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Mamm Genome. 2012 Apr;23(3-4):270-6. doi: 10.1007/s00335-011-9381-z. Epub 2011 Dec 15.

Conditional deletion of calcium-modulating cyclophilin ligand causes deafness in mice.

Author information

1
Research Animal Diagnostic Laboratory, Department of Veterinary Pathobiology, College of Veterinary Medicine, University of Missouri, 4011 Discovery Drive, Columbia, MO 65201, USA. brydae@missouri.edu

Abstract

Calcium-modulating cyclophilin ligand (Caml) is a ubiquitously expressed cytoplasmic protein that is involved in multiple signaling and developmental pathways. An observation in our laboratory of a protein-protein interaction between Caml and the cytoplasmic region of Cadherin23 led us to speculate that Caml might be important in the inner ear and play a role in the development and/or function of hair cells. To address this question, we generated a mouse line in which Caml expression was eliminated in Atoh1-expressing cells of the inner ear upon administration of tamoxifen. Tamoxifen was administered immediately after birth to neonates to assess the effect of loss of Caml in the inner ear during postnatal development. Hearing in treated animals was tested by auditory brain stem response (ABR) analysis and cochlear pathology was evaluated by light microscopy. Lack of Caml expression in the inner ear leads to severe loss of cochlear hair cells and complete deafness. Elucidating the role of Caml in the inner ear will aid our understanding of the molecular pathways important for auditory development and function.

PMID:
22170629
DOI:
10.1007/s00335-011-9381-z
[Indexed for MEDLINE]

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