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Disease Models & Mechanisms

Cilia

RESEARCH ARTICLE

Lack of whey acidic protein four disulphide core (WFDC) 2 protease inhibitor causes neonatal death from respiratory failure in mice
Kuniko Nakajima, Michio Ono, Uroš Radović, Selma Dizdarević, Shin-ichi Tomizawa, Kazushige Kuroha, Go Naganatsu, Ikue Hoshi, Risa Matsunaga, Takayuki Shirakawa, Takeyuki Kurosawa, Yasunari Miyazaki, Masahide Seki, Yutaka Suzuki, Haruhiko Koseki, Masataka Nakamura, Toshio Suda, Kazuyuki Ohbo

Disease Models & Mechanisms 2019 : dmm.040139 doi: 10.1242/dmm.040139 Published 27 September 2019
RESEARCH ARTICLE

Functional loss of Ccdc151 leads to hydrocephalus in a mouse model of primary ciliary dyskinesia
Francesco Chiani, Tiziana Orsini, Alessia Gambadoro, Miriam Pasquini, Sabrina Putti, Maurizio Cirilli, Olga Ermakova, Glauco P. Tocchini-Valentini

Disease Models & Mechanisms 2019 12: dmm038489 doi: 10.1242/dmm.038489 Published 2 August 2019

Summary: Ccdc151-knockout mice develop PCD with hydrocephalus, situs abnormalities and male infertility. Novel 3D microCT imaging analysis of the Ccdc151-lacZ reporter gene demonstrates Ccdc151 expression in ependymal cells.