Target intelligence / Profile preview

Cytoplasmic dynein 2 intermediate chain 1 (DYNC2I1)

Target
DYNC2I1
Molecular classification
Motor protein complex subunit, WD repeat protein family, Component of cytoplasmic dynein-2 complex, Other
01

Overview

Cytoplasmic dynein 2 intermediate chain 1 (DYNC2I1, also known as WDR60) is a non-catalytic, WD repeat-containing accessory protein that forms part of the cytoplasmic dynein-2 complex—a multisubunit motor protein responsible for retrograde cargo transport within cilia and flagella, in conjunction with the intraflagellar transport (IFT) system[1][2][4][5][6][7]. DYNC2I1 interacts with the heavy chain (DYNC2H1/DHC2) and another intermediate chain (WDR34) to mediate cargo movement toward the ciliary base, assuring proper ciliary assembly, signaling, and maintenance. Defects in this protein disrupt ciliary function, causing various ciliopathies—genetic disorders affecting the skeleton and other organs. DYNC2I1’s essential cellular roles include cell cycle regulation, signal transduction, and programmed cell death, highlighting its broad importance beyond ciliary biology[2][4][5][6]. **Notes:** - No drugs are currently known to directly target DYNC2I1. - While pathogenic mutations are clinically relevant (underlying role in genetic syndromes), this protein is not currently considered a drug target (therapeutic target) in the sense of receptors, enzymes, or classic drug modulators. - DYNC2I1 is often referred to interchangeably with "WDR60" in structural and genetic literature[2][7].

Other names
WDR60WD repeat-containing protein 60FAP163CFAP163DIC6SRPS6SRTD8testicular secretory protein Li 66FLJ10300
02

Biological functions

Retrograde ciliary protein traffickingIntraflagellar transport (IFT)Cilia and flagella formation and maintenanceCell cycle progressionSignal transductionApoptosisGene regulation
03

Disease associations

Short-rib thoracic dysplasia (SRTD8) with or without polydactylyJeune syndromeShort rib-polydactyly syndrome (Verma-Naumoff type)Bardet-Biedl syndrome (implicated in pathway)Other ciliopathies
04

Safety considerations

Mutations cause ciliopathies, especially skeletal dysplasias (short-rib thoracic dysplasia and polydactyly syndromes)Loss-of-function impairs cilia structure and function, affecting multiple organ systems

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