Literature DB >> 27916649

New horizons for cystic fibrosis treatment.

Isabelle Fajac1, Kris De Boeck2.   

Abstract

Cystic fibrosis is an inherited multi-system disease associated with chronic lung infection, malabsorption, salt loss syndromes, male infertility and leading to numerous comorbidities. The landscape in cystic fibrosis care has changed markedly with currently more adult patients than children in many countries. Over 2000 different mutations in the CFTR gene have been reported and the majority are extremely rare. Understanding how CFTR mutations translate to disturbed synthesis or function of the CFTR protein has opened the way to 'personalized' treatments to correct the basic defect. The first 2 drugs have reached the clinic: a CFTR potentiator to augment CFTR channel function, and the combination of this potentiator with a corrector to increase CFTR expression at the cell membrane. To obtain robust correction of CFTR expression at the cell membrane, combinations of correctors with additive efficacy are under investigation. Other mutation type-specific treatments under clinical investigation are premature stop codon-read through drugs and antisense oligonucleotides that correct the basic defect at the mRNA level. Restoring the defective gene by gene editing can already be achieved ex vivo. Mutation agnostic treatments are explored as well: stabilizing CFTR expression at the cell membrane, circumventing the CFTR channel by blocking or activating other ion channels, and gene therapy. Combinations of these therapies can be anticipated. The pipeline of corrective strategies under clinical investigation is increasing continuously and a rising number of pharmaceutical companies are entering the field.
Copyright © 2016 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  CFTR; CFTR modulator; Corrector; Cystic fibrosis; Gene therapy; Potentiator; Read-through agent

Mesh:

Substances:

Year:  2016        PMID: 27916649     DOI: 10.1016/j.pharmthera.2016.11.009

Source DB:  PubMed          Journal:  Pharmacol Ther        ISSN: 0163-7258            Impact factor:   12.310


  20 in total

1.  AJRCCM: 100-Year Anniversary. Progress along the Pathway of Discovery Leading to Treatment and Cure of Cystic Fibrosis.

Authors:  Bonnie W Ramsey; Michael J Welsh
Journal:  Am J Respir Crit Care Med       Date:  2017-05-01       Impact factor: 21.405

2.  Agonism of the TMEM16A calcium-activated chloride channel modulates airway smooth muscle tone.

Authors:  Jennifer Danielsson; Aisha S Kuforiji; Gene T Yocum; Yi Zhang; Dingbang Xu; George Gallos; Charles W Emala
Journal:  Am J Physiol Lung Cell Mol Physiol       Date:  2019-11-20       Impact factor: 5.464

3.  Deregulation of the growth hormone/insulin-like growth factor-1 axis in adults with cystic fibrosis.

Authors:  C Pascucci; R V De Biase; D Savi; S Quattrucci; A M Isidori; C Lubrano; L Gnessi; A Lenzi
Journal:  J Endocrinol Invest       Date:  2017-11-02       Impact factor: 4.256

4.  Pancreatic lipomatosis in cystic fibrosis: Rare manifestation of an uncommon disease.

Authors:  Harshal S Mandavdhare; Amit Kumar; Vishal Sharma; Surinder S Rana
Journal:  Intractable Rare Dis Res       Date:  2017-05

5.  The fractional excretion of sodium in patients with cystic fibrosis treated with oral sodium chloride.

Authors:  Majid Keivanfar; Sosan Daris; Mohsen Reisi; Mehryar Mehrkesh
Journal:  Am J Clin Exp Urol       Date:  2020-12-15

Review 6.  Ion Channel Modulators in Cystic Fibrosis.

Authors:  Martina Gentzsch; Marcus A Mall
Journal:  Chest       Date:  2018-05-08       Impact factor: 9.410

Review 7.  Beyond cystic fibrosis transmembrane conductance regulator therapy: a perspective on gene therapy and small molecule treatment for cystic fibrosis.

Authors:  Elena K Schneider-Futschik
Journal:  Gene Ther       Date:  2019-07-12       Impact factor: 5.250

8.  Clcn7F318L/+ as a new mouse model of Albers-Schönberg disease.

Authors:  J Caetano-Lopes; S G Lessard; S Hann; K Espinoza; K S Kang; K E Lim; D J Horan; H R Noonan; D Hu; R Baron; A G Robling; M L Warman
Journal:  Bone       Date:  2017-09-20       Impact factor: 4.626

9.  Mechanisms of KCNQ1 channel dysfunction in long QT syndrome involving voltage sensor domain mutations.

Authors:  Hui Huang; Georg Kuenze; Jarrod A Smith; Keenan C Taylor; Amanda M Duran; Arina Hadziselimovic; Jens Meiler; Carlos G Vanoye; Alfred L George; Charles R Sanders
Journal:  Sci Adv       Date:  2018-03-07       Impact factor: 14.136

10.  Thymosin α1 represents a potential potent single-molecule-based therapy for cystic fibrosis.

Authors:  Luigina Romani; Vasilis Oikonomou; Silvia Moretti; Rossana G Iannitti; Maria Cristina D'Adamo; Valeria R Villella; Marilena Pariano; Luigi Sforna; Monica Borghi; Marina M Bellet; Francesca Fallarino; Maria Teresa Pallotta; Giuseppe Servillo; Eleonora Ferrari; Paolo Puccetti; Guido Kroemer; Mauro Pessia; Luigi Maiuri; Allan L Goldstein; Enrico Garaci
Journal:  Nat Med       Date:  2017-04-10       Impact factor: 53.440

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