Modeling human neurodevelopmental diseases with brain organoids

被引:30
作者
Lu, Xiaoxiang [1 ]
Yang, Jiajie [1 ]
Xiang, Yangfei [1 ]
机构
[1] ShanghaiTech Univ, Sch Life Sci & Technol, Shanghai 201210, Peoples R China
基金
中国国家自然科学基金;
关键词
Brain organoids; Neurodevepmental diseases; Disease modeling; Stem cells; Gene editing; PLURIPOTENT STEM-CELLS; MOUSE MODEL; DOWN-SYNDROME; RETT-SYNDROME; CEREBRAL ORGANOIDS; SYNAPTIC PLASTICITY; CORTICAL-NEURONS; GENE-EXPRESSION; IN-VITRO; MICE;
D O I
10.1186/s13619-021-00103-6
中图分类号
Q813 [细胞工程];
学科分类号
摘要
Studying the etiology of human neurodevelopmental diseases has long been a challenging task due to the brain's complexity and its limited accessibility. Human pluripotent stem cells (hPSCs)-derived brain organoids are capable of recapitulating various features and functionalities of the human brain, allowing the investigation of intricate pathogenesis of developmental abnormalities. Over the past years, brain organoids have facilitated identifying disease-associated phenotypes and underlying mechanisms for human neurodevelopmental diseases. Integrating with more cutting-edge technologies, particularly gene editing, brain organoids further empower human disease modeling. Here, we review the latest progress in modeling human neurodevelopmental disorders with brain organoids.
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页数:13
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共 137 条
[1]   DYRKIA BAC transgenic mice show altered synaptic plasticity with learning and memory defects [J].
Ahn, Kyoung-Jin ;
Jeong, Hey Kyeong ;
Choi, Han-Saem ;
Ryoo, Soo-n Ryoo ;
Kim, Yeon Ju ;
Goo, Jun-Seo ;
Choi, Se-Young ;
Han, Jung-Soo ;
Ha, Ilho ;
Song, Woo-Joo .
NEUROBIOLOGY OF DISEASE, 2006, 22 (03) :463-472
[2]   Rett syndrome is caused by mutations in X-linked MECP2, encoding methyl-CpG-binding protein 2 [J].
Amir, RE ;
Van den Veyver, IB ;
Wan, M ;
Tran, CQ ;
Francke, U ;
Zoghbi, HY .
NATURE GENETICS, 1999, 23 (02) :185-188
[3]   Isogenic Pairs of Wild Type and Mutant Induced Pluripotent Stem Cell (iPSC) Lines from Rett Syndrome Patients as In Vitro Disease Model [J].
Ananiev, Gene ;
Williams, Emily Cunningham ;
Li, Hongda ;
Chang, Qiang .
PLOS ONE, 2011, 6 (09)
[4]   Generation of Functional Human 3D Cortico-Motor Assembloids [J].
Andersen, Jimena ;
Revah, Omer ;
Miura, Yuki ;
Thom, Nicholas ;
Amin, Neal D. ;
Kelley, Kevin W. ;
Singh, Mandeep ;
Chen, Xiaoyu ;
Thete, Mayuri Vijay ;
Walczak, Elisabeth M. ;
Vogel, Hannes ;
Fan, H. Christina ;
Pasca, Sergiu P. .
CELL, 2020, 183 (07) :1913-+
[5]   Global analysis of neurological disease: burden and benefit [J].
不详 .
LANCET NEUROLOGY, 2017, 16 (11) :857-857
[6]   FOXG1 is responsible for the congenital variant of Rett syndrome [J].
Ariani, Francesca ;
Hayek, Giuseppe ;
Rondinella, Dalila ;
Artuso, Rosangela ;
Mencarelli, Maria Antonietta ;
Spanhol-Rosseto, Ariele ;
Pollazzon, Marzia ;
Buoni, Sabrina ;
Spiga, Ottavia ;
Ricciardi, Sara ;
Meloni, Ilaria ;
Longo, Ilaria ;
Mari, Francesca ;
Broccoli, Vania ;
Zappella, Michele ;
Renieri, Alessandra .
AMERICAN JOURNAL OF HUMAN GENETICS, 2008, 83 (01) :89-93
[7]   Fused cerebral organoids model interactions between brain regions [J].
Bagley, Joshua A. ;
Reumann, Daniel ;
Bian, Shan ;
Levi-Strauss, Julie ;
Knoblich, Juergen A. .
NATURE METHODS, 2017, 14 (07) :743-+
[8]   CDK5RAP2 Regulates Centriole Engagement and Cohesion in Mice [J].
Barrera, Jose A. ;
Kao, Ling-Rong ;
Hammer, Robert E. ;
Seemann, Joachim ;
Fuchs, Jannon L. ;
Megraw, Timothy L. .
DEVELOPMENTAL CELL, 2010, 18 (06) :913-926
[9]   AutDB: a gene reference resource for autism research [J].
Basu, Saumyendra N. ;
Kollu, Ravi ;
Banerjee-Basu, Sharmila .
NUCLEIC ACIDS RESEARCH, 2009, 37 :D832-D836
[10]   The "Down Syndrome Critical Region" Is Sufficient in the Mouse Model to Confer Behavioral, Neurophysiological, and Synaptic Phenotypes Characteristic of Down Syndrome [J].
Belichenko, Nadia P. ;
Belichenko, Pavel V. ;
Kleschevnikov, Alexander M. ;
Salehi, Ahmad ;
Reeves, Roger H. ;
Mobley, William C. .
JOURNAL OF NEUROSCIENCE, 2009, 29 (18) :5938-5948