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A fluid form of deafness

Inner ear defects commonly underlie deafness. The inner ears of Foxi1^-/- deaf/circling mice, as reported on p. 2013, contain large, irregular cavities that replace the compartments of the inner ear. This phenotype, Hulander et al. suggest, is caused by a defect in inner ear fluid homeostasis that causes the endolymph compartment of the inner ear to expand and rupture during development. In support of this, they found that Pds expression is lost in the endolymphatic duct and sac — structures that reabsorb endolymph — of Foxi1 mutants, indicating that Foxi1 might be an upstream regulator of Pds. Pds encodes a chloride/iodide transporter believed to function in maintaining the ionic composition of endolymph. Moreover, human PDS mutations cause Pendred syndrome-associated deafness, which the Foxi1 phenotype resembles, leading the authors to suggest that FOXI1 mutations might also underlie other forms of Pendred syndrome-like deafness in humans.

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Lack of pendrin expression leads to deafness and expansion of the endolymphatic compartment in inner ears of Foxi1 null mutant mice

Malin Hulander, Amy E. Kiernan, Sandra Rodrigo Blomqvist, Peter Carlsson, Emma-Johanna Samuelsson, Bengt R. Johansson, Karen P. Steel, and Sven Enerbäck
Development 2003 130: 2013-2025. [Abstract] [Full Text]  

This Article Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Services Email this article to a friend Related articles in Development Similar articles in this journal Alert me to new issues of the journal Download to citation manager Citing Articles Citing Articles via Google Scholar Google Scholar Search for Related Content Social Bookmarking                         What's this?