De novo identification of mammalian ciliary motility proteins using cryo-EM

Structure of a microtubule-bound axonemal dynein

(2021) Travis Walton et al.   Nature Communications

The multi‐scale architecture of mammalian sperm flagella and implications for ciliary motility

(2021) Miguel Ricardo Leung et al.   EMBO JOURNAL

Remodeling and activation mechanisms of outer arm dyneins revealed by cryo‐EM

(2021) Shintaroh Kubo et al.   EMBO REPORTS

The morbid genome of ciliopathies: an update

(2020) Hanan E. Shamseldin et al.   GENETICS IN MEDICINE

Improved protein structure prediction using predicted interresidue orientations

(2020) Jianyi Yang et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

The inner junction complex of the cilia is an interaction hub that involves tubulin post-translational modifications

(2020) Ahmad Abdelzaher Zaki Khalifa et al.   eLife

C1orf194 deficiency leads to incomplete early embryonic lethality and dominant intermediate Charcot–Marie–Tooth disease in a knockout mouse model

(2020) Cheng Huang et al.   HUMAN MOLECULAR GENETICS

Right, left and cilia: How asymmetry is established

(2020) Rosie B Little et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

Derivation of Airway Basal Stem Cells from Human Pluripotent Stem Cells

(2020) Finn J. Hawkins et al.   Cell Stem Cell

Database resources of the National Center for Biotechnology Information

(2020) Eric W Sayers et al.   NUCLEIC ACIDS RESEARCH

UCSF ChimeraX : Structure visualization for researchers, educators, and developers

(2020) Eric F. Pettersen et al.   PROTEIN SCIENCE

UniProt: the universal protein knowledgebase in 2021

(2020) et al.   NUCLEIC ACIDS RESEARCH

Motile cilia and airway disease

(2020) Marie Legendre et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components

(2020) Takahiro Ide et al.   PLoS Genetics

Recent insight into intermediate filament structure

(2020) Sherif A. Eldirany et al.   CURRENT OPINION IN CELL BIOLOGY

Inner lumen proteins stabilize doublet microtubules in cilia and flagella

(2019) Mikito Owa et al.   Nature Communications

Taxon-specific expansion and loss of tektins inform metazoan ciliary diversity

(2019) Benjamin R. Bastin et al.   BMC EVOLUTIONARY BIOLOGY

Software tools for automated transmission electron microscopy

(2019) Martin Schorb et al.   NATURE METHODS

Mutations in C1orf194, encoding a calcium regulator, cause dominant Charcot-Marie-Tooth disease

(2019) Shun-Chang Sun et al.   BRAIN

Functional loss of Ccdc151 leads to hydrocephalus in a mouse model of primary ciliary dyskinesia

(2019) Francesco Chiani et al.   Disease Models & Mechanisms

MNS1 variant associated with situs inversus and male infertility

(2019) Joseph S. Leslie et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Structure of the Decorated Ciliary Doublet Microtubule

(2019) Meisheng Ma et al.   CELL

Cryo electron tomography with Volta phase plate reveals novel structural foundations of the 96-nm axonemal repeat in the pathogen Trypanosoma brucei

(2019) Simon Imhof et al.   eLife

Electron cryotomography of intact motile cilia defines the basal body to axoneme transition

(2019) Garrett A. Greenan et al.   JOURNAL OF CELL BIOLOGY

Clinical utility of NGS diagnosis and disease stratification in a multiethnic primary ciliary dyskinesia cohort

(2019) Mahmoud R. Fassad et al.   JOURNAL OF MEDICAL GENETICS

SWISS-MODEL: homology modelling of protein structures and complexes

(2018) Andrew Waterhouse et al.   NUCLEIC ACIDS RESEARCH

Asymmetric distribution and spatial switching of dynein activity generates ciliary motility

(2018) Jianfeng Lin et al.   SCIENCE

Crystal structure of a Ca2+-dependent regulator of flagellar motility reveals the open-closed structural transition

(2018) Tomoki Shojima et al.   Scientific Reports

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

(2018) Hiroshi Yamaguchi et al.   eLife

Automated map sharpening by maximization of detail and connectivity

(2018) Thomas C. Terwilliger et al.   Acta Crystallographica Section D-Structural Biology

Real-space refinement in PHENIX for cryo-EM and crystallography

(2018) Pavel V. Afonine et al.   Acta Crystallographica Section D-Structural Biology

Homozygous loss-of-function mutations in MNS1 cause laterality defects and likely male infertility

(2018) Asaf Ta-Shma et al.   PLoS Genetics

Insights into centriole geometry revealed by cryotomography of doublet and triplet centrioles

(2018) Garrett A Greenan et al.   eLife

The EVcouplings Python framework for coevolutionary sequence analysis

(2018) Thomas A Hopf et al.   BIOINFORMATICS

Whole-exome sequencing identifies a novel CCDC151 mutation, c.325G>T (p.E109X), in a patient with primary ciliary dyskinesia and situs inversus

