Genetic and Kidney Cyst Diseases

Sensory stimuli and cilium trafficking defects trigger the release of ciliary extracellular vesicles from multiple ciliary locations

This study identifies how sensory stimuli and ciliary trafficking defects trigger the release of ciliary extracellular vesicles from different ciliary regions in C. elegans.

Left-right cortical interactions drive intracellular pattern formation in the ciliate Tetrahymena

The paper investigates how left-right cortical interactions drive pattern formation in Tetrahymena by analyzing the role of HPO1 and Bcd1 in oral apparatus positioning.

Two functional forms of the Meckel-Gruber syndrome protein TMEM67 generated by proteolytic cleavage by ADAMTS9 mediate Wnt signaling and ciliogenesis

GABA-induced Ca2+ signaling in the primary cilium of neurons

This study shows that GABA receptor subtype 1 localizes to primary cilia of neurons and initiates ciliary Ca2+ signaling, distinct from its inhibitory role in other neuronal domains.

The RNA splicing factor PRPF8 is required for left-right organiser cilia differentiation and determination of cardiac left-right asymmetry via regulation of Arl13b splicing

This study identifies PRPF8 as essential for left-right organiser cilia function and cardiac asymmetry by regulating ARL13B splicing in mice and zebrafish.

Polycystin-1 acts as an atypical adhesion GPCR that responds to novel Wnt signaling and mechanical stimuli

This paper identifies Polycystin-1 as an atypical adhesion GPCR that activates a signaling pathway through Wnt ligands and mechanical stimuli, leading to GSK3 inhibition and ciliary removal.

Whole-Cell Proteomics Identifies Novel Regulators of Ciliogenesis Beyond the Axoneme

The paper uses whole-cell proteomics to identify novel regulators of ciliogenesis in Xenopus multiciliated cells, revealing 832 upregulated proteins involved in ciliary structures and function.

Ion channel function of polycystin-2/polycystin-1 heteromer revealed by structure-guided mutagenesis

The paper provides functional evidence supporting a model of PC1/PC2 heteromer ion channel function through structure-guided mutagenesis and electrophysiological analysis.

Borg3 controls septin recruitment for primary cilia formation

This study identifies Borg3 as an essential regulator of primary cilia formation by controlling septin dynamics through Cdc42 activation at the ciliary base.

Proximity proteomics of primary cilia in human hypothalamic neurons

The paper identifies ciliary proteins in human hypothalamic neurons and finds new therapeutic targets for obesity, including LPAR1, which mediates ciliary shortening.

SSNA1 organizes the distal luminal centriolar network and promotes ciliogenesis without microtubule association

This study identifies SSNA1 as a centriolar protein in the distal lumen that promotes ciliogenesis without microtubule association, challenging previous views of its function.

Synchronized Temporal-spatial Analysis via Microscopy and Phoshoproteomics (STAMP) of Quiescence

The paper develops STAMP to study temporal-spatial signaling in quiescence by synchronizing ciliogenesis and G0 transition with microscopy and phospho-proteomics, revealing transient phosphorylations in cellular functions.

C. elegans SAS-1 ensures centriole integrity and ciliary function, and operates with SSNA-1

This study shows SAS-1 is essential for centriole integrity and ciliary function in C. elegans and interacts with SSNA-1 to maintain these structures.

alms1 regulates the immune response and brain ageing in zebrafish

This study investigates the role of alms1 in zebrafish, showing its impact on immune response and brain ageing through transcriptomic analysis and CRISPR/Cas9 depletion.

The Dual Molecular Identity of Vestibular Kinocilia: Bridging Structural and Functional Traits of Primary and Motile Cilia

The paper identifies the kinocilium as a unique organelle with molecular features of both primary and motile cilia, showing it generates force in the hair bundle.

Regulation of FGF/MAPK signaling at the primary cilium base controls the proliferation-neurogenesis balance in human cerebellar organoids

This study shows that ciliary dysfunction in human cerebellar organoids leads to overactivation of FGF/MAPK signaling, impairing neurogenesis and increasing progenitor proliferation.

A Role for Importin α in Ciliogenesis and Cilia Length Regulation during Nephrogenesis

This study shows importin α is involved in ciliogenesis and cilia length regulation during kidney development in Xenopus laevis.

A differential requirement for ciliary transition zone proteins in human and mouse neural progenitor fate specification

This study compares the role of RPGRIP1L in human and mouse neural progenitor fate specification, finding distinct effects on motoneuron identity and SHH signaling.

Sorting at ciliary base and ciliary entry of BBSome, IFT-B and IFT-A

The paper uses single-molecule imaging in C. elegans to show how IFT subcomplexes reach the ciliary base and assemble into IFT trains.

Proteins with proximal-distal asymmetries in axoneme localisation control flagellum beat frequency

The paper identifies conserved proximal-distal asymmetries in flagellar axoneme proteins in trypanosomatids and shows their role in controlling flagellum beat frequency.

Methods to assess neuronal primary cilia electrochemical signaling.

The paper describes methods to assess neuronal primary cilia electrochemical signaling using electrophysiology and fluorescent sensors to study ion channels and GPCRs.

Kinesin-3 KIF14 Regulates Intraflagellar Transport Dynamics in Primary Cilia

This study identifies KIF14 as a non-canonical kinesin-3 motor that regulates intraflagellar transport in primary cilia by interacting with its motor domain and C-terminal CC1 domain.

Primary cilia shape postnatal astrocyte development through Sonic Hedgehog signaling

This study shows that astrocyte cilia regulate Sonic Hedgehog signaling during astrocyte development, affecting proliferation and morphology.

Catalytic activity of KMT5B promotes cilia tuft formation without affecting chromatin accessibility of ciliary genes

The paper shows that KMT5B promotes cilia tuft formation independently of chromatin accessibility changes at ciliary genes.

RPGR regulates motile cilia by interfering with actin dynamics

This study shows RPGR variants impair motile cilia in airway multiciliated cells by causing apical F-actin accumulation, leading to reduced ciliation, shorter cilia, and impaired cilia beat.

Decoupling shear stress and pressure effects in the biomechanics of autosomal dominant polycystic kidney disease using a perfused kidney-on-chip.

This study uses a kidney-on-chip model to decouple shear stress and pressure effects on tubule dilation in Pkd1-/- cells, revealing distinct mechanisms in different kidney cell types.

Cell-free Reconstitution Reveals Synergistic Stabilization of Microtubule Doublets by PACRG and FAP20

The paper uses cell-free assays to show PACRG and FAP20 synergistically stabilize microtubule doublets by reducing B-tubule depolymerization and increasing rescue frequency.

Collaborative role of two distinct cilium-specific cytoskeletal systems in driving Hedgehog-responsive transcription factor trafficking

This study identifies the collaborative role of IFT and KIF7 in regulating GLI2 transport within cilia to control Hedgehog signaling.

DYF-5 regulates intraflagellar transport by affecting train turnaround

This study investigates the role of DYF-5 in regulating intraflagellar transport (IFT) dynamics in C. elegans cilia, showing it is essential for IFT train turnaround at the ciliary tip.

Isoform-specific patterns of phosphorylation on axonemal dynein heavy chains

The paper identifies phosphorylation sites on axonemal dynein heavy chains across multiple ciliated eukaryotes, revealing kinase-specific patterns and cilium-specific modifications.