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PMID: 20308572 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Systematic discovery of nonobvious human disease models through orthologous phenotypes.

McGary KL, Park TJ, Woods JO, Cha HJ, Wallingford JB, Marcotte EM

Abstract

Biologists have long used model organisms to study human diseases, particularly when the model bears a close resemblance to the disease. We present a method that quantitatively and systematically identifies nonobvious equivalences between mutant phenotypes in different species, based on overlapping sets of orthologous genes from human, mouse, yeast, worm, and plant (212,542 gene-phenotype associations). These orthologous phenotypes, or phenologs, predict unique genes associated with diseases. Our method suggests a yeast model for angiogenesis defects, a worm model for breast cancer, mouse models of autism, and a plant model for the neural crest defects associated with Waardenburg syndrome, among others. Using these models, we show that SOX13 regulates angiogenesis, and that SEC23IP is a likely Waardenburg gene. Phenologs reveal functionally coherent, evolutionarily conserved gene networks-many predating the plant-animal divergence-capable of identifying candidate disease genes.

MeSH Terms
Animals Computational Biology Gene Expression Regulation Humans Neovascularization, Pathologic Phenotype
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
McGary Kriston L
Department of Molecular Cell and Developmental Biology, Center for Systems and Synthetic Biology, Institute for Cellular and Molecular Biology, University of Texas, Austin, TX 78712, USA.
Park Tae Joo
Woods John O
Cha Hye Ji
Wallingford John B
Marcotte Edward M
References (36)
36 references, click to expand
  1. lin-35 and lin-53, two genes that antagonize a C. elegans Ras pathway, encode proteins similar to Rb and its binding protein RbAp48.
    Cell. 1998 Dec 23;95(7):981-91 PMID: 9875852
  2. The chemical genomic portrait of yeast: uncovering a phenotype for all genes.
    Science. 2008 Apr 18;320(5874):362-5 PMID: 18420932
  3. Nondisjunction Mutants of the Nematode CAENORHABDITIS ELEGANS.
    Genetics. 1979 Jan;91(1):67-94 PMID: 17248881
  4. WormBase: a comprehensive data resource for Caenorhabditis biology and genomics.
    Nucleic Acids Res. 2005 Jan 1;33(Database issue):D383-9 PMID: 15608221
  5. McKusick's Online Mendelian Inheritance in Man (OMIM).
    Nucleic Acids Res. 2009 Jan;37(Database issue):D793-6 PMID: 18842627
  6. Homozygosity of chromosome 13 in retinoblastoma.
    N Engl J Med. 1984 Mar 1;310(9):550-3 PMID: 6694706
  7. Chemical rescue of cleft palate and midline defects in conditional GSK-3beta mice.
    Nature. 2007 Mar 1;446(7131):79-82 PMID: 17293880
  8. Regulation of gammadelta versus alphabeta T lymphocyte differentiation by the transcription factor SOX13.
    Science. 2007 Jan 12;315(5809):230-3 PMID: 17218525
  9. Prostate-specific membrane antigen regulates angiogenesis by modulating integrin signal transduction.
    Mol Cell Biol. 2006 Jul;26(14):5310-24 PMID: 16809768
  10. High expression of the HMG box factor sox-13 in arterial walls during embryonic development.
    Nucleic Acids Res. 1998 Jan 15;26(2):469-76 PMID: 9421502
  11. Worldwide distribution of Waardenburg syndrome.
    Ann Otol Rhinol Laryngol. 2003 Sep;112(9 Pt 1):817-20 PMID: 14535568
  12. SCMD: Saccharomyces cerevisiae Morphological Database.
    Nucleic Acids Res. 2004 Jan 1;32(Database issue):D319-22 PMID: 14681423
  13. On the Origin of Species by Means of Natural Selection, or the Preservation of Favoured Races in the Struggle for Life.
    Br Foreign Med Chir Rev. 1860 Apr;25(50):367-404 PMID: 30164232
  14. The genetic basis of a craniofacial disease provides insight into COPII coat assembly.
