ArticleGenetics2011
A modifier gene alleviates hypothyroidism-induced hearing impairment in Pou1f1dw dwarf mice.
Article in Genetics, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.
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Who cites it
7 citing papers in PubMed, 9 citations in OpenAlex.
- Heterozygous variants in SIX3 and POU1F1 cause pituitary hormone deficiency in mouse and man.Human molecular genetics · 2023Article
- Genetic variation in thyroid folliculogenesis influences susceptibility to hypothyroidism-induced hearing impairment.Mammalian genome : official journal of the International Mammalian Genome Society · 2019Article
- Genetics of Combined Pituitary Hormone Deficiency: Roadmap into the Genome Era.Endocrine reviews · 2016Review
- A QTL on Chr 5 modifies hearing loss associated with the fascin-2 variant of DBA/2J mice.Mammalian genome : official journal of the International Mammalian Genome Society · 2015Article
- Hearing impairment in hypothyroid dwarf mice caused by mutations of the thyroid peroxidase gene.Journal of the Association for Research in Otolaryngology : JARO · 2014Article
- Making sense with thyroid hormone--the role of T(3) in auditory development.Nature reviews. Endocrinology · 2013Review
- Genetic background of Prop1(df) mutants provides remarkable protection against hypothyroidism-induced hearing impairment.Journal of the Association for Research in Otolaryngology : JARO · 2012Article
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Authors and funding
7 authors at 2 institutions in 1 country.
Funding
Abstract
Thyroid hormone has pleiotropic effects on cochlear development, and genomic variation influences the severity of associated hearing deficits. DW/J-Pou1f1dw/dw mutant mice lack pituitary thyrotropin, which causes severe thyroid hormone deficiency and profound hearing impairment. To assess the genetic complexity of protective effects on hypothyroidism-induced hearing impairment, an F1 intercross was generated between DW/J-Pou1f1dw/+ carriers and an inbred strain with excellent hearing derived from Mus castaneus, CAST/EiJ. Approximately 24% of the (DW/J×CAST/EiJ) Pou1f1dw/dw F2 progeny had normal hearing. A genome scan revealed a locus on chromosome 2, named modifier of dw hearing, or Mdwh, that rescues hearing despite persistent hypothyroidism. This chromosomal region contains the modifier of tubby hearing 1 (Moth1) locus that encodes a protective allele of the microtubule-associated protein MTAP1A. DW/J-Pou1f1dw/+ carriers were crossed with the AKR strain, which also carries a protective allele of Mtap1a, and we found that AKR is not protective for hearing in the (DW/J×AKR) Pou1f1dw/dw F2 progeny. Thus, protective alleles of Mtap1a are not sufficient to rescue DW/J-Pou1f1dw/dw hearing. We expect that identification of protective modifiers will enhance our understanding of the mechanisms of hypothyroidism-induced hearing impairment.
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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.