Cited by 163Congenital heart defects researchCardiac electrophysiology and arrhythmiasCardiac Fibrosis and Remodeling

Genetic fate mapping demonstrates contribution of epicardium-derived cells to the annulus fibrosis of the mammalian heart

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Zhou, Bin, von Gise, Alexander, Ma, Qing, Hu, Yong Wu, Pu, William T.·2010·Developmental Biology

Abstract

The annulus fibrosis electrically insulates the atria and ventricles, allowing the timed sequential beating of these structures that is necessary for efficient heart function. Abnormal development of the annulus fibrosis leads to persistence of accessory electrical pathways from atria to ventricles, providing the anatomical substrate for re-entrant cardiac arrhythmias such as Wolff-Parkinson-White syndrome. To better understand the development of the annulus fibrosis and the etiology of these cardiac arrhythmias, we used Cre-LoxP technology to assess the contribution of epicardium derived cells (EPDCs) to the annulus fibrosis. We found that EPDCs migrated into the region of the forming annulus fibrosis, marked by the protein periostin. These EPDCs also stained positive for procollagen I, suggesting that the EPDCs themselves synthesize proteins of the annulus fibrosis. To further test the hypothesis that EPDCs contribute to cells that synthesize the annulus fibrosis, we purified genetically marked EPDCs from the atrioventricular region and measured gene expression by quantitative PCR. These EPDCs were highly enriched for mRNAs encoding periostin, procollagen I, fibronectin I, vimentin, discoidin domain receptor 2, and tenascin C, markers of fibroblasts and components of the annulus fibrosis. In addition, these EPDCs were highly enriched for Snail, Smad1, Slug, and Twist1, markers for epithelial-to-mesenchymal transition (EMT), and a metalloprotease, Mmp2, that contributes to cellular migration. Our work provides for the first time definitive evidence that epicardium contributes to formation of the mammalian annulus fibrosis through EMT. Abnormalities of this differentiation process may underlie development of some forms of re-entrant atrioventricular tachycardia.

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Crossref · 10.1016/j.ydbio.2009.12.007

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  • AV mesenchymal cells are also generated from the epicardium via EpiMT starting around E12.5.

    “AV mesenchymal cells are also generated from the epicardium via the epicardial-to-mesenchymal transition (EpiMT) starting around E12.5 (Fig. 1a).”

    From Cell diversity and plasticity during atrioventricular heart valve EMTs (2023) · cites this paper as [23]

    The Results report epicardium-derived cells at the atrioventricular junction with mesenchymal markers and evidence of epicardial-to-mesenchymal transition. The cited paper does not establish that this process starts around E12.5; its reported observations here are at E14.5 and later (page numbers unavailable in our copy). (LLM, conf=0.86)

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  • Via EpiMT, epicardial cells contribute to smooth muscle cells, vasculature, pericytes, and fibroblasts, including those of the AVC and annulus fibrosus.

    “Via EpiMT, epicardial cells contribute to smooth muscle cells, vasculature, pericytes, and fibroblasts, including those of the atrioventricular canal (AVC) and the annulus fibrosus5,6,23,24.”

    From Cell diversity and plasticity during atrioventricular heart valve EMTs (2023) · cites this paper as [23]

    The Introduction describes epicardial cells undergoing EMT and their descendants differentiating into smooth muscle, endothelial, and fibroblast lineages. The Results show epicardium-derived cells becoming fibroblasts at the atrioventricular junction and contributing to the annulus fibrosus. The copy does not identify pericytes as a lineage to which they contribute. Page numbers are unavailable in the indexed passages. (LLM, conf=0.85)

    Experimental — accuracy not yet measured

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Research works printed but not linked: 27

