CHARGE syndrome modeling using patient-iPSCs reveals defective migration of neural crest cells harboring CHD7 mutations

CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, and is characterized by a set of malformations that, on clinical grounds, were historically postulated to arise from defects in neural crest formation during embryogenesis. To better delineate neural crest defects in CHA...

Full description

Saved in:
Bibliographic Details
Published ineLife Vol. 6
Main Authors Okuno, Hironobu, Renault Mihara, Francois, Ohta, Shigeki, Fukuda, Kimiko, Kurosawa, Kenji, Akamatsu, Wado, Sanosaka, Tsukasa, Kohyama, Jun, Hayashi, Kanehiro, Nakajima, Kazunori, Takahashi, Takao, Wysocka, Joanna, Kosaki, Kenjiro, Okano, Hideyuki
Format Journal Article
LanguageEnglish
Published England eLife Sciences Publications Ltd 28.11.2017
eLife Sciences Publications, Ltd
Subjects
Biology
Cell migration
CHARGE syndrome
CHD7
Chromatin
Cloning
Defects
Developmental Biology
disease modeling
Embryogenesis
Experiments
Fibroblasts
Human Biology and Medicine
Hypotheses
induced pluripotent stem cells
Medicine
Mutation
Neural crest
Patients
Pluripotency
Software
Stem cells
Transplantation
human biology
stem cells
developmental biology
neural crest
CHD7
disease modeling
CHARGE syndrome
cell migration
induced pluripotent stem cells
medicine
human
Online AccessGet full text

Cover

Abstract CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, and is characterized by a set of malformations that, on clinical grounds, were historically postulated to arise from defects in neural crest formation during embryogenesis. To better delineate neural crest defects in CHARGE syndrome, we generated induced pluripotent stem cells (iPSCs) from two patients with typical syndrome manifestations, and characterized neural crest cells differentiated in vitro from these iPSCs (iPSC-NCCs). We found that expression of genes associated with cell migration was altered in CHARGE iPSC-NCCs compared to control iPSC-NCCs. Consistently, CHARGE iPSC-NCCs showed defective delamination, migration and motility in vitro, and their transplantation revealed overall defective migratory activity in the chick embryo. These results support the historical inference that CHARGE syndrome patients exhibit defects in neural crest migration, and provide the first successful application of patient-derived iPSCs in modeling craniofacial disorders.
AbstractList CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, and is characterized by a set of malformations that, on clinical grounds, were historically postulated to arise from defects in neural crest formation during embryogenesis. To better delineate neural crest defects in CHARGE syndrome, we generated induced pluripotent stem cells (iPSCs) from two patients with typical syndrome manifestations, and characterized neural crest cells differentiated in vitro from these iPSCs (iPSC-NCCs). We found that expression of genes associated with cell migration was altered in CHARGE iPSC-NCCs compared to control iPSC-NCCs. Consistently, CHARGE iPSC-NCCs showed defective delamination, migration and motility in vitro, and their transplantation revealed overall defective migratory activity in the chick embryo. These results support the historical inference that CHARGE syndrome patients exhibit defects in neural crest migration, and provide the first successful application of patient-derived iPSCs in modeling craniofacial disorders.
CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, CHD7, and is characterized by a set of malformations that, on clinical grounds, were historically postulated to arise from defects in neural crest formation during embryogenesis. To better delineate neural crest defects in CHARGE syndrome, we generated induced pluripotent stem cells (iPSCs) from two patients with typical syndrome manifestations, and characterized neural crest cells differentiated in vitro from these iPSCs (iPSC-NCCs). We found that expression of genes associated with cell migration was altered in CHARGE iPSC-NCCs compared to control iPSC-NCCs. Consistently, CHARGE iPSC-NCCs showed defective delamination, migration and motility in vitro, and their transplantation in ovo revealed overall defective migratory activity in the chick embryo. These results support the historical inference that CHARGE syndrome patients exhibit defects in neural crest migration, and provide the first successful application of patient-derived iPSCs in modeling craniofacial disorders.
CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, CHD7, and is characterized by a set of malformations that, on clinical grounds, were historically postulated to arise from defects in neural crest formation during embryogenesis. To better delineate neural crest defects in CHARGE syndrome, we generated induced pluripotent stem cells (iPSCs) from two patients with typical syndrome manifestations, and characterized neural crest cells differentiated in vitro from these iPSCs (iPSC-NCCs). We found that expression of genes associated with cell migration was altered in CHARGE iPSC-NCCs compared to control iPSC-NCCs. Consistently, CHARGE iPSC-NCCs showed defective delamination, migration and motility in vitro, and their transplantation in ovo revealed overall defective migratory activity in the chick embryo. These results support the historical inference that CHARGE syndrome patients exhibit defects in neural crest migration, and provide the first successful application of patient-derived iPSCs in modeling craniofacial disorders. CHARGE syndrome is a disease in which organs including the heart, eyes and ears may not develop properly. The cells that form the tissues affected by CHARGE syndrome develop in embryos from precursor cells called neural crest cells. Individuals with CHARGE syndrome also have mutations in a gene called CHD7 . However, it is difficult to examine how CHD7 mutations affect neural crest cells in embryos. In recent years, cell reprogramming techniques have made it possible to create induced pluripotent stem cells (iPSCs) from the specialized somatic cells found in the human body. These iPSCs can be developed into many different cell types, including neural crest cells. Okuno et al. created iPSCs from the skin cells of people with CHARGE syndrome, developed these cells into neural crest cells, and compared them with neural crest cells that were developed from the skin cells of people without CHARGE syndrome. The neural crest cells developed from people with CHARGE syndrome showed multiple abnormalities. For example, they were not able to move around correctly. This is an important observation because neural crest cells must move through tissues to form the various organs affected by CHARGE syndrome. Okuno et al. also observed changes in the activity of many genes other than CHD7 in the neural crest cells developed from CHARGE patients. Further research is now needed to find out which genes are the most important for restoring the normal activity of neural crest cells.
