Subunit composition of the human cytoplasmic dynein-2 complex

J Cell Sci. 2014 Nov 1;127(Pt 21):4774-87. doi: 10.1242/jcs.159038. Epub 2014 Sep 9.


Cytoplasmic dynein-2 is the motor for retrograde intraflagellar transport (IFT), and mutations in dynein-2 are known to cause skeletal ciliopathies. Here, we define for the first time the composition of the human cytoplasmic dynein-2 complex. We show that the proteins encoded by the ciliopathy genes WDR34 and WDR60 are bona fide dynein-2 intermediate chains and are both required for dynein-2 function. In addition, we identify TCTEX1D2 as a unique dynein-2 light chain that is itself required for cilia function. We define several subunits common to both dynein-1 and dynein-2, including TCTEX-1 (also known as DYNLT1) and TCTEX-3 (also known as DYNLT3), roadblock-1 (also known as DYNLRB1) and roadblock-2 (also known as DYNLRB2), and LC8-1 and LC8-2 light chains (DYNLL1 and DYNLL2, respectively). We also find that NudCD3 associates with dynein-2 as it does with dynein-1. By contrast, the common dynein-1 regulators dynactin, LIS1 (also known as PAFAH1B1) and BICD2 are not found in association with dynein-2. These data explain why mutations in either WDR34 or WDR60 cause disease, as well as identifying TCTEX1D2 as a candidate ciliopathy gene.

Keywords: Cilia; Dynein; Intraflagellar transport; Microtubule motor.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Biological Transport / physiology
  • Cell Line
  • Cilia / metabolism
  • Cytoplasmic Dyneins / metabolism*
  • Dyneins / genetics
  • Dyneins / metabolism
  • Humans
  • Immunoprecipitation


  • DYNLT1 protein, human
  • Cytoplasmic Dyneins
  • Dyneins