ArticlePloS one2015
Deficiency of angulin-2/ILDR1, a tricellular tight junction-associated membrane protein, causes deafness with cochlear hair cell degeneration in mice.
Article in PloS one, 2015. 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, 39 citations in OpenAlex.
- The roles of different gene expression regulators in acoustic variation in the intermediate horseshoe bat revealed by long-read and short-read RNA sequencing data.Current zoology · 2024Article
- Population dynamics of potentially harmful haplotypes: a pedigree analysis.BMC genomics · 2024Article
- Paracellular permeability and tight junction regulation in gut health and disease.Nature reviews. Gastroenterology & hepatology · 2023Review
- Alternative splicing in shaping the molecular landscape of the cochlea.Frontiers in cell and developmental biology · 2023Review
- Article
- ILDR1 promotes influenza A virus replication through binding to PLSCR1.Scientific reports · 2022Article
- Hearing loss in mice with disruption of auditory epithelial patterning in the cochlea.Frontiers in cell and developmental biology · 2022Article
- Claudin-9 constitutes tight junctions of folliculo-stellate cells in the anterior pituitary gland.Scientific reports · 2021Article
- Angulin-1 seals tricellular contacts independently of tricellulin and claudins.The Journal of cell biology · 2021Article
- Angulin-1 (LSR) Affects Paracellular Water Transport, However Only in Tight Epithelial Cells.International journal of molecular sciences · 2021Article
- The extracellular domain of angulin-1 and palmitoylation of its cytoplasmic region are required for angulin-1 assembly at tricellular contacts.The Journal of biological chemistry · 2020Article
- Striatin Is Required for Hearing and Affects Inner Hair Cells and Ribbon Synapses.Frontiers in cell and developmental biology · 2020Article
- Apoptotic Fragmentation of Tricellulin.International journal of molecular sciences · 2019Article
- Targeted Next Generation Sequencing Revealed a Novel HomozygousFrontiers in genetics · 2019Article
- Cell Biology of Tight Junction Barrier Regulation and Mucosal Disease.Cold Spring Harbor perspectives in biology · 2018Review
- A Novel p.G141R Mutation inNeural plasticity · 2018Article
- Angulin proteins ILDR1 and ILDR2 regulate alternative pre-mRNA splicing through binding to splicing factors TRA2A, TRA2B, or SRSF1.Scientific reports · 2017Article
- Tricellular junctions: how to build junctions at the TRICkiest points of epithelial cells.Molecular biology of the cell · 2017Review
- Identification of a novel frameshift mutation in the ILDR1 gene in a UAE family, mutations review and phenotype genotype correlation.PloS one · 2017Article
- Discovering the Unexpected with the Utilization of NGS in Diagnostics of Non-syndromic Hearing Loss Disorders: The Family Case ofFrontiers in genetics · 2017Article
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Authors and funding
4 authors at 3 institutions in 1 country.
Funding
Abstract
Tricellular tight junctions seal the extracellular spaces of tricellular contacts, where the vertices of three epithelial cells meet, and are required for the establishment of a strong barrier function of the epithelial cellular sheet. Angulins and tricellulin are known as specific protein components of tricellular tight junctions, where angulins recruit tricellulin. Mutations in the genes encoding angulin-2/ILDR1 and tricellulin have been reported to cause human hereditary deafness DFNB42 and DFNB49, respectively. To investigate the pathogenesis of DFNB42, we analyzed mice with a targeted disruption of Ildr1, which encodes angulin-2/ILDR1. Ildr1 null mice exhibited profound deafness. Hair cells in the cochlea of Ildr1 null mice develop normally, but begin to degenerate by two weeks after birth. Tricellulin localization at tricellular contacts of the organ of Corti in the cochlea was retained in Ildr1 null mice, but its distribution along the depth of tricellular contacts was affected. Interestingly, compensatory tricellular contact localization of angulin-1/LSR was observed in the organ of Corti in Ildr1 null mice although it was hardly detected in the organ of Corti in wild-type mice. The onset of hair cell degeneration in Ildr1 null mice was earlier than that in the reported Tric mutant mice, which mimic one of the tricellulin mutations in DFNB49 deafness. These results indicate that the angulin-2/ILDR1 deficiency causes the postnatal degenerative loss of hair cells in the cochlea, leading to human deafness DFNB42. Our data also suggest that angulin family proteins have distinct functions in addition to their common roles of tricellulin recruitment and that the function of angulin-2/ILDR1 for hearing cannot be substituted by angulin-1/LSR.
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