Targeting E3 Ubiquitin Ligases and Deubiquitinases in Ciliopathy and Cancer

Emerging Roles and Research Tools of Atypical Ubiquitination

(2020) Qiuling Huang et al.   PROTEOMICS

The primary cilium: Its role as a tumor suppressor organelle

(2020) Estanislao Peixoto et al.   BIOCHEMICAL PHARMACOLOGY

Small molecules that target the ubiquitin system

(2020) Hai Qiu Wu et al.   BIOCHEMICAL SOCIETY TRANSACTIONS

Prdx1 promotes the loss of primary cilia in esophageal squamous cell carcinoma

(2020) Qiongzhen Chen et al.   BMC CANCER

TACC3 promotes prostate cancer cell proliferation and restrains primary cilium formation

(2020) Yunkai Qie et al.   EXPERIMENTAL CELL RESEARCH

Cilia signaling and obesity

(2020) Staci E. Engle et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

Regulation of Deubiquitinating Enzymes by Post-Translational Modifications

(2020) Tanuza Das et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

Autosomal dominant polycystic kidney disease: Disrupted pathways and potential therapeutic interventions

(2019) Talieh Malekshahabi et al.   JOURNAL OF CELLULAR PHYSIOLOGY

Spatial proteomics: a powerful discovery tool for cell biology

(2019) Emma Lundberg et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

Primary cilia and their role in cancer (Review)

(2019) Michael Higgins et al.   Oncology Letters

Using Ubiquitin Binders to Decipher the Ubiquitin Code

(2019) Michael Mattern et al.   TRENDS IN BIOCHEMICAL SCIENCES

Cellular signalling by primary cilia in development, organ function and disease

(2019) Zeinab Anvarian et al.   Nature Reviews Nephrology

Autosomal dominant polycystic kidney disease

(2019) Emilie Cornec-Le Gall et al.   LANCET

Tubastatin�A, an inhibitor of HDAC6, enhances temozolomide‑induced apoptosis and reverses the malignant phenotype of glioblastoma cells

(2019) Alejandro Urdiciain et al.   INTERNATIONAL JOURNAL OF ONCOLOGY

Primary Cilium in Cancer Hallmarks

(2019) Lucilla Fabbri et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

Hypoxia/pseudohypoxia‐mediated activation of hypoxia‐inducible factor‐1α in cancer

(2019) Yoshihiro Hayashi et al.   CANCER SCIENCE

Malformations of Cerebral Cortex Development: Molecules and Mechanisms

(2019) Gordana Juric-Sekhar et al.   Annual Review of Pathology-Mechanisms of Disease

Assembling the adipose organ: adipocyte lineage segregation and adipogenesis in vivo

(2019) Zachary L. Sebo et al.   DEVELOPMENT

Identification of a USP9X Substrate NFX1-123 by SILAC-based Quantitative Proteomics

(2019) Xiangling Chen et al.   JOURNAL OF PROTEOME RESEARCH

Genome-Wide CRISPR-Cas9 Screens Expose Genetic Vulnerabilities and Mechanisms of Temozolomide Sensitivity in Glioblastoma Stem Cells

(2019) Graham MacLeod et al.   Cell Reports

CYLD Regulates Centriolar Satellites Proteostasis by Counteracting the E3 Ligase MIB1

(2019) Tiphaine Douanne et al.   Cell Reports

Primary cilia: biosensors of the male reproductive tract

(2019) Laura Girardet et al.   Andrology

The photoreceptor cilium and its diseases

(2019) Ruxandra Bachmann-Gagescu et al.   CURRENT OPINION IN GENETICS & DEVELOPMENT

Phosphorylation of USP15 and USP4 Regulates Localization and Spliceosomal Deubiquitination

(2019) Tanuza Das et al.   JOURNAL OF MOLECULAR BIOLOGY

Renal ciliopathies

(2019) Laura A Devlin et al.   CURRENT OPINION IN GENETICS & DEVELOPMENT

Making and breaking symmetry in development, growth and disease

(2019) Daniel T. Grimes   DEVELOPMENT

Sperm defects in primary ciliary dyskinesia and related causes of male infertility

(2019) Anu Sironen et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Subquinocin, a small molecule inhibitor of CYLD and USP-family deubiquitinating enzymes, promotes NF-κB signaling

(2019) Satoshi Yamanaka et al.   BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS

Ubiquitin-specific protease 8 (USP8/UBPy): a prototypic multidomain deubiquitinating enzyme with pleiotropic functions

(2019) Almut Dufner et al.   BIOCHEMICAL SOCIETY TRANSACTIONS

Cilia in cystic kidney and other diseases

(2019) Gregory J. Pazour et al.   CELLULAR SIGNALLING

Primary Cilia in Brain Development and Diseases

(2018) Yong Ha Youn et al.   AMERICAN JOURNAL OF PATHOLOGY

The cholangiocyte primary cilium in health and disease

(2018) Adrian P. Mansini et al.   BIOCHIMICA ET BIOPHYSICA ACTA-MOLECULAR BASIS OF DISEASE

EZH2-Mediated Primary Cilium Deconstruction Drives Metastatic Melanoma Formation

(2018) Daniel Zingg et al.   CANCER CELL

Inhibitors of protein–protein interactions (PPIs): an analysis of scaffold choices and buried surface area

(2018) Xu Ran et al.   CURRENT OPINION IN CHEMICAL BIOLOGY

The E3 ubiquitin ligase UBR5 regulates centriolar satellite stability and primary cilia

(2018) Robert F. Shearer et al.   MOLECULAR BIOLOGY OF THE CELL

Proteolysis to Identify Protease Substrates: Cleave to Decipher

(2018) Sonali R. Bhagwat et al.   PROTEOMICS

Ubiquitin E3 Ligase MARCH7 promotes proliferation and invasion of cervical cancer cells through VAV2‑RAC1‑CDC42 pathway

(2018) Jianguo Hu et al.   Oncology Letters

EGF receptor kinase suppresses ciliogenesis through activation of USP8 deubiquitinase

(2018) Kousuke Kasahara et al.   Nature Communications

Cilium structure, assembly, and disassembly regulated by the cytoskeleton

(2018) Mary Mirvis et al.   BIOCHEMICAL JOURNAL

The regulation of cilium assembly and disassembly in development and disease

(2018) Lei Wang et al.   DEVELOPMENT

The role of ubiquitination in the regulation of primary cilia assembly and disassembly

(2018) Delowar Hossain et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

The deubiquitinating enzyme USP14 controls ciliogenesis and hedgehog signalling

(2018) Massa Filomena et al.   HUMAN MOLECULAR GENETICS

Primary Cilia as Signaling Hubs in Health and Disease

(2018) Yuhei Nishimura et al.   Advanced Science

Stabilization of protein-protein interactions in drug discovery

(2017) Sebastian A. Andrei et al.   Expert Opinion on Drug Discovery

Mechanisms of regulation and diversification of deubiquitylating enzyme function

(2017) Pawel Leznicki et al.   JOURNAL OF CELL SCIENCE

Deubiquitylating enzymes and drug discovery: emerging opportunities

(2017) Jeanine A. Harrigan et al.   NATURE REVIEWS DRUG DISCOVERY

Genes and molecular pathways underpinning ciliopathies

(2017) Jeremy F. Reiter et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

HDAC6 inhibition suppresses chondrosarcoma by restoring the expression of primary cilia

(2017) Wei Xiang et al.   ONCOLOGY REPORTS

Cilia - The sensory antennae in the eye

(2017) Helen May-Simera et al.   PROGRESS IN RETINAL AND EYE RESEARCH

Primary cilia: Cell and molecular mechanosensors directing whole tissue function

(2017) Milos Spasic et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

The Cilium: Cellular Antenna and Central Processing Unit

(2017) Jarema J. Malicki et al.   TRENDS IN CELL BIOLOGY

Discovery, Diagnosis, and Etiology of Craniofacial Ciliopathies

(2017) Elizabeth N. Schock et al.   Cold Spring Harbor Perspectives in Biology

Cilia and Obesity

(2017) Christian Vaisse et al.   Cold Spring Harbor Perspectives in Biology

Photoreceptor Cilia and Retinal Ciliopathies

(2017) Kinga M. Bujakowska et al.   Cold Spring Harbor Perspectives in Biology

USP9X counteracts differential ubiquitination of NPHP5 by MARCH7 and BBS11 to regulate ciliogenesis

(2017) Arindam Das et al.   PLoS Genetics

Primary Ciliary Dyskinesia: An Update on Clinical Aspects, Genetics, Diagnosis, and Future Treatment Strategies

