ArticlePLoS genetics2013
Deletion of an X-inactivation boundary disrupts adjacent gene silencing.
Article in PLoS genetics, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers.
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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.
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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.
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Who cites it
28 citing papers in PubMed, 39 citations in OpenAlex.
- Escape from X-chromosome inactivation: from gene discovery to regulatory mechanisms.Biochemical Society transactions · 2026Review
- Aging promotes reactivation of the Barr body at distal chromosome regions.Nature aging · 2025Article
- CTCF-mediated insulation and chromatin environment modulate Car5b escape from X inactivation.BMC biology · 2025Article
- Multifaceted role of CTCF in X-chromosome inactivation.Chromosoma · 2024Review
- Review
- Refining the genomic determinants underlying escape from X-chromosome inactivation.NAR genomics and bioinformatics · 2023Article
- A lifelong duty: how Xist maintains the inactive X chromosome.Current opinion in genetics & development · 2022Review
- The Molecular and Nuclear Dynamics of X-Chromosome Inactivation.Cold Spring Harbor perspectives in biology · 2022Review
- Caregiver-reported characteristics of children diagnosed with pathogenic variants in KDM5C.American journal of medical genetics. Part A · 2021Article
- Contribution of genetic and epigenetic changes to escape from X-chromosome inactivation.Epigenetics & chromatin · 2021Article
- Contiguous erosion of the inactive X in human pluripotency concludes with global DNA hypomethylation.Cell reports · 2021Article
- Genes that escape from X-chromosome inactivation: Potential contributors to Klinefelter syndrome.American journal of medical genetics. Part C, Seminars in medical genetics · 2020Review
- Escape From X-Chromosome Inactivation: An Evolutionary Perspective.Frontiers in cell and developmental biology · 2019Review
- X Inactivation and Escape: Epigenetic and Structural Features.Frontiers in cell and developmental biology · 2019Review
- The eXceptional nature of the X chromosome.Human molecular genetics · 2018Review
- Human cis-acting elements regulating escape from X-chromosome inactivation function in mouse.Human molecular genetics · 2018Article
- When the Lyon(ized chromosome) roars: ongoing expression from an inactive X chromosome.Philosophical transactions of the Royal Society of London. Series B, Biological sciences · 2017Review
- Review
- Structural aspects of the inactive X chromosome.Philosophical transactions of the Royal Society of London. Series B, Biological sciences · 2017Review
- Genetic and epigenetic features direct differential efficiency of Xist-mediated silencing at X-chromosomal and autosomal locations.Nature communications · 2017Article
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
In mammalian females, genes on one X are largely silenced by X-chromosome inactivation (XCI), although some "escape" XCI and are expressed from both Xs. Escapees can closely juxtapose X-inactivated genes and provide a tractable model for assessing boundary function at epigenetically regulated loci. To delimit sequences at an XCI boundary, we examined female mouse embryonic stem cells carrying X-linked BAC transgenes derived from an endogenous escape locus. Previously we determined that large BACs carrying escapee Kdm5c and flanking X-inactivated transcripts are properly regulated. Here we identify two lines with truncated BACs that partially and completely delete the distal Kdm5c XCI boundary. This boundary is not required for escape, since despite integrating into regions that are normally X inactivated, transgenic Kdm5c escapes XCI, as determined by RNA FISH and by structurally adopting an active conformation that facilitates long-range preferential association with other escapees. Yet, XCI regulation is disrupted in the transgene fully lacking the distal boundary; integration site genes up to 350 kb downstream of the transgene now inappropriately escape XCI. Altogether, these results reveal two genetically separable XCI regulatory activities at Kdm5c. XCI escape is driven by a dominant element(s) retained in the shortest transgene that therefore lies within or upstream of the Kdm5c locus. Additionally, the distal XCI boundary normally plays an essential role in preventing nearby genes from escaping XCI.
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Registered trials
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.