Human CNS barrier-forming organoids with cerebrospinal fluid production

被引:300
作者
Pellegrini, Laura [1 ]
Bonfio, Claudia [1 ]
Chadwick, Jessica [1 ]
Begum, Farida [1 ]
Skehel, Mark [1 ]
Lancaster, Madeline A. [1 ]
机构
[1] MRC Lab Mol Biol, Francis Crick Ave, Cambridge CB2 0QH, England
基金
欧洲研究理事会; 英国医学研究理事会;
关键词
CENTRAL-NERVOUS-SYSTEM; CHOROID-PLEXUS; ALZHEIMERS-DISEASE; PROTEOMIC ANALYSIS; MATRIX METALLOPROTEINASES; CEREBRAL ORGANOIDS; APOLIPOPROTEIN-E; CSF BIOMARKER; TUMOR-MARKERS; SPINAL-FLUID;
D O I
10.1126/science.aaz5626
中图分类号
O [数理科学和化学]; P [天文学、地球科学]; Q [生物科学]; N [自然科学总论];
学科分类号
07 ; 0710 ; 09 ;
摘要
Cerebrospinal fluid (CSF) is a vital liquid, providing nutrients and signaling molecules and clearing out toxic by-products from the brain. The CSF is produced by the choroid plexus (ChP), a protective epithelial barrier that also prevents free entry of toxic molecules or drugs from the blood. Here, we establish human ChP organoids with a selective barrier and CSF-like fluid secretion in self-contained compartments. We show that this in vitro barrier exhibits the same selectivity to small molecules as the ChP in vivo and that ChP-CSF organoids can predict central nervous system (CNS) permeability of new compounds. The transcriptomic and proteomic signatures of ChP-CSF organoids reveal a high degree of similarity to the ChP in vivo. Finally, the intersection of single-cell transcriptomics and proteomic analysis uncovers key human CSF components produced by previously unidentified specialized epithelial subtypes.
引用
收藏
页码:159 / +
页数:58
相关论文
共 130 条
[11]   The single-cell transcriptional landscape of mammalian organogenesis [J].
Cao, Junyue ;
Spielmann, Malte ;
Qiu, Xiaojie ;
Huang, Xingfan ;
Ibrahim, Daniel M. ;
Hill, Andrew J. ;
Zhang, Fan ;
Mundlos, Stefan ;
Christiansen, Lena ;
Steemers, Frank J. ;
Trapnell, Cole ;
Shendure, Jay .
NATURE, 2019, 566 (7745) :496-+
[12]   Comparative proteomics of cerebrospinal fluid in neuropathologically-confirmed Alzheimer's disease and non-demented elderly subjects [J].
Castaño, EM ;
Roher, AE ;
Esh, CL ;
Kokjohn, TA ;
Beach, T .
NEUROLOGICAL RESEARCH, 2006, 28 (02) :155-163
[13]   Global Protein Differential Expression Profiling of Cerebrospinal Fluid Samples Pooled from Chinese Sporadic CJD and non-CJD Patients [J].
Chen, Cao ;
Xiao, Di ;
Zhou, Wei ;
Shi, Qi ;
Zhang, Hui-Fang ;
Zhang, Jin ;
Tian, Chan ;
Zhang, Jian-Zhong ;
Dong, Xiao-Ping .
MOLECULAR NEUROBIOLOGY, 2014, 49 (01) :290-302
[14]   Increased Albumin Oxidation in Cerebrospinal Fluid and Plasma from Alzheimer's Disease Patients [J].
Costa, Montserrat ;
Horrillo, Raquel ;
Maria Ortiz, Ana ;
Perez, Alba ;
Mestre, Anna ;
Ruiz, Agustin ;
Boada, Merce ;
Grancha, Salvador .
JOURNAL OF ALZHEIMERS DISEASE, 2018, 63 (04) :1395-1404
[15]   Direct and indirect roles of CNS dorsal midline cells in choroid plexus epithelia formation [J].
Currle, DS ;
Cheng, X ;
Hsu, CM ;
Monuki, ES .
DEVELOPMENT, 2005, 132 (15) :3549-3559
[16]  
Dani N., CELLULAR SPATIAL MAP
[17]   A cellular and spatial map of the choroid plexus across brain ventricles and ages [J].
Dani, Neil ;
Herbst, Rebecca H. ;
McCabe, Cristin ;
Green, Gilad S. ;
Kaiser, Karol ;
Head, Joshua P. ;
Cui, Jin ;
Shipley, Frederick B. ;
Jang, Ahram ;
Dionne, Danielle ;
Nguyen, Lan ;
Rodman, Christopher ;
Riesenfeld, Samantha J. ;
Prochazka, Jan ;
Prochazkova, Michaela ;
Sedlacek, Radislav ;
Zhang, Feng ;
Bryja, Vitezslav ;
Rozenblatt-Rosen, Orit ;
Habib, Naomi ;
Regev, Aviv ;
Lehtinen, Maria K. .
CELL, 2021, 184 (11) :3056-+
[18]   Preventing proteostasis diseases by selective inhibition of a phosphatase regulatory subunit [J].
Das, Indrajit ;
Krzyzosiak, Agnieszka ;
Schneider, Kim ;
Wrabetz, Lawrence ;
D'Antonio, Maurizio ;
Barry, Nicholas ;
Sigurdardottir, Anna ;
Bertolotti, Anne .
SCIENCE, 2015, 348 (6231) :239-242
[19]   Proteomes of Paired Human Cerebrospinal Fluid and Plasma: Relation to Blood-Brain Barrier Permeability in Older Adults [J].
Dayon, Loic ;
Cominetti, Ornella ;
Wojcik, Jerome ;
Galindo, Antonio Nunez ;
Oikonomidi, Aikaterini ;
Henry, Hugues ;
Migliavacca, Eugenia ;
Kussmann, Martin ;
Bowman, Gene L. ;
Popp, Julius .
JOURNAL OF PROTEOME RESEARCH, 2019, 18 (03) :1162-1174
[20]   Alzheimer disease pathology and the cerebrospinal fluid proteome [J].
Dayon, Loic ;
Galindo, Antonio Nunez ;
Wojcik, Jerome ;
Cominetti, Ornella ;
Corthesy, John ;
Oikonomidi, Aikaterini ;
Henry, Hugues ;
Kussmann, Martin ;
Migliavacca, Eugenia ;
Severin, India ;
Bowman, Gene L. ;
Popp, Julius .
ALZHEIMERS RESEARCH & THERAPY, 2018, 10