(2017) Virginia Mirra et al.   Frontiers in Pediatrics

Identification of Elongated Primary Cilia with Impaired Mechanotransduction in Idiopathic Scoliosis Patients

(2017) Niaz Oliazadeh et al.   Scientific Reports

De Novo Loss-of-Function Mutations in USP9X Cause a Female-Specific Recognizable Syndrome with Developmental Delay and Congenital Malformations

(2016) Margot R.F. Reijnders et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Recent advances in primary ciliary dyskinesia

(2016) Kazuhiko Takeuchi et al.   AURIS NASUS LARYNX

Systematic approaches to identify E3 ligase substrates

(2016) M. Iconomou et al.   BIOCHEMICAL JOURNAL

Regulation of primary cilia formation by the ubiquitin–proteasome system

(2016) Robert F. Shearer et al.   BIOCHEMICAL SOCIETY TRANSACTIONS

Genome-wide screen identifies novel machineries required for both ciliogenesis and cell cycle arrest upon serum starvation

(2016) Ji Hyun Kim et al.   BIOCHIMICA ET BIOPHYSICA ACTA-MOLECULAR CELL RESEARCH

The cilium: a cellular antenna with an influence on obesity risk

(2016) Edwin C. M. Mariman et al.   BRITISH JOURNAL OF NUTRITION

Drugging the undruggables: exploring the ubiquitin system for drug development

(2016) Xiaodong Huang et al.   CELL RESEARCH

Mechanisms of ciliogenesis suppression in dividing cells

(2016) Hidemasa Goto et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Mechanism of ciliary disassembly

(2016) Yinwen Liang et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

CPAP promotes timely cilium disassembly to maintain neural progenitor pool

(2016) Elke Gabriel et al.   EMBO JOURNAL

HDAC2 promotes loss of primary cilia in pancreatic ductal adenocarcinoma

(2016) Tetsuo Kobayashi et al.   EMBO REPORTS

Ndel1 suppresses ciliogenesis in proliferating cells by regulating the trichoplein–Aurora A pathway

(2016) Hironori Inaba et al.   JOURNAL OF CELL BIOLOGY

CYLD – a deubiquitylase that acts to fine-tune microtubule properties and functions

(2016) Yunfan Yang et al.   JOURNAL OF CELL SCIENCE

Motile and non-motile cilia in human pathology: from function to phenotypes

(2016) Hannah M Mitchison et al.   JOURNAL OF PATHOLOGY

USP8 suppresses death receptor-mediated apoptosis by enhancing FLIPL stability

(2016) M Jeong et al.   ONCOGENE

Genetics and biology of primary ciliary dyskinesia

(2016) Amjad Horani et al.   Paediatric Respiratory Reviews

Cilia in vertebrate left–right patterning

(2016) Agnik Dasgupta et al.   PHILOSOPHICAL TRANSACTIONS OF THE ROYAL SOCIETY B-BIOLOGICAL SCIENCES

Glia–neuron interactions in the mammalian retina

(2016) Elena Vecino et al.   PROGRESS IN RETINAL AND EYE RESEARCH

Axoneme Structure from Motile Cilia

(2016) Takashi Ishikawa   Cold Spring Harbor Perspectives in Biology

The Central Apparatus of Cilia and Eukaryotic Flagella

(2016) Thomas D. Loreng et al.   Cold Spring Harbor Perspectives in Biology

Ciliopathies

(2016) Daniela A. Braun et al.   Cold Spring Harbor Perspectives in Biology

Tethering of an E3 ligase by PCM1 regulates the abundance of centrosomal KIAA0586/Talpid3 and promotes ciliogenesis

(2016) Lei Wang et al.   eLife

Metabolic Regulation and Energy Homeostasis through the Primary Cilium

(2015) Edwin C. Oh et al.   Cell Metabolism

La FAM fatale: USP9X in development and disease

(2015) Mariyam Murtaza et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Proteomics of Primary Cilia by Proximity Labeling

(2015) David U. Mick et al.   DEVELOPMENTAL CELL

The tumor suppressor FBW7 controls ciliary length

(2015) A. S. Nikonova et al.   EMBO JOURNAL

Cell cycle-dependent ubiquitylation and destruction of NDE1 by CDK5-FBW7 regulates ciliary length

(2015) D. Maskey et al.   EMBO JOURNAL

Tubulin transport by IFT is upregulated during ciliary growth by a cilium-autonomous mechanism