(2018) Weizhi Zhang et al.   JOURNAL OF HUMAN GENETICS

Cilia-driven cerebrospinal fluid flow directs expression of urotensin neuropeptides to straighten the vertebrate body axis

(2018) Xiaoli Zhang et al.   NATURE GENETICS

EF-hand domain containing 2 (Efhc2) is crucial for distal segmentation of pronephros in zebrafish

(2018) Praveen Barrodia et al.   Cell and Bioscience

New tools for automated high-resolution cryo-EM structure determination in RELION-3

(2018) Jasenko Zivanov et al.   eLife

Functional characterization of tektin-1 in motile cilia and evidence for TEKT1 as a new candidate gene for motile ciliopathies

(2017) Rebecca Ryan et al.   HUMAN MOLECULAR GENETICS

Quantitative Proteomic Analysis of Human Airway Cilia Identifies Previously Uncharacterized Proteins of High Abundance

(2017) Kevin Blackburn et al.   JOURNAL OF PROTEOME RESEARCH

MotionCor2: anisotropic correction of beam-induced motion for improved cryo-electron microscopy

(2017) Shawn Q Zheng et al.   NATURE METHODS

Calaxin establishes basal body orientation and coordinates movement of monocilia in sea urchin embryos

(2017) Katsutoshi Mizuno et al.   Scientific Reports

TTC25 Deficiency Results in Defects of the Outer Dynein Arm Docking Machinery and Primary Ciliary Dyskinesia with Left-Right Body Asymmetry Randomization

(2016) Julia Wallmeier et al.   AMERICAN JOURNAL OF HUMAN GENETICS

A HomozygousNme7Mutation Is Associated withSitus Inversus Totalis

(2016) Orit Reish et al.   HUMAN MUTATION

Ccdc11 is a novel centriolar satellite protein essential for ciliogenesis and establishment of left–right asymmetry

(2016) Erica Silva et al.   MOLECULAR BIOLOGY OF THE CELL

Intermediate Filaments: Structure and Assembly

(2016) Harald Herrmann et al.   Cold Spring Harbor Perspectives in Biology

A method for high-throughput PCR-based genotyping of larval zebrafish tail biopsies

(2016) Robert N. Wilkinson et al.   BIOTECHNIQUES

PIERCE1 is critical for specification of left-right asymmetry in mice

(2016) Young Hoon Sung et al.   Scientific Reports

SnapShot: Motile Cilia

(2015) Feng Zhou et al.   CELL

A Zebrafish Loss-of-Function Model for Human CFAP53 Mutations Reveals Its Specific Role in Laterality Organ Function

(2015) Emily S. Noël et al.   HUMAN MUTATION

Mutations in CCDC11 , which Encodes a Coiled-Coil Containing Ciliary Protein, Causes Situs Inversus Due to Dysmotility of Monocilia in the Left-Right Organizer

(2015) Vijayashankaranarayanan Narasimhan et al.   HUMAN MUTATION

CTFFIND4: Fast and accurate defocus estimation from electron micrographs

(2015) Alexis Rohou et al.   JOURNAL OF STRUCTURAL BIOLOGY

Tissue-based map of the human proteome

(2015) M. Uhlen et al.   SCIENCE

Tools for macromolecular model building and refinement into electron cryo-microscopy reconstructions

(2015) Alan Brown et al.   ACTA CRYSTALLOGRAPHICA SECTION D-BIOLOGICAL CRYSTALLOGRAPHY

Characterization of Tetratricopeptide Repeat-Containing Proteins Critical for Cilia Formation and Function

(2015) Yanan Xu et al.   PLoS One

CCDC151 Mutations Cause Primary Ciliary Dyskinesia by Disruption of the Outer Dynein Arm Docking Complex Formation

(2014) Rim Hjeij et al.   AMERICAN JOURNAL OF HUMAN GENETICS

A human laterality disorder associated with a homozygous WDR16 deletion

(2014) Asaf Ta-Shma et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Structural mechanism of the dynein power stroke

(2014) Jianfeng Lin et al.   NATURE CELL BIOLOGY

Deciphering key features in protein structures with the new ENDscript server

(2014) Xavier Robert et al.   NUCLEIC ACIDS RESEARCH

CHOPCHOP: a CRISPR/Cas9 and TALEN web tool for genome editing

(2014) Tessa G. Montague et al.   NUCLEIC ACIDS RESEARCH

Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia

(2014) Jianfeng Lin et al.   Nature Communications

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

ARMC4 Mutations Cause Primary Ciliary Dyskinesia with Randomization of Left/Right Body Asymmetry

(2013) Rim Hjeij et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Loss-of-function mutations inCCDC114cause primary ciliary dyskinesia

