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MicroRNA 19a replacement partially rescues fin and cardiac defects in zebrafish model of Holt Oram syndrome
Holt-Oram Syndrome (HOS) is an autosomal dominant heart-hand syndrome caused by mutations in the TBX5 gene, a transcription factor capable of regulating hundreds of cardiac-specific genes through complex transcriptional networks. Here we show that, in zebrafish, modulation of a single miRNA is suffi...
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Published in: | Scientific reports 2015-12, Vol.5 (1), p.18240-18240, Article 18240 |
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creator | Chiavacci, Elena D'Aurizio, Romina Guzzolino, Elena Russo, Francesco Baumgart, Mario Groth, Marco Mariani, Laura D'Onofrio, Mara Arisi, Ivan Pellegrini, Marco Cellerino, Alessandro Cremisi, Federico Pitto, Letizia |
description | Holt-Oram Syndrome (HOS) is an autosomal dominant heart-hand syndrome caused by mutations in the TBX5 gene, a transcription factor capable of regulating hundreds of cardiac-specific genes through complex transcriptional networks. Here we show that, in zebrafish, modulation of a single miRNA is sufficient to rescue the morphogenetic defects generated by HOS. The analysis of miRNA-seq profiling revealed a decreased expression of miR-19a in Tbx5-depleted zebrafish embryos compared to the wild type. We revealed that the transcription of the miR-17-92 cluster, which harbors miR-19a, is induced by Tbx5 and that a defined dosage of miR-19a is essential for the correct development of the heart. Importantly, we highlighted that miR-19a replacement is able to rescue cardiac and pectoral fin defects and to increase the viability of HOS zebrafish embryos. We further observed that miR-19a replacement shifts the global gene expression profile of HOS-like zebrafish embryos towards the wild type condition, confirming the ability of miR-19a to rescue the Tbx5 phenotype. In conclusion our data demonstrate the importance of Tbx5/miR-19a regulatory circuit in heart development and provide a proof of principle that morphogenetic defects associated with HOS can be rescued by transient miRNA modulation. |
doi_str_mv | 10.1038/srep18240 |
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Here we show that, in zebrafish, modulation of a single miRNA is sufficient to rescue the morphogenetic defects generated by HOS. The analysis of miRNA-seq profiling revealed a decreased expression of miR-19a in Tbx5-depleted zebrafish embryos compared to the wild type. We revealed that the transcription of the miR-17-92 cluster, which harbors miR-19a, is induced by Tbx5 and that a defined dosage of miR-19a is essential for the correct development of the heart. Importantly, we highlighted that miR-19a replacement is able to rescue cardiac and pectoral fin defects and to increase the viability of HOS zebrafish embryos. We further observed that miR-19a replacement shifts the global gene expression profile of HOS-like zebrafish embryos towards the wild type condition, confirming the ability of miR-19a to rescue the Tbx5 phenotype. In conclusion our data demonstrate the importance of Tbx5/miR-19a regulatory circuit in heart development and provide a proof of principle that morphogenetic defects associated with HOS can be rescued by transient miRNA modulation.</description><identifier>ISSN: 2045-2322</identifier><identifier>EISSN: 2045-2322</identifier><identifier>DOI: 10.1038/srep18240</identifier><identifier>PMID: 26657204</identifier><language>eng</language><publisher>England: Nature Publishing Group</publisher><subject>Abnormalities, Multiple - genetics ; Abnormalities, Multiple - pathology ; Abnormalities, Multiple - therapy ; Animal Fins - embryology ; Animal Fins - pathology ; Animals ; Defects ; Embryonic Development - genetics ; Embryos ; Gene expression ; Gene Expression Regulation, Developmental ; Genetic Therapy - methods ; Genome-Wide Association Study ; Genomics ; Heart ; Heart Defects, Congenital - genetics ; Heart Defects, Congenital - pathology ; Heart Defects, Congenital - therapy ; Heart diseases ; Heart Septal Defects, Atrial - genetics ; Heart Septal Defects, Atrial - pathology ; Heart Septal Defects, Atrial - therapy ; Holt-Oram syndrome ; Lower Extremity Deformities, Congenital - genetics ; Lower Extremity Deformities, Congenital - pathology ; Lower Extremity Deformities, Congenital - therapy ; MicroRNAs - genetics ; miRNA ; Multigene Family ; Phenotype ; RNA Interference ; T-Box Domain Proteins - genetics ; TBX5 gene ; Upper Extremity Deformities, Congenital - genetics ; Upper Extremity Deformities, Congenital - pathology ; Upper Extremity Deformities, Congenital - therapy ; Zebrafish - embryology ; Zebrafish - genetics</subject><ispartof>Scientific reports, 2015-12, Vol.5 (1), p.18240-18240, Article 18240</ispartof><rights>Copyright Nature Publishing Group Dec 2015</rights><rights>Copyright © 2015, Macmillan Publishers Limited 2015 Macmillan Publishers Limited</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c403t-10280312529c1f03d4258b544c533e1cc555600468378ef3de465daf25e7013e3</citedby><cites>FETCH-LOGICAL-c403t-10280312529c1f03d4258b544c533e1cc555600468378ef3de465daf25e7013e3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.proquest.com/docview/1805462342/fulltextPDF?pq-origsite=primo$$EPDF$$P50$$Gproquest$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.proquest.com/docview/1805462342?pq-origsite=primo$$EHTML$$P50$$Gproquest$$Hfree_for_read</linktohtml><link.rule.ids>230,315,733,786,790,891,25783,27957,27958,37047,37048,44625,53827,53829,75483</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/26657204$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Chiavacci, Elena</creatorcontrib><creatorcontrib>D'Aurizio, Romina</creatorcontrib><creatorcontrib>Guzzolino, Elena</creatorcontrib><creatorcontrib>Russo, Francesco</creatorcontrib><creatorcontrib>Baumgart, Mario</creatorcontrib><creatorcontrib>Groth, Marco</creatorcontrib><creatorcontrib>Mariani, Laura</creatorcontrib><creatorcontrib>D'Onofrio, Mara</creatorcontrib><creatorcontrib>Arisi, Ivan</creatorcontrib><creatorcontrib>Pellegrini, Marco</creatorcontrib><creatorcontrib>Cellerino, Alessandro</creatorcontrib><creatorcontrib>Cremisi, Federico</creatorcontrib><creatorcontrib>Pitto, Letizia</creatorcontrib><title>MicroRNA 19a replacement partially rescues fin and cardiac defects in zebrafish model of Holt Oram syndrome</title><title>Scientific reports</title><addtitle>Sci Rep</addtitle><description>Holt-Oram Syndrome (HOS) is an autosomal dominant heart-hand syndrome caused by mutations in the TBX5 gene, a transcription factor capable of regulating hundreds of cardiac-specific genes through complex transcriptional networks. Here we show that, in zebrafish, modulation of a single miRNA is sufficient to rescue the morphogenetic defects generated by HOS. The analysis of miRNA-seq profiling revealed a decreased expression of miR-19a in Tbx5-depleted zebrafish embryos compared to the wild type. We revealed that the transcription of the miR-17-92 cluster, which harbors miR-19a, is induced by Tbx5 and that a defined dosage of miR-19a is essential for the correct development of the heart. Importantly, we highlighted that miR-19a replacement is able to rescue cardiac and pectoral fin defects and to increase the viability of HOS zebrafish embryos. We further observed that miR-19a replacement shifts the global gene expression profile of HOS-like zebrafish embryos towards the wild type condition, confirming the ability of miR-19a to rescue the Tbx5 phenotype. In conclusion our data demonstrate the importance of Tbx5/miR-19a regulatory circuit in heart development and provide a proof of principle that morphogenetic defects associated with HOS can be rescued by transient miRNA modulation.</description><subject>Abnormalities, Multiple - genetics</subject><subject>Abnormalities, Multiple - pathology</subject><subject>Abnormalities, Multiple - therapy</subject><subject>Animal Fins - embryology</subject><subject>Animal Fins - pathology</subject><subject>Animals</subject><subject>Defects</subject><subject>Embryonic Development - genetics</subject><subject>Embryos</subject><subject>Gene expression</subject><subject>Gene Expression Regulation, Developmental</subject><subject>Genetic Therapy - methods</subject><subject>Genome-Wide Association Study</subject><subject>Genomics</subject><subject>Heart</subject><subject>Heart Defects, Congenital - genetics</subject><subject>Heart Defects, Congenital - pathology</subject><subject>Heart Defects, Congenital - therapy</subject><subject>Heart diseases</subject><subject>Heart Septal Defects, Atrial - genetics</subject><subject>Heart Septal Defects, Atrial - pathology</subject><subject>Heart Septal Defects, Atrial - therapy</subject><subject>Holt-Oram syndrome</subject><subject>Lower Extremity Deformities, Congenital - genetics</subject><subject>Lower Extremity Deformities, Congenital - pathology</subject><subject>Lower Extremity Deformities, Congenital - therapy</subject><subject>MicroRNAs - genetics</subject><subject>miRNA</subject><subject>Multigene Family</subject><subject>Phenotype</subject><subject>RNA Interference</subject><subject>T-Box Domain Proteins - genetics</subject><subject>TBX5 gene</subject><subject>Upper Extremity Deformities, Congenital - genetics</subject><subject>Upper Extremity Deformities, Congenital - pathology</subject><subject>Upper Extremity Deformities, Congenital - therapy</subject><subject>Zebrafish - embryology</subject><subject>Zebrafish - genetics</subject><issn>2045-2322</issn><issn>2045-2322</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2015</creationdate><recordtype>article</recordtype><sourceid>PIMPY</sourceid><recordid>eNpdkV9LHDEUxUOxqFgf_AIl0Bf7sPXm38zsiyBiq2ArlPY5ZJMbNzYzWZNZYf30zbLrouYl4ebH4Zx7CDlh8I2B6M5KxgXruIQP5JCDVBMuON979T4gx6U8QD2KTyWb7pMD3jSqrcAh-fcz2Jx-_7qgbGpolYrGYo_DSBcmj8HEuKrTYpdYqA8DNYOj1mQXjKUOPdqx0Dp-xlk2PpQ57ZPDSJOn1ymO9C6bnpbV4HLq8RP56E0seLy9j8jf71d_Lq8nt3c_bi4vbidWghgnDHgHgvHq1jIPwkmuupmS0iohkFmrlGoAZNOJtkMvHMpGOeO5whaYQHFEzje6i-WsR2drmmyiXuTQm7zSyQT99mcIc32fnrRs2lYCVIHTrUBOjzX5qPtQLMZoBkzLolmrABhvFK_ol3foQ1rmocbTrAMlGy7kmvq6oequSy3M78ww0OsW9a7Fyn5-7X5HvnQm_gPLDJcL</recordid><startdate>20151214</startdate><enddate>20151214</enddate><creator>Chiavacci, Elena</creator><creator>D'Aurizio, Romina</creator><creator>Guzzolino, Elena</creator><creator>Russo, Francesco</creator><creator>Baumgart, Mario</creator><creator>Groth, Marco</creator><creator>Mariani, Laura</creator><creator>D'Onofrio, Mara</creator><creator>Arisi, Ivan</creator><creator>Pellegrini, Marco</creator><creator>Cellerino, Alessandro</creator><creator>Cremisi, Federico</creator><creator>Pitto, Letizia</creator><general>Nature Publishing Group</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88A</scope><scope>88E</scope><scope>88I</scope><scope>8FE</scope><scope>8FH</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BENPR</scope><scope>BHPHI</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>LK8</scope><scope>M0S</scope><scope>M1P</scope><scope>M2P</scope><scope>M7P</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>Q9U</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20151214</creationdate><title>MicroRNA 19a replacement partially rescues fin and cardiac defects in zebrafish model of Holt Oram syndrome</title><author>Chiavacci, Elena ; D'Aurizio, Romina ; Guzzolino, Elena ; Russo, Francesco ; Baumgart, Mario ; Groth, Marco ; Mariani, Laura ; D'Onofrio, Mara ; Arisi, Ivan ; Pellegrini, Marco ; Cellerino, Alessandro ; Cremisi, Federico ; Pitto, Letizia</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c403t-10280312529c1f03d4258b544c533e1cc555600468378ef3de465daf25e7013e3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2015</creationdate><topic>Abnormalities, Multiple - 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pathology</topic><topic>Lower Extremity Deformities, Congenital - therapy</topic><topic>MicroRNAs - genetics</topic><topic>miRNA</topic><topic>Multigene Family</topic><topic>Phenotype</topic><topic>RNA Interference</topic><topic>T-Box Domain Proteins - genetics</topic><topic>TBX5 gene</topic><topic>Upper Extremity Deformities, Congenital - genetics</topic><topic>Upper Extremity Deformities, Congenital - pathology</topic><topic>Upper Extremity Deformities, Congenital - therapy</topic><topic>Zebrafish - embryology</topic><topic>Zebrafish - genetics</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Chiavacci, Elena</creatorcontrib><creatorcontrib>D'Aurizio, Romina</creatorcontrib><creatorcontrib>Guzzolino, Elena</creatorcontrib><creatorcontrib>Russo, Francesco</creatorcontrib><creatorcontrib>Baumgart, Mario</creatorcontrib><creatorcontrib>Groth, Marco</creatorcontrib><creatorcontrib>Mariani, Laura</creatorcontrib><creatorcontrib>D'Onofrio, Mara</creatorcontrib><creatorcontrib>Arisi, Ivan</creatorcontrib><creatorcontrib>Pellegrini, Marco</creatorcontrib><creatorcontrib>Cellerino, Alessandro</creatorcontrib><creatorcontrib>Cremisi, Federico</creatorcontrib><creatorcontrib>Pitto, Letizia</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Biology Database (Alumni Edition)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Science Database (Alumni Edition)</collection><collection>ProQuest SciTech Collection</collection><collection>ProQuest Natural Science Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest Central</collection><collection>ProQuest