(2015) Julie M. Craft et al.   JOURNAL OF CELL BIOLOGY

The Prognostic Role of Ki-67/MIB-1 in Upper Urinary-Tract Urothelial Carcinomas: A Systematic Review and Meta-Analysis

(2015) Ye Lei et al.   JOURNAL OF ENDOUROLOGY

Identification of Salmonella Typhimurium Deubiquitinase SseL Substrates by Immunoaffinity Enrichment and Quantitative Proteomic Analysis

(2015) Ernesto S. Nakayasu et al.   JOURNAL OF PROTEOME RESEARCH

An siRNA-based functional genomics screen for the identification of regulators of ciliogenesis and ciliopathy genes

(2015) Gabrielle Wheway et al.   NATURE CELL BIOLOGY

Adipogenic Differentiation of hMSCs is Mediated by Recruitment of IGF-1r Onto the Primary Cilium Associated With Cilia Elongation

(2015) Melis T. Dalbay et al.   STEM CELLS

The demographics of the ubiquitin system

(2015) Michael J. Clague et al.   TRENDS IN CELL BIOLOGY

Developmental disruptions underlying brain abnormalities in ciliopathies

(2015) Jiami Guo et al.   Nature Communications

TALPID3 controls centrosome and cell polarity and the human ortholog KIAA0586 is mutated in Joubert syndrome (JBTS23)

(2015) Louise A Stephen et al.   eLife

Unmasking the ciliopathies: craniofacial defects and the primary cilium

(2015) Claudio R. Cortés et al.   Wiley Interdisciplinary Reviews-Developmental Biology

CYLD mediates ciliogenesis in multiple organs by deubiquitinating Cep70 and inactivating HDAC6

(2014) Yunfan Yang et al.   CELL RESEARCH

Tubulin glycylases are required for primary cilia, control of cell proliferation and tumor development in colon

(2014) C. Rocha et al.   EMBO JOURNAL

INPP5E interacts with AURKA, linking phosphoinositide signaling to primary cilium stability

(2014) O. V. Plotnikova et al.   JOURNAL OF CELL SCIENCE

Leptin-promoted cilia assembly is critical for normal energy balance

(2014) Yu Mi Han et al.   JOURNAL OF CLINICAL INVESTIGATION

Centriole amplification by mother and daughter centrioles differs in multiciliated cells

(2014) Adel Al Jord et al.   NATURE

Ubiquitination in disease pathogenesis and treatment

(2014) Doris Popovic et al.   NATURE MEDICINE

TGFβ signaling in establishing left–right asymmetry

(2014) Hidetaka Shiratori et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

Ubiquitin-proteasome system controls ciliogenesis at the initial step of axoneme extension

(2014) Kousuke Kasahara et al.   Nature Communications

From the cytoplasm into the cilium: Bon voyage

(2014) Jarema Malicki et al.   Organogenesis

HDAC6 Inhibition Restores Ciliary Expression and Decreases Tumor Growth

(2013) S. A. Gradilone et al.   CANCER RESEARCH

Cell cycle progression by the repression of primary cilia formation in proliferating cells

(2013) Hidemasa Goto et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Cluap1 localizes preferentially to the base and tip of cilia and is required for ciliogenesis in the mouse embryo

(2013) Yanick Botilde et al.   DEVELOPMENTAL BIOLOGY

Pathogenic NPHP5 mutations impair protein interaction with Cep290, a prerequisite for ciliogenesis

(2013) Marine Barbelanne et al.   HUMAN MOLECULAR GENETICS

USP33 regulates centrosome biogenesis via deubiquitination of the centriolar protein CP110

(2013) Ji Li et al.   NATURE

Primary cilia in neurodevelopmental disorders

(2013) Enza Maria Valente et al.   Nature Reviews Neurology

CEP192 interacts physically and functionally with the K63-deubiquitinase CYLD to promote mitotic spindle assembly

(2012) Maria Ana Gomez-Ferreria et al.   CELL CYCLE

BBS-Induced Ciliary Defect Enhances Adipogenesis, Causing Paradoxical Higher-Insulin Sensitivity, Glucose Usage, and Decreased Inflammatory Response

(2012) Vincent Marion et al.   Cell Metabolism

Left–right asymmetry in zebrafish

(2012) Takaaki Matsui et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Specific Small Molecule Inhibitors of Skp2-Mediated p27 Degradation

