Transport and barrier mechanisms that regulate ciliary compartmentalization and ciliopathies

AL Moran, L Louzao-Martinez, DP Norris… - Nature Reviews …, 2024 - nature.com
Primary cilia act as cell surface antennae, coordinating cellular responses to sensory inputs
and signalling molecules that regulate developmental and homeostatic pathways. Cilia are …

Applications of fluorescent protein tagging in structural studies of membrane proteins

AA Kermani - The FEBS Journal, 2024 - Wiley Online Library
Generating active, pure, and monodisperse protein remains a major bottleneck for structural
studies using X‐ray crystallography and cryo‐electron microscopy (cryo‐EM). The current …

Structural insight into the intraflagellar transport complex IFT-A and its assembly in the anterograde IFT train

Y Ma, J He, S Li, D Yao, C Huang, J Wu… - Nature Communications, 2023 - nature.com
Intraflagellar transport (IFT) trains, the polymers composed of two multi-subunit complexes,
IFT-A and IFT-B, carry out bidirectional intracellular transport in cilia, vital for cilia biogenesis …

Interactions between TULP3 tubby domain and ARL13B amphipathic helix promote lipidated protein transport to cilia

VR Palicharla, SH Hwang, BN Somatilaka… - Molecular biology of …, 2023 - Am Soc Cell Biol
The primary cilium is a nexus for cell signaling and relies on specific protein trafficking for
function. The tubby family protein TULP3 transports integral membrane proteins into cilia …

Compound heterozygous IFT81 variations in a skeletal ciliopathy patient cause Bardet–Biedl syndrome-like ciliary defects

K Tasaki, Z Zhou, Y Ishida, Y Katoh… - Human Molecular …, 2023 - academic.oup.com
Owing to their crucial roles in development and homeostasis, defects in cilia cause
ciliopathies with diverse clinical manifestations. The intraflagellar transport (IFT) machinery …

Defective airway intraflagellar transport underlies a combined motile and primary ciliopathy syndrome caused by IFT74 mutations

MR Fassad, N Rumman, K Junger… - Human Molecular …, 2023 - academic.oup.com
Ciliopathies are inherited disorders caused by defective cilia. Mutations affecting motile cilia
usually cause the chronic muco-obstructive sinopulmonary disease primary ciliary …

[HTML][HTML] Hot-wiring dynein-2 establishes roles for IFT-A in retrograde train assembly and motility

F Gonçalves-Santos, ARG De-Castro, DRM Rodrigues… - Cell Reports, 2023 - cell.com
Intraflagellar transport (IFT) trains, built around IFT-A and IFT-B complexes, are carried by
opposing motors to import and export ciliary cargo. While transported by kinesin-2 on …

The emerging functions of intraflagellar transport 52 in ciliary transport and ciliopathies

P Udupa, DK Ghosh - Traffic, 2024 - Wiley Online Library
Ciliary transport in eukaryotic cells is an intricate and conserved process involving the
coordinated assembly and functioning of a multiprotein intraflagellar transport (IFT) complex …

Molecular and structural perspectives on protein trafficking to the primary cilium membrane

V Reddy Palicharla… - Biochemical Society …, 2024 - portlandpress.com
The primary cilium is a dynamic subcellular compartment templated from the mother
centriole or basal body. Cilia are solitary and tiny, but remarkably consequential in cellular …

Programmed withdrawal of cilia maintenance followed by centriole capping leads to permanent cilia loss during cerebellar granule cell neurogenesis

S Constable, CM Ott, AL Lemire, K White, A Lim… - bioRxiv, 2023 - biorxiv.org
Primary cilia in brain neurons provide a privileged compartment for binding and responding
to extracellular ligands such as sonic hedgehog. Paradoxically, cilia in differentiating …