Skip NavigationSkip to Content
Return Return to Search Results

The abcc6a Gene Expression Is Required for Normal Zebrafish Development

  1. Author:
    Li, Q. L.
    Sadowski, S.
    Frank, M.
    Chai, C. L.
    Varadi, A.
    Ho, S. Y.
    Lou, H.
    Dean, M.
    Thisse, C.
    Thisse, B.
    Uitto, J.

  2. Author Address

    [Li, Qiaoli; Sadowski, Sara; Frank, Michael; Uitto, Jouni] Jefferson Med Coll, Dept Dermatol & Cutaneous Biol, Philadelphia, PA 19107 USA. [Li, Qiaoli; Sadowski, Sara; Frank, Michael; Uitto, Jouni] Thomas Jefferson Univ, Jefferson Inst Mol Med, Philadelphia, PA 19107 USA. [Chai, Chunli; Thisse, Christine; Thisse, Bernard] Univ Virginia, Sch Med, Dept Cell Biol, Charlottesville, VA 22908 USA. [Varadi, Andras] Hungarian Acad Sci, Inst Enzymol, Budapest, Hungary. [Ho, Shiu-Ying; Uitto, Jouni] Thomas Jefferson Univ, Dept Biochem & Mol Biol, Philadelphia, PA 19107 USA. [Lou, Hong] SAIC Frederick, Frederick, MD USA. [Dean, Michael] NCI FCRDC, Human Genet Sect, Expt Immunol Lab, Canc & Inflammat Program, Frederick, MD USA. [Chai, Chunli] Southwest Univ, Coll Biotechnol, Chongqing 40076, Peoples R China.;Uitto, J, Jefferson Med Coll, Dept Dermatol & Cutaneous Biol, 233 S,10th St,Suite 450 BLSB, Philadelphia, PA 19107 USA.;Jouni.uitto@jefferson.edu
    1. Year: 2010
    2. Date: Nov
  2. Journal: Journal of Investigative Dermatology
    1. 130
    2. 11
    3. Pages: 2561-2568
  4. Type of Article: Article
  5. ISSN: 0022-202X
  1. Abstract:

    Pseudoxanthoma elasticum (PXE) is caused by mutations in the ABCC6 gene, which encodes a putative efflux transporter, ABCC6. The zebrafish (Danio rerio) has two ABCC6-related sequences. To study the function of abcc6 during zebrafish development, the mRNA expression levels were measured using RT-PCR and in situ hybridization. The abcc6a showed a relatively high level of expression at 5 days post-fertilization (d.p.f.) and the expression was specific to the Kupffer's vesicles. The abcc6b expression was evident at 6 hours post-fertilization (h.p.f.) and remained high up to 8 d.p.f., corresponding to embryonic kidney proximal tubules. Morpholinos were designed to both genes to prevent pre-mRNA splicing and block translation. Injection of the abcc6a morpholinos into 1-4 cell zebrafish embryos decreased gene expression by 54-81%, and induced a phenotype, pericardial edema and curled tail associated with death at around 8 d.p.f. Microinjecting zebrafish embryos with full-length mouse Abcc6 mRNA together with the morpholino completely rescued this phenotype. No phenotypic changes were observed when the abcc6b gene morpholino was injected into embryos with knockdown efficiency of 100%. These results suggest that abcc6a is an essential gene for normal zebrafish development and provide insight into the function of ABCC6, the gene mutated in PXE.

    See More

  1. Keywords:

External Sources

  1. View Full Text
  2. DOI: 10.1038/jid.2010.174
  3. View record in Web of Science®
  4. WOS: 000282963600008

Library Notes

  1. Fiscal Year: FY2010-2011
NCI at Frederick

You are leaving a government website.

This external link provides additional information that is consistent with the intended purpose of this site. The government cannot attest to the accuracy of a non-federal site.

Linking to a non-federal site does not constitute an endorsement by this institution or any of its employees of the sponsors or the information and products presented on the site. You will be subject to the destination site's privacy policy when you follow the link.

ContinueCancel