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  1. Article ; Online: Intraflagellar transport: A critical player in photoreceptor development and the pathogenesis of retinal degenerative diseases.

    Gupta, Mohona / Pazour, Gregory J

    Cytoskeleton (Hoboken, N.J.)

    2023  

    Abstract: In vertebrate vision, photons are detected by highly specialized sensory cilia called outer segments. Photoreceptor outer segments form by remodeling the membrane of a primary cilium into a stack of flattened disks. Intraflagellar transport (IFT) is ... ...

    Abstract In vertebrate vision, photons are detected by highly specialized sensory cilia called outer segments. Photoreceptor outer segments form by remodeling the membrane of a primary cilium into a stack of flattened disks. Intraflagellar transport (IFT) is critical to the formation of most types of eukaryotic cilia including the outer segments. This review covers the state of knowledge of the role of IFT in the formation and maintenance of outer segments and the human diseases that result from mutations in genes encoding the IFT complex and associated motors.
    Language English
    Publishing date 2023-12-23
    Publishing country United States
    Document type Journal Article ; Review
    ZDB-ID 2534372-5
    ISSN 1949-3592 ; 1949-3584
    ISSN (online) 1949-3592
    ISSN 1949-3584
    DOI 10.1002/cm.21823
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    In stock of ZB MED Cologne/Königswinter

    Zs.A 1840: Show issues Location:
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  2. Article ; Online: Arih2 regulates Hedgehog signaling through smoothened ubiquitylation and ER-associated degradation.

    Lv, Bo / Zhang, Xiao-Ou / Pazour, Gregory J

    Journal of cell science

    2022  Volume 135, Issue 16

    Abstract: During Hedgehog signaling, the ciliary levels of Ptch1 and Smo are regulated by the pathway. At the basal state, Ptch1 localizes to cilia and prevents the ciliary accumulation and activation of Smo. Upon binding a Hedgehog ligand, Ptch1 exits cilia, ... ...

    Abstract During Hedgehog signaling, the ciliary levels of Ptch1 and Smo are regulated by the pathway. At the basal state, Ptch1 localizes to cilia and prevents the ciliary accumulation and activation of Smo. Upon binding a Hedgehog ligand, Ptch1 exits cilia, relieving inhibition of Smo. Smo then concentrates in cilia, becomes activated and activates downstream signaling. Loss of the ubiquitin E3 ligase Arih2 elevates basal Hedgehog signaling, elevates the cellular level of Smo and increases basal levels of ciliary Smo. Mice express two isoforms of Arih2 with Arih2α found primarily in the nucleus and Arih2β found on the cytoplasmic face of the endoplasmic reticulum (ER). Re-expression of ER-localized Arih2β but not nuclear-localized Arih2α rescues the Arih2 mutant phenotypes. When Arih2 is defective, protein aggregates accumulate in the ER and the unfolded protein response is activated. Arih2β appears to regulate the ER-associated degradation (ERAD) of Smo preventing excess and potentially misfolded Smo from reaching the cilium and interfering with pathway regulation.
    MeSH term(s) Animals ; Cilia/metabolism ; Endoplasmic Reticulum-Associated Degradation ; Hedgehog Proteins/metabolism ; Mice ; Receptors, G-Protein-Coupled/genetics ; Receptors, G-Protein-Coupled/metabolism ; Signal Transduction/physiology ; Smoothened Receptor/genetics ; Smoothened Receptor/metabolism ; Ubiquitin-Protein Ligases/metabolism ; Ubiquitination
    Chemical Substances Hedgehog Proteins ; Receptors, G-Protein-Coupled ; Smoothened Receptor ; Arih2 protein, mouse (EC 2.3.2.27) ; Ubiquitin-Protein Ligases (EC 2.3.2.27)
    Language English
    Publishing date 2022-08-22
    Publishing country England
    Document type Journal Article ; Research Support, N.I.H., Extramural
    ZDB-ID 2993-2
    ISSN 1477-9137 ; 0021-9533
    ISSN (online) 1477-9137
    ISSN 0021-9533
    DOI 10.1242/jcs.260299
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    In stock of ZB MED Cologne/Königswinter

    Zs.A 1200: Show issues Location:
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    Zs.MG 14: Show issues

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  3. Article ; Online: Ciliary Doublet Microtubules at Near-Atomic Resolution.

