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Cell Cycle. 2015;14(7):1116-25. doi: 10.4161/15384101.2014.985066.

Tctex1d2 associates with short-rib polydactyly syndrome proteins and is required for ciliogenesis.

Author information

1
a Department of Chemistry and Biochemistry ; University of California ; Los Angeles , CA USA.

Abstract

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.

KEYWORDS:

Cilia; Tctex1d2; Wdr60; ciliogenesis; dynein

PMID:
25830415
PMCID:
PMC4614626
DOI:
10.4161/15384101.2014.985066
[Indexed for MEDLINE]
Free PMC Article

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