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Decision letter: Dysregulation of sonic hedgehog signaling causes hearing loss in ciliopathy mouse models
[peer_review]
2020
unpublished
Defective primary cilia cause a range of diseases known as ciliopathies, including hearing loss. The etiology of hearing loss in ciliopathies, however, remains unclear. We analyzed cochleae from three ciliopathy mouse models exhibiting different ciliogenesis defects: Intraflagellar transport 88 (Ift88), Tbc1d32 (a.k.a. bromi), and Cilk1 (a.k.a. Ick) mutants. These mutants showed multiple developmental defects including shortened cochlear duct and abnormal apical patterning of the organ of
doi:10.7554/elife.56551.sa1
fatcat:y63e4ouypbcsppjkqxpixxues4