Evidence map›Paper›PMID 26763422›Full record

ArticleThe Journal of investigative dermatology2016

Correction of Hair Shaft Defects through Allele-Specific Silencing of Mutant Krt75.

Ying Liu, Elizabeth R Snedecor, Xu Zhang, Yanfeng Xu, Lan Huang, Evan C Jones, Lianfeng Zhang, Richard A Clark, Dennis R Roop, Chuan Qin and 1 more

Open access · bronzeAbstract read
In one paragraph

Article in The Journal of investigative dermatology, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
0.3field-weighted citation impact, top 39% 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

4 citing papers in PubMed, 7 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  4. Article
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

11 authors at 3 institutions in 2 countries.

Ying LiuInstitute of Laboratory Animal Science, Chinese Academy of Medical Science and Comparative Medical Center, Peking Union Medical College; Key Laboratory of Human Disease Comparative Medicine, Ministry of Health, Beijing, China.
Elizabeth R SnedecorDepartment of Pathology, Stony Brook University, Stony Brook, New York, USA; Graduate Program in Genetics, Stony Brook University, Stony Brook, New York, USA.
Xu ZhangInstitute of Laboratory Animal Science, Chinese Academy of Medical Science and Comparative Medical Center, Peking Union Medical College; Key Laboratory of Human Disease Comparative Medicine, Ministry of Health, Beijing, China.
Yanfeng XuInstitute of Laboratory Animal Science, Chinese Academy of Medical Science and Comparative Medical Center, Peking Union Medical College; Key Laboratory of Human Disease Comparative Medicine, Ministry of Health, Beijing, China.
Lan HuangInstitute of Laboratory Animal Science, Chinese Academy of Medical Science and Comparative Medical Center, Peking Union Medical College; Key Laboratory of Human Disease Comparative Medicine, Ministry of Health, Beijing, China.
Evan C JonesDepartment of Dermatology, Stony Brook University, Stony Brook, New York, USA.
Lianfeng ZhangInstitute of Laboratory Animal Science, Chinese Academy of Medical Science and Comparative Medical Center, Peking Union Medical College; Key Laboratory of Human Disease Comparative Medicine, Ministry of Health, Beijing, China.
Richard A ClarkDepartment of Dermatology, Stony Brook University, Stony Brook, New York, USA.
Dennis R RoopDepartment of Dermatology and Charles C. Gates Center for Regenerative Medicine and Stem Cell Biology, University of Colorado Anschutz Medical Campus, Aurora, Colorado, USA.
Chuan QinInstitute of Laboratory Animal Science, Chinese Academy of Medical Science and Comparative Medical Center, Peking Union Medical College; Key Laboratory of Human Disease Comparative Medicine, Ministry of Health, Beijing, China. Electronic address: qinchuan@pumc.edu.cn.
Jiang ChenInstitute of Laboratory Animal Science, Chinese Academy of Medical Science and Comparative Medical Center, Peking Union Medical College; Key Laboratory of Human Disease Comparative Medicine, Ministry of Health, Beijing, China; Department of Pathology, Stony Brook University, Stony Brook, New York, USA; Department of Dermatology, Stony Brook University, Stony Brook, New York, USA; Department of Dermatology and Charles C. Gates Center for Regenerative Medicine and Stem Cell Biology, University of Colorado Anschutz Medical Campus, Aurora, Colorado, USA. Electronic address: jiang.chen@stonybrook.edu.
Chinese Academy of Medical Sciences & Peking Union Medical College · CNStony Brook University · USUniversity of Colorado Anschutz Medical Campus · US

Funding

TARGETING ONCOGENE EXPRESSION TO SKIN IN TRANSGENIC MICER01CA052607 · NCI · UNIVERSITY OF COLORADO DENVER · PI ROOP, DENNIS · 1990 to 2015
$6.5M
Transgenic and Gene Targeting CoreP30AR057212 · NIAMS · UNIVERSITY OF COLORADO DENVER · PI NORRIS, DAVID A. · 2009 to 2018
$6.2M
Planar Cell Polarity Signaling in Hair Follicle FormationR01AR061485 · NIAMS · UNIVERSITY OF COLORADO DENVER · PI CHEN, JIANG · 2011 to 2021
$3.8M
Testing the Therapeutic Potential of iPS Cells for Inherited Skin DiseasesR01AR059947 · NIAMS · UNIVERSITY OF COLORADO DENVER · PI ROOP, DENNIS · 2012 to 2017
$2.1M
The consequences of loricrin deficiency on epidermal barrier functionR01AR060388 · NIAMS · UNIVERSITY OF COLORADO DENVER · PI REFAELI, YOSEF, ROOP, DENNIS · 2011 to 2015
$1.6M
NCI NIH HHS CA052607NCI NIH HHS R01 CA052607NIAMS NIH HHS AR057212NIAMS NIH HHS AR059947NIAMS NIH HHS AR060388NIAMS NIH HHS AR061485NIAMS NIH HHS P30 AR057212NIAMS NIH HHS R01 AR059947NIAMS NIH HHS R01 AR060388NIAMS NIH HHS R01 AR061485
6 · The paper itself

Abstract

Dominant mutations in keratin genes can cause a number of inheritable skin disorders characterized by intraepidermal blistering, epidermal hyperkeratosis, or abnormalities in skin appendages, such as nail plate dystrophy and structural defects in hair. Allele-specific silencing of mutant keratins through RNA interference is a promising therapeutic approach for suppressing the expression of mutant keratins and related phenotypes in the epidermis. However, its effectiveness on skin appendages remains to be confirmed in vivo. In this study, we developed allele-specific small interfering RNAs capable of selectively suppressing the expression of a mutant Krt75, which causes hair shaft structural defects characterized by the development of blebs along the hair shaft in mice. Hair regenerated from epidermal keratinocyte progenitor cells isolated from mutant Krt75 mouse models reproduced the blebbing phenotype when grafted in vivo. In contrast, mutant cells manipulated with a lentiviral vector expressing mutant Krt75-specific short hairpin RNA (shRNA) persistently suppressed this phenotype. The phenotypic correction was associated with a significant reduction of mutant Krt75 mRNA in the skin grafts. Thus, data obtained from this study demonstrated the feasibility of utilizing RNA interference to achieve durable correction of hair structural phenotypes through allele-specific silencing of mutant keratin genes.

Indexed as

Gene SilencingMutation, MissenseAllelesAnimalsDisease Models, AnimalGene Expression RegulationGenes, DominantHair FollicleKeratin-6KeratinsMiceMice, Mutant StrainsRandom AllocationReal-Time Polymerase Chain ReactionSensitivity and SpecificitySkin TransplantationKeratin-6KeratinsKrt75 protein, mouse

Identifiers

PMID26763422
PMCPMC4764097
OpenAlexW2221913703

What OpenQuestion holds

Textmetadata
LicenceTDM
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.