lucia</creator><creator>Gai, Xiaowu</creator><creator>Kim, Leo A.</creator><general>Elsevier Ltd</general><scope>6I.</scope><scope>AAFTH</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20171001</creationdate><title>Whole exome sequencing identification of novel candidate genes in patients with proliferative diabetic retinopathy</title><author>Ung, Cindy ; Sanchez, Angie V. ; Shen, Lishuang ; Davoudi, Samaneh ; Ahmadi, Tina ; Navarro-Gomez, Daniel ; Chen, Ching J. ; Hancock, Heather ; Penman, Alan ; Hoadley, Suzanne ; Consugar, Mark ; Restrepo, Carlos ; Shah, Vinay A. ; Arboleda-Velasquez, Joseph F. ; Sobrin, Lucia ; Gai, Xiaowu ; Kim, Leo A.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c463t-b6c997e00f6ae5aaf74282ba4928e5db4b1782f022fd433df5857be83488d8a73</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>Aberrant angiogenesis</topic><topic>Candidate genes</topic><topic>Diabetic retinopathy</topic><topic>Whole exome sequencing</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Ung, Cindy</creatorcontrib><creatorcontrib>Sanchez, Angie V.</creatorcontrib><creatorcontrib>Shen, Lishuang</creatorcontrib><creatorcontrib>Davoudi, Samaneh</creatorcontrib><creatorcontrib>Ahmadi, Tina</creatorcontrib><creatorcontrib>Navarro-Gomez, Daniel</creatorcontrib><creatorcontrib>Chen, Ching J.</creatorcontrib><creatorcontrib>Hancock, Heather</creatorcontrib><creatorcontrib>Penman, Alan</creatorcontrib><creatorcontrib>Hoadley, Suzanne</creatorcontrib><creatorcontrib>Consugar, Mark</creatorcontrib><creatorcontrib>Restrepo, Carlos</creatorcontrib><creatorcontrib>Shah, Vinay A.</creatorcontrib><creatorcontrib>Arboleda-Velasquez, Joseph F.</creatorcontrib><creatorcontrib>Sobrin, Lucia</creatorcontrib><creatorcontrib>Gai, Xiaowu</creatorcontrib><creatorcontrib>Kim, Leo A.</creatorcontrib><collection>ScienceDirect Open Access Titles</collection><collection>Elsevier:ScienceDirect:Open Access</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Vision research (Oxford)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Ung, Cindy</au><au>Sanchez, Angie V.</au><au>Shen, Lishuang</au><au>Davoudi, Samaneh</au><au>Ahmadi, Tina</au><au>Navarro-Gomez, Daniel</au><au>Chen, Ching J.</au><au>Hancock, Heather</au><au>Penman, Alan</au><au>Hoadley, Suzanne</au><au>Consugar, Mark</au><au>Restrepo, Carlos</au><au>Shah, Vinay A.</au><au>Arboleda-Velasquez, Joseph F.</au><au>Sobrin, Lucia</au><au>Gai, Xiaowu</au><au>Kim, Leo A.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Whole exome sequencing identification of novel candidate genes in patients with proliferative diabetic retinopathy</atitle><jtitle>Vision research (Oxford)</jtitle><addtitle>Vision Res</addtitle><date>2017-10-01</date><risdate>2017</risdate><volume>139</volume><spage>168</spage><epage>176</epage><pages>168-176</pages><issn>0042-6989</issn><eissn>1878-5646</eissn><abstract>Rare or novel gene variants in patients with proliferative diabetic retinopathy may contribute to disease development. We performed whole exome sequencing (WES) on patients at the phenotypic extremes of diabetic retinal complications: 57 patients diagnosed with proliferative diabetic retinopathy (PDR) as cases and 13 patients with no diabetic retinopathy despite at least 10years of type 2 diabetes as controls. Thirty-one out of the 57 cases and all 13 controls were from the African American Proliferative Diabetic Retinopathy Study (AA). The rest of the cases were of mixed ethnicities (ME). WES identified 721 candidate genes with rare or novel non-synonymous variants found in at least one case with PDR and not present in any controls. After filtering for genes with null alleles in greater than two cases, 28 candidate genes were identified in our ME cases and 16 genes were identified in our AA cases. Our analysis showed rare and novel variants within these genes that could contribute to the development of PDR, including rare non-synonymous variants in FAM132A, SLC5A9, ZNF600, and TMEM217. We also found previously unidentified variants in VEGFB and APOB. We found that VEGFB, VPS13B, PHF21A, NAT1, ZNF600, PKHD1L1 expression was reduced in human retinal endothelial cells (HRECs) cultured under high glucose conditions. In an exome sequence analysis of patients with PDR, we identified variants in genes that could contribute to pathogenesis. Six of these genes were further validated and found to have reduced expression in HRECs under high glucose conditions, suggestive of an important role in the development of PDR.</abstract><cop>England</cop><pub>Elsevier Ltd</pub><pmid>28431867</pmid><doi>10.1016/j.visres.2017.03.007</doi><tpages>9</tpages><oa>free_for_read</oa></addata></record> |
fulltext | fulltext |
identifier | ISSN: 0042-6989 |
ispartof | Vision research (Oxford), 2017-10, Vol.139, p.168-176 |
issn | 0042-6989 1878-5646 |
language | eng |
recordid | cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_5912887 |
source | Elsevier ScienceDirect Journals Complete; EZB-FREE-00999 freely available EZB journals |
subjects | Aberrant angiogenesis Candidate genes Diabetic retinopathy Whole exome sequencing |
title | Whole exome sequencing identification of novel candidate genes in patients with proliferative diabetic retinopathy |
url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-12-28T12%3A51%3A19IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_pubme&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Whole%20exome%20sequencing%20identification%20of%20novel%20candidate%20genes%20in%20patients%20with%20proliferative%20diabetic%20retinopathy&rft.jtitle=Vision%20research%20(Oxford)&rft.au=Ung,%20Cindy&rft.date=2017-10-01&rft.volume=139&rft.spage=168&rft.epage=176&rft.pages=168-176&rft.issn=0042-6989&rft.eissn=1878-5646&rft_id=info:doi/10.1016/j.visres.2017.03.007&rft_dat=%3Cproquest_pubme%3E1891145958%3C/proquest_pubme%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=1891145958&rft_id=info:pmid/28431867&rft_els_id=S0042698917300548&rfr_iscdi=true |