Journal
CELL CYCLE
Volume 14, Issue 7, Pages 1116-1125Publisher
TAYLOR & FRANCIS INC
DOI: 10.4161/15384101.2014.985066
Keywords
Cilia; ciliogenesis; dynein; Tctex1d2; Wdr60
Categories
Funding
- National Science Foundation [MCB1243645]
- [A P30 DK063491]
- Direct For Biological Sciences [1243645] Funding Source: National Science Foundation
- Div Of Molecular and Cellular Bioscience [1243645] Funding Source: National Science Foundation
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Short-rib polydactyly syndromes (SRPS) arise from mutations in genes involved in retrograde intraflagellar transport (IFT) and basal body homeostasis, which are critical for cilia assembly and function. Recently, mutations in WDR34 or WDR60 (candidate dynein intermediate chains) were identified in SRPS. We have identified and characterized Tctex1d2, which associates with Wdr34, Wdr60 and other dynein complex 1 and 2 subunits. Tctex1d2 and Wdr60 localize to the base of the cilium and their depletion causes defects in ciliogenesis. We propose that Tctex1d2 is a novel dynein light chain important for trafficking to the cilium and potentially retrograde IFT and is a new molecular link to understanding SRPS pathology.
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