RT info:eu-repo/semantics/article
T1 Analysis of Meis2 knockout mice reveals Sonic hedgehog‐mediated patterning of the cochlear duct
A1 Koo, Hei Yeun
A1 Oh, Jae Hwan
A1 Durán Alonso, María Beatriz
A1 Hernández, Iris López
A1 González‐Vallinas, Margarita
A1 Alonso, María Teresa
A1 Tena, Juan J.
A1 Gil‐Gálvez, Alejandro
A1 Giraldez, Fernando
A1 Bok, Jinwoong
A1 Schimmang, Thomas
K1 Cochlea
K1 Hair cell
K1 Inner ear
K1 Meis
K1 Otic vesicle
K1 Sonic hedgehog
AB Background: The mechanisms underlying the formation of complex structures such as during the outgrowth of the cochlear duct are still poorly understood.Results: We have analyzed the morphological and molecular changes associated with cochlear development in mouse mutants for the transcription factor Meis2, which show defective coiling of the cochlea. These morphological abnormalities were accompanied by the formation of ectopic and extra rows of sensory hair cells. Gene profiling of otic vesicles from Meis2 mutants revealed a dysregulation of genes that are potentially involved in Sonic hedgehog (Shh)-mediated patterning of the cochlear duct. Like in Shh mutants, Meis2 defective mice showed a loss of genes that are expressed in the apical part of the cochlear duct.Conclusions: Taken together, these data reveal that the loss of Meis2 leads to a phenotype that resembles Shh mutants, suggesting that Meis2 is instrumental for cochlear Shh signaling. The modulation of the same subset of genes provides an interesting insight into which Shh responsive genes are essential for outgrowth and patterning of the cochlear duct.
SN 1058-8388
YR 2025
FD 2025
LK https://uvadoc.uva.es/handle/10324/82165
UL https://uvadoc.uva.es/handle/10324/82165
LA eng
NO Dev Dyn, Abril 2025, vol. 254, no 4, p. 365-372.
NO Producción Científica
DS UVaDOC
RD 26-ene-2026