Mutations affecting the formation and function of the cardiovascular system in the zebrafish embryo

Didier Y R Stainier, Bernadette Fouquet, Jau Nian Chen, Kerri S. Warren, Brant M. Weinstein, Steffen E Meiler, Manzoor Ali P K Mohideen, Stephan C F Neuhauss, Liliana Solnica-Krezel, Alexander F. Schier, Fried Zwartkruis, Derek L. Stemple, Jarema Malicki, Wolfgang Driever, Mark C. Fishman

Research output: Contribution to journalArticle

429 Citations (Scopus)

Abstract

As part of a large-scale mutagenesis screen of the zebrafish genome, we have identified 58 mutations that affect the formation and function of the cardiovascular system. The cardiovascular system is particularly amenable for screening in the transparent zebrafish embryo because the heart and blood vessels are prominent and their function easily examined. We have classified the mutations affecting the heart into those that affect primarily either morphogenesis or function. Nine mutations clearly disrupt the formation of the heart. cloche deletes the endocardium. In cloche mutants, the myocardial layer forms in the absence of the endocardium but is dysmorphic and exhibits a weak contractility. Two loci, miles apart and bonnie and clyde, play a critical role in the fusion of the bilateral tubular primordia. Three mutations lead to an abnormally large heart and one to the formation of a diminutive, dysmorphic heart. We have found no mutation that deletes the myocardial cells altogether, but one, pandora, appears to eliminate the ventricle selectively. Seven mutations interfere with vascular integrity, as indicated by hemorrhage at particular sites. In terms of cardiac function, one large group exhibits a weak beat. In this group, five loci affect both chambers and seven a specific chamber (the atrium or ventricle). For example, the weak atrium mutation exhibits an atrium that becomes silent but has a normally beating ventricle. Seven mutations affect the rhythm of the heart causing, for example, a slow rate, a fibrillating pattern or an apparent block to conduction. In several other mutants, regurgitation of blood flow from ventricle to atrium is the most prominent abnormality, due either to the absence of valves or to poor coordination between the chambers with regard to the timing of contraction. The mutations identified in this screen point to discrete and critical steps in the formation and function of the heart and vasculature.

Original languageEnglish (US)
Pages (from-to)285-292
Number of pages8
JournalDevelopment
Volume123
StatePublished - Dec 1 1996
Externally publishedYes

Fingerprint

Zebrafish
Cardiovascular System
Embryonic Structures
Mutation
Endocardium
Blood Vessels
Morphogenesis
Mutagenesis
Genome
Hemorrhage

Keywords

  • Heart
  • Vasculature
  • Zebrafish

ASJC Scopus subject areas

  • Anatomy
  • Cell Biology

Cite this

Stainier, D. Y. R., Fouquet, B., Chen, J. N., Warren, K. S., Weinstein, B. M., Meiler, S. E., ... Fishman, M. C. (1996). Mutations affecting the formation and function of the cardiovascular system in the zebrafish embryo. Development, 123, 285-292.

Mutations affecting the formation and function of the cardiovascular system in the zebrafish embryo. / Stainier, Didier Y R; Fouquet, Bernadette; Chen, Jau Nian; Warren, Kerri S.; Weinstein, Brant M.; Meiler, Steffen E; Mohideen, Manzoor Ali P K; Neuhauss, Stephan C F; Solnica-Krezel, Liliana; Schier, Alexander F.; Zwartkruis, Fried; Stemple, Derek L.; Malicki, Jarema; Driever, Wolfgang; Fishman, Mark C.

In: Development, Vol. 123, 01.12.1996, p. 285-292.