Author Renault Mihara, Francois
Kosaki, Kenjiro
Sanosaka, Tsukasa
Hayashi, Kanehiro
Nakajima, Kazunori
Takahashi, Takao
Akamatsu, Wado
Kohyama, Jun
Wysocka, Joanna
Okuno, Hironobu
Fukuda, Kimiko
Kurosawa, Kenji
Ohta, Shigeki
Okano, Hideyuki
Author_xml – sequence: 1
  givenname: Hironobu
  orcidid: 0000-0003-1932-9482
  surname: Okuno
  fullname: Okuno, Hironobu
  organization: Department of Physiology, Keio University School of Medicine, Tokyo, Japan
– sequence: 2
  givenname: Francois
  surname: Renault Mihara
  fullname: Renault Mihara, Francois
  organization: Department of Physiology, Keio University School of Medicine, Tokyo, Japan
– sequence: 3
  givenname: Shigeki
  surname: Ohta
  fullname: Ohta, Shigeki
  organization: Department of Physiology, Keio University School of Medicine, Tokyo, Japan
– sequence: 4
  givenname: Kimiko
  surname: Fukuda
  fullname: Fukuda, Kimiko
  organization: Department of Biological Science, Tokyo Metropolitan University, Tokyo, Japan
– sequence: 5
  givenname: Kenji
  surname: Kurosawa
  fullname: Kurosawa, Kenji
  organization: Division of Medical Genetics, Kanagawa Children's Medical Center, Yokohama, Japan
– sequence: 6
  givenname: Wado
  surname: Akamatsu
  fullname: Akamatsu, Wado
  organization: Center for Genomic and Regenerative Medicine, Juntendo University School of Medicine, Tokyo, Japan
– sequence: 7
  givenname: Tsukasa
  surname: Sanosaka
  fullname: Sanosaka, Tsukasa
  organization: Department of Physiology, Keio University School of Medicine, Tokyo, Japan
– sequence: 8
  givenname: Jun
  surname: Kohyama
  fullname: Kohyama, Jun
  organization: Department of Physiology, Keio University School of Medicine, Tokyo, Japan
– sequence: 9
  givenname: Kanehiro
  surname: Hayashi
  fullname: Hayashi, Kanehiro
  organization: Department of Anatomy, Keio University School of Medicine, Tokyo, Japan
– sequence: 10
  givenname: Kazunori
  orcidid: 0000-0003-1864-9425
  surname: Nakajima
  fullname: Nakajima, Kazunori
  organization: Department of Anatomy, Keio University School of Medicine, Tokyo, Japan
– sequence: 11
  givenname: Takao
  surname: Takahashi
  fullname: Takahashi, Takao
  organization: Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan
– sequence: 12
  givenname: Joanna
  surname: Wysocka
  fullname: Wysocka, Joanna
  organization: Department of Chemical and Systems Biology, Stanford University School of Medicine, Stanford, United States
– sequence: 13
  givenname: Kenjiro
  surname: Kosaki
  fullname: Kosaki, Kenjiro
  organization: Center for Medical Genetics, Keio University School of Medicine, Tokyo, Japan
– sequence: 14
  givenname: Hideyuki
  orcidid: 0000-0001-7482-5935
  surname: Okano
  fullname: Okano, Hideyuki
  organization: Department of Physiology, Keio University School of Medicine, Tokyo, Japan
BackLink https://www.ncbi.nlm.nih.gov/pubmed/29179815$$D View this record in MEDLINE/PubMed
BookMark eNpdkktr3DAQgE1JaR7Nqfci6KUQnEq2ZEmXQnDTbGChpQ_oTcjSeOPFtraSvSX_vrO7aUiqgySkj08zoznNjsYwQpa9YfRSCsE_QN-1cFkwxviL7KSgguZU8V9HT_bH2XlKa4pDcqWYfpUdF5pJrZg4yf7Ui6tvN9ck3Y8-hgHIEDw6xxWZ027e2KmDccq7r9_rRCJswfaJeGjBTd0W8W4VEQkjCS0ZYY62Jy5CmoiDHsk7G5sQd6Z68UmSYZ72dHqdvWzRBOcP61n28_P1j3qRL7_c3NZXy9wJzqa8kbQCR52nrZSlckIp4ZgXvKSlaqtS2Yq2beUxF1FyUVgBnmpdSC99Q4Uqz7Lbg9cHuzab2A023ptgO7M_CHFlbJw614Px3BagFaqB8bKQumK-1EpZz_BJ16Dr48G1mZsBvMO6YLrPpM9vxu7OrMLWCEkFfhEK3j8IYvg9Y5HM0KVdnewIYU6G6UrrklMmEH33H7oOcxyxVEihTlMMD6mLA-ViSClC-xgMo2bXHwaW2B9m3x9Iv30a_yP7rxvKv0uFuHE
CitedBy_id crossref_primary_10_1038_s41598_020_74537_4
crossref_primary_10_1016_j_ymgmr_2019_100531
crossref_primary_10_1016_j_scr_2021_102497
crossref_primary_10_1016_j_ydbio_2018_05_013
crossref_primary_10_1089_scd_2018_0058
crossref_primary_10_1111_joa_12889
crossref_primary_10_1002_mgg3_1034
crossref_primary_10_1111_iep_12331
crossref_primary_10_15252_emmm_202012013
crossref_primary_10_1002_wsbm_1537
crossref_primary_10_26508_lsa_202101127
crossref_primary_10_3390_jdb8040026
crossref_primary_10_3389_fnmol_2019_00039
crossref_primary_10_1038_s41598_022_27293_6
crossref_primary_10_3389_fnmol_2019_00139
crossref_primary_10_3389_fcvm_2023_1249605
crossref_primary_10_1016_j_stemcr_2019_04_015
crossref_primary_10_1242_dev_200712
crossref_primary_10_3389_fcell_2021_706623
crossref_primary_10_1016_j_pnpbp_2017_12_013
crossref_primary_10_15252_embr_202050958
crossref_primary_10_1101_gad_301887_117
crossref_primary_10_1002_ajmg_c_31755
crossref_primary_10_1038_s41467_021_23327_1
crossref_primary_10_3389_fcell_2021_638674
crossref_primary_10_1016_j_scr_2020_101774
crossref_primary_10_1002_ajmg_c_31831
crossref_primary_10_3389_fcell_2022_994412
crossref_primary_10_1073_pnas_2212578120
crossref_primary_10_1093_jbmr_zjae024
crossref_primary_10_1007_s00795_019_00229_2
crossref_primary_10_1111_jcmm_18443
crossref_primary_10_1111_gbb_12839
crossref_primary_10_7554_eLife_62873
crossref_primary_10_1186_s12887_018_1181_0
crossref_primary_10_1016_j_reth_2021_08_001
crossref_primary_10_1016_j_semcdb_2021_11_024
crossref_primary_10_1038_s41419_021_04099_4
crossref_primary_10_2169_naika_108_2547
crossref_primary_10_1002_sctm_20_0361
crossref_primary_10_7554_eLife_64370
Cites_doi 10.1016/j.exer.2016.04.017
10.1038/nature08733
10.1016/j.celrep.2016.04.067
10.1089/gte.2006.10.244
10.1074/jbc.M506195200
10.1016/S0140-6736(83)90538-X
10.1093/database/baw100
10.1101/gad.1602907
10.1371/journal.pone.0016182
10.1242/dev.121590
10.1056/NEJM198907273210414
10.1093/bioinformatics/19.2.185
10.1016/j.ydbio.2014.01.021
10.1016/j.cell.2007.11.019
10.1038/ng.825
10.1247/csf.07035
10.1007/s00125-006-0271-y
10.1186/1756-6606-5-35
10.1038/ng1407
10.1093/hmg/ddt435
10.1016/S0925-4773(98)00190-7
10.1128/MCB.06257-11
10.1242/dev.01045
10.1242/dev.040048
10.1016/j.ydbio.2016.06.022
10.1371/journal.pgen.1004360
10.1371/journal.pgen.1001010
10.1002/bdra.20330
10.1016/j.ydbio.2010.04.010
10.1016/0045-6039(84)90049-6
10.1038/nbt1296-1675
10.1242/dev.122.12.3785
10.1002/ajmg.a.33323
10.1002/ajmg.a.33702
10.1158/0008-5472.CAN-08-2515
10.1242/dev.126920
10.1016/j.bbrc.2006.04.135
10.1038/nmeth.f.283
10.1016/j.stem.2013.07.002
10.1002/tera.1420310303
10.3791/903
10.1016/j.devcel.2011.10.012
10.1002/stem.1293
10.1523/JNEUROSCI.23-31-09996.2003
10.1136/jmg.2005.036160
10.1038/celldisc.2015.45
10.1016/j.stem.2012.07.006
10.1073/pnas.1212021109
10.1038/nprot.2010.35
10.1038/nature08320
10.1016/j.jpeds.2005.10.044
10.1172/JCI38019
10.1002/stem.1822
10.1093/hmg/ddw198
10.1002/ajmg.a.32179
10.1371/journal.pgen.1001023
10.1186/1750-1172-1-34
10.1038/nn.4258
ContentType Journal Article
Copyright 2017, Okuno et al. This work is licensed under the Creative Commons Attribution License ( https://creativecommons.org/licenses/by/3.0/ ) (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.
