Use of 3D Organoids as a Model to Study Idiopathic Form of Parkinson's Disease

被引:57
作者
Chlebanowska, Paula [1 ]
Tejchman, Anna [1 ]
Sulkowski, Maciej [1 ]
Skrzypek, Klaudia [1 ]
Majka, Marcin [1 ]
机构
[1] Jagiellonian Univ, Fac Med, Inst Pediat, Dept Transplantat,Med Coll, Wielicka 265, PL-30663 Krakow, Poland
关键词
organoid; Parkinson's disease; iPS cells; 3D model; idiopathic; LMX1A; TH; FOXA2; PTX3; PLURIPOTENT STEM-CELLS; IPS CELLS; HUMAN ES; PATHOGENESIS; SURVIVAL; ROLES; FOXA2; NURR1;
D O I
10.3390/ijms21030694
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
Organoids are becoming particularly popular in modeling diseases that are difficult to reproduce in animals, due to anatomical differences in the structure of a given organ. Thus, they are a bridge between the in vitro and in vivo models. Human midbrain is one of the structures that is currently being intensively reproduced in organoids for modeling Parkinson's disease (PD). Thanks to three-dimensional (3D) architecture and the use of induced pluripotent stem cells (iPSCs) differentiation into organoids, it has been possible to recapitulate a complicated network of dopaminergic neurons. In this work, we present the first organoid model for an idiopathic form of PD. iPSCs were generated from peripheral blood mononuclear cells of healthy volunteers and patients with the idiopathic form of PD by transduction with Sendai viral vector. iPSCs were differentiated into a large multicellular organoid-like structure. The mature organoids displayed expression of neuronal early and late markers. Interestingly, we observed statistical differences in the expression levels of LIM homeobox transcription factor alpha (early) and tyrosine hydroxylase (late) markers between organoids from PD patient and healthy volunteer. The obtained results show immense potential for the application of 3D human organoids in studying the neurodegenerative disease and modeling cellular interactions within the human brain.
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页数:13
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共 33 条
  • [1] Foxa2: The rise and fall of dopamine neurons
    Arenas, Ernest
    [J]. CELL STEM CELL, 2008, 2 (02) : 110 - 112
  • [2] Do polymorphisms in transcription factors LMX1A and LMX1B influence the risk for Parkinson's disease?
    Bergman, Olle
    Hayenkansson, Anna
    Westberg, Lars
    Belin, Andrea Carmine
    Sydow, Olof
    Olson, Lars
    Holmberg, Bjorn
    Fratiglioni, Laura
    Baeckman, Lars
    Eriksson, Elias
    Nissbrandt, Hans
    [J]. JOURNAL OF NEURAL TRANSMISSION, 2009, 116 (03) : 333 - 338
  • [3] Patient-specific induced pluripotent stem-cell-derived models of LEOPARD syndrome
    Carvajal-Vergara, Xonia
    Sevilla, Ana
    D'Souza, Sunita L.
    Ang, Yen-Sin
    Schaniel, Christoph
    Lee, Dung-Fang
    Yang, Lei
    Kaplan, Aaron D.
    Adler, Eric D.
    Rozov, Roye
    Ge, YongChao
    Cohen, Ninette
    Edelmann, Lisa J.
    Chang, Betty
    Waghray, Avinash
    Su, Jie
    Pardo, Sherly
    Lichtenbelt, Klaske D.
    Tartaglia, Marco
    Gelb, Bruce D.
    Lemischka, Ihor R.
    [J]. NATURE, 2010, 465 (7299) : 808 - U12
  • [4] Highly efficient neural conversion of human ES and iPS cells by dual inhibition of SMAD signaling
    Chambers, Stuart M.
    Fasano, Christopher A.
    Papapetrou, Eirini P.
