Literature DB >> 27111774

Modeling the autistic cell: iPSCs recapitulate developmental principles of syndromic and nonsyndromic ASD.

Lihi Ben-Reuven1, Orly Reiner1.   

Abstract

The opportunity to model autism spectrum disorders (ASD) through generation of patient-derived induced pluripotent stem cells (iPSCs) is currently an emerging topic. Wide-scale research of altered brain circuits in syndromic ASD, including Rett Syndrome, Fragile X Syndrome, Angelman's Syndrome and sporadic Schizophrenia, was made possible through animal models. However, possibly due to species differences, and to the possible contribution of epigenetics in the pathophysiology of these diseases, animal models fail to recapitulate many aspects of ASD. With the advent of iPSCs technology, 3D cultures of patient-derived cells are being used to study complex neuronal phenotypes, including both syndromic and nonsyndromic ASD. Here, we review recent advances in using iPSCs to study various aspects of the ASD neuropathology, with emphasis on the efforts to create in vitro model systems for syndromic and nonsyndromic ASD. We summarize the main cellular activity phenotypes and aberrant genetic interaction networks that were found in iPSC-derived neurons of syndromic and nonsyndromic autistic patients.
© 2016 Japanese Society of Developmental Biologists.

Entities:  

Keywords:  autism; autism spectrum disorder; brain development; disease model; induced pluripotent stem cell

Mesh:

Year:  2016        PMID: 27111774     DOI: 10.1111/dgd.12280

Source DB:  PubMed          Journal:  Dev Growth Differ        ISSN: 0012-1592            Impact factor:   2.053


  3 in total

Review 1.  Stem cell therapy in autism: recent insights.

Authors:  Dario Siniscalco; Suresh Kannan; Neomar Semprún-Hernández; Adrien A Eshraghi; Anna Lisa Brigida; Nicola Antonucci
Journal:  Stem Cells Cloning       Date:  2018-10-23

Review 2.  Human in vitro models for understanding mechanisms of autism spectrum disorder.

Authors:  Aaron Gordon; Daniel H Geschwind
Journal:  Mol Autism       Date:  2020-04-16       Impact factor: 7.509

3.  Dysfunction of NMDA receptors in neuronal models of an autism spectrum disorder patient with a DSCAM mutation and in Dscam-knockout mice.

Authors:  Chae-Seok Lim; Min Jung Kim; Ja Eun Choi; Md Ariful Islam; You-Kyung Lee; Yinyi Xiong; Kyu-Won Shim; Jung-Eun Yang; Ro Un Lee; Jiah Lee; Pojeong Park; Ji-Hye Kwak; Hyunhyo Seo; Chul Hoon Kim; Jae-Hyung Lee; Yong-Seok Lee; Su-Kyeong Hwang; Kyungmin Lee; Jin-A Lee; Bong-Kiun Kaang
Journal:  Mol Psychiatry       Date:  2021-07-12       Impact factor: 15.992

  3 in total

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