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Details on Person Dynein arm assembly factor (DNAAF) 11 (also known as LRRC6) ...

Class:IdSummation:9969049
_displayNameDynein arm assembly factor (DNAAF) 11 (also known as LRRC6) ...
_timestamp2025-12-12 20:35:27
created[InstanceEdit:9969048] Rothfels, Karen, 2025-10-20
modified[InstanceEdit:9976669] Rothfels, Karen, 2025-12-12
textDynein arm assembly factor (DNAAF) 11 (also known as LRRC6) and ZMYND10 (also known as DNAAF7) are proteins that contribute to the cytoplasmic assembly of inner and outer dynein arms (IDAs and ODAs, respectively) of the motile axoneme prior to the translocation of IDAs and ODAs to the site of axoneme extension at the plasma membrane (Mali et al, 2018; reviewed in King, 2021; King, 2023; Desai et al, 2018). ZMYND10 interacts with HSP90:FKBP8 in mouse testes and human tracheal endothelial cells (Mali et al, 2018; Cho et al, 2018) and additionally interacts with other DNAAFs and with structural components of the inner and outer dynein arms (Zariwala et al, 2013; Moore et al, 2013; Cho et al, 2018).
DNAAF11/LRRC6 has a p23-like domain, suggesting it may interact with HSP90. Based on the role of p23 proteins in coordinating the release of client proteins with the hydrolysis of ATP during the HSP90 cycle, DNAAF11/LRRC6 may play a role in release of client proteins from the HSP90 chaperone system to the R2TP-like chaperone complexes, though this remains to be explicitly demonstrated (Zariwala et al, 2013; Kott et al, 2012; Horani et al, 2013; Inaba et al, 2016; Li et al, 2021; reviewed in Li and Buchner, 2013; Biebl and Buchner, 2023; Biebl and Buchner, 2019; Fabczak and Osinka, 2019; Lynham and Houry, 2022).
Mutations in both DNAAF11/LRRC6 and ZMYND10/DNAAF7 are associated with primary ciliary dyskinesia (PCD), a genetically heterogeneous disorder characterized by mislocalization of components of the dynein arms and consequent defects or absence of ciliary beating (reviewed in Despotes et al, 2024). Mutations in DNAAF11/LRRC6 and ZMYMD10 affect assembly of both inner and outer dynein arms (Moore et al, 2013; Zariwala et al, 2013; Kott et al, 2012; Li et al, 2021; reviewed in Legendre et al, 2021). Consistent with this, ZMYND10/DNAAF7 has been shown to interact with the ODA light chain DNAL1 (Cho et al, 2018); while mutation of DNAAF11/LRRC6 has been shown to cause mislocalization of IDA heavy chains DNAH7 and DNAH1, ODA intermediate chain DNAI1, and ODA heavy chains DNAH8, DNAH9, DNAH10 and DNAH17 (Horani et al, 2013; Li et al, 2021; Kott et al, 2012). The mechanistic details of how ZMYND10/DNAAF7 and DNAAF11/LRRC6 contribute to the assembly of these proteins into the mature ODA and IDA are not clear however.
In addition to its role in the cytoplasmic assembly of ODAs and IDAs, ZMYND10/DNAAF7 and DNAAF11/LRRC6 have also been shown to contribute to multiciliogenesis through their role in promoting nuclear localization of the key transcriptional regulator FOXJ1 (Kim et al, 2023).
(summation)[Reaction:9969495] DNAAF11:ZMYND10 bind HSP90 dimer:FKBP8 [Homo sapiens]
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