Dync2h1

Summary

Gene Symbol: Dync2h1
Description: dynein cytoplasmic 2 heavy chain 1
Alias: 4432416O06Rik, AI448217, D030010H02Rik, D330044F14Rik, DHC1b, DHC2, Dnchc2, b2b414Clo, mKIAA1997, cytoplasmic dynein 2 heavy chain 1, cytoplasmic dynein heavy chain 2, dynein cytoplasmic heavy chain 2, dynein heavy chain 11, dynein heavy chain 2, dynein heavy chain isotype 1B, mDHC11
Species: mouse
Products:     Dync2h1

Top Publications

  1. Huangfu D, Anderson K. Cilia and Hedgehog responsiveness in the mouse. Proc Natl Acad Sci U S A. 2005;102:11325-30 pubmed
    ..b>Dnchc2 and Kif3a, essential subunits of the retrograde and anterograde IFT motors, are also required for both formation ..
  2. May S, Ashique A, Karlen M, Wang B, Shen Y, Zarbalis K, et al. Loss of the retrograde motor for IFT disrupts localization of Smo to cilia and prevents the expression of both activator and repressor functions of Gli. Dev Biol. 2005;287:378-89 pubmed
    ..These data indicate that primary cilia act as specialized signal transduction organelles required for coupling Smo activity to the biochemical processing of Gli3 protein. ..
  3. Liem K, He M, Ocbina P, Anderson K. Mouse Kif7/Costal2 is a cilia-associated protein that regulates Sonic hedgehog signaling. Proc Natl Acad Sci U S A. 2009;106:13377-82 pubmed publisher
    ..We conclude that Kif7 is a core regulator of Shh signaling that may also act as a ciliary motor. ..
  4. Liem K, Ashe A, He M, Satir P, Moran J, Beier D, et al. The IFT-A complex regulates Shh signaling through cilia structure and membrane protein trafficking. J Cell Biol. 2012;197:789-800 pubmed publisher
  5. Tuson M, He M, Anderson K. Protein kinase A acts at the basal body of the primary cilium to prevent Gli2 activation and ventralization of the mouse neural tube. Development. 2011;138:4921-30 pubmed publisher
  6. Ocbina P, Eggenschwiler J, Moskowitz I, Anderson K. Complex interactions between genes controlling trafficking in primary cilia. Nat Genet. 2011;43:547-53 pubmed publisher
    ..Inactivation of the retrograde ciliary motor by mutations in DYNC2H1 causes skeletal dysplasias that have strongly variable expressivity...
  7. Ocbina P, Tuson M, Anderson K. Primary cilia are not required for normal canonical Wnt signaling in the mouse embryo. PLoS ONE. 2009;4:e6839 pubmed publisher
    ..We also analyzed mutant embryos with abnormal primary cilia due to defects in retrograde IFT (Dync2h1)...
  8. Ocbina P, Anderson K. Intraflagellar transport, cilia, and mammalian Hedgehog signaling: analysis in mouse embryonic fibroblasts. Dev Dyn. 2008;237:2030-8 pubmed publisher
    ..SmoA1, activates an Hh reporter in wild-type MEFs, but not in MEFs derived from embryos that lack IFT172 or the Dync2h1 subunit of the retrograde IFT motor...
  9. Perrone C, Tritschler D, Taulman P, Bower R, Yoder B, Porter M. A novel dynein light intermediate chain colocalizes with the retrograde motor for intraflagellar transport at sites of axoneme assembly in chlamydomonas and Mammalian cells. Mol Biol Cell. 2003;14:2041-56 pubmed
    ..work identified a novel dynein light intermediate chain (D2LIC) that colocalized with the mammalian cDHC1b homolog DHC2 in the centrosomal region of cultured cells...

More Information

Publications17

  1. Beckers A, Alten L, Viebahn C, Andre P, Gossler A. The mouse homeobox gene Noto regulates node morphogenesis, notochordal ciliogenesis, and left right patterning. Proc Natl Acad Sci U S A. 2007;104:15765-70 pubmed
    ..Our results establish Noto as an essential regulator of node morphogenesis and ciliogenesis in the posterior notochord, and suggest Noto acts upstream of Foxj1 and Rfx3...
  2. Neesen J, Koehler M, Kirschner R, Steinlein C, Kreutzberger J, Engel W, et al. Identification of dynein heavy chain genes expressed in human and mouse testis: chromosomal localization of an axonemal dynein gene. Gene. 1997;200:193-202 pubmed
    ..Two of these clones, gHDHC7 and gMDHC7, are homologous genes encoding axonemal inner arm dyneins. While the human clone is assigned to 3p21, the mouse gene maps to chromosome 14. ..
  3. Quesnel Vallieres M, Dargaei Z, Irimia M, Gonatopoulos Pournatzis T, Ip J, Wu M, et al. Misregulation of an Activity-Dependent Splicing Network as a Common Mechanism Underlying Autism Spectrum Disorders. Mol Cell. 2016;64:1023-1034 pubmed publisher
    ..Collectively, our results provide evidence that misregulation of an nSR100-dependent splicing network controlled by changes in neuronal activity is causally linked to a substantial fraction of autism cases. ..
  4. Alten L, Schuster Gossler K, Beckers A, Groos S, Ulmer B, Hegermann J, et al. Differential regulation of node formation, nodal ciliogenesis and cilia positioning by Noto and Foxj1. Development. 2012;139:1276-84 pubmed publisher
    ..Thus, the function of Foxj1 in vertebrate organs of asymmetry is conserved, and Noto regulates node morphogenesis and the posterior localization of cilia on node cells independently of Foxj1. ..
  5. Goggolidou P, Stevens J, Agueci F, Keynton J, Wheway G, Grimes D, et al. ATMIN is a transcriptional regulator of both lung morphogenesis and ciliogenesis. Development. 2014;141:3966-77 pubmed publisher
    ..embryonic cilia exhibited shortening and bulging, highly similar to the characterised retrograde IFT phenotype of Dync2h1. Depletion of ATMIN or DYNLL1 in cultured cells recapitulated the in vivo ciliogenesis phenotypes and expression ..
  6. Pfister K, Fisher E, Gibbons I, Hays T, Holzbaur E, McIntosh J, et al. Cytoplasmic dynein nomenclature. J Cell Biol. 2005;171:411-3 pubmed
    ..This nomenclature recognizes the two distinct cytoplasmic dynein complexes and has the flexibility to accommodate the discovery of new subunits and isoforms. ..
  7. Bonnafe E, Touka M, AitLounis A, Baas D, Barras E, Ucla C, et al. The transcription factor RFX3 directs nodal cilium development and left-right asymmetry specification. Mol Cell Biol. 2004;24:4417-27 pubmed
    ..In conclusion, RFX3 is essential for the differentiation of nodal monocilia and hence for LR body axis determination. ..
  8. Bay S, Caspary T. What are those cilia doing in the neural tube?. Cilia. 2012;1:19 pubmed publisher
    ..Here, we discuss the links between cilia and Shh signaling, as well as suggesting additional roles for cilia, and mechanisms for their placement, in the neural tube. ..