Reported Adverse Events in a Multicenter Cohort of Patients Ages 6-18 Years with Cystic Fibrosis and at Least One F508del Allele Receiving Elexacaftor/Tezacaftor/Ivacaftor

J Pediatr. 2024 Nov:274:114176. doi: 10.1016/j.jpeds.2024.114176. Epub 2024 Jun 28.

Abstract

Objective: The objective of this study was to describe reported adverse events (AEs) associated with elexacaftor/tezacaftor/ivacaftor (ETI) in a pediatric sample with cystic fibrosis (CF) aged 6-18 years, with at least one F508del variant, followed at multiple Italian CF centers.

Study design: This was a retrospective, multicenter, observational study. All children receiving ETI therapy from October 2019 to December 2023 were included. We assessed the prevalence and type of any reported potential drug-related AEs, regardless of discontinuation necessity. Persistent AEs were defined as those continuing at the end of the observation period.

Results: Among 608 patients on ETI, 109 (17.9%) reported at least 1 AE. The majority (n = 85, 77.9%) were temporary, with a median duration of 11 days (range 1-441 days). Only 7 (1.1%) patients permanently discontinued treatment, suggesting good overall safety of ETI. The most common AEs leading to discontinuation were transaminase elevations (temporary 14.1%, persistent 25.9%) and urticaria (temporary 41.2%, persistent 7.4%). Creatinine phosphokinase elevation was uncommon. No significant differences in AEs were observed based on sex, age groups (6-11 vs 12-18 years), or genotype. Pre-existing CF-related liver disease was associated with an increased risk of transaminase elevations. We identified significant variability in the percentage of reported AEs (ANOVA P value .026).

Conclusions: This real-world study highlights significant variability in reported AEs. Our findings suggest that ETI is a safe and well-tolerated therapy in children and adolescents with CF. However, further long-term safety and effectiveness investigations are warranted.

Keywords: Kaftrio; Trikafta; benign intracranial hypertension; children; management; rash; transaminases.

Publication types

  • Multicenter Study
  • Observational Study

MeSH terms

  • Adolescent
  • Alleles
  • Aminophenols* / adverse effects
  • Aminophenols* / therapeutic use
  • Benzodioxoles* / adverse effects
  • Benzodioxoles* / therapeutic use
  • Child
  • Cystic Fibrosis Transmembrane Conductance Regulator* / genetics
  • Cystic Fibrosis* / drug therapy
  • Cystic Fibrosis* / genetics
  • Drug Combinations*
  • Female
  • Humans
  • Indoles* / adverse effects
  • Italy
  • Male
  • Pyrazoles / adverse effects
  • Pyridines / adverse effects
  • Pyrroles / adverse effects
  • Pyrrolidines
  • Quinolones* / adverse effects
  • Quinolones* / therapeutic use
  • Retrospective Studies

Substances

  • Benzodioxoles
  • Aminophenols
  • Cystic Fibrosis Transmembrane Conductance Regulator
  • Indoles
  • Quinolones
  • Drug Combinations
  • Pyridines
  • Pyrazoles
  • Pyrroles
  • elexacaftor
  • Pyrrolidines