2017, Okuno et al 2017 Okuno et al
Copyright_xml – notice: 2017, Okuno et al. This work is licensed under the Creative Commons Attribution License ( https://creativecommons.org/licenses/by/3.0/ ) (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.
– notice: 2017, Okuno et al 2017 Okuno et al
DBID NPM
AAYXX
CITATION
3V.
7X7
7XB
88E
88I
8FE
8FH
8FI
8FJ
8FK
ABUWG
AFKRA
AZQEC
BBNVY
BENPR
BHPHI
CCPQU
DWQXO
FYUFA
GHDGH
GNUQQ
HCIFZ
K9.
LK8
M0S
M1P
M2P
M7P
PIMPY
PQEST
PQQKQ
PQUKI
PRINS
Q9U
7X8
5PM
DOA
DOI 10.7554/elife.21114
DatabaseName PubMed
CrossRef
ProQuest Central (Corporate)
Health & Medical Collection
ProQuest Central (purchase pre-March 2016)
Medical Database (Alumni Edition)
Science Database (Alumni Edition)
ProQuest SciTech Collection
ProQuest Natural Science Collection
Hospital Premium Collection
Hospital Premium Collection (Alumni Edition)
ProQuest Central (Alumni) (purchase pre-March 2016)
ProQuest Central (Alumni)
ProQuest Central
ProQuest Central Essentials
Biological Science Collection
ProQuest Central
Natural Science Collection
ProQuest One Community College
ProQuest Central Korea
Health Research Premium Collection
Health Research Premium Collection (Alumni)
ProQuest Central Student
SciTech Premium Collection
ProQuest Health & Medical Complete (Alumni)
Biological Sciences
Health & Medical Collection (Alumni Edition)
PML(ProQuest Medical Library)
ProQuest Science Journals
Biological Science Database
Publicly Available Content Database
ProQuest One Academic Eastern Edition (DO NOT USE)
ProQuest One Academic
ProQuest One Academic UKI Edition
ProQuest Central China
ProQuest Central Basic
MEDLINE - Academic
PubMed Central (Full Participant titles)
Directory of Open Access Journals
DatabaseTitle PubMed
CrossRef
Publicly Available Content Database
ProQuest Central Student
ProQuest Central Essentials
ProQuest Health & Medical Complete (Alumni)
ProQuest Central (Alumni Edition)
SciTech Premium Collection
ProQuest One Community College
ProQuest Natural Science Collection
ProQuest Central China
ProQuest Central
Health Research Premium Collection
Health and Medicine Complete (Alumni Edition)
Natural Science Collection
ProQuest Central Korea
Biological Science Collection
ProQuest Medical Library (Alumni)
ProQuest Science Journals (Alumni Edition)
ProQuest Biological Science Collection
ProQuest Central Basic
ProQuest Science Journals
ProQuest One Academic Eastern Edition
ProQuest Hospital Collection
Health Research Premium Collection (Alumni)
Biological Science Database
ProQuest SciTech Collection
ProQuest Hospital Collection (Alumni)
ProQuest Health & Medical Complete
ProQuest Medical Library
ProQuest One Academic UKI Edition
ProQuest One Academic
ProQuest Central (Alumni)
MEDLINE - Academic
DatabaseTitleList PubMed
MEDLINE - Academic


CrossRef
Publicly Available Content Database
Database_xml – sequence: 1
  dbid: DOA
  name: Directory of Open Access Journals
  url: https://www.doaj.org/
  sourceTypes: Open Website
– sequence: 2
  dbid: NPM
  name: PubMed
  url: https://proxy.k.utb.cz/login?url=http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed
  sourceTypes: Index Database
– sequence: 3
  dbid: BENPR
  name: ProQuest Central
  url: https://www.proquest.com/central
  sourceTypes: Aggregation Database
DeliveryMethod fulltext_linktorsrc
Discipline Medicine
Biology
EISSN 2050-084X
ExternalDocumentID oai_doaj_org_article_d4a2e98430e14327961d3988ad1c1dcb
10_7554_eLife_21114
29179815
Genre Journal Article
GrantInformation_xml – fundername: ;
  grantid: Grant-in-Aid for the Encouragement of Young Medical Scientists
– fundername: ;
  grantid: A Grant-in-Aid for Young Scientists (B)
– fundername: ;
  grantid: A Grant -in-Aid for the Global COE program
– fundername: ;
  grantid: Project for Realization of Regenerative Medicine and Support for Core Institutes for iPSC research
– fundername: ;
  grantid: A Grant-in-Aid for Scientifc Reserch on Innovative Areas
– fundername: ;
  grantid: Support for Core Institutes fro iPS Cell Research
GroupedDBID 3V.
53G
5VS
7X7
88E
88I
8FE
8FH
8FI
8FJ
AAFWJ
AAKDD
ABUWG
ACGFO
ACGOD
ACPRK
ADBBV
ADRAZ
AENEX
AFKRA
ALIPV
ALMA_UNASSIGNED_HOLDINGS
AOIJS
AZQEC
BAWUL
BBNVY
BCNDV
BENPR
BHPHI
BPHCQ
BVXVI
CCPQU
DIK
DWQXO
EMOBN
FRP
FYUFA
GNUQQ
GROUPED_DOAJ
GX1
HCIFZ
HMCUK
HYE
IAO
IEA
IHR
INH
INR
ISR
ITC
KQ8
LK8
M1P
M2P
M48
M7P
M~E
NPM
NQS
OK1
PGMZT
PIMPY
PQQKQ
PROAC
PSQYO
RHF
RHI
RNS
RPM
UKHRP
AAYXX
CITATION
7XB
8FK
K9.
PQEST
PQUKI
PRINS
Q9U
7X8
5PM
AFPKN
ID FETCH-LOGICAL-c541t-b706ec0cd0f7738c5885c1d543038f638a60ff6d81553452a5ed09927d7db0583
IEDL.DBID RPM
ISSN 2050-084X
IngestDate Tue Oct 22 15:08:08 EDT 2024
Tue Sep 17 21:27:38 EDT 2024
Fri Oct 25 13:37:10 EDT 2024
Tue Dec 10 13:11:36 EST 2024
Fri Dec 06 01:27:54 EST 2024
Wed Oct 16 00:58:27 EDT 2024
IsDoiOpenAccess false
IsOpenAccess true
IsPeerReviewed true
IsScholarly true
Keywords human biology
stem cells
developmental biology
neural crest
CHD7
disease modeling
CHARGE syndrome
cell migration
induced pluripotent stem cells
medicine
human
Language English
License This article is distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use and redistribution provided that the original author and source are credited.
