The French Compassionate Program of elexacaftor-tezacaftor-ivacaftor in people with cystic fibrosis with advanced lung disease and no F508del


Journal

The European respiratory journal
ISSN: 1399-3003
Titre abrégé: Eur Respir J
Pays: England
ID NLM: 8803460

Informations de publication

Date de publication:
16 Feb 2023
Historique:
received: 20 12 2022
accepted: 08 02 2023
entrez: 16 2 2023
pubmed: 17 2 2023
medline: 17 2 2023
Statut: aheadofprint

Résumé

The European Medicines Agency has approved the cystic fibrosis transmembrane conductance regulator (CFTR) modulator combination elexacaftor-tezacaftor-ivacaftor (ETI) for people with cystic fibrosis (pwCF) carrying at least one F508del variant. The United States Food and Drug Administration (FDA) also approved ETI for pwCF carrying one of 177 rare variants. An observational study was conducted to evaluate the effectiveness of ETI in pwCF with advanced lung disease that were not eligible to ETI in Europe. All patients with no F508del variant and advanced lung disease (defined as having a percent predicted forced expiratory volume (ppFEV Among the first 84 pwCF included in the program, ETI was effective in 45 (54%) and 39 (46%) were considered to be non-responders. Among the responders 22/45 (49%) carried a Clinical benefits were observed in a large subset of pwCF with advanced lung disease and

Sections du résumé

BACKGROUND BACKGROUND
The European Medicines Agency has approved the cystic fibrosis transmembrane conductance regulator (CFTR) modulator combination elexacaftor-tezacaftor-ivacaftor (ETI) for people with cystic fibrosis (pwCF) carrying at least one F508del variant. The United States Food and Drug Administration (FDA) also approved ETI for pwCF carrying one of 177 rare variants.
METHODS METHODS
An observational study was conducted to evaluate the effectiveness of ETI in pwCF with advanced lung disease that were not eligible to ETI in Europe. All patients with no F508del variant and advanced lung disease (defined as having a percent predicted forced expiratory volume (ppFEV
RESULTS RESULTS
Among the first 84 pwCF included in the program, ETI was effective in 45 (54%) and 39 (46%) were considered to be non-responders. Among the responders 22/45 (49%) carried a
CONCLUSION CONCLUSIONS
Clinical benefits were observed in a large subset of pwCF with advanced lung disease and

Identifiants

pubmed: 36796836
pii: 13993003.02437-2022
doi: 10.1183/13993003.02437-2022
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

Copyright ©The authors 2023. For reproduction rights and permissions contact permissions@ersnet.org.

Auteurs

Pierre-Régis Burgel (PR)

Université Paris-Cité, Institut Cochin, Inserm U1016, Paris, France pierre-regis.burgel@aphp.fr.
Respiratory Medicine and Cystic Fibrosis National Reference Center; Cochin Hospital; Assistance Publique Hôpitaux de Paris (AP-HP), Paris, France.
ERN-Lung CF network, Frankfurt, Germany.

Isabelle Sermet-Gaudelus (I)

ERN-Lung CF network, Frankfurt, Germany.
Centre de de Référence Maladies Rares, Mucoviscidose et affections liées à CFTR, Pneumologie Pédiatrique et Allergologie, Hôpital Necker Enfants Malades, Assistance Publique-Hôpitaux de Paris, Paris, France.
Université Paris-Cité, Institut Necker Enfants Malades, INSERM U1151, Paris, France.

Isabelle Durieu (I)

ERN-Lung CF network, Frankfurt, Germany.
Centre de référence Adulte de la Mucoviscidose, Service de médecine interne, Hospices civils de Lyon, Pierre Bénite, France.
Université de Lyon, Équipe d'Accueil Health Services and Performance Research (HESPER) 7425, Lyon, France.

Reem Kanaan (R)

Respiratory Medicine and Cystic Fibrosis National Reference Center; Cochin Hospital; Assistance Publique Hôpitaux de Paris (AP-HP), Paris, France.
ERN-Lung CF network, Frankfurt, Germany.

Julie Macey (J)

Respiratory Medicine and Cystic Fibrosis Center, CHU de Bordeaux, Bordeaux, France.

Dominique Grenet (D)

CRCM - Centre de Transplantation Pulmonaire. Service de pneumologie, hôpital Foch, Suresnes, France.

Michele Porzio (M)

Department of Respiratory Medicine and Cystic Fibrosis Center, Federation of Translational Medicine of Strasbourg (FMTS), University Hospitals, Strasbourg, France.

Nathalie Coolen-Allou (N)

Centre Hospitalier Universitaire Félix Guyon, Saint Denis, La Réunion, France.

Raphael Chiron (R)

Cystic Fibrosis Center, Hôpital Arnaud de Villeneuve, Centre Hospitalier Universitaire de Montpellier, Montpellier, France.

Christophe Marguet (C)

Pediatric Respiratory Disease and Cystic Fibrosis Center, Hospital, UNIROUEN, Inserm EA 2656, Rouen University Hospital, Normandie Univ, Rouen, France.

Benoit Douvry (B)

Service de Pneumologie, Centre Hospitalier Intercommunal, FHU SENEC, Créteil, France.

Nadine Dufeu (N)

Department of Respiratory Medicine and Lung Transplantation, Aix Marseille Univ, APHM, Hôpital Nord, Marseille, France.

Isabelle Danner-Boucher (I)

Service de Pneumologie, L'Institut Du Thorax, CHU Nantes, Nantes, France.

Pierre Foucaud (P)

Association Vaincre la Mucoviscidose, Paris, France.

Lydie Lemonnier (L)

Association Vaincre la Mucoviscidose, Paris, France.

Emmanuelle Girodon (E)

APHP.Centre-Université de Paris Cité, Service de Médecine Génomique des Maladies de Système et d'Organe, Hôpital Cochin, Paris, France.

Jennifer Da Silva (J)

Respiratory Medicine and Cystic Fibrosis National Reference Center; Cochin Hospital; Assistance Publique Hôpitaux de Paris (AP-HP), Paris, France.
ERN-Lung CF network, Frankfurt, Germany.

Clémence Martin (C)

Université Paris-Cité, Institut Cochin, Inserm U1016, Paris, France.
Respiratory Medicine and Cystic Fibrosis National Reference Center; Cochin Hospital; Assistance Publique Hôpitaux de Paris (AP-HP), Paris, France.
ERN-Lung CF network, Frankfurt, Germany.

Classifications MeSH