Literature DB >> 20641042

Human embryonic stem cells as models for aneuploid chromosomal syndromes.

Juan-Carlos Biancotti1, Kavita Narwani, Nicole Buehler, Berhan Mandefro, Tamar Golan-Lev, Ofra Yanuka, Amander Clark, David Hill, Nissim Benvenisty, Neta Lavon.   

Abstract

Syndromes caused by chromosomal aneuploidies are widely recognized genetic disorders in humans and often lead to spontaneous miscarriage. Preimplantation genetic screening is used to detect chromosomal aneuploidies in early embryos. Our aim was to derive aneuploid human embryonic stem cell (hESC) lines that may serve as models for human syndromes caused by aneuploidies. We have established 25 hESC lines from blastocysts diagnosed as aneuploid on day 3 of their in vitro development. The hESC lines exhibited morphology and expressed markers typical of hESCs. They demonstrated long-term proliferation capacity and pluripotent differentiation. Karyotype analysis revealed that two-third of the cell lines carry a normal euploid karyotype, while one-third remained aneuploid throughout the derivation, resulting in eight hESC lines carrying either trisomy 13 (Patau syndrome), 16, 17, 21 (Down syndrome), X (Triple X syndrome), or monosomy X (Turner syndrome). On the basis of the level of single nucleotide polymorphism heterozygosity in the aneuploid chromosomes, we determined whether the aneuploidy originated from meiotic or mitotic chromosomal nondisjunction. Gene expression profiles of the trisomic cell lines suggested that all three chromosomes are actively transcribed. Our analysis allowed us to determine which tissues are most affected by the presence of a third copy of either chromosome 13, 16, 17 or 21 and highlighted the effects of trisomies on embryonic development. The results presented here suggest that aneuploid embryos can serve as an alternative source for either normal euploid or aneuploid hESC lines, which represent an invaluable tool to study developmental aspects of chromosomal abnormalities in humans.

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Year:  2010        PMID: 20641042     DOI: 10.1002/stem.483

Source DB:  PubMed          Journal:  Stem Cells        ISSN: 1066-5099            Impact factor:   6.277


  38 in total

1.  Targeting pancreatic progenitor cells in human embryonic stem cell differentiation for the identification of novel cell surface markers.

Authors:  Bettina Fishman; Hanna Segev; Oded Kopper; Jonathan Nissenbaum; Margarita Schulman; Nissim Benvenisty; Joseph Itskovitz-Eldor; Danny Kitsberg
Journal:  Stem Cell Rev Rep       Date:  2012-09       Impact factor: 5.739

2.  Epigenetic changes in the developing brain: Effects on behavior.

Authors:  Eric B Keverne; Donald W Pfaff; Inna Tabansky
Journal:  Proc Natl Acad Sci U S A       Date:  2015-06-02       Impact factor: 11.205

Review 3.  Genetic and epigenetic stability of human pluripotent stem cells.

Authors:  Riikka J Lund; Elisa Närvä; Riitta Lahesmaa
Journal:  Nat Rev Genet       Date:  2012-09-11       Impact factor: 53.242

4.  Establishment, characterization, and differentiation of a karyotypically normal human embryonic stem cell line from a trisomy-affected embryo.

Authors:  Arundhati Mandal; Sheena Mathew; Debapriya Saha; Chandra Viswanathan
Journal:  In Vitro Cell Dev Biol Anim       Date:  2012-12-14       Impact factor: 2.416

5.  Gene expression changes in the MAPK pathway in both Fragile X and Down syndrome human neural progenitor cells.

Authors:  Erin L McMillan; Allison L Kamps; Samuel S Lake; Clive N Svendsen; Anita Bhattacharyya
Journal:  Am J Stem Cells       Date:  2012-06-03

Review 6.  Investigating human disease using stem cell models.

Authors:  Jared L Sterneckert; Peter Reinhardt; Hans R Schöler
Journal:  Nat Rev Genet       Date:  2014-07-29       Impact factor: 53.242

Review 7.  Achilles' heel of pluripotent stem cells: genetic, genomic and epigenetic variations during prolonged culture.

Authors:  Paola Rebuzzini; Maurizio Zuccotti; Carlo Alberto Redi; Silvia Garagna
Journal:  Cell Mol Life Sci       Date:  2016-03-09       Impact factor: 9.261

Review 8.  Modeling Alzheimer's disease with human induced pluripotent stem (iPS) cells.

Authors:  Alison E Mungenast; Sandra Siegert; Li-Huei Tsai
Journal:  Mol Cell Neurosci       Date:  2015-12-04       Impact factor: 4.314

9.  Deficits in human trisomy 21 iPSCs and neurons.

Authors:  Jason P Weick; Dustie L Held; George F Bonadurer; Matthew E Doers; Yan Liu; Chelsie Maguire; Aaron Clark; Joshua A Knackert; Katharine Molinarolo; Michael Musser; Lin Yao; Yingnan Yin; Jianfeng Lu; Xiaoqing Zhang; Su-Chun Zhang; Anita Bhattacharyya
Journal:  Proc Natl Acad Sci U S A       Date:  2013-05-28       Impact factor: 11.205

10.  Altered hematopoiesis in trisomy 21 as revealed through in vitro differentiation of isogenic human pluripotent cells.

Authors:  Glenn A Maclean; Tobias F Menne; Guoji Guo; Danielle J Sanchez; In-Hyun Park; George Q Daley; Stuart H Orkin
Journal:  Proc Natl Acad Sci U S A       Date:  2012-10-08       Impact factor: 11.205

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