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Effect of the CFTR-modulating Triple Therapy Elexacaftor - Tezacaftor - Ivacaftor

Recruiting
Conditions
Cystic Fibrosis
Interventions
Registration Number
NCT05576324
Lead Sponsor
University of Erlangen-Nürnberg Medical School
Brief Summary

The aim of this study is to investigate the frequency distribution, cytokine profile and function of peripheral, mononuclear leukocyte populations (monocytes, NK cells, T/B lymphocytes) and their correlation to clinical and biochemical parameters in patients with cystic fibrosis receiving CFTR modulatory triple therapy consisting of elexacaftor, tezacaftor and ivacaftor and to compare it with patients without CFTR modulatory therapy and healthy control subjects.

Detailed Description

The therapy of cystic fibrosis usually consists of an inhalative therapy with hy-pertonic saline and other mucolytics (e.g. dornase alpha) for secretolysis as well as a pancreatic enzyme replacement therapy. In recent years, however, the introduction of novel drugs, the so-called CFTR modulators, has revolutionized the previous treatment concept of a symptom-oriented therapy. Ivacaftor, which was approved by the FDA in 2012 for the treatment of patients with G551D mutation, causes a prolongation of the opening probability of the CFTR channel (CFTR potentiator) and was able to show a significant improvement in lung function in studies. By combining ivacaftor with the CFTR corrector lumacaftor, which improves the processing of the CFTR channel in the endoplasmic reticulum as well as its incorporation into the cell membrane, this therapeutic strategy has also been successfully tested for use in patients with F508del homozygous mutation. Also, the combination of ivacaftor with another CFTR corrector, tezacaftor, was approved for the treatment of patients with F508del heterozygous mutations in which the second mutation was classified as a mutation with residual activity and was able to show an increase in FEV1. The efficacy of this therapeutic approach was further enhanced by the combination of ivacaftor as a CFTR potentiator with tezacaftor and a next-generation CFTR corrector, elexacaftor; in the pivotal study, an improvement in FEV1 of an average of 14 points in untreated patients and 11 points in ivacaftor/tezacaftor-pretreated patients was demonstrated, as well as a significant decrease in hospitalizations due to pulmonary exacerbation. Since September 2020 in the European Union, this combination has been approved under the trade name Kaftrio® for the treatment of patients with F508del homozygous mutation or F508del heterozygous mutation and minimal function mutation. This form of therapy is based on a concept that comes closest to a causal therapy. In April 2021, the EMA granted approval for the drug for all patients older than 12 years and with evidence of at least one F508del mutation. In addition, the manufacturer applied for an extension of the approval in the EU for children aged 6-11 years based on the also very positive study results and received a positive decision from the European Medicines Agency (EMA) in November 2021. However, in addition to the clear role of the CFTR channel in epithelial tissues, it has been increasingly shown in recent years that the CFTR channel is also expressed by a variety of immune cells of the innate as well as the acquired immune system, such as neutrophils, macrophages, monocytes, and B and T lymphocytes. Its absence or dysfunction in cystic fibrosis seems to trigger a disturbed regulation or an exaggerated reaction of various immune responses.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
130
Inclusion Criteria
  • Patients (m/f/d) with molecularly genetically confirmed cystic fibrosis aged 6 years and older.
  • Do not meet any of the exclusion criteria
  • Written informed consent
  • For study arm "Kaftrio® ongoing": Kaftrio® therapy for at least 6 months
  • For study arm "Kaftrio® longitudinal": no Kaftrio® therapy started yet
Exclusion Criteria
  • Use of inhaled or systemic glucocorticoids as part of a permanent medication regimen
  • Pregnancy

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Arm && Interventions
GroupInterventionDescription
under ETIElexacaftor / Ivacaftor / TezacaftorPatients with diagnosed CF already receiving ETI therapy for 6 months
longitudinalElexacaftor / Ivacaftor / TezacaftorInclusion of patients with diagnosed CF prior ETI therapy, follow-up visit after 6 months
Primary Outcome Measures
NameTimeMethod
Peripheral Blood Immunograms6 months ETI

Relative and absolute peripheral blood immune cell count as determined by multicolor flow cytometry

Secondary Outcome Measures
NameTimeMethod
Respiratory function test (FEV1, FVC)6 months ETI

Respiratory function test (FEV1, FVC) as measured by bodyplethysmography

Prothrombin time6 months ETI

Prothrombin time as defined by Quick percent

Coagulation factors6 months ETI

Vitamin K- dependent coagulation factors (II, VII, IX, X) as measured in %

Plasmatic glutamate dehydrogenase6 months ETI

Plasmatic glutamate dehydrogenase (defined in U/l)

Serum bile acids6 months ETI

Level of serum bile acids as measured by mass spectrometry

Plasmatic bilirubin6 months ETI

Total and direct plasmatic bilirubin as defined in mg/dl

Plasmatic creatinin6 months ETI

Plasmatic creatinin (defined in mg/dl)

Ageprior ETI

Patients' age in years

Functional pulmonary magnetic resonance imaging6 months ETI

Ventilation defect, perfusion defect, combined defects in longitudinal pediatric cohort (6-11 yrs)

Liver transaminases6 months ETI

Aspartate/Alanine aminotransferase (AST, ALt) as defined in U/l

Plasmatic C-reactive proteine6 months ETI

Plasmatic C-reactive proteine (defined in mg/l)

Plasmatic cholinesterase6 months ETI

Plasmatic cholinesterase (defined in U/l)

Sweat chloride6 months ETI

Sweat chloride level (defined in mmol/l)

BMI6 months ETI

BMI in kg/m\^2

Microbial colonization status6 months ETI

Microbial colonization status as defined by microbiological reports

Shear Wave Velocity (SWV)6 months ETI

Difference of mean/median SWV in treated vs. untreated patients as measured by Acoustic Radiation Force Impulse Imaging (ARFI)

Plasma electrolytes6 months ETI

Plasma electrolytes (Na, Cl) as defined by mmol/l

Plasmatic albumine6 months ETI

Plasma levels of albumine (defined in g/dl)

Attenuation Coefficient (AC)6 months ETI

Difference of mean/median AC in treated vs. untreated pediatric patients (6-11 yrs) as measured by Ultrasound-guided attenuation parameter (UGAP)

Blood cell count6 months ETI

Blood cell count as defined in x10\^3/µl

Erythrocytoid hemoglobin A1c6 months ETI

Erythrocytoid hemoglobin A1c in %

Serum immunoglobulins6 months ETI

Serum immunoglobulins G, A, M, E (defined in g/l)

Individual concomitant medication regime6 months ETI

Individual concomitant medication regime

Neutrophilic dihydrorhodamine assay6 months ETI

Assay for determination of neutrophilic reactive oxygen species (measured as stimulation index)

Trial Locations

Locations (1)

University Hospital Erlange, Department of Pediatrics

🇩🇪

Erlangen, Bavaria, Germany

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