ArticlePLoS genetics2008
The novel mouse mutation Oblivion inactivates the PMCA2 pump and causes progressive hearing loss.
Article in PLoS genetics, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 41 papers.
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41 citing papers in PubMed, 68 citations in OpenAlex.
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- Hair cell apoptosis and deafness inProceedings of the National Academy of Sciences of the United States of America · 2025Article
- The Role of Calcium Signaling in Sensorineural Hearing Loss.International journal of medical sciences · 2025Review
- Deafness causing neuroplastin missense variants fail to promote plasma membrane CaThe Journal of biological chemistry · 2024Article
- Absence of oncomodulin increases susceptibility to noise-induced outer hair cell death and alters mitochondrial morphology.Frontiers in neurology · 2024Article
- Treatment of monogenic and digenic dominant genetic hearing loss by CRISPR-Cas9 ribonucleoprotein delivery in vivo.Nature communications · 2023Article
- The ataxia-linked E1081Q mutation affects the sub-plasma membrane CaCell death & disease · 2022Article
- Identification and characterisation of spontaneous mutations causing deafness from a targeted knockout programme.BMC biology · 2022Article
- Crosstalk among Calcium ATPases: PMCA, SERCA and SPCA in Mental Diseases.International journal of molecular sciences · 2021Review
- Primary Active CaCold Spring Harbor perspectives in biology · 2020Review
- Building and repairing the stereocilia cytoskeleton in mammalian auditory hair cells.Hearing research · 2019Review
- Tonotopy in calcium homeostasis and vulnerability of cochlear hair cells.Hearing research · 2019Review
- Research and Discussion on the Relationships between Noise-Induced Hearing Loss and ATP2B2 Gene Polymorphism.International journal of genomics · 2019Article
- De novo and inherited loss-of-function variants of ATP2B2 are associated with rapidly progressive hearing impairment.Human genetics · 2019Article
- Structure of the human plasma membrane CaNature communications · 2018Article
- Characterizations of PMCA2-interacting complex and its role as a calcium oxalate crystal-binding protein.Cellular and molecular life sciences : CMLS · 2018Article
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Authors and funding
12 authors at 4 institutions in 3 countries.
Funding
Abstract
Progressive hearing loss is common in the human population, but we have few clues to the molecular basis. Mouse mutants with progressive hearing loss offer valuable insights, and ENU (N-ethyl-N-nitrosourea) mutagenesis is a useful way of generating models. We have characterised a new ENU-induced mouse mutant, Oblivion (allele symbol Obl), showing semi-dominant inheritance of hearing impairment. Obl/+ mutants showed increasing hearing impairment from post-natal day (P)20 to P90, and loss of auditory function was followed by a corresponding base to apex progression of hair cell degeneration. Obl/Obl mutants were small, showed severe vestibular dysfunction by 2 weeks of age, and were completely deaf from birth; sensory hair cells were completely degenerate in the basal turn of the cochlea, although hair cells appeared normal in the apex. We mapped the mutation to Chromosome 6. Mutation analysis of Atp2b2 showed a missense mutation (2630C-->T) in exon 15, causing a serine to phenylalanine substitution (S877F) in transmembrane domain 6 of the PMCA2 pump, the resident Ca(2+) pump of hair cell stereocilia. Transmembrane domain mutations in these pumps generally are believed to be incompatible with normal targeting of the protein to the plasma membrane. However, analyses of hair cells in cultured utricular maculae of Obl/Obl mice and of the mutant Obl pump in model cells showed that the protein was correctly targeted to the plasma membrane. Biochemical and biophysical characterisation showed that the pump had lost a significant portion of its non-stimulated Ca(2+) exporting ability. These findings can explain the progressive loss of auditory function, and indicate the limits in our ability to predict mechanism from sequence alone.
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