Research output: Contribution to journalArticle

Stainier, DYR, Fouquet, B, Chen, JN, Warren, KS, Weinstein, BM, Meiler, SE, Mohideen, MAPK, Neuhauss, SCF, Solnica-Krezel, L, Schier, AF, Zwartkruis, F, Stemple, DL, Malicki, J, Driever, W & Fishman, MC 1996, 'Mutations affecting the formation and function of the cardiovascular system in the zebrafish embryo', Development, vol. 123, pp. 285-292.
Stainier DYR, Fouquet B, Chen JN, Warren KS, Weinstein BM, Meiler SE et al. Mutations affecting the formation and function of the cardiovascular system in the zebrafish embryo. Development. 1996 Dec 1;123:285-292.
Stainier, Didier Y R ; Fouquet, Bernadette ; Chen, Jau Nian ; Warren, Kerri S. ; Weinstein, Brant M. ; Meiler, Steffen E ; Mohideen, Manzoor Ali P K ; Neuhauss, Stephan C F ; Solnica-Krezel, Liliana ; Schier, Alexander F. ; Zwartkruis, Fried ; Stemple, Derek L. ; Malicki, Jarema ; Driever, Wolfgang ; Fishman, Mark C. / Mutations affecting the formation and function of the cardiovascular system in the zebrafish embryo. In: Development. 1996 ; Vol. 123. pp. 285-292.
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abstract = "As part of a large-scale mutagenesis screen of the zebrafish genome, we have identified 58 mutations that affect the formation and function of the cardiovascular system. The cardiovascular system is particularly amenable for screening in the transparent zebrafish embryo because the heart and blood vessels are prominent and their function easily examined. We have classified the mutations affecting the heart into those that affect primarily either morphogenesis or function. Nine mutations clearly disrupt the formation of the heart. cloche deletes the endocardium. In cloche mutants, the myocardial layer forms in the absence of the endocardium but is dysmorphic and exhibits a weak contractility. Two loci, miles apart and bonnie and clyde, play a critical role in the fusion of the bilateral tubular primordia. Three mutations lead to an abnormally large heart and one to the formation of a diminutive, dysmorphic heart. We have found no mutation that deletes the myocardial cells altogether, but one, pandora, appears to eliminate the ventricle selectively. Seven mutations interfere with vascular integrity, as indicated by hemorrhage at particular sites. In terms of cardiac function, one large group exhibits a weak beat. In this group, five loci affect both chambers and seven a specific chamber (the atrium or ventricle). For example, the weak atrium mutation exhibits an atrium that becomes silent but has a normally beating ventricle. Seven mutations affect the rhythm of the heart causing, for example, a slow rate, a fibrillating pattern or an apparent block to conduction. In several other mutants, regurgitation of blood flow from ventricle to atrium is the most prominent abnormality, due either to the absence of valves or to poor coordination between the chambers with regard to the timing of contraction. The mutations identified in this screen point to discrete and critical steps in the formation and function of the heart and vasculature.",
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AU - Zwartkruis, Fried

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N2 - As part of a large-scale mutagenesis screen of the zebrafish genome, we have identified 58 mutations that affect the formation and function of the cardiovascular system. The cardiovascular system is particularly amenable for screening in the transparent zebrafish embryo because the heart and blood vessels are prominent and their function easily examined. We have classified the mutations affecting the heart into those that affect primarily either morphogenesis or function. Nine mutations clearly disrupt the formation of the heart. cloche deletes the endocardium. In cloche mutants, the myocardial layer forms in the absence of the endocardium but is dysmorphic and exhibits a weak contractility. Two loci, miles apart and bonnie and clyde, play a critical role in the fusion of the bilateral tubular primordia. Three mutations lead to an abnormally large heart and one to the formation of a diminutive, dysmorphic heart. We have found no mutation that deletes the myocardial cells altogether, but one, pandora, appears to eliminate the ventricle selectively. Seven mutations interfere with vascular integrity, as indicated by hemorrhage at particular sites. In terms of cardiac function, one large group exhibits a weak beat. In this group, five loci affect both chambers and seven a specific chamber (the atrium or ventricle). For example, the weak atrium mutation exhibits an atrium that becomes silent but has a normally beating ventricle. Seven mutations affect the rhythm of the heart causing, for example, a slow rate, a fibrillating pattern or an apparent block to conduction. In several other mutants, regurgitation of blood flow from ventricle to atrium is the most prominent abnormality, due either to the absence of valves or to poor coordination between the chambers with regard to the timing of contraction. The mutations identified in this screen point to discrete and critical steps in the formation and function of the heart and vasculature.

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