Central Essentials</collection><collection>Biological Science Collection</collection><collection>AUTh Library subscriptions: ProQuest Central</collection><collection>Natural Science Collection</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>SciTech Premium Collection</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Biological Sciences</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>PML(ProQuest Medical Library)</collection><collection>Science Database</collection><collection>Biological Science Database</collection><collection>Publicly Available Content Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Scientific reports</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Chiavacci, Elena</au><au>D'Aurizio, Romina</au><au>Guzzolino, Elena</au><au>Russo, Francesco</au><au>Baumgart, Mario</au><au>Groth, Marco</au><au>Mariani, Laura</au><au>D'Onofrio, Mara</au><au>Arisi, Ivan</au><au>Pellegrini, Marco</au><au>Cellerino, Alessandro</au><au>Cremisi, Federico</au><au>Pitto, Letizia</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>MicroRNA 19a replacement partially rescues fin and cardiac defects in zebrafish model of Holt Oram syndrome</atitle><jtitle>Scientific reports</jtitle><addtitle>Sci Rep</addtitle><date>2015-12-14</date><risdate>2015</risdate><volume>5</volume><issue>1</issue><spage>18240</spage><epage>18240</epage><pages>18240-18240</pages><artnum>18240</artnum><issn>2045-2322</issn><eissn>2045-2322</eissn><notes>ObjectType-Article-1</notes><notes>SourceType-Scholarly Journals-1</notes><notes>ObjectType-Feature-2</notes><notes>content type line 23</notes><notes>Present address: Institute of Molecular Life Sciences University of Zurich, 8057, Zurich.</notes><abstract>Holt-Oram Syndrome (HOS) is an autosomal dominant heart-hand syndrome caused by mutations in the TBX5 gene, a transcription factor capable of regulating hundreds of cardiac-specific genes through complex transcriptional networks. Here we show that, in zebrafish, modulation of a single miRNA is sufficient to rescue the morphogenetic defects generated by HOS. The analysis of miRNA-seq profiling revealed a decreased expression of miR-19a in Tbx5-depleted zebrafish embryos compared to the wild type. We revealed that the transcription of the miR-17-92 cluster, which harbors miR-19a, is induced by Tbx5 and that a defined dosage of miR-19a is essential for the correct development of the heart. Importantly, we highlighted that miR-19a replacement is able to rescue cardiac and pectoral fin defects and to increase the viability of HOS zebrafish embryos. We further observed that miR-19a replacement shifts the global gene expression profile of HOS-like zebrafish embryos towards the wild type condition, confirming the ability of miR-19a to rescue the Tbx5 phenotype. In conclusion our data demonstrate the importance of Tbx5/miR-19a regulatory circuit in heart development and provide a proof of principle that morphogenetic defects associated with HOS can be rescued by transient miRNA modulation.</abstract><cop>England</cop><pub>Nature Publishing Group</pub><pmid>26657204</pmid><doi>10.1038/srep18240</doi><tpages>1</tpages><oa>free_for_read</oa></addata></record> |
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subjects | Abnormalities, Multiple - genetics Abnormalities, Multiple - pathology Abnormalities, Multiple - therapy Animal Fins - embryology Animal Fins - pathology Animals Defects Embryonic Development - genetics Embryos Gene expression Gene Expression Regulation, Developmental Genetic Therapy - methods Genome-Wide Association Study Genomics Heart Heart Defects, Congenital - genetics Heart Defects, Congenital - pathology Heart Defects, Congenital - therapy Heart diseases Heart Septal Defects, Atrial - genetics Heart Septal Defects, Atrial - pathology Heart Septal Defects, Atrial - therapy Holt-Oram syndrome Lower Extremity Deformities, Congenital - genetics Lower Extremity Deformities, Congenital - pathology Lower Extremity Deformities, Congenital - therapy MicroRNAs - genetics miRNA Multigene Family Phenotype RNA Interference T-Box Domain Proteins - genetics TBX5 gene Upper Extremity Deformities, Congenital - genetics Upper Extremity Deformities, Congenital - pathology Upper Extremity Deformities, Congenital - therapy Zebrafish - embryology Zebrafish - genetics |
title | MicroRNA 19a replacement partially rescues fin and cardiac defects in zebrafish model of Holt Oram syndrome |
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