Document Detail

Secreted factor R-Spondin 2 is involved in refinement of patterning of the mammalian cochlea.
MedLine Citation:
PMID:  23192966     Owner:  NLM     Status:  MEDLINE    
BACKGROUND: In the cochlea, patterning of the organ of Corti is tightly regulated to produce a single row of sound-detecting inner hair cells and three rows of outer hair cells, which amplify and refine the signal. The recently identified R-Spondin family of signaling molecules usually act as co-activators of Wnt signaling; it is thought that they regulate turnover of Wnt receptors at the membrane. We sought to test whether R-Spondins function in the developing cochlea.
RESULTS: Expression analysis of all four members of the R-Spondin family showed that only R-Spondin2 (Rspo2) is expressed in the cochlea during development of the sensory epithelium. Examination of an Rspo2(-/-) mouse showed that loss of Rspo2 results in an additional single row of outer hair cells and disruption of peripheral innervation pattern. Addition of Rspo2 recombinant protein to organotypic cochlear cultures resulted in a small but significant decrease in the number of outer hair cells.
CONCLUSIONS: Rspo2 is required to limit the number of outer hair cells to three rows and for optimal arrangement of peripheral nerve fibers. The Rspo2 gain- and loss-of-function studies show that in the ear, Rspo2 function is not consistent with its assigned role as a Wnt potentiator.
J F Mulvaney; A Yatteau; W W Sun; B Jacques; K Takubo; T Suda; W Yamada; A Dabdoub
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural    
Journal Detail:
Title:  Developmental dynamics : an official publication of the American Association of Anatomists     Volume:  242     ISSN:  1097-0177     ISO Abbreviation:  Dev. Dyn.     Publication Date:  2013 Feb 
Date Detail:
Created Date:  2013-01-23     Completed Date:  2013-07-05     Revised Date:  2014-02-04    
Medline Journal Info:
Nlm Unique ID:  9201927     Medline TA:  Dev Dyn     Country:  United States    
Other Details:
Languages:  eng     Pagination:  179-88     Citation Subset:  IM    
Copyright Information:
Copyright © 2012 Wiley Periodicals, Inc.
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MeSH Terms
Cochlea / embryology*,  metabolism
DNA Primers / genetics
Fluorescent Antibody Technique
Gene Expression Regulation, Developmental / genetics,  physiology*
Hair Cells, Auditory / physiology
Image Processing, Computer-Assisted
In Situ Hybridization
Mice, Knockout
Microscopy, Confocal
Morphogenesis / physiology*
Reverse Transcriptase Polymerase Chain Reaction
Thrombospondins / genetics,  metabolism*
Grant Support
Reg. No./Substance:
0/DNA Primers; 0/RSPO2 protein, mouse; 0/Thrombospondins

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