(2013) DH Wu et al.   CLINICAL GENETICS

The coiled-coil domain containing protein CCDC151 is required for the function of IFT-dependent motile cilia in animals

(2013) Julie Jerber et al.   HUMAN MOLECULAR GENETICS

Combined exome and whole-genome sequencing identifies mutations inARMC4as a cause of primary ciliary dyskinesia with defects in the outer dynein arm

(2013) Alexandros Onoufriadis et al.   JOURNAL OF MEDICAL GENETICS

Collaboration gets the most out of software

(2013) Andrew Morin et al.   eLife

Exome Sequencing Identifies Mutations in CCDC114 as a Cause of Primary Ciliary Dyskinesia

(2012) Michael R. Knowles et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Splice-Site Mutations in the Axonemal Outer Dynein Arm Docking Complex Gene CCDC114 Cause Primary Ciliary Dyskinesia

(2012) Alexandros Onoufriadis et al.   AMERICAN JOURNAL OF HUMAN GENETICS

A human laterality disorder associated with recessiveCCDC11mutation

(2012) Zeev Perles et al.   JOURNAL OF MEDICAL GENETICS

NIH Image to ImageJ: 25 years of image analysis

(2012) Caroline A Schneider et al.   NATURE METHODS

Calaxin drives sperm chemotaxis by Ca2+-mediated direct modulation of a dynein motor

(2012) K. Mizuno et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Atomic structure of the vimentin central  -helical domain and its implications for intermediate filament assembly

(2012) A. A. Chernyatina et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Two rotating cilia in the node cavity are sufficient to break left–right symmetry in the mouse embryo

(2012) Kyosuke Shinohara et al.   Nature Communications

Fluid flow and interlinked feedback loops establish left–right asymmetric decay of Cerl2 mRNA

(2012) Tetsuya Nakamura et al.   Nature Communications

MNS1 Is Essential for Spermiogenesis and Motile Ciliary Functions in Mice

(2012) Jian Zhou et al.   PLoS Genetics

Overview of theCCP4 suite and current developments

(2011) Martyn D. Winn et al.   ACTA CRYSTALLOGRAPHICA SECTION D-BIOLOGICAL CRYSTALLOGRAPHY

CMView: Interactive contact map visualization and analysis

(2011) C. Vehlow et al.   BIOINFORMATICS

Pkd1l1 establishes left-right asymmetry and physically interacts with Pkd2

(2011) S. Field et al.   DEVELOPMENT

Characterization and subcellular localization of Tektin 3 in rat spermatozoa

(2011) Hiroe Takiguchi et al.   MOLECULAR REPRODUCTION AND DEVELOPMENT

Fast, scalable generation of high-quality protein multiple sequence alignments using Clustal Omega

(2011) F. Sievers et al.   Molecular Systems Biology

Cryo-electron tomography reveals conserved features of doublet microtubules in flagella

(2011) D. Nicastro et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Deletion of the Parkin co-regulated gene causes defects in ependymal ciliary motility and hydrocephalus in the quakingviable mutant mouse

(2010) G. R. Wilson et al.   HUMAN MOLECULAR GENETICS

Atomic structure of vimentin coil 2

(2010) Stefan Nicolet et al.   JOURNAL OF STRUCTURAL BIOLOGY

Situs Inversus in Dpcd/Poll–/–, Nme7–/–, and Pkd1l1–/– Mice

(2010) P. Vogel et al.   VETERINARY PATHOLOGY

Molecular replacement withMOLREP

(2009) Alexei Vagin et al.   ACTA CRYSTALLOGRAPHICA SECTION D-BIOLOGICAL CRYSTALLOGRAPHY

MolProbity: all-atom structure validation for macromolecular crystallography

(2009) Vincent B. Chen et al.   ACTA CRYSTALLOGRAPHICA SECTION D-BIOLOGICAL CRYSTALLOGRAPHY

Efhc1 deficiency causes spontaneous myoclonus and increased seizure susceptibility

(2009) Toshimitsu Suzuki et al.   HUMAN MOLECULAR GENETICS

A novel neuronal calcium sensor family protein, calaxin, is a potential Ca2+-dependent regulator for the outer arm dynein of metazoan cilia and flagella

(2008) Katsutoshi Mizuno et al.   BIOLOGY OF THE CELL

Tektin 3 is required for progressive sperm motility in mice

(2008) Angshumoy Roy et al.   MOLECULAR REPRODUCTION AND DEVELOPMENT

The tektin family of microtubule-stabilizing proteins

(2008) Linda A Amos   GENOME BIOLOGY

Tektin5, a new Tektin family member, is a component of the middle piece of flagella in rat spermatozoa

(2007) Emi Murayama et al.   MOLECULAR REPRODUCTION AND DEVELOPMENT

High-resolution in situ hybridization to whole-mount zebrafish embryos

(2007) Christine Thisse et al.   Nature Protocols