Standards of care for CFTR variant-specific therapy (including modulators) for people with cystic fibrosis
Publication date
2023-01
Authors
Southern, Kevin W.
Castellani, Carlo
Lammertyn, Elise
Smyth, Alan
VanDevanter, Donald
van Koningsbruggen-Rietschel, Silke
Barben, Jürg
Bevan, Amanda
Brokaar, Edwin
Collins, Sarah
Editors
Advisors
Supervisors
Document Type
Article
Metadata
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License
cc_by
Abstract
Cystic fibrosis (CF) has entered the era of variant-specific therapy, tailored to the genetic variants in the Cystic Fibrosis Transmembrane Conductance Regulator (CFTR) gene. CFTR modulators, the first variant-specific therapy available, have transformed the management of CF. The latest standards of care from the European CF Society (2018) did not include guidance on variant-specific therapy, as CFTR modulators were becoming established as a novel therapy. We have produced interim standards to guide healthcare professionals in the provision of variant-specific therapy for people with CF. Here we provide evidence-based guidance covering the spectrum of care, established using evidence from systematic reviews and expert opinion. Statements were reviewed by key stakeholders using Delphi methodology, with agreement (≥80%) achieved for all statements after one round of consultation. Issues around accessibility are discussed and there is clear consensus that all eligible people with CF should have access to variant-specific therapy.
Keywords
CFTR, CFTR modulators, Cystic fibrosis, Guidelines, Variant-specific therapy, Pediatrics, Perinatology, and Child Health, Pulmonary and Respiratory Medicine
Citation
Southern, K W, Castellani, C, Lammertyn, E, Smyth, A, VanDevanter, D, van Koningsbruggen-Rietschel, S, Barben, J, Bevan, A, Brokaar, E, Collins, S, Connett, G J, Daniels, T W V, Davies, J, Declercq, D, Gartner, S, Gramegna, A, Hamilton, N, Hauser, J, Kashirskaya, N, Kessler, L, Lowdon, J, Makukh, H, Martin, C, Morrison, L, Nazareth, D, Noordhoek, J, O'Neill, C, Owen, E, Oxley, H, Raraigh, K S, Raynal, C, Robinson, K, Roehmel, J, Schwarz, C, Sermet, I, Shteinberg, M, Sinha, I, Takawira, C, van Mourik, P, Verkleij, M, Waller, M D & Duff, A 2023, 'Standards of care for CFTR variant-specific therapy (including modulators) for people with cystic fibrosis', Journal of Cystic Fibrosis, vol. 22, no. 1, pp. 17-30. https://doi.org/10.1016/j.jcf.2022.10.002