    Pazour, Gregory J / King, Stephen M

    Cell

    2019  Volume 179, Issue 4, Page(s) 805–807

    Abstract: The beauty of the eukaryotic cilium has been appreciated since electron microscopy first revealed its 9-fold symmetry. In this issue of Cell, Ma et al. use cryoelectron microscopy and modeling to define doublet microtubules at near-atomic resolution, ... ...

    Abstract The beauty of the eukaryotic cilium has been appreciated since electron microscopy first revealed its 9-fold symmetry. In this issue of Cell, Ma et al. use cryoelectron microscopy and modeling to define doublet microtubules at near-atomic resolution, revealing an intricate array of proteins decorating the inner and outer surfaces.
    MeSH term(s) Cilia ; Cryoelectron Microscopy ; Microscopy, Electron ; Microtubules ; Proteins
    Chemical Substances Proteins
    Language English
    Publishing date 2019-10-29
    Publishing country United States
    Document type Journal Article ; Comment
    ZDB-ID 187009-9
    ISSN 1097-4172 ; 0092-8674
    ISSN (online) 1097-4172
    ISSN 0092-8674
    DOI 10.1016/j.cell.2019.10.013
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    In stock of ZB MED Cologne/Königswinter

    Zs.A 1167: Show issues Location:
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    Zs.MG 58: Show issues

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  4. Article ; Online: X Caps the Phosphate for Phospho-Rab GTPase Recognition in Ciliogenesis and Parkinson's Disease.

    Struck, Michael W / Pazour, Gregory J / Lambright, David G

    Structure (London, England : 1993)

    2020  Volume 28, Issue 4, Page(s) 385–387

    Abstract: Some Rab GTPases, after activation by GDP to GTP exchange, are phosphorylated by the LRRK2 kinase implicated in Parkinson's disease. In the current issue of Structure, Waschbüsch et al. (2020) investigate the structural basis for recognition of active ... ...

    Abstract Some Rab GTPases, after activation by GDP to GTP exchange, are phosphorylated by the LRRK2 kinase implicated in Parkinson's disease. In the current issue of Structure, Waschbüsch et al. (2020) investigate the structural basis for recognition of active phospho-Rab GTPases by the RH2 domain of the effector protein RILPL2.
    MeSH term(s) Humans ; Leucine-Rich Repeat Serine-Threonine Protein Kinase-2 ; Parkinson Disease ; Phosphates ; Phosphorylation ; rab GTP-Binding Proteins
    Chemical Substances Phosphates ; LRRK2 protein, human (EC 2.7.11.1) ; Leucine-Rich Repeat Serine-Threonine Protein Kinase-2 (EC 2.7.11.1) ; RAB8A protein, human (EC 3.6.1.-.) ; rab GTP-Binding Proteins (EC 3.6.5.2)
    Language English
    Publishing date 2020-04-07
    Publishing country United States
    Document type Journal Article ; Comment
    ZDB-ID 1213087-4
    ISSN 1878-4186 ; 0969-2126
    ISSN (online) 1878-4186
    ISSN 0969-2126
    DOI 10.1016/j.str.2020.03.005
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    In stock of ZB MED Cologne/Königswinter

    Zs.A 4141: Show issues Location:
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  5. Article: The Tumor-Associated Calcium Signal Transducer 2 (TACSTD2) oncogene is upregulated in pre-cystic epithelial cells revealing a new target for polycystic kidney disease.

    Smith, Abigail O / Frantz, William Tyler / Preval, Kenley M / Edwards, Yvonne J K / Ceol, Craig J / Jonassen, Julie A / Pazour, Gregory J

    medRxiv : the preprint server for health sciences

    2023  

    Abstract: Polycystic kidney disease (PKD) is an important cause of end stage renal disease, but treatment options are limited. While later stages of the disease have been extensively studied, mechanisms driving the initial conversion of renal tubules into cysts ... ...

    Abstract Polycystic kidney disease (PKD) is an important cause of end stage renal disease, but treatment options are limited. While later stages of the disease have been extensively studied, mechanisms driving the initial conversion of renal tubules into cysts are not understood. To identify factors that promote the initiation of cysts we deleted polycystin-2 (
    One sentence summary: The oncogene, tumor-associated calcium signal transducer 2 (Tacstd2) mRNA increased in abundance shortly after Pkd2 loss and may be a driver of cyst initiation in polycystic kidney disease.
    Language English
    Publishing date 2023-12-04
    Publishing country United States
    Document type Preprint
    DOI 10.1101/2023.12.04.23299387
    Database MEDical Literature Analysis and Retrieval System OnLINE

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  6. Article ; Online: Rab34 is necessary for early stages of intracellular ciliogenesis.

