MedPath

Study of Ivacaftor in Cystic Fibrosis Subjects Aged 6 to 11 Years With the G551D Mutation

Phase 3
Completed
Conditions
Cystic Fibrosis
Interventions
Drug: Ivacaftor
Drug: Placebo
Registration Number
NCT00909727
Lead Sponsor
Vertex Pharmaceuticals Incorporated
Brief Summary

The purpose of this study was to evaluate the efficacy and safety of ivacaftor in subjects with cystic fibrosis aged 6 to 11 years who have the G551D mutation in the cystic fibrosis transmembrane conductance regulator (CFTR) gene. Ivacaftor is a potent and selective potentiator of wild-type, G551D, F508del, and R117H forms of human CFTR protein. Potentiators are pharmacological agents that increase the chloride ion transport properties of the channel in the presence of cyclic adenosine monophosphate (AMP)-dependent protein kinase A (PKA) activation.

Detailed Description

This is a Phase 3, 2-part, randomized, double-blind, placebo-controlled, parallel group multicenter study of orally administered ivacaftor in subjects with cystic fibrosis (CF) 6 to 11 years of age who have the G551D-CFTR mutation and a forced expiratory volume in 1 second (FEV1) between 90% and 105% predicted (using Knudson standards).

Based on in vitro studies and pharmacologic, pharmacokinetic (PK), and safety profiles, ivacaftor was selected for clinical development as a possible treatment for patients with CF. Patients with the G551D mutation were the targeted population for this study because ivacaftor is a potentiator of the gating effect of the CFTR protein, and the most prevalent mutation with a gating defect in CF is the G551D mutation.

This study was conducted in 2 parts. Part A was conducted to analyze the PK properties of ivacaftor and to determine the most appropriate dose to administer to subjects in Part B of this study. Part B explored the safety and efficacy of ivacaftor over long-term treatment in subjects 6 to 11 years of age.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
52
Inclusion Criteria
  • Weighing at least 15 kg
  • Confirmed diagnosis of cystic fibrosis (CF) and G551D mutation in at least 1 allele
  • Forced expiratory volume in 1 second (FEV1) of 40% to 105% (inclusive) of predicted normal for age, gender, and height (Knudson standards) at Screening
  • Able to swallow tablets
  • As judged by the investigator, parent or legal guardian and subject must have been able to understand protocol requirements, restrictions, and instructions, and the parent or legal guardian should have been able to ensure that the subject complied with, and was likely to complete, the study as planned
  • Parent or legal guardian must have signed the informed consent form and corresponding assent must be obtained from the subject
  • Willing to use at least 1 highly effective birth control method during the study
  • No clinically significant abnormalities that would have interfered with the study assessments, as judged by the investigator
Read More
Exclusion Criteria
  • History of any illness or condition that might confound the results of the study or pose an additional risk in administering study drug to the subject
  • Acute respiratory infection, pulmonary exacerbation, or changes in therapy for pulmonary disease within 4 weeks of Day 1 of the study
  • Abnormal liver function ≥ 3x the upper limit of normal
  • Abnormal renal function at Screening
  • History of solid organ or hematological transplantation
  • Ongoing participation in another therapeutic clinical study or prior participation in an investigational drug study within 30 days prior to Screening
  • Use of inhaled hypertonic saline treatment
  • Concomitant use of any inhibitors or inducers of cytochrome P450 3A4 (CYP 3A4)
Read More

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
150 mg Ivacaftor q12hIvacaftorSubjects who received 150 mg of ivacaftor q12h for up to 48 weeks.
PlaceboPlaceboSubjects who received placebo every 12 hours (q12h) for up to 48 weeks.
Primary Outcome Measures
NameTimeMethod
Absolute Change From Baseline in Percent Predicted Forced Expiratory Volume in 1 Second (FEV1) Through Week 24baseline through 24 weeks

Spirometry (as measured by FEV1) is a standardized assessment to evaluate lung function that is the most widely used endpoint in cystic fibrosis studies.

Secondary Outcome Measures
NameTimeMethod
Absolute Change From Baseline in Percent Predicted Forced Expiratory Volume in 1 Second (FEV1) Through Week 48baseline through 48 weeks

Spirometry (as measured by FEV1) is a standardized assessment to evaluate lung function that is the most widely used endpoint in cystic fibrosis studies.

