ArticleThe Journal of cell biology2017
The BEACH-containing protein WDR81 coordinates p62 and LC3C to promote aggrephagy.
Article in The Journal of cell biology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 38 papers.
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Who cites it
38 citing papers in PubMed, 74 citations in OpenAlex.
- WDR81 Mutation in Two Siblings: A Case Report and Review of Literature.Molecular syndromology · 2026Article
- Article
- Sex does not influence neuronal autophagosome biogenesis throughout aging in mice.Molecular biology of the cell · 2025Article
- Betaine enhances SCAPs chondrogenic differentiation and promotes cartilage repair in TMJOA through WDR81.Stem cell research & therapy · 2025Article
- Expression of Autophagy-Related Proteins in Microlissencephaly Associated with a Novel Variant in theMolecular syndromology · 2025Article
- Liquid-liquid phase separation: an emerging perspective on the tumorigenesis, progression, and treatment of tumors.Frontiers in immunology · 2025Review
- The role of autophagy in brain health and disease: Insights into exosome and autophagy interactions.Heliyon · 2024Review
- Physiological functions of ULK1/2.Journal of molecular biology · 2024Review
- LRBA, a BEACH protein mutated in human immune deficiency, is widely expressed in epithelia, exocrine and endocrine glands, and neurons.Scientific reports · 2024Article
- The mechanisms and roles of selective autophagy in mammals.Nature reviews. Molecular cell biology · 2023Review
- A Pharmacological Review of Tanshinones, Naturally Occurring Monomers fromOxidative medicine and cellular longevity · 2023Review
- LC3 subfamily in cardiolipin-mediated mitophagy: a comparison of the LC3A, LC3B and LC3C homologs.Autophagy · 2022Article
- Manipulating autophagic degradation in human diseases: from mechanisms to interventions.Life medicine · 2022Review
- The Multifaceted Roles of Autophagy in Infectious, Obstructive, and Malignant Airway Diseases.Biomedicines · 2022Review
- The caspase-6-p62 axis modulates p62 droplets based autophagy in a dominant-negative manner.Cell death and differentiation · 2022Article
- Endosomal trafficking defects alter neural progenitor proliferation and cause microcephaly.Nature communications · 2022Article
- Machine learning identification of cuproptosis and necroptosis-associated molecular subtypes to aid in prognosis assessment and immunotherapy response prediction in low-grade glioma.Frontiers in genetics · 2022Article
- Spatial and temporal localization of SPIRRIG and WAVE/SCAR reveal roles for these proteins in actin-mediated root hair development.The Plant cell · 2021Article
- A Novel Homozygous Frameshift WDR81 Mutation associated with Microlissencephaly, Corpus Callosum Agenesis, and Pontocerebellar Hypoplasia.Journal of pediatric genetics · 2021Article
- TFG binds LC3C to regulate ULK1 localization and autophagosome formation.The EMBO journal · 2021Article
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Authors and funding
12 authors at 3 institutions in 2 countries.
Funding
Abstract
Autophagy-dependent clearance of ubiquitinated and aggregated proteins is critical to protein quality control, but the underlying mechanisms are not well understood. Here, we report the essential role of the BEACH (beige and Chediak-Higashi) and WD40 repeat-containing protein WDR81 in eliminating ubiquitinated proteins through autophagy. WDR81 associates with ubiquitin (Ub)-positive protein foci, and its loss causes accumulation of Ub proteins and the autophagy cargo receptor p62. WDR81 interacts with p62, facilitating recognition of Ub proteins by p62. Furthermore, WDR81 interacts with LC3C through canonical LC3-interacting regions in the BEACH domain, promoting LC3C recruitment to ubiquitinated proteins. Inactivation of LC3C or defective autophagy results in accumulation of Ub protein aggregates enriched for WDR81. In mice, WDR81 inactivation causes accumulation of p62 bodies in cortical and striatal neurons in the brain. These data suggest that WDR81 coordinates p62 and LC3C to facilitate autophagic removal of Ub proteins, and provide important insights into CAMRQ2 syndrome, a WDR81-related developmental disorder.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.