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https://www.readbyqxmd.com/read/28935136/coordinating-the-uncoordinated-unc119-trafficking-in-cilia
#1
REVIEW
Francesca Jean, David Pilgrim
Constructing the distinct subcellular environment of the cilium relies in a large part upon intraflagellar transport (IFT) proteins, which traffic cargo both to and within the cilium. However, evidence from the last 10 years suggests that IFT alone is not sufficient to generate the ciliary environment. One essential factor is UNC119, which interacts with known IFT molecular switches to transport ciliary cargos. Despite its apparent importance in ciliary trafficking though, human UNC119 mutations have only rarely been associated with diseases commonly linked with ciliopathies...
September 6, 2017: European Journal of Cell Biology
https://www.readbyqxmd.com/read/28931721/neuropeptidergic-integration-of-behavior-in-trichoplax-adhaerens-an-animal-without-synapses
#2
Adriano Senatore, Thomas S Reese, Carolyn L Smith
Trichoplax adhaerens is a flat, millimeter-sized marine animal that adheres to surfaces and grazes on algae. Trichoplax displays a repertoire of different feeding behaviors despite the apparent absence of a true nervous system with electrical or chemical synapses. It glides along surfaces to find food, propelled by beating cilia on cells at its ventral surface, and pauses during feeding by arresting ciliary beating. We found that when endomorphin-like peptides are applied to an animal, ciliary beating is arrested, mimicking natural feeding pauses...
September 15, 2017: Journal of Experimental Biology
https://www.readbyqxmd.com/read/28929469/chondrocyte-expansion-is-associated-with-loss-of-primary-cilia-and-disrupted-hedgehog-signalling
#3
C L Thompson, J C Plant, A K Wann, C L Bishop, P Novak, H M Mitchison, P L Beales, J P Chapple, M M Knight
Tissue engineering-based therapies targeting cartilage diseases, such as osteoarthritis, require in vitro expansion of articular chondrocytes. A major obstacle for these therapies is the dedifferentiation and loss of phenotype accompanying chondrocyte expansion. Recent studies suggest that manipulation of hedgehog signalling may be used to promote chondrocyte re-differentiation. Hedgehog signalling requires the primary cilium, a microtubule-based signalling compartment, the integrity of which is linked to the cytoskeleton...
September 20, 2017: European Cells & Materials
https://www.readbyqxmd.com/read/28927861/primary-cilia-formation-is-diminished-in-schizophrenia-and-bipolar-disorder-a-possible-marker-for-these-psychiatric-diseases
#4
Jesús Muñoz-Estrada, Alejandra Lora-Castellanos, Isaura Meza, Salvador Alarcón Elizalde, Gloria Benítez-King
Primary cilium (PC) is a microtubule-rich organelle that protrudes from the plasma membrane and acts as a cellular antenna sensing extracellular signals during brain development. DISC1 (Disrupted-in-Schizophrenia-1) is involved in PC formation and is considered a risk factor for neuropsychiatric disorders. We have previously described altered subcellular distribution of DISC1 and an aberrant microtubule organization in olfactory neuronal precursors (ONP) obtained from schizophrenia (SCZ) and bipolar disorder (BD) patients...
September 16, 2017: Schizophrenia Research
https://www.readbyqxmd.com/read/28927423/primary-cilia-modulate-tlr4-mediated-inflammatory-responses-in-hippocampal-neurons
#5
Hyunjung Baek, Hyo Jung Shin, Jwa-Jin Kim, Nara Shin, Sena Kim, Min-Hee Yi, Enji Zhang, Jinpyo Hong, Joon Won Kang, Yonghyun Kim, Cuk-Seong Kim, Dong Woon Kim
BACKGROUND: The primary cilium is an organelle that can act as a master regulator of cellular signaling. Despite the presence of primary cilia in hippocampal neurons, their function is not fully understood. Recent studies have demonstrated that the primary cilium influences interleukin (IL)-1β-induced NF-κB signaling, ultimately mediating the inflammatory response. We, therefore, investigated ciliary function and NF-κB signaling in lipopolysaccharide (LPS)-induced neuroinflammation in conjunction with ciliary length analysis...
