Composition, organization and mechanisms of the transition zone, a gate for the cilium

K Park, MR Leroux - EMBO reports, 2022 - embopress.org
The cilium evolved to provide the ancestral eukaryote with the ability to move and sense its
environment. Acquiring these functions required the compartmentalization of a dynein …

Cilia-localized counterregulatory signals as drivers of renal cystogenesis

RV Walker, A Maranto, VR Palicharla… - Frontiers in Molecular …, 2022 - frontiersin.org
Primary cilia play counterregulatory roles in cystogenesis—they inhibit cyst formation in the
normal renal tubule but promote cyst growth when the function of polycystins is impaired …

GPR161 structure uncovers the redundant role of sterol-regulated ciliary cAMP signaling in the Hedgehog pathway

N Hoppe, S Harrison, SH Hwang, Z Chen… - Nature Structural & …, 2024 - nature.com
The orphan G protein-coupled receptor (GPCR) GPR161 plays a central role in
development by suppressing Hedgehog signaling. The fundamental basis of how GPR161 …

Permanent deconstruction of intracellular primary cilia in differentiating granule cell neurons

CM Ott, S Constable, TM Nguyen, K White… - Journal of Cell …, 2024 - rupress.org
Primary cilia on granule cell neuron progenitors in the developing cerebellum detect sonic
hedgehog to facilitate proliferation. Following differentiation, cerebellar granule cells …

Context-dependent ciliary regulation of hedgehog pathway repression in tissue morphogenesis

SH Hwang, KA White, BN Somatilaka, B Wang… - PLoS …, 2023 - journals.plos.org
A fundamental problem in tissue morphogenesis is identifying how subcellular signaling
regulates mesoscale organization of tissues. The primary cilium is a paradigmatic organelle …

Interactome analysis of human phospholipase D and phosphatidic acid-associated protein network

RE Kattan, H Han, G Seo, B Yang, Y Lin… - Molecular & Cellular …, 2022 - ASBMB
Mammalian phospholipase D (PLD) enzyme family consists of six members. Among them,
PLD1/2/6 catalyzes phosphatidic acid (PA) production, while PLD3/4/5 has no catalytic …

Ciliary signaling in stem cells in health and disease: Hedgehog pathway and beyond

IS Shimada, Y Kato - Seminars in Cell & Developmental Biology, 2022 - Elsevier
The primary cilium is a hair-like sensory compartment that protrudes from the cellular
surface. The primary cilium is enriched in a variety of signaling molecules that regulate …

Ciliary and extraciliary Gpr161 pools repress hedgehog signaling in a tissue-specific manner

SH Hwang, BN Somatilaka, K White, S Mukhopadhyay - Elife, 2021 - elifesciences.org
The role of compartmentalized signaling in primary cilia during tissue morphogenesis is not
well understood. The cilia localized G protein-coupled receptor, Gpr161, represses …

Positive regulation of Hedgehog signaling via phosphorylation of GLI2/GLI3 by DYRK2 kinase

S Yoshida, A Kawamura, K Aoki… - Proceedings of the …, 2024 - pnas.org
Hedgehog (Hh) signaling, an evolutionarily conserved pathway, plays an essential role in
development and tumorigenesis, making it a promising drug target. Multiple negative …

A cAMP signalosome in primary cilia drives gene expression and kidney cyst formation

JN Hansen, F Kaiser, P Leyendecker, B Stüven… - EMBO …, 2022 - embopress.org
The primary cilium constitutes an organelle that orchestrates signal transduction
independently from the cell body. Dysregulation of this intricate molecular architecture leads …