Literature DB >> 31570318

The future of cystic fibrosis care: a global perspective.

Scott C Bell1, Marcus A Mall2, Hector Gutierrez3, Milan Macek4, Susan Madge5, Jane C Davies6, Pierre-Régis Burgel7, Elizabeth Tullis8, Claudio Castaños9, Carlo Castellani10, Catherine A Byrnes11, Fiona Cathcart5, Sanjay H Chotirmall12, Rebecca Cosgriff13, Irmgard Eichler14, Isabelle Fajac7, Christopher H Goss15, Pavel Drevinek16, Philip M Farrell17, Anna M Gravelle18, Trudy Havermans19, Nicole Mayer-Hamblett20, Nataliya Kashirskaya21, Eitan Kerem22, Joseph L Mathew23, Edward F McKone24, Lutz Naehrlich25, Samya Z Nasr26, Gabriela R Oates3, Ciaran O'Neill27, Ulrike Pypops28, Karen S Raraigh29, Steven M Rowe3, Kevin W Southern30, Sheila Sivam31, Anne L Stephenson8, Marco Zampoli32, Felix Ratjen33.   

Abstract

The past six decades have seen remarkable improvements in health outcomes for people with cystic fibrosis, which was once a fatal disease of infants and young children. However, although life expectancy for people with cystic fibrosis has increased substantially, the disease continues to limit survival and quality of life, and results in a large burden of care for people with cystic fibrosis and their families. Furthermore, epidemiological studies in the past two decades have shown that cystic fibrosis occurs and is more frequent than was previously thought in populations of non-European descent, and the disease is now recognised in many regions of the world. The Lancet Respiratory Medicine Commission on the future of cystic fibrosis care was established at a time of great change in the clinical care of people with the disease, with a growing population of adult patients, widespread genetic testing supporting the diagnosis of cystic fibrosis, and the development of therapies targeting defects in the cystic fibrosis transmembrane conductance regulator (CFTR), which are likely to affect the natural trajectory of the disease. The aim of the Commission was to bring to the attention of patients, health-care professionals, researchers, funders, service providers, and policy makers the various challenges associated with the changing landscape of cystic fibrosis care and the opportunities available for progress, providing a blueprint for the future of cystic fibrosis care. The discovery of the CFTR gene in the late 1980s triggered a surge of basic research that enhanced understanding of the pathophysiology and the genotype-phenotype relationships of this clinically variable disease. Until recently, available treatments could only control symptoms and restrict the complications of cystic fibrosis, but advances in CFTR modulator therapies to address the basic defect of cystic fibrosis have been remarkable and the field is evolving rapidly. However, CFTR modulators approved for use to date are highly expensive, which has prompted questions about the affordability of new treatments and served to emphasise the considerable gap in health outcomes for patients with cystic fibrosis between high-income countries, and low-income and middle-income countries (LMICs). Advances in clinical care have been multifaceted and include earlier diagnosis through the implementation of newborn screening programmes, formalised airway clearance therapy, and reduced malnutrition through the use of effective pancreatic enzyme replacement and a high-energy, high-protein diet. Centre-based care has become the norm in high-income countries, allowing patients to benefit from the skills of expert members of multidisciplinary teams. Pharmacological interventions to address respiratory manifestations now include drugs that target airway mucus and airway surface liquid hydration, and antimicrobial therapies such as antibiotic eradication treatment in early-stage infections and protocols for maintenance therapy of chronic infections. Despite the recent breakthrough with CFTR modulators for cystic fibrosis, the development of novel mucolytic, anti-inflammatory, and anti-infective therapies is likely to remain important, especially for patients with more advanced stages of lung disease. As the median age of patients with cystic fibrosis increases, with a rapid increase in the population of adults living with the disease, complications of cystic fibrosis are becoming increasingly common. Steps need to be taken to ensure that enough highly qualified professionals are present in cystic fibrosis centres to meet the needs of ageing patients, and new technologies need to be adopted to support communication between patients and health-care providers. In considering the future of cystic fibrosis care, the Commission focused on five key areas, which are discussed in this report: the changing epidemiology of cystic fibrosis (section 1); future challenges of clinical care and its delivery (section 2); the building of cystic fibrosis care globally (section 3); novel therapeutics (section 4); and patient engagement (section 5). In panel 1, we summarise key messages of the Commission. The challenges faced by all stakeholders in building and developing cystic fibrosis care globally are substantial, but many opportunities exist for improved care and health outcomes for patients in countries with established cystic fibrosis care programmes, and in LMICs where integrated multidisciplinary care is not available and resources are lacking at present. A concerted effort is needed to ensure that all patients with cystic fibrosis have access to high-quality health care in the future.
Copyright © 2020 Elsevier Ltd. All rights reserved.

