作者
Yuechuan Xue, Zachary Gursky, Brittany Monte, Sunil Koundal, Xiaodan Liu, Hedok Lee, Tatyana V Michurina, Kennelia A Mellanson, Lucy Zhao, Alice Nemajerova, Kristopher T Kahle, Ken-Ichi Takemaru, Grigori Enikolopov, Natalia I Peunova, Helene Benveniste
发表日期
2022/3/5
期刊
Fluids and Barriers of the CNS
卷号
19
期号
1
页码范围
20
出版商
BioMed Central
简介
Background
Hydrocephalus (increased ventricular size due to CSF accumulation) is a common finding in human ciliopathies and in mouse models with genetic depletion of the multiciliated cell (MCC) cilia machinery. However, the contribution of MCC to CSF dynamics and, the mechanism by which impaired MCC function leads to hydrocephalus remains poorly understood. The aim of our study was to examine if defects in MCC ciliogenesis and cilia-generated CSF flow impact central nervous system (CNS) fluid homeostasis including glymphatic transport and solute waste drainage.
Methods
We used two distinct mouse models of MCC ciliopathy: MCC-specific CEP164 conditional knockout mice (FOXJ1-Cre;CEP164fl/fl (N = 10), 3-month-old) and p73 knock-out (p73−/− (N = 8), 5-month-old) mice. Age-matched, wild-type littermates for each of the mutants served as controls. Glymphatic transport and solute …
学术搜索中的文章
Y Xue, Z Gursky, B Monte, S Koundal, X Liu, H Lee… - Fluids and Barriers of the CNS, 2022