    Stuck, Michael W / Chong, Weng Man / Liao, Jung-Chi / Pazour, Gregory J

    Current biology : CB

    2021  Volume 31, Issue 13, Page(s) 2887–2894.e4

    Abstract: Primary cilia are sensory organelles present on most vertebrate cells and are critical for development and health. Ciliary dysfunction is associated with a large class of human pathologies collectively known as ciliopathies. These include cystic kidneys, ...

    Abstract Primary cilia are sensory organelles present on most vertebrate cells and are critical for development and health. Ciliary dysfunction is associated with a large class of human pathologies collectively known as ciliopathies. These include cystic kidneys, blindness, obesity, skeletal malformations, and other organ anomalies. Using a proximity biotinylation with Ift27 as bait, we identified the small guanosine triphosphatase (GTPase) Rab34 as a ciliary protein. Rab34 localizes to the centrosomes near the mother centriole, the axoneme of developed cilia, and highly dynamic tubule structures in the centrosomal region. Rab34 is required for cilia formation in fibroblasts, where we find that Rab34 loss blocks ciliogenesis at an early step of ciliary vesicle formation. In inner medullary collecting duct (IMCD3) epithelial cells, the requirement is more complex, with Rab34 needed in cells grown at low density but becoming less important as cell density increases. Ciliogenesis can proceed by an internal pathway where cilia form in the cytoplasm before being displayed on the ciliary surface or cilia can assemble by an external pathway where the centriole docks on the plasma membrane before ciliary assembly. Fibroblasts are thought to use the internal pathway, although IMCD3 cells are thought to use the external pathway. However, we find that IMCD3 cells can use the internal assembly pathway and significant numbers of internally assembling cilia are observed in low-density cells. Together, our work indicates that Rab34 is required for internal assembly of cilia, but not for cilia built on the cell surface.
    MeSH term(s) Animals ; Axoneme/metabolism ; Cell Line ; Centrioles/metabolism ; Centrosome/metabolism ; Cilia/metabolism ; Epithelial Cells/cytology ; Epithelial Cells/metabolism ; Mice ; rab GTP-Binding Proteins/metabolism
    Chemical Substances Rab34 protein, mouse (EC 3.6.1.-) ; rab GTP-Binding Proteins (EC 3.6.5.2)
    Language English
    Publishing date 2021-05-13
    Publishing country England
    Document type Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 1071731-6
    ISSN 1879-0445 ; 0960-9822
    ISSN (online) 1879-0445
    ISSN 0960-9822
    DOI 10.1016/j.cub.2021.04.018
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    In stock of ZB MED Cologne/Königswinter

    Zs.A 3208: Show issues Location:
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    Jg. 1995 - 2021: Lesesall (2.OG)
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  7. Article: Allelic Diversity in the Serum Amyloid A2 Gene and Amyloid A Amyloidosis in a Breeding Colony of Zebra Finches (

    Shientag, Lisa J / Cabrera, Oscar A / Pazour, Gregory J

    Comparative medicine

    2019  Volume 69, Issue 5, Page(s) 425–431

    Abstract: A high incidence of amyloid A (AA) amyloidosis was observed in the research breeding colony of zebra finches at our institution. Some birds with hepatic AA amyloidosis were asymptomatic for comorbid conditions frequently associated with the development ... ...

    Abstract A high incidence of amyloid A (AA) amyloidosis was observed in the research breeding colony of zebra finches at our institution. Some birds with hepatic AA amyloidosis were asymptomatic for comorbid conditions frequently associated with the development of AA amyloidosis, whereas other birds with comorbid conditions failed to develop AA amyloidosis, suggesting a potential genetic component to the disease. Sequencing the
    MeSH term(s) Amyloidosis/diagnosis ; Amyloidosis/genetics ; Animals ; Female ; Finches ; Male ; Serum Amyloid A Protein/metabolism
    Chemical Substances Serum Amyloid A Protein
    Language English
    Publishing date 2019-08-28
    Publishing country United States
    Document type Journal Article ; Research Support, Non-U.S. Gov't
    ZDB-ID 2006425-1
    ISSN 1532-0820 ; 0023-6764
    ISSN 1532-0820 ; 0023-6764
    DOI 10.30802/AALAS-CM-18-000139
    Database MEDical Literature Analysis and Retrieval System OnLINE

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    Zs.B 601: Show issues Location:
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  8. Article: Ciliary Doublet Microtubules at Near-Atomic Resolution

    Pazour, Gregory J / King, Stephen M

    Cell. 2019 Oct. 31, v. 179, no. 4

    2019  

    Abstract: The beauty of the eukaryotic cilium has been appreciated since electron microscopy first revealed its 9-fold symmetry. In this issue of Cell, Ma et al. use cryoelectron microscopy and modeling to define doublet microtubules at near-atomic resolution, ... ...

