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1000 Titel
  • MicroRNA 19a replacement partially rescues fin and cardiac defects in zebrafish model of Holt Oram syndrome
1000 Autor/in
  1. Chiavacci, Elena |
  2. D’Aurizio, Romina |
  3. Guzzolino, Elena |
  4. Russo, Francesco |
  5. Baumgart, Mario |
  6. Groth, Marco |
  7. Mariani, Laura |
  8. D’Onofrio, Mara |
  9. Arisi, Ivan |
  10. Pellegrini, Marco |
  11. Cellerino, Alessandro |
  12. Cremisi, Federico |
  13. Pitto, Letizia |
1000 Erscheinungsjahr 2015
1000 Art der Datei
1000 Publikationstyp
  1. Artikel |
1000 Online veröffentlicht
  • 2015-12-14
1000 Erschienen in
1000 Quellenangabe
  • 5:18240 (2015)
1000 FRL-Sammlung
1000 Copyrightjahr
  • 2015
1000 Verlagsversion
  • http://dx.doi.org/10.1038/srep18240 |
  • https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4677400/ |
1000 Publikationsstatus
1000 Begutachtungsstatus
1000 Sprache der Publikation
1000 Abstract/Summary
  • 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.
1000 Sacherschließung
lokal RNAi
lokal Disease model
1000 Fachgruppe
  1. Biologie |
1000 Fächerklassifikation (DDC)
1000 Liste der Beteiligten
  1. https://frl.publisso.de/adhoc/creator/Q2hpYXZhY2NpLCBFbGVuYQ==|https://frl.publisso.de/adhoc/creator/ROKAmUF1cml6aW8sIFJvbWluYQ==|https://frl.publisso.de/adhoc/creator/R3V6em9saW5vLCBFbGVuYQ==|https://frl.publisso.de/adhoc/creator/UnVzc28sIEZyYW5jZXNjbw==|http://orcid.org/0000-0001-8172-1595|http://orcid.org/0000-0002-9199-8990|https://frl.publisso.de/adhoc/creator/TWFyaWFuaSwgTGF1cmE=|https://frl.publisso.de/adhoc/creator/ROKAmU9ub2ZyaW8sIE1hcmE=|https://frl.publisso.de/adhoc/creator/QXJpc2ksIEl2YW4=|https://frl.publisso.de/adhoc/creator/UGVsbGVncmluaSwgTWFyY28=|http://orcid.org/0000-0003-3834-0097|https://frl.publisso.de/adhoc/creator/Q3JlbWlzaSwgRmVkZXJpY28=|https://frl.publisso.de/adhoc/creator/UGl0dG8sIExldGl6aWE=
1000 Förderer
  1. Italian Ministry for Instruction University and Research (MIUR) |
  2. CNR organizations |
  3. IIT-IFC Laboratory of Integrative Systems Medicine (LISM) |
1000 Fördernummer
  1. -
  2. -
  3. -
1000 Förderprogramm
  1. Flagship project InterOmics (PB. P05)
  2. Flagship project InterOmics (PB. P05)
  3. -
1000 Dateien
1000 Förderung
  1. 1000 joinedFunding-child
    1000 Förderer Italian Ministry for Instruction University and Research (MIUR) |
    1000 Förderprogramm Flagship project InterOmics (PB. P05)
    1000 Fördernummer -
  2. 1000 joinedFunding-child
    1000 Förderer CNR organizations |
    1000 Förderprogramm Flagship project InterOmics (PB. P05)
    1000 Fördernummer -
  3. 1000 joinedFunding-child
    1000 Förderer IIT-IFC Laboratory of Integrative Systems Medicine (LISM) |
    1000 Förderprogramm -
    1000 Fördernummer -
1000 Objektart article
1000 Beschrieben durch
1000 @id frl:6404540.rdf
1000 Erstellt am 2017-09-21T13:31:03.810+0200
1000 Erstellt von 122
1000 beschreibt frl:6404540
1000 Bearbeitet von 218
1000 Zuletzt bearbeitet Mon May 28 11:29:59 CEST 2018
1000 Objekt bearb. Mon May 28 11:29:58 CEST 2018
1000 Vgl. frl:6404540
1000 Oai Id
  1. oai:frl.publisso.de:frl:6404540 |
1000 Sichtbarkeit Metadaten public
1000 Sichtbarkeit Daten public
1000 Gegenstand von

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