Role of the Novel Hsp90 Co-Chaperones in Dynein Arms’ Preassembly

CFAP300: Mutations in Slavic Primary Ciliary Dyskinesia Patients and a Role in Ciliary Dynein Arms Trafficking

(2019) Ewa Zietkiewicz et al.   AMERICAN JOURNAL OF RESPIRATORY CELL AND MOLECULAR BIOLOGY

Binding properties of the quaternary assembly protein SPAG1

(2019) Marie-Eve Chagot et al.   BIOCHEMICAL JOURNAL

WDR92 Is Required for Axonemal Dynein Heavy Chain Stability in Cytoplasm

(2019) Ramila S. Patel-King et al.   MOLECULAR BIOLOGY OF THE CELL

Structural mechanism for regulation of the AAA-ATPases RUVBL1-RUVBL2 in the R2TP co-chaperone revealed by cryo-EM

(2019) Hugo Muñoz-Hernández et al.   Science Advances

Clinical and genetic analysis of patients with primary ciliary dyskinesia caused by novel DNAAF3 mutations

(2019) Zhuoyao Guo et al.   JOURNAL OF HUMAN GENETICS

Mutations in C11orf70 Cause Primary Ciliary Dyskinesia with Randomization of Left/Right Body Asymmetry Due to Defects of Outer and Inner Dynein Arms

(2018) Inga M. Höben et al.   AMERICAN JOURNAL OF HUMAN GENETICS

C11orf70 Mutations Disrupting the Intraflagellar Transport-Dependent Assembly of Multiple Axonemal Dyneins Cause Primary Ciliary Dyskinesia

(2018) Mahmoud R. Fassad et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Ciliary dynein motor preassembly is regulated by Wdr92 in association with HSP90 co-chaperone, R2TP

(2018) Petra zur Lage et al.   JOURNAL OF CELL BIOLOGY

Establishment of the early cilia preassembly protein complex during motile ciliogenesis

(2018) Amjad Horani et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Deep Structural Analysis of RPAP3 and PIH1D1, Two Components of the HSP90 Co-chaperone R2TP Complex

(2018) Julien Henri et al.   STRUCTURE

RPAP3 provides a flexible scaffold for coupling HSP90 to the human R2TP co-chaperone complex

(2018) Fabrizio Martino et al.   Nature Communications

The RPAP3-Cterminal domain identifies R2TP-like quaternary chaperones

(2018) Chloé Maurizy et al.   Nature Communications

ZMYND10 stabilizes intermediate chain proteins in the cytoplasmic pre-assembly of dynein arms

(2018) Kyeong Jee Cho et al.   PLoS Genetics

Systematic studies of all PIH proteins in zebrafish reveal their distinct roles in axonemal dynein assembly

(2018) Hiroshi Yamaguchi et al.   eLife

ZMYND10 functions in a chaperone relay during axonemal dynein assembly

(2018) Girish R Mali et al.   eLife

A liquid-like organelle at the root of motile ciliopathy

(2018) Ryan L Huizar et al.   eLife

DNAAF1 links heart laterality with the AAA+ ATPase RUVBL1 and ciliary intraflagellar transport

(2017) Verity L Hartill et al.   HUMAN MOLECULAR GENETICS

The HSP90 chaperone machinery

(2017) Florian H. Schopf et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

Genes and molecular pathways underpinning ciliopathies

(2017) Jeremy F. Reiter et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

R2TP/Prefoldin-like component RUVBL1/RUVBL2 directly interacts with ZNHIT2 to regulate assembly of U5 small nuclear ribonucleoprotein

(2017) Philippe Cloutier et al.   Nature Communications

X-linked primary ciliary dyskinesia due to mutations in the cytoplasmic axonemal dynein assembly factor PIH1D3

(2017) Chiara Olcese et al.   Nature Communications

Primary Ciliary Dyskinesia

(2016) Michael R. Knowles et al.   CLINICS IN CHEST MEDICINE

Transport of the outer dynein arm complex to cilia requires a cytoplasmic protein Lrrc6

(2016) Yasuko Inaba et al.   GENES TO CELLS

Motile and non-motile cilia in human pathology: from function to phenotypes

(2016) Hannah M Mitchison et al.   JOURNAL OF PATHOLOGY

Fine-Tuning Motile Cilia and Flagella: Evolution of the Dynein Motor Proteins from Plants to Humans at High Resolution

(2016) Martin Kollmar   MOLECULAR BIOLOGY AND EVOLUTION

A prefoldin-associated WD-repeat protein (WDR92) is required for the correct architectural assembly of motile cilia

(2016) Ramila S. Patel-King et al.   MOLECULAR BIOLOGY OF THE CELL

Axoneme Structure from Motile Cilia

(2016) Takashi Ishikawa   Cold Spring Harbor Perspectives in Biology

Mutations in the Motile Cilia Gene DNAAF1 are Associated with Neural Tube Defects in Humans

(2016) C. Miao et al.   G3-Genes Genomes Genetics

ZMYND10 - Mutation Analysis in Slavic Patients with Primary Ciliary Dyskinesia

(2016) Małgorzata Kurkowiak et al.   PLoS One

Diagnosis and management of primary ciliary dyskinesia

(2014) J. S. Lucas et al.   ARCHIVES OF DISEASE IN CHILDHOOD

DrosophilaSpag Is the Homolog of RNA Polymerase II-associated Protein 3 (RPAP3) and Recruits the Heat Shock Proteins 70 and 90 (Hsp70 and Hsp90) during the Assembly of Cellular Machineries

(2014) Nour El Houda Benbahouche et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Pih1d3 is required for cytoplasmic preassembly of axonemal dynein in mouse sperm

