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Article

Dysregulation of sonic hedgehog signaling causes hearing loss in ciliopathy mouse models

31 Dec 2020

Abstract excerpt

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...

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PMID: 33382037
DOI: 10.7554/elife.56551

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