Genomic, transcriptomic, and protein landscape profile of CFTR and cystic fibrosis.
Journal
Human genetics
ISSN: 1432-1203
Titre abrégé: Hum Genet
Pays: Germany
ID NLM: 7613873
Informations de publication
Date de publication:
Mar 2021
Mar 2021
Historique:
received:
16
05
2020
accepted:
25
07
2020
pubmed:
1
8
2020
medline:
9
3
2021
entrez:
1
8
2020
Statut:
ppublish
Résumé
Cystic Fibrosis (CF) is caused most often by removal of amino acid 508 (Phe508del, deltaF508) within CFTR, yet dozens of additional CFTR variants are known to give rise to CF and many variants in the genome are known to contribute to CF pathology. To address CFTR coding variants, we developed a sequence-to-structure-to-dynamic matrix for all amino acids of CFTR using 233 vertebrate species, CFTR structure within a lipid membrane, and 20 ns of molecular dynamic simulation to assess known variants from the CFTR1, CFTR2, ClinVar, TOPmed, gnomAD, and COSMIC databases. Surprisingly, we identify 18 variants of uncertain significance within CFTR from diverse populations that are heritable and a likely cause of CF that have been understudied due to nonexistence in Caucasian populations. In addition, 15 sites within the genome are known to modulate CF pathology, where we have identified one genome region (chr11:34754985-34836401) that contributes to CF through modulation of expression of a noncoding RNA in epithelial cells. These 15 sites are just the beginning of understanding comodifiers of CF, where utilization of eQTLs suggests many additional genomics of CFTR expressing cells that can be influenced by genomic background of CFTR variants. This work highlights that many additional insights of CF genetics are needed, particularly as pharmaceutical interventions increase in the coming years.
Identifiants
pubmed: 32734384
doi: 10.1007/s00439-020-02211-w
pii: 10.1007/s00439-020-02211-w
pmc: PMC7855842
mid: NIHMS1616738
doi:
Substances chimiques
CFTR protein, human
0
Cystic Fibrosis Transmembrane Conductance Regulator
126880-72-6
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
423-439Subventions
Organisme : NIEHS NIH HHS
ID : K01 ES025435
Pays : United States
Organisme : NIH HHS
ID : K01-ES025435
Pays : United States
Organisme : NIH HHS
ID : K01-ES025435
Pays : United States
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