Literature DB >> 32734384

Genomic, transcriptomic, and protein landscape profile of CFTR and cystic fibrosis.

Morgan Sanders1, James M J Lawlor2, Xiaopeng Li1, John N Schuen3, Susan L Millard3, Xi Zhang4, Leah Buck1,5, Bethany Grysko6, Katie L Uhl1, David Hinds1,2, Cynthia L Stenger5, Michele Morris2, Neil Lamb2, Hara Levy4, Caleb Bupp6, Jeremy W Prokop7,8.   

Abstract

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.

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Year:  2020        PMID: 32734384      PMCID: PMC7855842          DOI: 10.1007/s00439-020-02211-w

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  74 in total

1.  SIFT: Predicting amino acid changes that affect protein function.

Authors:  Pauline C Ng; Steven Henikoff
Journal:  Nucleic Acids Res       Date:  2003-07-01       Impact factor: 16.971

2.  MUC4 expression is regulated by cystic fibrosis transmembrane conductance regulator in pancreatic adenocarcinoma cells via transcriptional and post-translational mechanisms.

Authors:  A P Singh; S C Chauhan; M Andrianifahanana; N Moniaux; J L Meza; M C Copin; I van Seuningen; M A Hollingsworth; J P Aubert; S K Batra
Journal:  Oncogene       Date:  2006-06-26       Impact factor: 9.867

3.  Classification of CFTR mutation classes.

Authors:  Fernando Augusto Lima Marson; Carmen Sílvia Bertuzzo; José Dirceu Ribeiro
Journal:  Lancet Respir Med       Date:  2016-07-01       Impact factor: 30.700

4.  Genotyping microarray for the detection of more than 200 CFTR mutations in ethnically diverse populations.

Authors:  Iris Schrijver; Eneli Oitmaa; Andres Metspalu; Phyllis Gardner
Journal:  J Mol Diagn       Date:  2005-08       Impact factor: 5.568

5.  Cystic fibrosis mutations for p.F508del compound heterozygotes predict sweat chloride levels and pancreatic sufficiency.

Authors:  R Sebro; H Levy; K Schneck; D Dimmock; B A Raby; C L Cannon; U Broeckel; N J Risch
Journal:  Clin Genet       Date:  2011-11-29       Impact factor: 4.438

6.  Improving physical realism, stereochemistry, and side-chain accuracy in homology modeling: Four approaches that performed well in CASP8.

Authors:  Elmar Krieger; Keehyoung Joo; Jinwoo Lee; Jooyoung Lee; Srivatsan Raman; James Thompson; Mike Tyka; David Baker; Kevin Karplus
Journal:  Proteins       Date:  2009

7.  Bronchoalveolar lavage fluid surfactant protein-A and surfactant protein-D are inversely related to inflammation in early cystic fibrosis.

Authors:  Terry L Noah; Paula C Murphy; Jorien J Alink; Margaret W Leigh; William M Hull; Mildred T Stahlman; Jeffrey A Whitsett
Journal:  Am J Respir Crit Care Med       Date:  2003-06-26       Impact factor: 21.405

8.  De novo transcript sequence reconstruction from RNA-seq using the Trinity platform for reference generation and analysis.

Authors:  Brian J Haas; Alexie Papanicolaou; Moran Yassour; Manfred Grabherr; Philip D Blood; Joshua Bowden; Matthew Brian Couger; David Eccles; Bo Li; Matthias Lieber; Matthew D MacManes; Michael Ott; Joshua Orvis; Nathalie Pochet; Francesco Strozzi; Nathan Weeks; Rick Westerman; Thomas William; Colin N Dewey; Robert Henschel; Richard D LeDuc; Nir Friedman; Aviv Regev
Journal:  Nat Protoc       Date:  2013-07-11       Impact factor: 13.491

9.  The chitinase-like protein YKL-40 modulates cystic fibrosis lung disease.

Authors:  Andreas Hector; Michael S D Kormann; Ines Mack; Philipp Latzin; Carmen Casaulta; Elisabeth Kieninger; Zhe Zhou; Ali Ö Yildirim; Alexander Bohla; Nikolaus Rieber; Matthias Kappler; Barbara Koller; Ernst Eber; Olaf Eickmeier; Stefan Zielen; Oliver Eickelberg; Matthias Griese; Marcus A Mall; Dominik Hartl
Journal:  PLoS One       Date:  2011-09-20       Impact factor: 3.240

10.  Genetic association and transcriptome integration identify contributing genes and tissues at cystic fibrosis modifier loci.

Authors:  Jiafen Gong; Fan Wang; Bowei Xiao; Naim Panjwani; Fan Lin; Katherine Keenan; Julie Avolio; Mohsen Esmaeili; Lin Zhang; Gengming He; David Soave; Scott Mastromatteo; Zeynep Baskurt; Sangook Kim; Wanda K O'Neal; Deepika Polineni; Scott M Blackman; Harriet Corvol; Garry R Cutting; Mitchell Drumm; Michael R Knowles; Johanna M Rommens; Lei Sun; Lisa J Strug
Journal:  PLoS Genet       Date:  2019-02-26       Impact factor: 5.917

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