    Abstract The beauty of the eukaryotic cilium has been appreciated since electron microscopy first revealed its 9-fold symmetry. In this issue of Cell, Ma et al. use cryoelectron microscopy and modeling to define doublet microtubules at near-atomic resolution, revealing an intricate array of proteins decorating the inner and outer surfaces.
    Keywords cryo-electron microscopy ; microtubules ; proteins
    Language English
    Dates of publication 2019-1031
    Size p. 805-807.
    Publishing place Elsevier Inc.
    Document type Article
    ZDB-ID 187009-9
    ISSN 1097-4172 ; 0092-8674
    ISSN (online) 1097-4172
    ISSN 0092-8674
    DOI 10.1016/j.cell.2019.10.013
    Database NAL-Catalogue (AGRICOLA)

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    In stock of ZB MED Bonn / Germany

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  9. Article ; Online: c-JUN n-Terminal Kinase (JNK) Signaling in Autosomal Dominant Polycystic Kidney Disease.

    Smith, Abigail O / Jonassen, Julie A / Preval, Kenley M / Davis, Roger J / Pazour, Gregory J

    Journal of cellular signaling

    2021  Volume 3, Issue 1, Page(s) 62–78

    Abstract: Polycystic kidney disease is an inherited degenerative disease in which the uriniferous tubules are replaced by expanding fluid-filled cysts that ultimately destroy organ function. Autosomal dominant polycystic kidney disease (ADPKD) is the most common ... ...

    Abstract Polycystic kidney disease is an inherited degenerative disease in which the uriniferous tubules are replaced by expanding fluid-filled cysts that ultimately destroy organ function. Autosomal dominant polycystic kidney disease (ADPKD) is the most common form, afflicting approximately 1 in 1,000 people and is caused by mutations in the transmembrane proteins polycystin-1 (Pkd1) and polycystin-2 (Pkd2). The mechanisms by which polycystin mutations induce cyst formation are not well understood, however pro-proliferative signaling must be involved for tubule epithelial cell number to increase over time. We recently found that the stress-activated mitogen-activated protein kinase (MAPK) pathway c-Jun N-terminal kinase (JNK) pathway is activated in cystic disease and genetically removing JNK reduces cyst growth driven by a loss of Pkd2. This review covers the current state of knowledge of signaling in ADPKD with an emphasis on the JNK pathway.
    Language English
    Publishing date 2021-05-27
    Publishing country United States
    Document type Journal Article
    ZDB-ID 3040876-3
    ISSN 2692-0638 ; 2692-0638
    ISSN (online) 2692-0638
    ISSN 2692-0638
    DOI 10.33696/Signaling.3.068
    Database MEDical Literature Analysis and Retrieval System OnLINE

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  10. Article: Loss of Primary Cilia Protein IFT20 Dysregulates Lymphatic Vessel Patterning in Development and Inflammation.

    Paulson, Delayna / Harms, Rebecca / Ward, Cody / Latterell, Mackenzie / Pazour, Gregory J / Fink, Darci M

    Frontiers in cell and developmental biology

    2021  Volume 9, Page(s) 672625

    Abstract: Microenvironmental signals produced during development or inflammation stimulate lymphatic endothelial cells to undergo lymphangiogenesis, in which they sprout, proliferate, and migrate to expand the vascular network. Many cell types detect changes in ... ...

    Abstract Microenvironmental signals produced during development or inflammation stimulate lymphatic endothelial cells to undergo lymphangiogenesis, in which they sprout, proliferate, and migrate to expand the vascular network. Many cell types detect changes in extracellular conditions via primary cilia, microtubule-based cellular protrusions that house specialized membrane receptors and signaling complexes. Primary cilia are critical for receipt of extracellular cues from both ligand-receptor pathways and physical forces such as fluid shear stress. Here, we report the presence of primary cilia on immortalized mouse and primary adult human dermal lymphatic endothelial cells
    Language English
    Publishing date 2021-05-14
    Publishing country Switzerland
    Document type Journal Article
    ZDB-ID 2737824-X
    ISSN 2296-634X
    ISSN 2296-634X
    DOI 10.3389/fcell.2021.672625
    Database MEDical Literature Analysis and Retrieval System OnLINE

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