club CFC

Missense Variants in the Second Transmembrane Domain of TMEM17 Disrupt Its Stability and Function and Lead to a Wide Phenotypic Spectrum of Ciliopathies Missense variants in TMEM17 disrupt its localization and function at the ciliary transition zone, leading to a wide range of ciliopathy phenotypes, from OFD6 and Joubert syndromes to Meckel syndrome.

Congratulations to Lucile Boutaud, Sophie Thomas and colleagues @institutimagine.bsky.social ! #cilia #ciliopathies

onlinelibrary.wiley.com/doi/10.1111/...

An inducible multiciliated cell line resolves proteome dynamics and identifies CDK7 as a conserved regulator. New study from Camille Boutin @camboutin.bsky.social, Laurent Kodjabachian @ibdm.bsky.social @univ-amu.fr and colleagues: rupress.org/jcb/article/...

#Cilia #Technology #Development

Our next afternoon meeting is organized by @alexbnmrh.bsky.social on Wednesday March 18 @institutimagine.bsky.social ! Happy to see you soon there...
#cilia #ciliopathies #flagella

𝗠𝗲𝗲𝘁 𝗼𝘂𝗿 𝗸𝗲𝘆𝗻𝗼𝘁𝗲 𝘀𝗽𝗲𝗮𝗸𝗲𝗿𝘀 𝗳𝗼𝗿 #𝗖𝗶𝗹𝗶𝗮𝟮𝟬𝟮𝟳!

We are grateful for their participation and look forward to welcoming the cilia community on 8-12, March 2027.

Wishing you all a happy and inspiring 2026! 🎉

#Cilia #Milan

From limitless primary basal cells for #PCD therapies, to the TOGORAM1 complex, cytoskeleton & ciliopathies, to #PrimaryCilia diversity in brain #microvasculature, to #ExtrasynapticSignaling at neuronal cilia by in situ proteomics, to an epigenetic 'eraser' controlling #cilia stability... 🔥 /fin

As a Christmas present, our Jove paper with @ebertiaux.bsky.social is out and available for everyone ! Check this out & thanks @centriolelab.bsky.social for the nice highlight

I wish a happy new year to all members of the CFC and all the other fans of centrioles, cilia and flagella!
Hope to see you soon in 2026, ALexandre

C2CD3 localizes to a ring structure observed in the lumen of the distal centriole by in situ cryo-electron tomography. Top: Confocal image of an expanded mouse photoreceptor cell immunolabeled for tubulin (magenta) and C2CD3 (green). DC, daughter centriole; BB, basal body; TZ, transition zone; CC, connecting cilium. Scale bar: 500 nm. Middle: In situ cryo-tomogram slices taken along the longitudinal axis of the centriole and connecting cilium. White arrowheads indicate microtubule triplets, black arrowheads indicate microtubule doublets, orange arrowheads mark the luminal ring structure, and yellow highlights the membrane. Scale bar: 50 nm. Bottom: Model of the architecture of the distal region of the human centriole.

How does C2CD3 contribute to distal appendage formation in centrioles? @ebertiaux.bsky.social @centriolelab.bsky.social show that C2CD3 architecturally organizes the distal #centriole, bridging the luminal distal ring complex & peripheral appendage sites @plosbiology.org 🧪 plos.io/496XZuH

Primary cilia as architects of the neocortex: Roles in brain development, function, and microcephaly In this perspective article, Thomsen et al. discuss how primary cilia coordinate neocortical development, function, and regeneration. They highlight emerging links between ciliary control of neural pr...

Our last review is out today!
It was a fantastic team effort with my friends in Copenhagen, especially Søren Christensen @sorentc.bsky.social and Lars Allan Larsen. A very special thank you to Oskar for his excellent work on the final version! #cilia #microcephaly

www.cell.com/developmenta...

Arp2/3-dependent regulation of ciliogenesis governs adaptive distal tubular epithelial cell states in kidney disease Proteinuria causes ciliary elongation and actin cytoskeleton remodeling, revealing a key mechanism in kidney disease progression.

Very interesting paper!

Arp2/3-dependent regulation of ciliogenesis governs adaptive distal tubular epithelial cell states in kidney disease | Science Advances www.science.org/doi/10.1126/...

Another fantastic technology development paper by @davidbreslowlab.bsky.social. Being able to perform microscopy-based CRISPR screens on fixed cells is big. And TZMP1 is a fascinating transition zone proteins. We may be missing many more of these #microproteins. Congrats to all authors!

Hello everyone, just to announce that our next afternoon symposium on #cilia #flagella and #ciliopathies will take place @institutimagine.bsky.social on Wednesday afternoon March 18 2026. Save the date!

www.biorxiv.org/content/10.1...

A conserved mechanism for the retrieval of polyubiquitinated proteins from cilia CFAP36, a conserved cilia-specific ubiquitin reader, partners with the ARL3 GTPase to enable the selective and directional retrieval of polyubiquitinated proteins from cilia, a process essential for m...

www.cell.com/cell/abstrac...

www.jci.org/articles/vie...

A CRISPR activation screen reveals a cilia disassembly pathway mutated in focal cortical dysplasia A gain-of-function screen uncovers a cilia disassembly pathway linked to genes somatically mutated in neurodevelopmental disease.

www.science.org/doi/10.1126/...

Great issue! Bonus: includes Daniel's work on incorporation of tagged tubulin in flagella!

Drosophila Alms1 proteins are general regulators of #centriolar cartwheel assembly, exhibiting distinct centriolar localization patterns and enabling Plk4-Ana2 interaction
#RefereedPreprint c/o @reviewcommons.org by Véronique Morel, Bénédicte Durand and colleagues
www.embopress.org/doi/full/10....

The protein KIF7 plays a crucial role in shaping the brain’s cortex. By fine-tuning Sonic Hedgehog signalling, it guides neuron development and connectivity. When KIF7 is missing, brain wiring is disrupted. #Neuroscience
buff.ly/ZHSAeIE

Thank you @jcb.org Cynthia and Yameng for this commentary on our IFT paper!

Our collaboration with @mbonhivers.bsky.social is now out in @plosbiology.org! 🎉Using U-ExM, we mapped the formation of the flagellar pocket collar in T. brucei. Huge thanks to everyone involved — one step closer to #PhDone!

#ExM #oneringtorulethemall #Bilbo1 #trypanosoma #protistsonsky

Many thanks to all participants to the CFC2025 meeting last week for having made it such a vibrant experience! So much exciting research on #cilia

Special thanks to our keynote lectures for fantastic talks! @micromotility.bsky.social @mick-lab.bsky.social

🎆 Join us for an unmissable finale to 2025! 🎆 We're thrilled to bring our #cilia & #centrosome community together 25/11/2025 15:00-17:45 GMT! Our 53rd @bscb-official.bsky.social @gensocuk.bsky.social @ukcilianetwork.bsky.social e-symposium schedule is live: www.eventbrite.co.uk/e/bscb-genso... /1

Detail from the graphical abstract: mouse with deletion of the gene Glis2, Glis2 interaction network and the link to DNA damage. Generated with biorender.com.

Fresh online: our paper on Glis2 - project started years ago, and it’s finally out! Congratulations and a big thank you to Lena, Gisela, and the whole team!
tinyurl.com/Glis2

#Cilia #nephrology #ciliopathy
#DDR #nephrolab @ajpseditor.bsky.social @uniklinikkoeln.bsky.social @cecad.bsky.social

Primary #cilia dysfunction as in Bardet-Biedl syndrome compromises normal adipogenic differentiation in favor of fibrogenic transition, predisposing to obesity
@wachtenlab.bsky.social and collaborators
www.embopress.org/doi/full/10....

The Neuronal Primary Cilium is a Key Regulator of Homeostatic Plasticity The capacity of neurons to maintain stable activity levels through homeostatic plasticity is essential for proper brain function. Primary cilia, which are non-motile, antenna-like organelles projectin...

New #cilia preprint alert

Our latest manuscript is now live on bioRxiv:
"The Neuronal Primary Cilium is a Key Regulator of Homeostatic Plasticity"

We hope you enjoy reading it!

www.biorxiv.org/content/10.1...

Spotlight: Yameng Huang and Cynthia He discuss recent advances from Araujo Alves et al. @bastinlab.bsky.social (rupress.org/jcb/article/...), describing how intraflagellar transport trains in T. brucei selectively associate with axonemal doublet #microtubules. rupress.org/jcb/article/...

#Cilia

How do trypanosome IFT trains choose special tracks? Yameng Huang and Cynthia He (National University of Singapore) discuss recent advances from @bastinlab.bsky.social (rupress.org/jcb/article/...), in Spotlight: rupress.org/jcb/article/...

#Biochemistry #Organelles #Cilia #Flagella #Trypanosomes

So proud and happy to share that our study on the #proteome, heterogeneity, and cell-type specificity of primary #cilia is published now at Cell 🎉

Big thank you to all the many people that made this possible, at the Lundberg Lab, Human Protein Atlas, Lindstrand lab, and Hsien-Sheu lab.