Evidence map›Paper›PMID 30421281›Full record

ArticleStem cell reviews and reports2019

Generation of Isogenic Controls for In Vitro Disease Modelling of X-Chromosomal Disorders.

Lisa Hinz, Stephanie D Hoekstra, Kyoko Watanabe, Danielle Posthuma, Vivi M Heine

Open access · hybridAbstract read
In one paragraph

Article in Stem cell reviews and reports, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed, 1 pooled it
2.0field-weighted citation impact, top 12% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

12 citing papers in PubMed, 1 synthesis or guideline pooled it, 23 citations in OpenAlex.

  1. Meta-Analysis IdentifiesInternational journal of molecular sciences · 2022
    Pooled it
  2. Review
  3. Article
  4. Article
  5. Review
  6. Review
  7. CRISPR/Cas9 andInternational journal of molecular sciences · 2023
    Article
  8. Review
  9. Stem Cell-Based Therapeutic Approaches in Genetic Diseases.Advances in experimental medicine and biology · 2023
    Article
  10. Review
  11. Article
  12. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

5 authors at 1 institution in 2 countries.

Lisa HinzComplex Trait Genetics, Center for Neurogenomics and Cognitive Research, Amsterdam Neuroscience, Vrije Universiteit Amsterdam, Boelelaan 1085, 1081HV, Amsterdam, The Netherlands.
Stephanie D HoekstraComplex Trait Genetics, Center for Neurogenomics and Cognitive Research, Amsterdam Neuroscience, Vrije Universiteit Amsterdam, Boelelaan 1085, 1081HV, Amsterdam, The Netherlands.
Kyoko WatanabeComplex Trait Genetics, Center for Neurogenomics and Cognitive Research, Amsterdam Neuroscience, Vrije Universiteit Amsterdam, Boelelaan 1085, 1081HV, Amsterdam, The Netherlands.
Danielle PosthumaComplex Trait Genetics, Center for Neurogenomics and Cognitive Research, Amsterdam Neuroscience, Vrije Universiteit Amsterdam, Boelelaan 1085, 1081HV, Amsterdam, The Netherlands.
Vivi M HeineComplex Trait Genetics, Center for Neurogenomics and Cognitive Research, Amsterdam Neuroscience, Vrije Universiteit Amsterdam, Boelelaan 1085, 1081HV, Amsterdam, The Netherlands. vm.heine@vumc.nl.ORCID 0000-0003-4416-3875
Amsterdam Neuroscience · NL

Funding

EPA EP-C-14-005
6 · The paper itself

Abstract

Generation of proper controls is crucial in induced pluripotent stem cell (iPSC) studies. X-chromosomal disorders offer the potential to develop isogenic controls due to random X-chromosomal inactivation (XCI). However, the generation of such lines is currently hampered by skewed X-inactivation in fibroblast lines and X-chromosomal reactivation (XCR) after reprogramming. Here we describe a method to generate a pure iPSC population with respect to the specific inactivated X-chromosome (Xi). We used fibroblasts from Rett patients, who all have a causal mutation in the X-linked MeCP2 gene. Pre-sorting these fibroblasts followed by episomal reprogramming, allowed us to overcome skewness in fibroblast lines and to retain the X-chromosomal state, which was unpredictable with lentiviral reprogramming. This means that fibroblast pre-sorting followed by episomal reprogramming can be used to reliably generate iPSC lines with specified X-chromosomal phenotype such as Rett syndrome.

Indexed as

Cellular ReprogrammingX Chromosome InactivationChromosome DisordersChromosomes, Human, XFemaleFibroblastsHumansInduced Pluripotent Stem CellsIn Vitro TechniquesMethyl-CpG-Binding Protein 2MutationPhenotypeRett SyndromeRNA, Long NoncodingMECP2 protein, humanMethyl-CpG-Binding Protein 2RNA, Long NoncodingXIST non-coding RNAEpisomal reprogrammingiPSCsIsogenic controlRett syndromeX-chromosome

Identifiers

PMID30421281
PMCPMC6441401
OpenAlexW2901028297

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.