  1. BECKER AE ANDERSON RH DURRER D WELLENS HJ 1978 The anatomical substrates of wolff-parkinson-white syndrome. A clinicopathologic correlation in seven patients Circulation 57 870 879 639209
  2. BUCKINGHAM M MEILHAC S ZAFFRAN S 2005 Building the mammalian heart from two sources of myocardial cells Nat Rev Genet 6 826 835 16304598
  3. CAI CL MARTIN JC SUN Y CUI L WANG L OUYANG K YANG L BU L LIANG X ZHANG X STALLCUP WB DENTON CP MCCULLOCH A CHEN J EVANS SM 2008 A myocardial lineage derives from Tbx18 epicardial cells Nature 454 104 108 18480752 PMC5540369
  4. FEIL R WAGNER J METZGER D CHAMBON P 1997 Regulation of Cre recombinase activity by mutated estrogen receptor ligand-binding domains Biochem Biophys Res Commun 237 752 757 9299439
  5. GITTENBERGER-DE GROOT AC VRANCKEN PEETERS MP MENTINK MM GOURDIE RG POELMANN RE 1998 Epicardium-derived cells contribute a novel population to the myocardial wall and the atrioventricular cushions Circ Res 82 1043 1052 9622157
  6. KERN CB HOFFMAN S MORENO R DAMON BJ NORRIS RA KRUG EL MARKWALD RR MJAATVEDT CH 2005 Immunolocalization of chick periostin protein in the developing heart Anat Rec A Discov Mol Cell Evol Biol 284 415 423 15803479
  7. KOLDITZ DP WIJFFELS MC BLOM NA VAN DER LAARSE A HAHURIJ ND LIE-VENEMA H MARKWALD RR POELMANN RE SCHALIJ MJ GITTENBERGER-DE GROOT AC 2008 Epicardium-derived cells in development of annulus fibrosis and persistence of accessory pathways Circulation 117 1508 1517 18332266
  8. KOLDITZ DP WIJFFELS MC BLOM NA VAN DER LAARSE A MARKWALD RR SCHALIJ MJ GITTENBERGER-DE GROOT AC 2007 Persistence of functional atrioventricular accessory pathways in postseptated embryonic avian hearts: implications for morphogenesis and functional maturation of the cardiac conduction system Circulation 115 17 26 17190860
  9. KRUZYNSKA-FREJTAG A MACHNICKI M ROGERS R MARKWALD RR CONWAY SJ 2001 Periostin (an osteoblast-specific factor) is expressed within the embryonic mouse heart during valve formation Mech Dev 103 183 188 11335131
  10. LAVINE KJ YU K WHITE AC ZHANG X SMITH C PARTANEN J ORNITZ DM 2005 Endocardial and epicardial derived FGF signals regulate myocardial proliferation and differentiation in vivo Dev Cell 8 85 95 15621532
  11. LI WE WALDO K LINASK KL CHEN T WESSELS A PARMACEK MS KIRBY ML LO CW 2002 An essential role for connexin43 gap junctions in mouse coronary artery development Development 129 2031 2042 11934868
  12. LIE-VENEMA H ERALP I MARKWALD RR VAN DEN AKKER NM WIJFFELS MC KOLDITZ DP VAN DER LAARSE A SCHALIJ MJ POELMANN RE BOGERS AJ GITTENBERGER-DE GROOT AC 2008 Periostin expression by epicardium-derived cells is involved in the development of the atrioventricular valves and fibrous heart skeleton Differentiation 76 809 819 18294225
  13. MA Q ZHOU B PU WT 2008 Reassessment of Isl1 and Nkx2-5 cardiac fate maps using a Gata4-based reporter of Cre activity Dev Biol 323 98 104 18775691 PMC2655699
  14. MANI SA GUO W LIAO MJ EATON EN AYYANAN A ZHOU AY BROOKS M REINHARD F ZHANG CC SHIPITSIN M CAMPBELL LL POLYAK K BRISKEN C YANG J WEINBERG RA 2008 The epithelial-mesenchymal transition generates cells with properties of stem cells Cell 133 704 715 18485877 PMC2728032
  15. MERKI E ZAMORA M RAYA A KAWAKAMI Y WANG J ZHANG X BURCH J KUBALAK SW KALIMAN P BELMONTE JC CHIEN KR RUIZ-LOZANO P 2005 Epicardial retinoid X receptor alpha is required for myocardial growth and coronary artery formation Proc Natl Acad Sci U S A 102 18455 18460 16352730 PMC1317903
  16. MUZUMDAR MD TASIC B MIYAMICHI K LI L LUO L 2007 A global double-fluorescent Cre reporter mouse Genesis 45 593 605 17868096
  17. NORRIS RA MORENO-RODRIGUEZ RA SUGI Y HOFFMAN S AMOS J HART MM POTTS JD GOODWIN RL MARKWALD RR 2008 Periostin regulates atrioventricular valve maturation Dev Biol 316 200 213 18313657 PMC2386672
  18. PEREZ-POMARES JM PHELPS A SEDMEROVA M CARMONA R GONZALEZ-IRIARTE M MUNOZ-CHAPULI R WESSELS A 2002 Experimental studies on the spatiotemporal expression of WT1 and RALDH2 in the embryonic avian heart: a model for the regulation of myocardial and valvuloseptal development by epicardially derived cells (EPDCs) Dev Biol 247 307 326 12086469
  19. SNIDER P HINTON RB MORENO-RODRIGUEZ RA WANG J ROGERS R LINDSLEY A LI F INGRAM DA MENICK D FIELD L FIRULLI AB MOLKENTIN JD MARKWALD R CONWAY SJ 2008 Periostin is required for maturation and extracellular matrix stabilization of noncardiomyocyte lineages of the heart Circ Res 102 752 760 18296617 PMC2754697
  20. SORIANO P 1999 Generalized lacZ expression with the ROSA26 Cre reporter strain Nat Genet 21 70 71 9916792
  21. VOOIJS M JONKERS J BERNS A 2001 A highly efficient ligand-regulated Cre recombinase mouse line shows that LoxP recombination is position dependent EMBO Rep 2 292 297 11306549 PMC1083861
  22. WILM B IPENBERG A HASTIE ND BURCH JB BADER DM 2005 The serosal mesothelium is a major source of smooth muscle cells of the gut vasculature Development 132 5317 5328 16284122
  23. WU H LEE SH GAO J LIU X IRUELA-ARISPE ML 1999 Inactivation of erythropoietin leads to defects in cardiac morphogenesis Development 126 3597 3605 10409505
  24. ZAMORA M MANNER J RUIZ-LOZANO P 2007 Epicardium-derived progenitor cells require beta-catenin for coronary artery formation Proc Natl Acad Sci U S A 104 18109 18114 17989236 PMC2084304
  25. ZHOU B MA Q KONG SW HU Y CAMPBELL PH MCGOWAN FX ACKERMAN KG WU B ZHOU B TEVOSIAN SG PU WT 2009 Fog2 is critical for cardiac function and maintenance of coronary vasculature in the adult mouse heart J Clin Invest 119 1462 1476 19411759 PMC2689123
  26. ZHOU B MA Q RAJAGOPAL S WU SM DOMIAN I RIVERA-FELICIANO J JIANG D VON GISE A IKEDA S CHIEN KR PU WT 2008a Epicardial progenitors contribute to the cardiomyocyte lineage in the developing heart Nature 454 109 113 18568026 PMC2574791
  27. ZHOU B VON GISE A MA Q RIVERA-FELICIANO J PU WT 2008b Nkx2-5- and Isl1-expressing cardiac progenitors contribute to proepicardium Biochem Biophys Res Commun 375 450 453 18722343 PMC2610421