    Dev Cell. 2007 Nov;13(5):623-634 PMID: 17981132
  15. The mouse genome database (MGD): new features facilitating a model system.
    Nucleic Acids Res. 2007 Jan;35(Database issue):D630-7 PMID: 17135206
  16. Automatic clustering of orthologs and in-paralogs from pairwise species comparisons.
    J Mol Biol. 2001 Dec 14;314(5):1041-52 PMID: 11743721
  17. Disruption of Bardet-Biedl syndrome ciliary proteins perturbs planar cell polarity in vertebrates.
    Nat Genet. 2005 Oct;37(10):1135-40 PMID: 16170314
  18. Deep homology and the origins of evolutionary novelty.
    Nature. 2009 Feb 12;457(7231):818-23 PMID: 19212399
  19. The Saccharomyces cerevisiae GSK-3 beta homologs.
    Curr Drug Targets. 2006 Nov;7(11):1455-65 PMID: 17100585
  20. The pallid gene encodes a novel, syntaxin 13-interacting protein involved in platelet storage pool deficiency.
    Nat Genet. 1999 Nov;23(3):329-32 PMID: 10610180
  21. Statins and cancer prevention.
    Nat Rev Cancer. 2005 Dec;5(12):930-42 PMID: 16341084
  22. Targeted deletion of BMK1/ERK5 in adult mice perturbs vascular integrity and leads to endothelial failure.
    J Clin Invest. 2004 Apr;113(8):1138-48 PMID: 15085193
  23. Ultrastructure of developing melanosomes in C57 black and pallid mice.
    Dev Biol. 1970 Oct;23(2):261-75 PMID: 5476812
  24. X chromosomal abnormalities in basal-like human breast cancer.
    Cancer Cell. 2006 Feb;9(2):121-32 PMID: 16473279
  25. Conserved pathways within bacteria and yeast as revealed by global protein network alignment.
    Proc Natl Acad Sci U S A. 2003 Sep 30;100(20):11394-9 PMID: 14504397
  26. Cranio-lenticulo-sutural dysplasia is caused by a SEC23A mutation leading to abnormal endoplasmic-reticulum-to-Golgi trafficking.
    Nat Genet. 2006 Oct;38(10):1192-7 PMID: 16980979
  27. SIRT1 controls endothelial angiogenic functions during vascular growth.
    Genes Dev. 2007 Oct 15;21(20):2644-58 PMID: 17938244
  28. Erk5 null mice display multiple extraembryonic vascular and embryonic cardiovascular defects.
    Proc Natl Acad Sci U S A. 2002 Jul 9;99(14):9248-53 PMID: 12093914
  29. Distinguishing homologous from analogous proteins.
    Syst Zool. 1970 Jun;19(2):99-113 PMID: 5449325
  30. An improved, bias-reduced probabilistic functional gene network of baker's yeast, Saccharomyces cerevisiae.
    PLoS One. 2007 Oct 03;2(10):e988 PMID: 17912365
  31. Involvement of protein kinase CK2 in angiogenesis and retinal neovascularization.
    Invest Ophthalmol Vis Sci. 2004 Dec;45(12):4583-91 PMID: 15557471
  32. The emerging conceptual framework of evolutionary developmental biology.
    Nature. 2002 Feb 14;415(6873):757-64 PMID: 11845200
  33. GSK3 at the edge: regulation of developmental specification and cell polarization.
    Curr Drug Targets. 2006 Nov;7(11):1411-9 PMID: 17100581
  34. Broad network-based predictability of Saccharomyces cerevisiae gene loss-of-function phenotypes.
    Genome Biol. 2007;8(12):R258 PMID: 18053250
  35. The sequential activation of the yeast HOG and SLT2 pathways is required for cell survival to cell wall stress.
    Mol Biol Cell. 2008 Mar;19(3):1113-24 PMID: 18184748
  36. Saccharomyces Genome Database (SGD) provides secondary gene annotation using the Gene Ontology (GO).
    Nucleic Acids Res. 2002 Jan 1;30(1):69-72 PMID: 11752257
Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
1091-6490
Published
2010-04-06
Epub
2010-00-22
Pages
6544-9
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC2851946
Subset
IM
Grants
NIGMS NIH HHS · R01 GM067779 · United States
NIGMS NIH HHS · R01 GM074104 · United States
NIGMS NIH HHS · R01 GM088624 · United States
Howard Hughes Medical Institute · United States
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