(2012) Lily Wu et al.   CHEMISTRY & BIOLOGY

Identification of a novel Wnt5a-CK1ε-Dvl2-Plk1-mediated primary cilia disassembly pathway

(2012) Kyung Ho Lee et al.   EMBO JOURNAL

The base of the cilium: roles for transition fibres and the transition zone in ciliary formation, maintenance and compartmentalization

(2012) Jeremy F Reiter et al.   EMBO REPORTS

Trichoplein and Aurora A block aberrant primary cilia assembly in proliferating cells

(2012) Akihito Inoko et al.   JOURNAL OF CELL BIOLOGY

Comprehensive profiling of proteome changes upon sequential deletion of deubiquitylating enzymes

(2012) Jon W. Poulsen et al.   Journal of Proteomics

Centrosome Fine Ultrastructure of the Osteocyte Mechanosensitive Primary Cilium

(2012) R.E. Uzbekov et al.   MICROSCOPY AND MICROANALYSIS

Calmodulin activation of Aurora-A kinase (AURKA) is required during ciliary disassembly and in mitosis

(2012) Olga V. Plotnikova et al.   MOLECULAR BIOLOGY OF THE CELL

The deubiquitinase USP9X suppresses pancreatic ductal adenocarcinoma

(2012) Pedro A. Pérez-Mancera et al.   NATURE

Fluid flow and interlinked feedback loops establish left–right asymmetric decay of Cerl2 mRNA

(2012) Tetsuya Nakamura et al.   Nature Communications

Left–right asymmetry in the level of active Nodal protein produced in the node is translated into left–right asymmetry in the lateral plate of mouse embryos

(2011) Aiko Kawasumi et al.   DEVELOPMENTAL BIOLOGY

Trichoplein controls microtubule anchoring at the centrosome by binding to Odf2 and ninein

(2011) M. Ibi et al.   JOURNAL OF CELL SCIENCE

Ciliopathies

(2011) Friedhelm Hildebrandt et al.   NEW ENGLAND JOURNAL OF MEDICINE

A primary cilia-dependent etiology for midline facial disorders

(2010) Samantha A. Brugmann et al.   HUMAN MOLECULAR GENETICS

SCFCyclin F controls centrosome homeostasis and mitotic fidelity through CP110 degradation

(2010) Vincenzo D’Angiolella et al.   NATURE

The ubiquitin conjugation system is involved in the disassembly of cilia and flagella

(2009) Kaiyao Huang et al.   JOURNAL OF CELL BIOLOGY

Growth arrest induces primary-cilium formation and sensitizes IGF-1-receptor signaling during differentiation induction of 3T3-L1 preadipocytes

(2009) D. Zhu et al.   JOURNAL OF CELL SCIENCE

Enhanced Stat3 Activation in POMC Neurons Provokes Negative Feedback Inhibition of Leptin and InsulinSignaling in Obesity

(2009) M. B. Ernst et al.   JOURNAL OF NEUROSCIENCE

Dual and opposing roles of primary cilia in medulloblastoma development

(2009) Young-Goo Han et al.   NATURE MEDICINE

Primary cilia can both mediate and suppress Hedgehog pathway–dependent tumorigenesis

(2009) Sunny Y Wong et al.   NATURE MEDICINE

Transient ciliogenesis involving Bardet-Biedl syndrome proteins is a fundamental characteristic of adipogenic differentiation

(2009) Vincent Marion et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Intraflagellar transport and the generation of dynamic, structurally and functionally diverse cilia

(2009) Michael A. Silverman et al.   TRENDS IN CELL BIOLOGY

Left-Right Determination: Involvement of Molecular Motor KIF3, Cilia, and Nodal Flow

(2009) N. Hirokawa et al.   Cold Spring Harbor Perspectives in Biology

Tripartite Motif Protein 32 Facilitates Cell Growth and Migration via Degradation of Abl-Interactor 2

(2008) S. Kano et al.   CANCER RESEARCH

Activation of Stat3 Signaling in AgRP Neurons Promotes Locomotor Activity

(2008) Andrea Mesaros et al.   Cell Metabolism

Role of Hedgehog Signaling in Ovarian Cancer

(2008) R. Bhattacharya et al.   CLINICAL CANCER RESEARCH

Inhibition of neural crest migration underlies craniofacial dysmorphology and Hirschsprung's disease in Bardet-Biedl syndrome

(2008) J. L. Tobin et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA