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Daple deficiency causes hearing loss in adult mice by inducing defects in cochlear stereocilia and apical microtubules.
Ozono, Yoshiyuki; Tamura, Atsushi; Nakayama, Shogo; Herawati, Elisa; Hanada, Yukiko; Ohata, Kazuya; Takagishi, Maki; Takahashi, Masahide; Imai, Takao; Ohta, Yumi; Oshima, Kazuo; Sato, Takashi; Inohara, Hidenori; Tsukita, Sachiko.
Afiliación
  • Ozono Y; Laboratory of Biological Science, Graduate School of Frontier Biosciences and Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Tamura A; Department of Otorhinolaryngology-Head and Neck Surgery, Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Nakayama S; Laboratory of Biological Science, Graduate School of Frontier Biosciences and Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan. atamura@biosci.med.osaka-u.ac.jp.
  • Herawati E; Department of Pharmacology, Teikyo University, 2-11-1 Kaga, Itabashi, Tokyo, 173-8605, Japan. atamura@biosci.med.osaka-u.ac.jp.
  • Hanada Y; Advanced Comprehensive Research Organization (ACRO), Teikyo University, 2-21-1 Kaga, Itabashi, Tokyo, 173-0003, Japan. atamura@biosci.med.osaka-u.ac.jp.
  • Ohata K; Laboratory of Biological Science, Graduate School of Frontier Biosciences and Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Takagishi M; Laboratory of Biological Science, Graduate School of Frontier Biosciences and Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Takahashi M; Department of Biology, Faculty of Mathematics and Natural Science, Universitas Sebelas Maret, Jalan Ir. Sutami 36 A, Surakarta, 57126, Indonesia.
  • Imai T; Department of Otorhinolaryngology-Head and Neck Surgery, Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Ohta Y; Department of Neuroscience and Cell Biology, Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Oshima K; Department of Otorhinolaryngology-Head and Neck Surgery, Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Sato T; Department of Neuroscience and Cell Biology, Graduate School of Medicine, Osaka University, 2-2 Yamadaoka, Suita, Osaka, 565-0871, Japan.
  • Inohara H; Department of Pathology, Graduate School of Medicine, Nagoya University, 65 Tsurumai, Showa, Nagoya, Aichi, 466-8550, Japan.
  • Tsukita S; Department of Pathology, Graduate School of Medicine, Nagoya University, 65 Tsurumai, Showa, Nagoya, Aichi, 466-8550, Japan.
Sci Rep ; 11(1): 20224, 2021 10 12.
Article en En | MEDLINE | ID: mdl-34642354
The V-shaped arrangement of hair bundles on cochlear hair cells is critical for auditory sensing. However, regulation of hair bundle arrangements has not been fully understood. Recently, defects in hair bundle arrangement were reported in postnatal Dishevelled-associating protein (ccdc88c, alias Daple)-deficient mice. In the present study, we found that adult Daple-/- mice exhibited hearing disturbances over a broad frequency range through auditory brainstem response testing. Consistently, distorted patterns of hair bundles were detected in almost all regions, more typically in the basal region of the cochlear duct. In adult Daple-/- mice, apical microtubules were irregularly aggregated, and the number of microtubules attached to plasma membranes was decreased. Similar phenotypes were manifested upon nocodazole treatment in a wild type cochlea culture without affecting the microtubule structure of the kinocilium. These results indicate critical role of Daple in hair bundle arrangement through the orchestration of apical microtubule distribution, and thereby in hearing, especially at high frequencies.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteínas Portadoras / Cóclea / Estereocilios / Pérdida Auditiva / Microtúbulos Tipo de estudio: Etiology_studies Límite: Animals Idioma: En Revista: Sci Rep Año: 2021 Tipo del documento: Article País de afiliación: Japón Pais de publicación: Reino Unido
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteínas Portadoras / Cóclea / Estereocilios / Pérdida Auditiva / Microtúbulos Tipo de estudio: Etiology_studies Límite: Animals Idioma: En Revista: Sci Rep Año: 2021 Tipo del documento: Article País de afiliación: Japón Pais de publicación: Reino Unido