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Year:  2019        PMID: 31570318      PMCID: PMC8862661          DOI: 10.1016/S2213-2600(19)30337-6

Source DB:  PubMed          Journal:  Lancet Respir Med        ISSN: 2213-2600            Impact factor:   30.700


  488 in total

1.  Non-invasive prenatal diagnosis (NIPD) of cystic fibrosis: an optimized protocol using MEMO fluorescent PCR to detect the p.Phe508del mutation.

Authors:  C Guissart; C Dubucs; C Raynal; A Girardet; F Tran Mau Them; V Debant; C Rouzier; A Boureau-Wirth; E Haquet; J Puechberty; E Bieth; D Dupin Deguine; P Khau Van Kien; M P Brechard; V Pritchard; M Koenig; M Claustres; M C Vincent
Journal:  J Cyst Fibros       Date:  2016-12-28       Impact factor: 5.482

2.  A standardized approach to estimating survival statistics for population-based cystic fibrosis registry cohorts.

Authors:  Jenna Sykes; Sanja Stanojevic; Christopher H Goss; Bradley S Quon; Bruce C Marshall; Kristofer Petren; Josh Ostrenga; Aliza Fink; Alexander Elbert; Anne L Stephenson
Journal:  J Clin Epidemiol       Date:  2015-10-03       Impact factor: 6.437

Review 3.  Emerging role of cystic fibrosis transmembrane conductance regulator - an epithelial chloride channel in gastrointestinal cancers.

Authors:  Yuning Hou; Xiaoqing Guan; Zhe Yang; Chunying Li
Journal:  World J Gastrointest Oncol       Date:  2016-03-15

Review 4.  Ion Channel Modulators in Cystic Fibrosis.

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

5.  Efficacy and safety of ivacaftor in patients aged 6 to 11 years with cystic fibrosis with a G551D mutation.

Authors:  Jane C Davies; Claire E Wainwright; Gerard J Canny; Mark A Chilvers; Michelle S Howenstine; Anne Munck; Jochen G Mainz; Sally Rodriguez; Haihong Li; Karl Yen; Claudia L Ordoñez; Richard Ahrens
Journal:  Am J Respir Crit Care Med       Date:  2013-06-01       Impact factor: 21.405

Review 6.  Standard versus biofilm antimicrobial susceptibility testing to guide antibiotic therapy in cystic fibrosis.

Authors:  Valerie Waters; Felix Ratjen
Journal:  Cochrane Database Syst Rev       Date:  2017-10-05

7.  Bias in CFTR screening panels.

Authors:  Patrick R Sosnay; Carlo Castellani; Christopher M Penland; Johanna M Rommens; Michelle Lewis; Karen S Raraigh; Mary Corey; Garry R Cutting
Journal:  Genet Med       Date:  2015-10-29       Impact factor: 8.822

8.  Tele-Exercise as a Promising Tool to Promote Exercise in Children With Cystic Fibrosis.

Authors:  Jen Jen Chen; Dan M Cooper; Fadia Haddad; Anna Sladkey; Eliezer Nussbaum; Shlomit Radom-Aizik
Journal:  Front Public Health       Date:  2018-09-28

Review 9.  Recent progress in translational cystic fibrosis research using precision medicine strategies.

Authors:  Deborah M Cholon; Martina Gentzsch
Journal:  J Cyst Fibros       Date:  2017-10-04       Impact factor: 5.482

10.  International Committee on Mental Health in Cystic Fibrosis: Cystic Fibrosis Foundation and European Cystic Fibrosis Society consensus statements for screening and treating depression and anxiety.