Absolute Change From Baseline in Cystic Fibrosis Questionnaire-Revised (CFQ-R) Through Week 24 and Week 48 (Respiratory Domain Score, Children)baseline through 24 weeks and 48 weeks

The CFQ-R is a health-related quality of life measure for subjects with cystic fibrosis. Each domain is scored from 0 (worst) to 100 (best). A difference of at least 4 points in the respiratory domain score of the CFQ-R is considered a minimal clinically important difference (MCID).

Absolute Change From Baseline in Sweat Chloride Concentration Through Week 24 and Week 48baseline through 24 weeks and 48 weeks

The sweat chloride (quantitative pilocarpine iontophoresis) test is a standard diagnostic tool for cystic fibrosis (CF), serving as an indicator of cystic fibrosis transmembrane conductance regulator (CFTR) activity.

Absolute Change From Baseline in Weight at Week 24 and Week 48baseline to 24 weeks and 48 weeks

As malnutrition is common in patients with cystic fibrosis (CF) because of increased energy expenditures due to lung disease and fat malabsorption, body weight is an important clinical measure of nutritional status.

Trial Locations

Locations (29)

The Children's Mercy Hospital

🇺🇸

Kansas City, Missouri, United States

Children's Memorial Hospital

🇺🇸

Chicago, Illinois, United States

Riley Hospital for Children

🇺🇸

Indianapolis, Indiana, United States

Children's Hospital of Michigan

🇺🇸

Detroit, Michigan, United States

The Cystic Fibrosis Center of Chicago

🇺🇸

Glenview, Illinois, United States

Helen DeVos Children's Hospital Spectrum Health Hospitals

🇺🇸

Grand Rapids, Michigan, United States

University of Minnesota

🇺🇸

Minneapolis, Minnesota, United States

East Tennessee Children's Hospital Pediatric Pulmonary and Respiratory Care

🇺🇸

Knoxville, Tennessee, United States

University of Iowa Department of Pediatrics

🇺🇸

Iowa City, Iowa, United States

Princess Margaret Hospital for Children

🇦🇺

Subiaco, Western Australia, Australia

University of Alabama

🇺🇸

Birmingham, Alabama, United States

Emory Cystic Fibrosis Center

🇺🇸

Atlanta, Georgia, United States

Massachusetts General Hospital

🇺🇸

Boston, Massachusetts, United States

Children's Hospital Boston

🇺🇸

Boston, Massachusetts, United States

University of Michigan

🇺🇸

Ann Arbor, Michigan, United States

University of Nebraska Medical Center Pediatric Pulmonary/ CF

🇺🇸

Omaha, Nebraska, United States

University of Utah Pediatric Pulmonology

🇺🇸

Salt Lake City, Utah, United States

The Children's Hospital Westmead

🇦🇺

Westmead, New South Wales, Australia

Royal Children's Hospital Brisbane

🇦🇺

Herston, Queensland, Australia

University of Virginia Pediatric Respiratory Medicine

🇺🇸

Charlottesville, Virginia, United States

Royal Children's Hospital Melbourne

🇦🇺

Parkville, Victoria, Australia

Our Lady's Children's Hospital

🇮🇪

Dublin, Ireland

Hôpital Robert Debré - Service de gastro-entérologiemucoviscidose et nutrition

🇫🇷

Paris, France

Kinder- und Jugendklinik Universitätsklinikum Erlangen

🇩🇪

Erlangen, Germany

British Columbia Children's Hospital

🇨🇦

Vancouver, British Columbia, Canada

Hospital for Sick Children CF Center

🇨🇦

Toronto, Ontario, Canada

Mukoviszidose-Zentrum am Klinikum der Friedrich-Schiller-Universität Jena, Klinik für Kinder- und Jugendmedizin

🇩🇪

Jena, Germany

Dept of Gene Therapy, Imperial College London

🇬🇧

London, United Kingdom

The National Children's Hospital

🇮🇪

Dublin, Ireland

© Copyright 2025. All Rights Reserved by MedPath