Cited by

Sorted by citations

Authors
Zhou, B., von Gise, A., Ma, Q., Hu, Y. W., & Pu, W. T.
Affiliations
Boston Children's Hospital (US)
Corresponding author
  • Zhou, Bin
  • Pu, William T.
Venue
Developmental Biology
Publisher
Elsevier BV
Published
2010-02
Volume
338
Issue
2
Pages
251-261
ISSN
0012-1606 (ISSN-L)
OpenAlex
W2024971066
Cited by
163
References
0
Citations by year
2026: 4 · 2025: 3 · 2024: 8 · 2023: 6 · 2022: 5 · 2021: 6 · 2020: 12 · 2019: 10 · 2018: 15 · 2017: 8 · 2016: 13 · 2015: 12 · 2014: 10 · 2013: 13 · 2012: 17
FWCI
5.11
Type
journal article
Language
en
Open access
Locations
  • Developmental Biology (publishedVersion)
  • PubMed (publishedVersion)
  • PubMed Central (acceptedVersion)
Funders
NIH (1R01HL094683)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Congenital heart defects research
  • Health Sciences › Medicine › Cardiology and Cardiovascular Medicine › Cardiac electrophysiology and arrhythmias
  • Health Sciences › Medicine › Cardiology and Cardiovascular Medicine › Cardiac Fibrosis and Remodeling
Keywords
epicardium, epicardial-derived cells, atrioventricular junction, periostin, epithelial-mesenchymal transition, cardiac fibroblasts, type I procollagen, cardiac arrhythmias, Wolff-Parkinson-White syndrome, atrioventricular reentrant tachycardia, heart development, cell migration, MMP-2
MeSH terms
Animals, Arrhythmias, Cardiac, Cell Movement, Fibrosis, Heart Atria, Heart Conduction System, Heart Ventricles, Myocardium, Pericardium, RNA, Messenger, Gene Expression Profiling, Mice, Cardiac Electrophysiology

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