Authors:  Alexandra L Quittner; Janice Abbott; Anna M Georgiopoulos; Lutz Goldbeck; Beth Smith; Sarah E Hempstead; Bruce Marshall; Kathryn A Sabadosa; Stuart Elborn
Journal:  Thorax       Date:  2015-10-09       Impact factor: 9.139

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  147 in total

Review 1.  Building global development strategies for cf therapeutics during a transitional cftr modulator era.

Authors:  N Mayer-Hamblett; S van Koningsbruggen-Rietschel; D P Nichols; D R VanDevanter; J C Davies; T Lee; A G Durmowicz; F Ratjen; M W Konstan; K Pearson; S C Bell; J P Clancy; J L Taylor-Cousar; K De Boeck; S H Donaldson; D G Downey; P A Flume; P Drevinek; C H Goss; I Fajac; A S Magaret; B S Quon; S M Singleton; J M VanDalfsen; G Z Retsch-Bogart
Journal:  J Cyst Fibros       Date:  2020-06-07       Impact factor: 5.482

Review 2.  Hyperinflammation and airway surface liquid dehydration in cystic fibrosis: purinergic system as therapeutic target.

Authors:  Thiago Inácio Teixeira do Carmo; Victor Emanuel Miranda Soares; Jonatha Wruck; Fernanda Dos Anjos; Débora Tavares de Resende E Silva; Sarah Franco Vieira de Oliveira Maciel; Margarete Dulce Bagatini
Journal:  Inflamm Res       Date:  2021-04-27       Impact factor: 4.575

3.  Succinate links mitochondria to deadly bacteria in cystic fibrosis.

Authors:  Speranza Esposito; Valeria Rachela Villella; Federica Rossin; Antonella Tosco; Valeria Raia; Alessandro Luciani
Journal:  Ann Transl Med       Date:  2019-12

4.  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

5.  Triple Therapy for Cystic Fibrosis Phe508del-Gating and -Residual Function Genotypes.

Authors:  Peter J Barry; Marcus A Mall; Antonio Álvarez; Carla Colombo; Karin M de Winter-de Groot; Isabelle Fajac; Kimberly A McBennett; Edward F McKone; Bonnie W Ramsey; Sivagurunathan Sutharsan; Jennifer L Taylor-Cousar; Elizabeth Tullis; Neil Ahluwalia; Lucy S Jun; Samuel M Moskowitz; Valentin Prieto-Centurion; Simon Tian; David Waltz; Fengjuan Xuan; Yaohua Zhang; Steven M Rowe; Deepika Polineni
Journal:  N Engl J Med       Date:  2021-08-26       Impact factor: 91.245

6.  Quantification of Cystic Fibrosis Lung Disease with Radiomics-based CT Scores.

Authors:  Guillaume Chassagnon; Evangelia I Zacharaki; Sébastien Bommart; Pierre-Régis Burgel; Raphael Chiron; Séverine Dangeard; Nikos Paragios; Clémence Martin; Marie-Pierre Revel
Journal:  Radiol Cardiothorac Imaging       Date:  2020-12-17

Review 7.  Accelerated Approval or Risk Reduction? How Response Biomarkers Advance Therapeutics through Clinical Trials in Cystic Fibrosis.

Authors:  N Mayer-Hamblett; D R VanDevanter
Journal:  Trends Mol Med       Date:  2020-08-28       Impact factor: 11.951

Review 8.  Pseudomonas aeruginosa adaptation and evolution in patients with cystic fibrosis.

Authors:  Elio Rossi; Ruggero La Rosa; Jennifer A Bartell; Rasmus L Marvig; Janus A J Haagensen; Lea M Sommer; Søren Molin; Helle Krogh Johansen
Journal:  Nat Rev Microbiol       Date:  2020-11-19       Impact factor: 60.633

Review 9.  [Evidence-based treatment of cystic fibrosis].

Authors:  F C Ringshausen; T Hellmuth; A-M Dittrich
Journal:  Internist (Berl)       Date:  2020-12       Impact factor: 0.743

Review 10.  Cystic Fibrosis: Emergence of Highly Effective Targeted Therapeutics and Potential Clinical Implications.

Authors:  Marcus A Mall; Nicole Mayer-Hamblett; Steven M Rowe
Journal:  Am J Respir Crit Care Med       Date:  2020-05-15       Impact factor: 21.405

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