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

被引：24

作者：

Gholkar, Ankur A. ^{[1
]}

Senese, Silvia ^{[1
]}

Lo, Yu-Chen ^{[1
,2
]}

Capri, Joseph ^{[3
]}

Deardorff, William J. ^{[1
]}

Dharmarajan, Harish ^{[1
]}

Contreras, Ely ^{[1
]}

Hodara, Emmanuelle ^{[1
]}

Whitelegge, Julian P. ^{[3
,4
]}

Jackson, Peter K. ^{[5
]}

Torres, Jorge Z. ^{[1
,4
,6
]}

机构：

[1] Univ Calif Los Angeles, Dept Chem & Biochem, Los Angeles, CA 90024 USA

[2] Univ Calif Los Angeles, Program Bioengn, Los Angeles, CA USA

[3] Univ Calif Los Angeles, David Geffen Sch Med, Jane & Terry Semel Inst Neurosci & Human Behav, Pasarow Mass Spectrometry Lab, Los Angeles, CA 90095 USA

[4] Univ Calif Los Angeles, Inst Mol Biol, Los Angeles, CA 90024 USA

[5] Stanford Univ, Dept Microbiol & Immunol, Sch Med, Baxter Lab Stem Cell Biol, Stanford, CA 94305 USA

[6] Univ Calif Los Angeles, Jonsson Comprehens Canc Ctr, Los Angeles, CA 90024 USA

来源：

CELL CYCLE | 2015年 / 14卷 / 07期

基金：

美国国家科学基金会;

关键词：

Cilia; ciliogenesis; dynein; Tctex1d2; Wdr60; DYNEIN INTERMEDIATE CHAIN; RETROGRADE INTRAFLAGELLAR TRANSPORT; CYTOPLASMIC DYNEIN; PROTEOMIC ANALYSIS; LIGHT-CHAIN; MUTATIONS; DISEASE; CILIUM; GENES; DISORDER;

D O I：

10.4161/15384101.2014.985066

中图分类号：

Q2 [细胞生物学];

学科分类号：

071009 ; 090102 ;

摘要：

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.

引用

页码：1116 / 1125

页数：10

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