September 19, 2017: Journal of Neuroinflammation
https://www.readbyqxmd.com/read/28924414/cilium-incarnatum-externum
#6
Taru Dewan, Chetna Sharma, Harshika Chawla, Renu Singhania, Ananya Chatterjee
A 25-year-old woman presented with a history of a cosmetically bothersome bulge over her left eyelid. She was diagnosed with cilium incarnatum externum. The subcutaneous cilium was surgically removed and examined.
2017: Digital Journal of Ophthalmology: DJO
https://www.readbyqxmd.com/read/28916710/apc-sets-the-wnt-tone-necessary-for-cerebral-cortical-progenitor-development
#7
Naoki Nakagawa, Jingjun Li, Keiko Yabuno-Nakagawa, Tae-Yeon Eom, Martis Cowles, Tavien Mapp, Robin Taylor, E S Anton
Adenomatous polyposis coli (APC) regulates the activity of β-catenin, an integral component of Wnt signaling. However, the selective role of the APC-β-catenin pathway in cerebral cortical development is unknown. Here we genetically dissected the relative contributions of APC-regulated β-catenin signaling in cortical progenitor development, a necessary early step in cerebral cortical formation. Radial progenitor-specific inactivation of the APC-β-catenin pathway indicates that the maintenance of appropriate β-catenin-mediated Wnt tone is necessary for the orderly differentiation of cortical progenitors and the resultant formation of the cerebral cortex...
September 15, 2017: Genes & Development
https://www.readbyqxmd.com/read/28913352/new-roles-of-the-primary-cilium-in-autophagy
#8
REVIEW
Yenniffer Ávalos, Daniel Peña-Oyarzun, Mauricio Budini, Eugenia Morselli, Alfredo Criollo
The primary cilium is a nonmotile organelle that emanates from the surface of multiple cell types and receives signals from the environment to regulate intracellular signaling pathways. The presence of cilia, as well as their length, is important for proper cell function; shortened, elongated, or absent cilia are associated with pathological conditions. Interestingly, it has recently been shown that the molecular machinery involved in autophagy, the process of recycling of intracellular material to maintain cellular and tissue homeostasis, participates in ciliogenesis...
2017: BioMed Research International
https://www.readbyqxmd.com/read/28901584/the-primary-cilium-as-a-signaling-nexus-for-growth-plate-function-and-subsequent-skeletal-development
#9
REVIEW
Emily R Moore, Christopher R Jacobs
The primary cilium is a solitary, antenna-like sensory organelle with many important roles in cartilage and bone development, maintenance, and function. The primary cilium's potential role as a signaling nexus in the growth plate makes it an attractive therapeutic target for diseases and disorders associated with bone development and maintenance. Many signaling pathways that are mediated by the cilium - such as Hh, Wnt, Ihh/PTHrP, TGFβ, BMP, FGF, and Notch - are also known to influence endochondral ossification, primarily by directing growth plate formation and chondrocyte behavior...
September 13, 2017: Journal of Orthopaedic Research: Official Publication of the Orthopaedic Research Society
https://www.readbyqxmd.com/read/28900213/copine-7-binds-to-the-cell-surface-receptor-nucleolin-and-regulates-ciliogenesis-and-dspp-expression-during-odontoblast-differentiation
#10
You-Mi Seo, Su-Jin Park, Hye-Kyung Lee, Joo-Cheol Park
Tooth development is a progressive process regulated by interactions between epithelial and mesenchymal tissues. Our previous studies showed that copine-7 (Cpne7), a dental epithelium-derived protein, is a signalling molecule that is secreted by preameloblasts and regulates the differentiation of preodontoblasts into odontoblasts. However, the mechanisms involved in the translocation of Cpne7 from preameloblasts to preodontoblasts and the functions of Cpne7 during odontogenesis are poorly understood. Here, we showed that the internalization of Cpne7 was mediated primarily by caveolae...
