WD60/FAP163 is a dynein intermediate chain required for retrograde intraflagellar transport in cilia

Abstract

WD60/FAP163 is identified as an additional intermediate chain of the dynein that powers retrograde intraflagellar transport. Lack of WD60/FAP163 results in a severe ciliary assembly defect.

The intraflagellar transport dynein complex of trypanosomes is made of a heterodimer of dynein heavy chains and of light and intermediate chains of distinct functions

“…Clear homologues of DIC5/FAP133 and of the other intermediate dynein chain, WD60/FAP163, could not be identified in C. elegans (Rompolas et al. , 2007; Patel-King et al. , 2013).…”

Dynein and intraflagellar transport

“…The sequence of a second dynein intermediate chain (D1bIC1/FAP163, encoded by DIC6 ) is closely related to that of D1bIC2 (Patel-King et al, 2013). Biochemical analysis showed that it is in the same complex as D1bIC2 and LC8 and is transported by IFT in Chlamydomonas .…”

Whole exome sequencing is an efficient, sensitive and specific method for determining the genetic cause of short‐rib thoracic dystrophies

“…In addition, the discovery of novel disease‐associated genes can shed further light on biological processes disrupted in disease states. For example, two of the genes identified by analysis of the current cohort, WDR60 and WDR34 , are homologous to genes in lower organisms that have been implicated as components of cytoplasmic dynein motors . As the vertebrate cytoplasmic dynein‐2 motor responsible for retrograde IFT is not well‐defined at this stage, these data provide valuable insight into the structure of this complex.…”