    Tomishima, Mark
    Sadelain, Michel
    Studer, Lorenz
    [J]. NATURE BIOTECHNOLOGY, 2009, 27 (03) : 275 - 280
  • [5] NURR1 in Parkinson disease-from pathogenesis to therapeutic potential
    Decressac, Mickael
    Volakakis, Nikolaos
    Bjorklund, Anders
    Perlmann, Thomas
    [J]. NATURE REVIEWS NEUROLOGY, 2013, 9 (11) : 629 - 636
  • [6] Nurr1-Based Therapies for Parkinson's Disease
    Dong, Jie
    Li, Song
    Mo, Jing-Lin
    Cai, Huai-Bin
    Le, Wei-Dong
    [J]. CNS NEUROSCIENCE & THERAPEUTICS, 2016, 22 (05) : 351 - 359
  • [7] Lmx1a and Lmx1b regulate mitochondrial functions and survival of adult midbrain dopaminergic neurons
    Doucet-Beaupre, Helene
    Gilbert, Catherine
    Profes, Marcos Schaan
    Chabrat, Audrey
    Pacelli, Consiglia
    Giguere, Nicolas
    Rioux, Veronique
    Charest, Julien
    Deng, Qiaolin
    Laguna, Ariadna
    Ericson, Johan
    Perlmann, Thomas
    Ang, Siew-Lan
    Cicchetti, Francesca
    Parent, Martin
    Trudeau, Louis-Eric
    Levesque, Martin
    [J]. PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA, 2016, 113 (30) : E4387 - E4396
  • [8] Drug Screening for ALS Using Patient-Specific Induced Pluripotent Stem Cells
    Egawa, Naohiro
    Kitaoka, Shiho
    Tsukita, Kayoko
    Naitoh, Motoko
    Takahashi, Kazutoshi
    Yamamoto, Takuya
    Adachi, Fumihiko
    Kondo, Takayuki
    Okita, Keisuke
    Asaka, Isao
    Aoi, Takashi
    Watanabe, Akira
    Yamada, Yasuhiro
    Morizane, Asuka
    Takahashi, Jun
    Ayaki, Takashi
    Ito, Hidefumi
    Yoshikawa, Katsuhiro
    Yamawaki, Satoko
    Suzuki, Shigehiko
    Watanabe, Dai
    Hioki, Hiroyuki
    Kaneko, Takeshi
    Makioka, Kouki
    Okamoto, Koichi
    Takuma, Hiroshi
    Tamaoka, Akira
    Hasegawa, Kazuko
    Nonaka, Takashi
    Hasegawa, Masato
    Kawata, Akihiro
    Yoshida, Minoru
    Nakahata, Tatsutoshi
    Takahashi, Ryosuke
    Marchetto, Maria C. N.
    Gage, Fred H.
    Yamanaka, Shinya
    Inoue, Haruhisa
    [J]. SCIENCE TRANSLATIONAL MEDICINE, 2012, 4 (145)
  • [9] Mechanisms of hyperexcitability in Alzheimer's disease hiPSC-derived neurons and cerebral organoids vs isogenic controls
    Ghatak, Swagata
    Dolatabadi, Nima
    Trudler, Dorit
    Zhang, XiaoTong
    Wu, Yin
    Mohata, Madhav
    Ambasudhan, Rajesh
    Talantova, Maria
    Lipton, Stuart A.
    [J]. ELIFE, 2019, 8
  • [10] Synapse alterations precede neuronal damage and storage pathology in a human cerebral organoid model of CLN3-juvenile neuronal ceroid lipofuscinosis
    Gomez-Giro, Gemma
    Arias-Fuenzalida, Jonathan
    Jarazo, Javier
    Zeuschner, Dagmar
    Ali, Muhammad
    Possemis, Nina
    Bolognin, Silvia
    Halder, Rashi
    Jager, Christian
    Kuper, Willemijn F. E.
    van Hasselt, Peter M.
    Zaehres, Holm
    del Sol, Antonio
    van der Putten, Herman
    Scholer, Hans R.
    Schwamborn, Jens C.
    [J]. ACTA NEUROPATHOLOGICA COMMUNICATIONS, 2019, 7 (01)