LinkModel DirectLink
MergedId FETCHMERGED-LOGICAL-c541t-b706ec0cd0f7738c5885c1d543038f638a60ff6d81553452a5ed09927d7db0583
Notes ObjectType-Article-1
SourceType-Scholarly Journals-1
ObjectType-Feature-2
content type line 23
ORCID 0000-0003-1932-9482
0000-0001-7482-5935
0000-0003-1864-9425
OpenAccessLink https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5705211/
PMID 29179815
PQID 1970590961
PQPubID 2045579
ParticipantIDs doaj_primary_oai_doaj_org_article_d4a2e98430e14327961d3988ad1c1dcb
pubmedcentral_primary_oai_pubmedcentral_nih_gov_5705211
proquest_miscellaneous_1969934015
proquest_journals_1970590961
crossref_primary_10_7554_eLife_21114
pubmed_primary_29179815
PublicationCentury 2000
PublicationDate 2017-11-28
PublicationDateYYYYMMDD 2017-11-28
PublicationDate_xml – month: 11
  year: 2017
  text: 2017-11-28
  day: 28
PublicationDecade 2010
PublicationPlace England
PublicationPlace_xml – name: England
– name: Cambridge
PublicationTitle eLife
PublicationTitleAlternate Elife
PublicationYear 2017
Publisher eLife Sciences Publications Ltd
eLife Sciences Publications, Ltd
Publisher_xml – sequence: 0
  name: eLife Sciences Publications Ltd
– name: eLife Sciences Publications, Ltd
– name: eLife Sciences Publications Ltd
References 24491819 - Dev Biol. 2014 May 1;389(1):28-38
19693009 - Nature. 2009 Sep 17;461(7262):402-6
26928066 - Nat Neurosci. 2016 May;19(5):678-89
20360764 - Nat Protoc. 2010 Apr;5(4):688-701
23933087 - Cell Stem Cell. 2013 Oct 3;13(4):403-18
23039195 - Mol Brain. 2012 Oct 06;5:35
24875170 - PLoS Genet. 2014 May 29;10 (5):e1004360
19078945 - J Vis Exp. 2008 Oct 20;(20):null
17149726 - Birth Defects Res A Clin Mol Teratol. 2007 Jan;79(1):50-7
2747765 - N Engl J Med. 1989 Jul 27;321(4):262
16959034 - Orphanet J Rare Dis. 2006 Sep 07;1:34
10096060 - Mech Dev. 1999 Jan;80(1):15-27
27418670 - Hum Mol Genet. 2016 Aug 15;25(16):3539-3554
22891335 - Proc Natl Acad Sci U S A. 2012 Sep 11;109(37):E2441-50
21271641 - Am J Med Genet A. 2011 Feb;155A(2):270-9
16615981 - J Pediatr. 2006 Mar;148(3):410-4
19487820 - J Clin Invest. 2009 Jun;119(6):1438-49
20657659 - PLoS Genet. 2010 Jul 15;6(7):e1001010
17253929 - Genet Test. 2006 Winter;10(4):244-51
19351860 - Cancer Res. 2009 Apr 15;69(8):3713-20
25922521 - Development. 2015 May 1;142(9):1555-60
6136666 - Lancet. 1983 Aug 27;2(8348):513
16752183 - Diabetologia. 2006 Jul;49(7):1647-55
21249204 - PLoS One. 2011 Jan 13;6(1):e16182
24026680 - Hum Mol Genet. 2014 Jan 15;23(2):434-48
20186815 - Am J Med Genet A. 2010 Mar;152A(3):674-86
12538238 - Bioinformatics. 2003 Jan 22;19(2):185-93
17974916 - Genes Dev. 2007 Nov 1;21(21):2747-61
21532573 - Nat Genet. 2011 Jun;43(6):607-11
14602813 - J Neurosci. 2003 Nov 5;23(31):9996-10001
27462442 - Cell Discov. 2016 Feb 09;2:15045
6207939 - Cell Differ. 1984 Aug;14(3):223-30
16169932 - J Med Genet. 2006 Mar;43(3):211-217
15300250 - Nat Genet. 2004 Sep;36(9):955-7
9634850 - Nat Biotechnol. 1996 Dec;14(13):1675-80
9012500 - Development. 1996 Dec;122(12):3785-97
18035408 - Cell. 2007 Nov 30;131(5):861-72
20663816 - Development. 2010 Aug;137(16):2605-21
18432839 - Curr Protoc Immunol. 2001 May;Chapter 7:Unit 7.28
16707099 - Biochem Biophys Res Commun. 2006 Jul 7;345(3):926-32
16115881 - J Biol Chem. 2005 Oct 21;280(42):35228-37
20399765 - Dev Biol. 2010 Aug 15;344(2):543-54
23193063 - Stem Cells. 2013 Mar;31(3):458-66
27126352 - Exp Eye Res. 2017 Mar;156:10-21
15105373 - Development. 2004 May;131(9):2137-47
18241060 - Am J Med Genet A. 2008 Mar 1;146A(5):585-92
27374120 - Database (Oxford). 2016 Jul 03;2016
20038952 - Nat Methods. 2010 Jan;7(1):25-7
18227620 - Cell Struct Funct. 2008;33(1):61-73
27210753 - Cell Rep. 2016 May 31;15(9):2076-88
26718006 - Development. 2016 Feb 1;143(3):504-15
27341758 - Dev Biol. 2016 Sep 1;417(1):77-90
20657823 - PLoS Genet. 2010 Jul 15;6(7):e1001023
4012643 - Teratology. 1985 Jun;31(3):331-6
20130577 - Nature. 2010 Feb 18;463(7283):958-62
22118769 - Dev Cell. 2011 Dec 13;21(6):1026-37
22290434 - Mol Cell Biol. 2012 Apr;32(7):1237-47
25183173 - Stem Cells. 2015 Jan;33(1):196-210
22981823 - Cell Stem Cell. 2012 Nov 2;11(5):633-48
Takahashi (bib54) 2007; 131
Maj (bib32) 2016; 417
Minoux (bib37) 2010; 137
Bolstad (bib11) 2003; 19
Chen (bib13) 2009; 69
Lee (bib30) 2010; 7
Lockhart (bib31) 1996; 14
Engelen (bib16) 2011; 43
Sanlaville (bib48) 2006; 43
Ohta (bib40) 2011; 6
Mercurio (bib34) 2004; 131
Bajpai (bib6) 2010; 463
Blake (bib10) 2006; 1
Raviv (bib44) 2014; 10
Schnetz (bib49) 2010; 6
Steventon (bib51) 2014; 389
Aramaki (bib3) 2006; 148
Zhao (bib61) 2007; 21
Acloque (bib1) 2009; 119
Heishi (bib20) 2006; 49
Mobley (bib38) 2001
Rouillard (bib47) 2016; 2016
Asad (bib5) 2016; 25
Kimura (bib24) 2008; 33
Berlin (bib8) 2012; 32
Okita (bib41) 2013; 31
Rosa (bib46) 1983; 2
Imaizumi (bib21) 2012; 5
Suemori (bib52) 2006; 345
Psychoyos (bib42) 2008; 20
Lamar (bib27) 2012; 109
Tabata (bib53) 2003; 23
Ninkovic (bib39) 2013; 13
Rada-Iglesias (bib43) 2012; 11
Kraft (bib25) 1989; 321
Aramaki (bib2) 2007; 79
Aramaki (bib4) 2006; 10
Grinchuk (bib17) 2005; 280
Blake (bib9) 2008; 146A
Cvekl (bib15) 2017; 156
Gómez-Skarmeta (bib18) 1999; 80
Lee (bib29) 2009; 461
Warnes (bib59) 2015
Jones (bib23) 2015; 33
Richardson (bib45) 2016; 15
Barriga (bib7) 2015; 142
Carmona-Fontaine (bib12) 2011; 21
Martin (bib33) 2010; 6
Zentner (bib60) 2010; 152A
Cordero (bib14) 2011; 155A
Millet (bib36) 1996; 122
Vissers (bib57) 2004; 36
He (bib19) 2016; 19
Kulesa (bib26) 2010; 344
Micucci (bib35) 2014; 23
Siebert (bib50) 1985; 31
Wang (bib58) 2016; 143
Imaizumi (bib22) 2012; 5
Thakurela (bib55) 2016; 2
Lee (bib28) 2010; 5
Tucker (bib56) 1984; 14
References_xml – volume: 156
  start-page: 10
  year: 2017
  ident: bib15
  article-title: PAX6: 25th anniversary and more to learn
  publication-title: Experimental Eye Research
  doi: 10.1016/j.exer.2016.04.017
  contributor:
    fullname: Cvekl
– volume: 463
  start-page: 958
  year: 2010
  ident: bib6
  article-title: CHD7 cooperates with PBAF to control multipotent neural crest formation
  publication-title: Nature
  doi: 10.1038/nature08733
  contributor:
    fullname: Bajpai
– volume: 15
  start-page: 2076
  year: 2016
  ident: bib45
  article-title: Leader Cells Define Directionality of Trunk, but Not Cranial, Neural Crest Cell Migration
  publication-title: Cell Reports
  doi: 10.1016/j.celrep.2016.04.067
  contributor:
    fullname: Richardson
– volume: 10
  start-page: 244
  year: 2006
  ident: bib4
  article-title: Screening for CHARGE syndrome mutations in the CHD7 gene using denaturing high-performance liquid chromatography
  publication-title: Genetic Testing
  doi: 10.1089/gte.2006.10.244
  contributor:
    fullname: Aramaki
– start-page: 7
  volume-title: Current Protocols in Immunology
  year: 2001
  ident: bib38
  contributor:
    fullname: Mobley
– volume: 280
  start-page: 35228
  year: 2005
  ident: bib17
  article-title: The Optimedin gene is a downstream target of Pax6
  publication-title: The Journal of Biological Chemistry
  doi: 10.1074/jbc.M506195200
  contributor:
    fullname: Grinchuk
– volume: 2
  start-page: 513
  year: 1983
  ident: bib46
  article-title: Teratogenicity of isotretinoin
  publication-title: The Lancet
  doi: 10.1016/S0140-6736(83)90538-X
  contributor:
    fullname: Rosa
– volume: 2016
  start-page: baw100
  year: 2016
  ident: bib47
  article-title: The harmonizome: a collection of processed datasets gathered to serve and mine knowledge about genes and proteins