(2014) Fenglan Dong et al.   JOURNAL OF CELL BIOLOGY

Structural Basis for Phosphorylation-Dependent Recruitment of Tel2 to Hsp90 by Pih1

(2014) Mohinder Pal et al.   STRUCTURE

Functional Diversity of Axonemal Dyneins as Assessed by in Vitro and in Vivo Motility Assays ofChlamydomonasMutants

(2014) Ritsu Kamiya et al.   ZOOLOGICAL SCIENCE

MCIDAS mutations result in a mucociliary clearance disorder with reduced generation of multiple motile cilia

(2014) Mieke Boon et al.   Nature Communications

Phosphorylation-Dependent PIH1D1 Interactions Define Substrate Specificity of the R2TP Cochaperone Complex

(2014) Zuzana Hořejší et al.   Cell Reports

HEATR2 Plays a Conserved Role in Assembly of the Ciliary Motile Apparatus

(2014) Christine P. Diggle et al.   PLoS Genetics

Zebrafish Ciliopathy Screen Plus Human Mutational Analysis Identifies C21orf59 and CCDC65 Defects as Causing Primary Ciliary Dyskinesia

(2013) Christina Austin-Tse et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Mutations in SPAG1 Cause Primary Ciliary Dyskinesia Associated with Defective Outer and Inner Dynein Arms

(2013) Michael R. Knowles et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Mutations in ZMYND10, a Gene Essential for Proper Axonemal Assembly of Inner and Outer Dynein Arms in Humans and Flies, Cause Primary Ciliary Dyskinesia

(2013) Daniel J. Moore et al.   AMERICAN JOURNAL OF HUMAN GENETICS

ZMYND10 Is Mutated in Primary Ciliary Dyskinesia and Interacts with LRRC6

(2013) Maimoona A. Zariwala et al.   AMERICAN JOURNAL OF HUMAN GENETICS

DYX1C1 is required for axonemal dynein assembly and ciliary motility

(2013) Aarti Tarkar et al.   NATURE GENETICS

LRRC6 Mutation Causes Primary Ciliary Dyskinesia with Dynein Arm Defects

(2013) Amjad Horani et al.   PLoS One

Reptin/Ruvbl2 is a Lrrc6/Seahorse interactor essential for cilia motility

(2013) L. Zhao et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Structure, Function and Regulation of the Hsp90 Machinery

(2013) Johannes Buchner et al.   Biomedical Journal

Whole-Exome Capture and Sequencing Identifies HEATR2 Mutation as a Cause of Primary Ciliary Dyskinesia

(2012) Amjad Horani et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Loss-of-Function Mutations in LRRC6 , a Gene Essential for Proper Axonemal Assembly of Inner and Outer Dynein Arms, Cause Primary Ciliary Dyskinesia

(2012) Esther Kott et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Cilia, KIF3 molecular motor and nodal flow

(2012) Nobutaka Hirokawa et al.   CURRENT OPINION IN CELL BIOLOGY

Mutations in axonemal dynein assembly factor DNAAF3 cause primary ciliary dyskinesia

(2012) Hannah M Mitchison et al.   NATURE GENETICS

The R2TP complex: Discovery and functions

(2011) Yoshito Kakihara et al.   BIOCHIMICA ET BIOPHYSICA ACTA-MOLECULAR CELL RESEARCH

Molecular chaperones in protein folding and proteostasis

(2011) F. Ulrich Hartl et al.   NATURE

Ciliogenesis: building the cell's antenna

(2011) Hiroaki Ishikawa et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

PIH1D1, a subunit of R2TP complex, inhibits doxorubicin-induced apoptosis

(2010) Mika Inoue et al.   BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS

Discrete PIH proteins function in the cytoplasmic preassembly of different subsets of axonemal dyneins

(2010) Ryosuke Yamamoto et al.   JOURNAL OF CELL BIOLOGY

CK2 Phospho-Dependent Binding of R2TP Complex to TEL2 Is Essential for mTOR and SMG1 Stability

(2010) Zuzana Hořejší et al.   MOLECULAR CELL

HSP90 and Its R2TP/Prefoldin-like Cochaperone Are Involved in the Cytoplasmic Assembly of RNA Polymerase II

(2010) Séverine Boulon et al.   MOLECULAR CELL

Loss-of-Function Mutations in the Human Ortholog of Chlamydomonas reinhardtii ODA7 Disrupt Dynein Arm Assembly and Cause Primary Ciliary Dyskinesia

(2009) Philippe Duquesnoy et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Deletions and Point Mutations of LRRC50 Cause Primary Ciliary Dyskinesia Due to Dynein Arm Defects

(2009) Niki Tomas Loges et al.   AMERICAN JOURNAL OF HUMAN GENETICS

A novel role for DYX1C1, a chaperone protein for both Hsp70 and Hsp90, in breast cancer

(2009) Yuxin Chen et al.   JOURNAL OF CANCER RESEARCH AND CLINICAL ONCOLOGY

Left-Right Determination: Involvement of Molecular Motor KIF3, Cilia, and Nodal Flow

(2009) N. Hirokawa et al.   Cold Spring Harbor Perspectives in Biology

Molecular cloning of novel Monad binding protein containing tetratricopeptide repeat domains

(2008) Yuki Itsuki et al.   FEBS LETTERS

ODA16 aids axonemal outer row dynein assembly through an interaction with the intraflagellar transport machinery

(2008) Noveera T. Ahmed et al.   JOURNAL OF CELL BIOLOGY

Ktu/PF13 is required for cytoplasmic pre-assembly of axonemal dyneins

(2008) Heymut Omran et al.   NATURE

Tissue expression patterns identify mouse cilia genes

(2007) Timothy S. McClintock et al.   PHYSIOLOGICAL GENOMICS