Literature DB >> 28406713

An Observational Study of Outcomes and Tolerances in Patients with Cystic Fibrosis Initiated on Lumacaftor/Ivacaftor.

Mark T Jennings1, Rebecca Dezube1, Shruti Paranjape2, Natalie E West1, Gina Hong3, Andrew Braun1, Jonathan Grant4, Christian A Merlo1, Noah Lechtzin1.   

Abstract

RATIONALE: In July 2015, the U.S. Food and Drug Administration approved lumacaftor/ivacaftor for use in patients with cystic fibrosis (CF). This drug targets the primary defect in the CFTR protein that is conferred by the F508del CFTR mutation.
OBJECTIVE: As there is limited experience with this therapy outside of clinical trials, this study aims to examine the clinical experience of this new drug in a population with CF.
RESULTS: Retrospective cohort study of individuals followed at the Johns Hopkins CF Center who initiated treatment with lumacaftor/ivacaftor. Patients were followed from 1 year before drug initiation to up to 11 months postinitiation. Key exclusion criteria include previous exposure to lumacaftor/ivacaftor through participation in a clinical trial. Of 116 individuals identified who started lumacaftor/ivacaftor treatment, 46 (39.7%) reported adverse effects related to lumacaftor/ivacaftor, with the vast majority (82.2%) being pulmonary adverse effects, and 20 (17.2%) discontinued lumacaftor/ivacaftor because of adverse effects. The mean change in FEV1% predicted was 0.11% (range: -39% to +20%; P = 0.9). Nineteen individuals had an FEV1% predicted of 40% or less before treatment, and there was a higher percentage of patients in this subgroup who reported adverse effects (57.9%) and a higher percentage of patients who discontinued lumacaftor/ivacaftor (31.6%). Female sex was associated with a higher odds of drug discontinuation (adjusted odds ratio, 3.12, 95% confidence interval, 1.04-9.38).
CONCLUSIONS: This study highlights the prevalence of adverse effects in a CF population newly exposed to lumacaftor/ivacaftor and demonstrates a relatively high rate of drug intolerance.

Entities:  

Keywords:  CFTR modulators; cystic fibrosis; post-marketing study

Mesh:

Substances:

Year:  2017        PMID: 28406713     DOI: 10.1513/AnnalsATS.201701-058OC

Source DB:  PubMed          Journal:  Ann Am Thorac Soc        ISSN: 2325-6621


  20 in total

Review 1.  Safety and efficacy of treatment with lumacaftor in combination with ivacaftor in younger patients with cystic fibrosis.

Authors:  Pi Chun Cheng; Stamatia Alexiou; Ronald C Rubenstein
Journal:  Expert Rev Respir Med       Date:  2019-04-08       Impact factor: 3.772

2.  Whole-blood transcriptomic responses to lumacaftor/ivacaftor therapy in cystic fibrosis.

Authors:  Benjamin T Kopp; James Fitch; Lisa Jaramillo; Chandra L Shrestha; Frank Robledo-Avila; Shuzhong Zhang; Sabrina Palacios; Fred Woodley; Don Hayes; Santiago Partida-Sanchez; Octavio Ramilo; Peter White; Asuncion Mejias
Journal:  J Cyst Fibros       Date:  2019-08-29       Impact factor: 5.482

3.  Lipid Nanoparticle-Delivered Chemically Modified mRNA Restores Chloride Secretion in Cystic Fibrosis.

Authors:  Ema Robinson; Kelvin D MacDonald; Kai Slaughter; Madison McKinney; Siddharth Patel; Conroy Sun; Gaurav Sahay
Journal:  Mol Ther       Date:  2018-06-15       Impact factor: 11.454

4.  Tezacaftor/Ivacaftor in Subjects with Cystic Fibrosis and F508del/F508del-CFTR or F508del/G551D-CFTR.

Authors:  Scott H Donaldson; Joseph M Pilewski; Matthias Griese; Jon Cooke; Lakshmi Viswanathan; Elizabeth Tullis; Jane C Davies; Julie A Lekstrom-Himes; Linda T Wang
Journal:  Am J Respir Crit Care Med       Date:  2018-01-15       Impact factor: 21.405

Review 5.  RNA delivery biomaterials for the treatment of genetic and rare diseases.

Authors:  Weiyu Zhao; Xucheng Hou; Olivia G Vick; Yizhou Dong
Journal:  Biomaterials       Date:  2019-06-20       Impact factor: 12.479

Review 6.  An Update on CFTR Modulators as New Therapies for Cystic Fibrosis.

Authors:  John A King; Anna-Louise Nichols; Sian Bentley; Siobhan B Carr; Jane C Davies
Journal:  Paediatr Drugs       Date:  2022-05-16       Impact factor: 3.022

Review 7.  Toward inclusive therapy with CFTR modulators: Progress and challenges.

Authors:  Jennifer Guimbellot; Jyoti Sharma; Steven M Rowe
Journal:  Pediatr Pulmonol       Date:  2017-09-07

8.  Quantitative Method for the Analysis of Ivacaftor, Hydroxymethyl Ivacaftor, Ivacaftor Carboxylate, Lumacaftor, and Tezacaftor in Plasma and Sputum Using Liquid Chromatography With Tandem Mass Spectrometry and Its Clinical Applicability.

Authors:  Steffie E M Vonk; Marloes van der Meer-Vos; Lieuwe D J Bos; Anne H Neerincx; Christof J Majoor; Anke-Hilse Maitland-van der Zee; Ron A A Mathôt; E Marleen Kemper
Journal:  Ther Drug Monit       Date:  2021-08-01       Impact factor: 3.681

Review 9.  Epithelial vectorial ion transport in cystic fibrosis: Dysfunction, measurement, and pharmacotherapy to target the primary deficit.

Authors:  Lucy A Clunes; Naia McMillan-Castanares; Neil Mehta; Afia Mesadieu; Jorge Rodriguez; Mary Maj; Mark T Clunes
Journal:  SAGE Open Med       Date:  2020-06-25

10.  Prolonged co-treatment with HGF sustains epithelial integrity and improves pharmacological rescue of Phe508del-CFTR.

Authors:  Ana M Matos; Andreia Gomes-Duarte; Márcia Faria; Patrícia Barros; Peter Jordan; Margarida D Amaral; Paulo Matos
Journal:  Sci Rep       Date:  2018-08-29       Impact factor: 4.379

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