  publication-title: Database
  doi: 10.1093/database/baw100
  contributor:
    fullname: Rouillard
– volume: 21
  start-page: 2747
  year: 2007
  ident: bib61
  article-title: Inactivation of YAP oncoprotein by the Hippo pathway is involved in cell contact inhibition and tissue growth control
  publication-title: Genes & Development
  doi: 10.1101/gad.1602907
  contributor:
    fullname: Zhao
– volume: 6
  year: 2011
  ident: bib40
  article-title: Generation of human melanocytes from induced pluripotent stem cells
  publication-title: PLoS One
  doi: 10.1371/journal.pone.0016182
  contributor:
    fullname: Ohta
– volume: 142
  start-page: 1555
  year: 2015
  ident: bib7
  article-title: Animal models for studying neural crest development: is the mouse different?
  publication-title: Development
  doi: 10.1242/dev.121590
  contributor:
    fullname: Barriga
– volume: 321
  year: 1989
  ident: bib25
  article-title: Embryonic retinoid concentrations after maternal intake of isotretinoin
  publication-title: The New England Journal of Medicine
  doi: 10.1056/NEJM198907273210414
  contributor:
    fullname: Kraft
– volume: 19
  start-page: 185
  year: 2003
  ident: bib11
  article-title: A comparison of normalization methods for high density oligonucleotide array data based on variance and bias
  publication-title: Bioinformatics
  doi: 10.1093/bioinformatics/19.2.185
  contributor:
    fullname: Bolstad
– volume: 389
  start-page: 28
  year: 2014
  ident: bib51
  article-title: Neural crest and placode interaction during the development of the cranial sensory system
  publication-title: Developmental Biology
  doi: 10.1016/j.ydbio.2014.01.021
  contributor:
    fullname: Steventon
– volume: 131
  start-page: 861
  year: 2007
  ident: bib54
  article-title: Induction of pluripotent stem cells from adult human fibroblasts by defined factors
  publication-title: Cell
  doi: 10.1016/j.cell.2007.11.019
  contributor:
    fullname: Takahashi
– volume: 43
  start-page: 607
  year: 2011
  ident: bib16
  article-title: Sox2 cooperates with Chd7 to regulate genes that are mutated in human syndromes
  publication-title: Nature Genetics
  doi: 10.1038/ng.825
  contributor:
    fullname: Engelen
– volume: 33
  start-page: 61
  year: 2008
  ident: bib24
  article-title: The organization of histone H3 modifications as revealed by a panel of specific monoclonal antibodies
  publication-title: Cell Structure and Function
  doi: 10.1247/csf.07035
  contributor:
    fullname: Kimura
– volume: 49
  start-page: 1647
  year: 2006
  ident: bib20
  article-title: Global gene expression analysis in liver of obese diabetic db/db mice treated with metformin
  publication-title: Diabetologia
  doi: 10.1007/s00125-006-0271-y
  contributor:
    fullname: Heishi
– volume: 5
  start-page: 35
  year: 2012
  ident: bib22
  article-title: Mitochondrial dysfunction associated with increased oxidative stress and α-synuclein accumulation in PARK2 iPSC-derived neurons and postmortem brain tissue
  publication-title: Molecular Brain
  doi: 10.1186/1756-6606-5-35
  contributor:
    fullname: Imaizumi
– volume: 36
  start-page: 955
  year: 2004
  ident: bib57
  article-title: Mutations in a new member of the chromodomain gene family cause CHARGE syndrome
  publication-title: Nature Genetics
  doi: 10.1038/ng1407
  contributor:
    fullname: Vissers
– volume: 23
  start-page: 434
  year: 2014
  ident: bib35
  article-title: CHD7 and retinoic acid signaling cooperate to regulate neural stem cell and inner ear development in mouse models of CHARGE syndrome
  publication-title: Human Molecular Genetics
  doi: 10.1093/hmg/ddt435
  contributor:
    fullname: Micucci
– volume: 80
  start-page: 15
  year: 1999
  ident: bib18
  article-title: Xenopus brain factor-2 controls mesoderm, forebrain and neural crest development
  publication-title: Mechanisms of Development
  doi: 10.1016/S0925-4773(98)00190-7
  contributor:
    fullname: Gómez-Skarmeta
– year: 2015
  ident: bib59
  article-title: gplots: Various R Programming Tools for Plotting Data
  contributor:
    fullname: Warnes
– volume: 32
  start-page: 1237
  year: 2012
  ident: bib8
  article-title: Phosphorylation of BRN2 modulates its interaction with the Pax3 promoter to control melanocyte migration and proliferation
  publication-title: Molecular and Cellular Biology
  doi: 10.1128/MCB.06257-11
  contributor:
    fullname: Berlin
– volume: 131
  start-page: 2137
  year: 2004
  ident: bib34
  article-title: Connective-tissue growth factor modulates WNT signalling and interacts with the WNT receptor complex
  publication-title: Development
  doi: 10.1242/dev.01045
  contributor:
    fullname: Mercurio
– volume: 137
  start-page: 2605
  year: 2010
  ident: bib37
  article-title: Molecular mechanisms of cranial neural crest cell migration and patterning in craniofacial development
  publication-title: Development
  doi: 10.1242/dev.040048
  contributor:
    fullname: Minoux
– volume: 417
  start-page: 77
  year: 2016
  ident: bib32
  article-title: Controlled levels of canonical Wnt signaling are required for neural crest migration
  publication-title: Developmental Biology
  doi: 10.1016/j.ydbio.2016.06.022
  contributor:
    fullname: Maj
– volume: 10
  year: 2014
  ident: bib44
  article-title: PAX6 regulates melanogenesis in the retinal pigmented epithelium through feed-forward regulatory interactions with MITF
  publication-title: PLoS Genetics
  doi: 10.1371/journal.pgen.1004360
  contributor:
    fullname: Raviv
– volume: 6
  year: 2010
  ident: bib33
  article-title: Chromatin remodeling in development and disease: focus on CHD7
  publication-title: PLoS Genetics
  doi: 10.1371/journal.pgen.1001010
  contributor:
    fullname: Martin
– volume: 79
  start-page: 50
  year: 2007
  ident: bib2
  article-title: Embryonic expression profile of chickenCHD7, the ortholog of the causative gene for CHARGE syndrome
  publication-title: Birth Defects Research Part A: Clinical and Molecular Teratology
  doi: 10.1002/bdra.20330
  contributor:
    fullname: Aramaki
– volume: 344
  start-page: 543
  year: 2010
  ident: bib26
  article-title: Cranial neural crest migration: new rules for an old road
  publication-title: Developmental Biology
  doi: 10.1016/j.ydbio.2010.04.010
  contributor:
    fullname: Kulesa
– volume: 14
  start-page: 223
  year: 1984
  ident: bib56
  article-title: Identical reactivity of monoclonal antibodies HNK-1 and NC-1: conservation in vertebrates on cells derived from the neural primordium and on some leukocytes
  publication-title: Cell Differentiation
  doi: 10.1016/0045-6039(84)90049-6
  contributor:
    fullname: Tucker
– volume: 14
  start-page: 1675
  year: 1996
  ident: bib31
  article-title: Expression monitoring by hybridization to high-density oligonucleotide arrays
  publication-title: Nature Biotechnology
  doi: 10.1038/nbt1296-1675
  contributor:
    fullname: Lockhart
– volume: 122
  start-page: 3785
  year: 1996
  ident: bib36
  article-title: The caudal limit of Otx2 gene expression as a marker of the midbrain/hindbrain boundary: a study using in situ hybridisation and chick/quail homotopic grafts
  publication-title: Development
  doi: 10.1242/dev.122.12.3785
  contributor:
    fullname: Millet
– volume: 152A
  start-page: 674
  year: 2010
  ident: bib60
  article-title: Molecular and phenotypic aspects of CHD7 mutation in CHARGE syndrome
  publication-title: American Journal of Medical Genetics Part A
