ArticleeLife2020
LUZP1, a novel regulator of primary cilia and the actin cytoskeleton, is a contributing factor in Townes-Brocks Syndrome.
Article in eLife, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers.
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Who cites it
21 citing papers in PubMed, 40 citations in OpenAlex.
- Multi-Modal Kinome Profiling Discovers Mesenchymal-Like Polarity Networks that Underly Directed Hepatocellular Carcinoma Cell Migration.bioRxiv : the preprint server for biology · 2026Article
- NF‑κB‑driven LUZP1 promotes metastasis and chemoresistance in head and neck squamous cell carcinoma.Oncology reports · 2026Article
- CEP76 impairment at the centrosome-cilium interface contributes to a spectrum of ciliopathies.Science advances · 2025Article
- LUZP1 Regulates Dendritic Spine Maturation and Synaptic Plasticity in the Hippocampal Dentate Gyrus of Mice.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025Article
- TurboID Labeling and Analysis of Proteins in the Primary Cilium.Bio-protocol · 2025Article
- Extrahepatic and Circulating miR-122: Diagnostic Implications and Future Directions.MicroRNA (Shariqah, United Arab Emirates) · 2025Review
- Actin cytoskeletal regulation of ciliogenesis in development and disease.Developmental dynamics : an official publication of the American Association of Anatomists · 2024Review
- Disruption of KLHL6 Fuels Oncogenic Antigen Receptor Signaling in B-Cell Lymphoma.Blood cancer discovery · 2024Article
- Leucine zipper protein 1 attenuates pressure overload-induced cardiac hypertrophy through inhibiting Stat3 signaling.Journal of advanced research · 2024Article
- LUZP1 regulates the maturation of contractile actomyosin bundles.Cellular and molecular life sciences : CMLS · 2024Article
- Maternal and Paternal Dietary Quality and Dietary Inflammation Associations with Offspring DNA Methylation and Epigenetic Biomarkers of Aging in the Lifeways Cross-Generation Study.The Journal of nutrition · 2023Article
- Primary cilia and actin regulatory pathways in renal ciliopathies.Frontiers in nephrology · 2023Review
- Circular RNA circFIRRE drives osteosarcoma progression and metastasis through tumorigenic-angiogenic coupling.Molecular cancer · 2022Article
- Identification of STAU1 as a regulator of HBV replication by TurboID-based proximity labeling.iScience · 2022Article
- Roles of the actin cytoskeleton in ciliogenesis.Journal of cell science · 2022Review
- Proximity Biotin Labeling Reveals Kaposi's Sarcoma-Associated Herpesvirus Interferon Regulatory Factor Networks.Journal of virology · 2021Article
- The Circ_0001367/miR-545-3p/LUZP1 Axis Regulates Cell Proliferation, Migration and Invasion in Glioma Cells.Frontiers in oncology · 2021Article
- SALL1 Modulates CBX4 Stability, Nuclear Bodies, and Regulation of Target Genes.Frontiers in cell and developmental biology · 2021Article
- LUZP1 Controls Cell Division, Migration and Invasion Through Regulation of the Actin Cytoskeleton.Frontiers in cell and developmental biology · 2021Article
- Freeing the brake: Proliferation needs primary cilium to disassemble.Journal of biosciences · 2020Review
Corrections and comments
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Authors and funding
18 authors at 6 institutions in 2 countries.
Funding
Abstract
Primary cilia are sensory organelles crucial for cell signaling during development and organ homeostasis. Cilia arise from centrosomes and their formation and function is governed by numerous factors. Through our studies on Townes-Brocks Syndrome (TBS), a rare disease linked to abnormal cilia formation in human fibroblasts, we uncovered the leucine-zipper protein LUZP1 as an interactor of truncated SALL1, a dominantly-acting protein causing the disease. Using TurboID proximity labeling and pulldowns, we show that LUZP1 associates with factors linked to centrosome and actin filaments. Here, we show that LUZP1 is a cilia regulator. It localizes around the centrioles and to actin cytoskeleton. Loss of LUZP1 reduces F-actin levels, facilitates ciliogenesis and alters Sonic Hedgehog signaling, pointing to a key role in cytoskeleton-cilia interdependency. Truncated SALL1 increases the ubiquitin proteasome-mediated degradation of LUZP1. Together with other factors, alterations in LUZP1 may be contributing to TBS etiology.
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Registered trials
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