September 12, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28887322/kinesin-2-motors-adapt-their-stepping-behavior-for-processive-transport-on-axonemes-and-microtubules
#11
Willi L Stepp, Georg Merck, Felix Mueller-Planitz, Zeynep Ökten
Two structurally distinct filamentous tracks, namely singlet microtubules in the cytoplasm and axonemes in the cilium, serve as railroads for long-range transport processes in vivo In all organisms studied so far, the kinesin-2 family is essential for long-range transport on axonemes. Intriguingly, in higher eukaryotes, kinesin-2 has been adapted to work on microtubules in the cytoplasm as well. Here, we show that heterodimeric kinesin-2 motors distinguish between axonemes and microtubules. Unlike canonical kinesin-1, kinesin-2 takes directional, off-axis steps on microtubules, but it resumes a straight path when walking on the axonemes...
September 8, 2017: EMBO Reports
https://www.readbyqxmd.com/read/28877879/uninephrectomy-and-apical-fluid-shear-stress-decrease-enac-abundance-in-collecting-duct-principal-cells
#12
Thomas Ernandez, Khalil Udwan, Alexandra Chassot, Pierre-Yves Martin, Eric Feraille
Acute nephron reduction such as after living kidney donation may increase the risk of hypertension. Uninephrectomy induces major hemodynamic changes in the remaining kidney resulting in rapid increase of single nephron GFR and fluid delivery in the distal nephron. Decreased sodium (Na) fractional reabsorption after the distal tubule has been reported after uninephrectomy in animals preserving volume homeostasis. In the present study, we thought to specifically explore the effect of unilateral nephrectomy on ENaC subunits expression in mice...
September 6, 2017: American Journal of Physiology. Renal Physiology
https://www.readbyqxmd.com/read/28877752/spen-a-new-player-in-primary-cilia-formation-and-cell-migration-in-breast-cancer
#13
Stéphanie Légaré, Catherine Chabot, Mark Basik
BACKGROUND: The primary cilium is a microtubule-based and nonmotile organelle functioning as a cellular antenna that is involved in the regulation of cell proliferation, differentiation, and migration. In breast cancer cells, the primary cilium is a structure that decreases in incidence with increasing degrees of transformation and may be biologically more important in estrogen receptor (ERα)-negative breast cancer cells. Split ends (SPEN) is an ERα corepressor that we have identified as a tumor suppressor protein in ERα-positive breast cancer cells whose hormone-independent roles in breast cancer have never been explored...
September 6, 2017: Breast Cancer Research: BCR
https://www.readbyqxmd.com/read/28871046/loss-of-ocrl-increases-ciliary-pi-4-5-p2-in-oculocerebrorenal-syndrome-of-lowe
#14
Philipp P Prosseda, Na Luo, Biao Wang, Jorge A Alvarado, Yang Hu, Yang Sun
Lowe syndrome is a rare X-linked disorder characterized by bilateral congenital cataracts and glaucoma, mental retardation, and proximal renal tubular dysfunction. Mutations in OCRL1, an inositol polyphosphate 5-phosphatase that dephosphorylates PI(4,5)P2, cause Lowe syndrome. Previously we showed that OCRL localizes to the primary cilium, which has a distinct membrane phospholipid composition, but disruption of phosphoinositides in the ciliary membrane poorly understood. Here we demonstrate that cilia from Lowe syndrome patient fibroblasts exhibit increased levels of PI(4,5)P2 and decreased levels of PI4P...
September 4, 2017: Journal of Cell Science
https://www.readbyqxmd.com/read/28870737/chondrocyte-primary-cilium-is-mechanosensitive-and-responds-to-low-intensity-ultrasound-by-altering-its-length-and-orientation
#15
Anuradha Subramanian, Gaurav Budhiraja, Neety Sahu
The primary cilium has been implicated in mechanotransduction, however, its mechanosensory role in transducing signals under low-intensity ultrasound (LIUS) which has the potential to repair fractures and cartilage, remains uninvestigated. This study examined the impact of continuous-wave US, at the cell resonance frequency of 5.0MHz and pressure amplitude of either 14 or 60kPa, on the incidence, length and orientation of primary cilium in bovine articular chondrocytes. Visualization of primary cilium with acetylated α-tubulin staining demonstrated that the primary cilium was elongated, bent under US and these changes were reversible...
September 8, 2017: International Journal of Biochemistry & Cell Biology
https://www.readbyqxmd.com/read/28868293/the-kidney-in-bardet-biedl-syndrome-possible-pathogenesis-of-urine-concentrating-defect
#16
REVIEW
Miriam Zacchia, Valentina Di Iorio, Francesco Trepiccione, Marianna Caterino, Giovambattista Capasso
BACKGROUND: The ciliopathies are a growing number of disorders caused by mutations in genes involved in the function of the primary cilium. Bardet-Biedl syndrome (BBS) belongs to this group of disorders. In this setting, kidney dysfunction is highly variable, and urine concentrating defect, a common feature of multiple ciliopathies, has been described as the most frequent defect. Here we review the mechanism of urine concentration and describe the possible mechanism underling this defect in ciliopathies and especially BBS, based on the current body of literature...
July 2017: Kidney Diseases
https://www.readbyqxmd.com/read/28854412/wide-spread-cone-shaped-epiphyses-in-two-saudi-siblings-with-ellis-van-creveld-syndrome
#17
Abeer Al-Fardan, Mohammad M Al-Qattan
INTRODUCTION: Ellis-van Creveld (EVC) syndrome is one of the rarest ciliopathy syndromes. It is caused by mutations of the EVC and EVC2 genes which encode the EVC proteins present in the basal body of the primary cilium. PRESENTATION OF CASES: We report on a Saudi family with two affected children. Gene analysis revealed a homozygous c.2T >A in exon 1 of the EVC gene. The most interesting finding in our patients was the wide - spread cone-shaped epiphyses in the hands and feet...
August 24, 2017: International Journal of Surgery Case Reports
https://www.readbyqxmd.com/read/28852774/mechanisms-of-ciliary-targeting-entering-importins-and-rabs
#18
REVIEW
Lei Lu, Viswanadh Madugula
Primary cilium is a rod-like plasma membrane protrusion that plays important roles in sensing the cellular environment and initiating corresponding signaling pathways. The sensory functions of the cilium critically depend on the unique enrichment of ciliary residents, which is maintained by the ciliary diffusion barrier. It is still unclear how ciliary cargoes specifically enter the diffusion barrier and accumulate within the cilium. In this review, the organization and trafficking mechanism of the cilium are compared to those of the nucleus, which are much better understood at the moment...
August 29, 2017: Cellular and Molecular Life Sciences: CMLS
https://www.readbyqxmd.com/read/28846093/super-resolution-microscopy-reveals-that-disruption-of-ciliary-transition-zone-architecture-causes-joubert%C3%A2-syndrome
#19
Xiaoyu Shi, Galo Garcia Iii, Julie C Van De Weghe, Ryan McGorty, Gregory J Pazour, Dan Doherty, Bo Huang, Jeremy F Reiter
Ciliopathies, including nephronophthisis (NPHP), Meckel syndrome (MKS) and Joubert syndrome (JBTS), can be caused by mutations affecting components of the transition zone, a domain near the base of the cilium that controls the protein composition of its membrane. We defined the three-dimensional arrangement of key proteins in the transition zone using two-colour stochastic optical reconstruction microscopy (STORM). NPHP and MKS complex components form nested rings comprised of nine-fold doublets. JBTS-associated mutations in RPGRIP1L or TCTN2 displace certain transition-zone proteins...
August 28, 2017: Nature Cell Biology
https://www.readbyqxmd.com/read/28843978/primary-cilia-cell-and-molecular-mechanosensors-directing-whole-tissue-function
#20
REVIEW
Milos Spasic, Christopher R Jacobs
Primary cilia are immotile, microtubule-based organelles extending from the surface of nearly every mammalian cell. Mechanical stimulation causes deflection of the primary cilium, initiating downstream signaling cascades to the rest of the cell. The cilium forms a unique subcellular microdomain, and defects in ciliary protein composition or physical structure have been associated with a myriad of human pathologies. In this review, we discuss the importance of ciliary mechanotransduction at the cell and tissue level, and how furthering our molecular understanding of primary cilia mechanobiology may lead to therapeutic strategies to treat human diseases...
August 24, 2017: Seminars in Cell & Developmental Biology
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