  doi: 10.1002/ajmg.a.33323
  contributor:
    fullname: Zentner
– volume: 155A
  start-page: 270
  year: 2011
  ident: bib14
  article-title: Cranial neural crest cells on the move: their roles in craniofacial development
  publication-title: American Journal of Medical Genetics Part A
  doi: 10.1002/ajmg.a.33702
  contributor:
    fullname: Cordero
– volume: 69
  start-page: 3713
  year: 2009
  ident: bib13
  article-title: Combined integrin phosphoproteomic analyses and small interfering RNA--based functional screening identify key regulators for cancer cell adhesion and migration
  publication-title: Cancer Research
  doi: 10.1158/0008-5472.CAN-08-2515
  contributor:
    fullname: Chen
– volume: 143
  start-page: 504
  year: 2016
  ident: bib58
  article-title: Yap and Taz play a crucial role in neural crest-derived craniofacial development
  publication-title: Development
  doi: 10.1242/dev.126920
  contributor:
    fullname: Wang
– volume: 345
  start-page: 926
  year: 2006
  ident: bib52
  article-title: Efficient establishment of human embryonic stem cell lines and long-term maintenance with stable karyotype by enzymatic bulk passage
  publication-title: Biochemical and Biophysical Research Communications
  doi: 10.1016/j.bbrc.2006.04.135
  contributor:
    fullname: Suemori
– volume: 7
  start-page: 25
  year: 2010
  ident: bib30
  article-title: Induced pluripotent stem cell technology for the study of human disease
  publication-title: Nature Methods
  doi: 10.1038/nmeth.f.283
  contributor:
    fullname: Lee
– volume: 13
  start-page: 403
  year: 2013
  ident: bib39
  article-title: The BAF complex interacts with Pax6 in adult neural progenitors to establish a neurogenic cross-regulatory transcriptional network
  publication-title: Cell Stem Cell
  doi: 10.1016/j.stem.2013.07.002
  contributor:
    fullname: Ninkovic
– volume: 31
  start-page: 331
  year: 1985
  ident: bib50
  article-title: Pathologic features of the CHARGE association: support for involvement of the neural crest
  publication-title: Teratology
  doi: 10.1002/tera.1420310303
  contributor:
    fullname: Siebert
– volume: 20
  year: 2008
  ident: bib42
  article-title: Method for culture of early chick embryos ex vivo (new culture)
  publication-title: Journal of Visualized Experiments
  doi: 10.3791/903
  contributor:
    fullname: Psychoyos
– volume: 21
  start-page: 1026
  year: 2011
  ident: bib12
  article-title: Complement fragment C3a controls mutual cell attraction during collective cell migration
  publication-title: Developmental Cell
  doi: 10.1016/j.devcel.2011.10.012
  contributor:
    fullname: Carmona-Fontaine
– volume: 31
  start-page: 458
  year: 2013
  ident: bib41
  article-title: An efficient nonviral method to generate integration-free human-induced pluripotent stem cells from cord blood and peripheral blood cells
  publication-title: Stem Cells
  doi: 10.1002/stem.1293
  contributor:
    fullname: Okita
– volume: 23
  start-page: 9996
  year: 2003
  ident: bib53
  article-title: Multipolar migration: the third mode of radial neuronal migration in the developing cerebral cortex
  publication-title: Journal of Neuroscience
  doi: 10.1523/JNEUROSCI.23-31-09996.2003
  contributor:
    fullname: Tabata
– volume: 43
  start-page: 211
  year: 2006
  ident: bib48
  article-title: Phenotypic spectrum of CHARGE syndrome in fetuses with CHD7 truncating mutations correlates with expression during human development
  publication-title: Journal of Medical Genetics
  doi: 10.1136/jmg.2005.036160
  contributor:
    fullname: Sanlaville
– volume: 2
  start-page: 15045
  year: 2016
  ident: bib55
  article-title: Mapping gene regulatory circuitry of Pax6 during neurogenesis
  publication-title: Cell Discovery
  doi: 10.1038/celldisc.2015.45
  contributor:
    fullname: Thakurela
– volume: 11
  start-page: 633
  year: 2012
  ident: bib43
  article-title: Epigenomic annotation of enhancers predicts transcriptional regulators of human neural crest
  publication-title: Cell Stem Cell
  doi: 10.1016/j.stem.2012.07.006
  contributor:
    fullname: Rada-Iglesias
– volume: 109
  start-page: E2441
  year: 2012
  ident: bib27
  article-title: The Hippo pathway target, YAP, promotes metastasis through its TEAD-interaction domain
  publication-title: PNAS
  doi: 10.1073/pnas.1212021109
  contributor:
    fullname: Lamar
– volume: 5
  start-page: 688
  year: 2010
  ident: bib28
  article-title: Derivation of neural crest cells from human pluripotent stem cells
  publication-title: Nature Protocols
  doi: 10.1038/nprot.2010.35
  contributor:
    fullname: Lee
– volume: 461
  start-page: 402
  year: 2009
  ident: bib29
  article-title: Modelling pathogenesis and treatment of familial dysautonomia using patient-specific iPSCs
  publication-title: Nature
  doi: 10.1038/nature08320
  contributor:
    fullname: Lee
– volume: 148
  start-page: 410
  year: 2006
  ident: bib3
  article-title: Phenotypic spectrum of CHARGE syndrome with CHD7 mutations
  publication-title: The Journal of Pediatrics
  doi: 10.1016/j.jpeds.2005.10.044
  contributor:
    fullname: Aramaki
– volume: 119
  start-page: 1438
  year: 2009
  ident: bib1
  article-title: Epithelial-mesenchymal transitions: the importance of changing cell state in development and disease
  publication-title: Journal of Clinical Investigation
  doi: 10.1172/JCI38019
  contributor:
    fullname: Acloque
– volume: 33
  start-page: 196
  year: 2015
  ident: bib23
  article-title: CHD7 maintains neural stem cell quiescence and prevents premature stem cell depletion in the adult hippocampus
  publication-title: Stem Cells
  doi: 10.1002/stem.1822
  contributor:
    fullname: Jones
– volume: 5
  year: 2012
  ident: bib21
  article-title: Mitochondrial dysfunction associated with increased oxidative stress and α-synuclein accumulation in PARK2 iPSC-derived neurons and postmortem brain tissue
  publication-title: Molecular Brain
  doi: 10.1186/1756-6606-5-35
  contributor:
    fullname: Imaizumi
– volume: 25
  start-page: 3539
  year: 2016
  ident: bib5
  article-title: Rescue of neural crest-derived phenotypes in a zebrafish CHARGE model by Sox10 downregulation
  publication-title: Human Molecular Genetics
  doi: 10.1093/hmg/ddw198
  contributor:
    fullname: Asad
– volume: 146A
  start-page: 585
  year: 2008
  ident: bib9
  article-title: Cranial nerve manifestations in CHARGE syndrome
  publication-title: American Journal of Medical Genetics Part A
  doi: 10.1002/ajmg.a.32179
  contributor:
    fullname: Blake
– volume: 6
  year: 2010
  ident: bib49
  article-title: CHD7 targets active gene enhancer elements to modulate ES cell-specific gene expression
  publication-title: PLoS Genetics
  doi: 10.1371/journal.pgen.1001023
  contributor:
    fullname: Schnetz
– volume: 1
  year: 2006
  ident: bib10
  article-title: CHARGE syndrome
  publication-title: Orphanet Journal of Rare Diseases
  doi: 10.1186/1750-1172-1-34
  contributor:
    fullname: Blake
– volume: 19
  start-page: 678
  year: 2016
  ident: bib19
  article-title: Chd7 cooperates with Sox10 and regulates the onset of CNS myelination and remyelination
  publication-title: Nature Neuroscience
  doi: 10.1038/nn.4258
  contributor:
    fullname: He
SSID ssj0000748819
Score 2.41146
Snippet CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, and is characterized by a set of malformations that, on clinical grounds, were...
CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, CHD7, and is characterized by a set of malformations that, on clinical grounds,...
CHARGE syndrome is caused by heterozygous mutations in the chromatin remodeler, CHD7, and is characterized by a set of malformations that, on clinical grounds,...
SourceID doaj
pubmedcentral
proquest
crossref
pubmed
SourceType Open Website
Open Access Repository
Aggregation Database
Index Database
SubjectTerms Biology
Cell migration
CHARGE syndrome
CHD7
Chromatin
Cloning
Defects
Developmental Biology
disease modeling
Embryogenesis
Experiments
Fibroblasts
Human Biology and Medicine
Hypotheses
induced pluripotent stem cells
Medicine
Mutation
Neural crest
Patients
Pluripotency
Software
Stem cells
Transplantation
SummonAdditionalLinks – databaseName: Directory of Open Access Journals
  dbid: DOA
  link: http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwrV1LTxsxELYqTlwqELQNBGQkrlvWXnvtPYbwiKq2qtoicVv5tUkObFAe4u8zY2-ipELi0uva0s5-n-2Z8drfEHJpRQEJV7BZ0Ciq3TRlZnkhMtyBFFoKpQJuDfz4WY4exLdH-bhV6gvPhCV54ATclReGh0qLIg_g2rmqSuaLSmvjmWPe2bj65nwrmYprsIKByap0IU-By7wK36dN-ArpDhM7Ligq9b8VXv57SnLL7dwdkI9dvEgHyc5D8iG0R-RlOBr8vr-la7kBGuvZgBOieIx9TDux1Gz6689wQVGkCQYZ9aFJqxt9mo4T8XTWUFS0hDc4LNJBcR9_QSdmbuPBPDoc3Sj6tEr_6xfH5OHu9u9wlHUVFDInBVtmVuVlcLnzeaNUoZ3UWgJqEtAsdANTz5Q50OM1Vg8SkhsZPISMXHnlbS518YnstbM2fCE0SGOcFpwZcGgAuLUut0pJ64RxopQ9crkGtX5OQhk1JBiIfR2xryP2PXKNgG-6oLp1fACc1x3n9Xuc90h_TVfdTblFzSqFF2mhb49cbJphsiBypg2zFfYpIR6DlBLM_ZzY3VjCK9Ruwxa1w_uOqbst7XQSBbmlwivQ7OR_fNsp2ecYOTCWcd0ne8v5KpxB3LO053GIvwJq4gIC
  priority: 102
  providerName: Directory of Open Access Journals
– databaseName: Health & Medical Collection
  dbid: 7X7
  link: http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwfV3db9MwELdgCMTLBOOrY0NG2mtY7Nix84RG2agQQwiY1LfIX-n6sGQ0rab999w5bqET4jW2lJPvzr_z-fw7Qo6sKODAFWwWNJJqN02ZWV6IDDOQQkuhVMDUwPnXcnIhPk_lNCXc-lRWud4T40btO4c58mNWKXwnWZXs_fWvDLtG4e1qaqFxnzxgHKAc7FlN1SbHAvCoAfGGZ3kKgPM4fJk34R0cepjYAqLI1_-vIPNureRf4HP2hOymqJGeDGp-Su6Fdo88HPpI3u6RR-fphvwZuRlPTr5_OqVrJgIaW90APlGscJ_RxKOazb_9GPcU-ZvA_qgPzbDx0av5bLAJ2jUUyS7htw77d1BM8ff00ixsrNmj48lHRa9Ww1V-_5xcnJ3-HE-y1Fwhc1KwZWZVXgaXO583ShXaSa2lY14KwDTdgFeaMgfNeY2NhYTkRgYP0SRXXnmbS128IDtt14ZXhAZpjNOCMwNYBwGLtS63SknrhHGilCNytF7p-nrg0Kjh7IEKqaNC6qiQEfmAWthMQeLr-KFbzOrkR7UXhodKg5QBIj2uwB58UWltPAPpnR2Rg7UO6-SNff3Hdkbk7WYY_AhXzrShW-GcEkI1OG2CuC8HlW8k4RXSuuGI2jKGLVG3R9r5ZeTqlgpfR7P9_4v1mjzmGC4wlnF9QHaWi1U4hGBnad9Ei_4NBJ__7w
  priority: 102
  providerName: ProQuest
– databaseName: Scholars Portal Open Access Journals
  dbid: M48
  link: http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwfV3dT9swELcQ06a9IMY-KLDJk3hNFzt27DxNrINV0zohoBJvkb9SKkG69UOM_353TlKtCO01dpST7y73O_v8O0KOrcgg4Qo2CRpJtasqTyzPRII7kEJLoVTArYHRz3w4Ft-v5fUW6Zpxtgu4eDK1w35S4_lt_8_vh8_g8IBf-wqi4afwY1qFPmQy2ND6GYeQiLVdoxbnx1-yAjtlRXM_7_E7GxEpEvc_hTYfF03-E4XOdslOCx_pSaPvV2Qr1HvkedNQ8mGPvBi1R-Wvyf1geHLx7ZR2lAQ09ryBQEWx1H1CW0LVZHp-OVhQJHKCBaE-VM0fkN5NJ41x0FlFkfUSPuuwkQfFvf4FvTFzG4v36GD4VdG7VXOmv3hDxmenV4Nh0nZZSJwUbJlYlebBpc6nlVKZdlJr6ZiXAoKbrsA9TZ6CCr3GDkNCciODB1jJlVfeplJnb8l2PavDPqFBGuO04MxA0APkYq1LrVLSOmGcyGWPHHcrXf5qyDRKSEJQIWVUSBkV0iNfUAvrKciAHR_M5pOydajSC8NDoUHKAJCPqyJnPiu0Np6B9M72yFGnw7KzqpIVCi_bwtwe-bgeBofClTN1mK1wTg6YDdJOEPddo_K1JLxAfjccURvGsCHq5kg9vYmk3VLhNWl28H-xDslLjriBsYTrI7K9nK_Ce0A9S_shWvRfok8CcA
  priority: 102
  providerName: Scholars Portal
Title CHARGE syndrome modeling using patient-iPSCs reveals defective migration of neural crest cells harboring CHD7 mutations
URI https://www.ncbi.nlm.nih.gov/pubmed/29179815
https://www.proquest.com/docview/1970590961
https://search.proquest.com/docview/1969934015
https://pubmed.ncbi.nlm.nih.gov/PMC5705211
https://doaj.org/article/d4a2e98430e14327961d3988ad1c1dcb
Volume 6
hasFullText 1
inHoldings 1
isFullTextHit
isPrint
link http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwnV1Lb9swDBba7rLLsGGvbF2gAb06sWzJko-tly4YliLoViA3Qy-nBhanyAP7-yNlO0iGnXbxwZJgQh8lUjT1kZArw1M4cHkTeYWk2lWVRSZJeYQRSK4El9JjaGB2l00f-LeFWJwR0d-FCUn71tSj5tdq1NSPIbfyaWXHfZ7YeD4rhMQrp2x8Ts7B_B4d0cP2K0EnWd7exZNgLcf-e135EQxgWIUnyZGjC8vgHhmiwNf_Lyfz71zJI-Nz-5K86LxGet1K94qc-eY1-V1Mr--_TmhPOkBDVRswRRST2Ze0o0yN6vmPYkuRqglUjTpftXscXdXLFn66rijyWsIXLJbqoBjN39JHvTEhPY8W0y-SrvbtX_vtG_JwO_lZTKOujkJkBWe7yMg48za2Lq6kTJUVSgnLnOBgvlQFC1BnMYDkFNYQ4iLRwjtwHBPppDOxUOlbctGsG_-eUC-0toonTINZA9_EGBsbKYWxXFueiQG56ie1fGrpMko4ZiAMZYChDDAMyA1O-KELclyHF-vNsuyQLh3Xic8VSOnBqUtknjGX5kppx0B6awbksoer7BbetmS5xOu00HdAPh-aYcngzOnGr_fYJwOvDA6WIO67Ft2DJL12DIg8wf1E1NMW0NJAy91p5Yf_HvmRPE_QaWAsStQludht9v4TuDw7MwRFX8gheXYzuZvfD0PgAJ4zroZB-f8AomMHDg
link.rule.ids 230,314,727,780,784,864,885,2102,12056,21388,24318,27924,27925,31719,31720,33744,33745,43310,43805,53791,53793,73745,74302
linkProvider National Library of Medicine
linkToHtml http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwfV3fb9MwELagE7AXBINBYYCR9hoWp3bsPKGtdARoq2ls0t4i_0rXhyWjaTXx33OXuIUixGtsKSd_d77z-fwdIYeGD-DA5U3kFZJql2UamWTAI8xAciW4lB5TA5Npml_yr1fiKiTcmlBWud4T243a1RZz5Ecsk_hOMkvZx9sfEXaNwtvV0ELjPtlB5nTRIzsno-nZ-SbLAg5Sgc_rHuZJcJ1Hfjwv_Qc49jC-5Ypaxv5_hZl_V0v-4X5On5DHIW6kxx3QT8k9X-2RB10nyZ975OEk3JE_I3fD_Pj884iuuQho2-wGPBTFGvcZDUyq0fzs-7ChyOAEGkidL7utj97MZ51W0LqkSHcJv7XYwYNikr-h13ph2qo9Osw_SXqz6i7zm-fk8nR0Mcyj0F4hsoKzZWRknHobWxeXUg6UFUoJy5zg4NVUCXap0xiwcwpbC3GRaOEdxJOJdNKZWKjBPulVdeVfEuqF1lbxhGnwdhCyGGNjI6UwlmvLU9Enh-uVLm47Fo0CTh8ISNECUrSA9MkJorCZgtTX7Yd6MSuCJRWO68RnCqT0EOslEjTCDTKltGMgvTV9crDGsAj22BS_tadP3m-GwZJw5XTl6xXOSSFYg_MmiPuig3wjSZIhsRuOyC1l2BJ1e6SaX7ds3ULi-2j26v9ivSOP8ovJuBh_mX57TXYTDB4YixJ1QHrLxcq_gdBnad4G_f4FirMEVA
linkToPdf http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwfV3fb9MwELagExMvCAaMwgAj7TU0duzYeUJb11Jgq6rBpL1F8Y90fVgymlaI_567xC0UIV5jSzn57nyfz-fvCDk2IoEDlzeR10iqXZZpZHgiIsxACi2FUh5TAxfTdHIlPl_L61D_1ISyys2e2G7UrraYIx-wTOE7ySxlgzKURczOxh_uvkfYQQpvWkM7jftkD6JizHtk73Q0nV1uMy4QLDXEv-6RnoIwOvDni9K_hyMQEzthqWXv_xfk_Lty8o9QNH5MHgUMSU86pT8h93x1QB50XSV_HpD9i3Bf_pT8GE5OLj-O6IaXgLaNbyBaUax3n9PAqhotZl-HDUU2J7BG6nzZbYP0djHvLITWJUXqS_itxW4eFBP-Db0plqat4KPDyZmit-vuYr95Rq7Go2_DSRRaLURWCraKjIpTb2Pr4lKpRFuptbTMSVjIRJfgo0Uagx6dxjZDQvJCegfYkiunnImlTp6TXlVX_gWhXhaF1YKzAiIfwBdjbGyUksaKwopU9snxZqXzu45RI4eTCCokbxWStwrpk1PUwnYK0mC3H-rlPA9elTtRcJ9pkNID7uMKrMMlmdaFYyC9NX1ytNFhHnyzyX9bUp-82w6DV-HKFZWv1zgnBeAGZ08Q97BT-VYSniHJG46oHWPYEXV3pFrctMzdUuFbafby_2K9Jftg2vn5p-mXV-QhRxzBWMT1Eemtlmv_GlDQyrwJ5v0LhrsIgQ
openUrl ctx_ver=Z39.88-2004&ctx_enc=info%3Aofi%2Fenc%3AUTF-8&rfr_id=info%3Asid%2Fsummon.serialssolutions.com&rft_val_fmt=info%3Aofi%2Ffmt%3Akev%3Amtx%3Ajournal&rft.genre=article&rft.atitle=CHARGE+syndrome+modeling+using+patient-iPSCs+reveals+defective+migration+of+neural+crest+cells+harboring+CHD7+mutations&rft.jtitle=eLife&rft.au=Okuno+Hironobu&rft.au=Renault%2C+Mihara+Francois&rft.au=Ohta+Shigeki&rft.au=Fukuda+Kimiko&rft.date=2017-11-28&rft.pub=eLife+Sciences+Publications+Ltd&rft.eissn=2050-084X&rft.volume=6&rft_id=info:doi/10.7554%2FeLife.21114&rft.externalDBID=HAS_PDF_LINK
thumbnail_l http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/lc.gif&issn=2050-084X&client=summon
thumbnail_m http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/mc.gif&issn=2050-084X&client=summon
thumbnail_s http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/sc.gif&issn=2050-084X&client=summon