Essential role of lysyl oxidases in notochord development.

Essential role of lysyl oxidases in notochord development. Research Abstract Details 

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Essential role of lysyl oxidases in notochord development. Abstract Text:

John M Gansner,Bryce A Mendelsohn,Keith A Hultman,Stephen L Johnson,Jonathan D Gitlin,

Recent studies reveal a critical role for copper in the development of the zebrafish notochord, suggesting that specific cuproenzymes are required for the structural integrity of the notochord sheath. We now demonstrate that beta-aminopropionitrile, a known inhibitor of the copper-dependent lysyl oxidases, causes notochord distortion in the zebrafish embryo identical to that seen in copper deficiency. Characterization of the zebrafish lysyl oxidase genes reveals eight unique sequences, several of which are expressed in the developing notochord. Specific gene knockdown demonstrates that loss of loxl1 results in notochord distortion, and that loxl1 and loxl5b have overlapping roles in notochord formation. Interestingly, while notochord abnormalities are not observed following partial knockdown of loxl1 or loxl5b alone, in each case this markedly sensitizes developing embryos to notochord distortion if copper availability is diminished. Likewise, partial knockdown of the lysyl oxidase substrate col2a1 results in notochord distortion when combined with reduced copper availability or partial knockdown of loxl1 or loxl5b. These data reveal a complex interplay of gene expression and nutrient availability critical to notochord development. They also provide insight into specific genetic and nutritional factors that may play a role in the pathogenesis of structural birth defects of the axial skeleton.

Essential role of lysyl oxidases in notochord development. Publishing Authors By Initials

JM Gansner,BA Mendelsohn,KA Hultman,SL Johnson,JD Gitlin,

For similar proteins: fish proteins: zebrafish proteins research abstracts see: proteins: fish proteins: zebrafish proteins research

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Essential role of lysyl oxidases in notochord development. Journal Published:

PUBLICATION TYPE: Research Support, N.I.H., Extr

Journal: Developmental biology

VOLUME: 307

Page Numbers: 202-13

Journal Abbreviation: Dev. Biol.

ISSN: 0012-1606

DAY: 1

MONTH: 05

YEAR: 2007

Essential role of lysyl oxidases in notochord development. Information

Number of References:

LANGUAGE: eng

NlmUniqueID: 372762

Essential role of lysyl oxidases in notochord development. Keywords Mesh Terms:

KEYWORDS: Zebrafish Proteins

MESH TERMS: metabolism

Chemical & Substance for Abstract: Essential role of lysyl oxidases in notochord development. Information

Substance Name: Protein-Lysine 6-Oxidase

Registry Number: EC 1.4.3.13

Grant and Affiliation Information for Essential role of lysyl oxidases in notochord development.

AFFILIATION: Department of Pediatrics, Washington University School of Medicine, St. Louis, Missouri 63110, USA.

Country: United States

AGENCY: United States NIGMS

GRANT: T32 GM07200

ACRONYM: GM

MEDLINETA: Dev Biol

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