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A Prospective Study to Evaluate Biological and Clinical Effects of Significantly Corrected CFTR Function

Completed
Conditions
Cystic Fibrosis
Registration Number
NCT04038047
Lead Sponsor
Nicole Hamblett
Brief Summary

This is a prospective, multi-center observational study. The study is designed to measure the clinical effectiveness of elexacaftor, tezacaftor and ivacaftor (ETI) triple combination therapy in people with one or more copies of the F508del mutation, study the effects of ETI across a number of CF disease manifestations, and collect specimens for future research. Subjects in the study will have one "before TCT" visit within 30 days before initiation of the therapy and five "after TCT" visits over a 30-month follow-up period. Participants who have participated in the original PROMISE cohort have the option of participating in a long-term extension with annual visits performed at the 42- and 54-month timepoints. The durability of the clinical and biological changes in PROMISE can be assessed with extended follow-up, which would enable the sub-studies to consider potential clinical consequences of the biological or physiological effects being studied. This work will help to inform long term prognosis and feasibility of certain clinical trials outcomes for interventional studies and may be useful when considering research priorities in drug development. Most participating sites have been divided into sub-study groups; each sub-study group has specific non-optional procedures conducted in addition to the "Core" procedures. Finally, there is one optional procedure (transient elastography) that will be offered to subjects at certain sites. The duration of participation for each subject is 30 months (with an additional 24 months if participants agree to the optional long-term extension). NOTE: FDA has reviewed the New Drug Application (NDA) for elexacaftor, tezacaftor and ivacaftor and has granted approval.

Detailed Description

Cystic fibrosis (CF) is an autosomal recessive genetic disorder caused by mutations in the gene encoding the cystic fibrosis transmembrane conductance regulator (CFTR) protein. In people with CF, this manifests as dysfunction in multiple organ systems including the lungs, pancreas, liver, intestines, skin and others.

While nearly 2000 mutations have been described, the most common disease-causing CFTR mutation is F508del, which is found in \>85% of patients followed in the US CF Patient Registry. Two CFTR corrector drugs plus the potentiator ivacaftor have been developed as a triple combination therapy for CF patients with one or two copies of the F508del mutation. We predict that over 90% of CF patients (initially age 12 y/o and above) will be eligible for highly effective CFTR modulator therapy in the U.S.

The PROMISE study is designed to measure the direct and indirect CFTR-dependent anion secretion by collecting and analyzing clinical research outcomes and biomarkers on a large number of patients both before and after they begin treatment with elexacaftor, tezacaftor and ivacaftor triple combination therapy (TCT). This study will investigate the impact of TCT across a wide range of CF disease manifestations and organ systems. While specific biomarkers of special interest have been selected for detailed analysis in this study, an additional important goal is to collect blood, urine, stool, and airway epithelial cell specimens for long-term storage in a biorepository to enable future research. These samples can be made available for research beyond the current scope of work. The PROMISE study will provide a coordinated collection of clinical research outcomes data that can be linked with these specimens.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
490
Inclusion Criteria
  1. All genders within the age limit of the FDA approved indication for elexacaftor, tezacaftor and ivacaftor triple combination therapy (TCT) at Day 1.
  2. Diagnosis of CF.
  3. CFTR mutations consistent with the FDA approved indication for elexacaftor, tezacaftor and ivacaftor triple combination therapy (TCT).
  4. Physician intent to prescribe elexacaftor, tezacaftor and ivacaftor triple combination therapy (TCT).
  5. Willing to fast for 8 hours prior to all study visits (for subjects on overnight enteric tube feedings, willing to hold the feeding for at least 8 hours).
  6. Able to perform the testing and procedures required for this study, as judged by the investigator.
  7. Enrolled in the Cystic Fibrosis Foundation Patient Registry.
  8. Clinically stable with no significant changes in health status within the 14 days prior to Visit 1.
Exclusion Criteria
  1. Use of any TCT within the 180 days prior to Visit 1.
  2. Any acute use of antibiotics (oral, inhaled or IV) or systemic corticosteroids within the 2 weeks prior to Visit 1 for lower respiratory tract symptoms.
  3. Initiation of any new chronic therapy (e.g., ibuprofen, Pulmozyme®, hypertonic saline, azithromycin, inhaled tobramycin, Cayston®, Kalydeco, Orkambi®, Symdeko®) within the 4 weeks prior to Visit 1.
  4. Use of an investigational agent within the 28 days prior to Visit 1.
  5. Use of chronic oral corticosteroids (equivalent to 10 mg. or more per day of prednisone) within the 28 days prior to Visit 1.
  6. Treatment for nontuberculous mycobacterial (NTM) infection, consisting of ≥ two antibiotics (oral, IV, and/or inhaled) within the 28 days prior to Visit 1.
  7. History of lung or liver transplantation, or listing for organ transplantation.

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
Sweat Chloride at 6 months6 months

Change in sweat chloride from Baseline to 6 months.

Sweat Chloride at 30 months30 months

Change sweat chloride from Baseline to 30 months.

Forced expiratory volume at one second (FEV1) at 6 months6 months

Change in FEV1 from Baseline to 6 months.

Forced expiratory volume at one second (FEV1) at 30 months30 months

Change in FEV1 from Baseline to 30 months.

Secondary Outcome Measures
NameTimeMethod
Weight at 6 Months6 months

Change in weight from Baseline to 6 months.

BMI at 6 Months6 months

Change in BMI from Baseline to 6 months.

Cystic Fibrosis Questionnaire Revised (CFQ-R) at 6 Months6 months

Change in CFQ-R (respiratory domain) from Baseline to 6 months.

BMI at 30 months30 months

Change in BMI from Baseline to 30 months.

Cystic Fibrosis Questionnaire Revised (CFQ-R) at 30 months30 months

Change in CFQ-R (respiratory domain) from Baseline to 30 months.

Weight at 30 Months30 months

Change in weight from Baseline to 30 months.

Trial Locations

Locations (56)

Ann & Robert H. Lurie Children's Hospital of Chicago

🇺🇸

Chicago, Illinois, United States

Riley Hospital for Children

🇺🇸

Indianapolis, Indiana, United States

Cincinnati Children's Hospital Medical Center

🇺🇸

Cincinnati, Ohio, United States

Massachusetts General Hospital

🇺🇸

Boston, Massachusetts, United States

Boston Children's Hospital, Brigham & Women's Hospital

🇺🇸

Boston, Massachusetts, United States

University Hospitals Case Medical Center/Rainbow Babies and Children's Hospital

🇺🇸

Cleveland, Ohio, United States

Cleveland Clinic Cystic Fibrosis Program

🇺🇸

Cleveland, Ohio, United States

Baylor College of Medicine

🇺🇸

Houston, Texas, United States

University of Washington Medical Center

🇺🇸

Seattle, Washington, United States

Oklahoma Cystic Fibrosis Center

🇺🇸

Oklahoma City, Oklahoma, United States

Intermountain Cystic Fibrosis Center

🇺🇸

Salt Lake City, Utah, United States

Emory University

🇺🇸

Atlanta, Georgia, United States

University of Arkansas for Medical Sciences

🇺🇸

Little Rock, Arkansas, United States

Yale University School of Medicine

🇺🇸

New Haven, Connecticut, United States

Children's Healthcare of Atlanta and Emory University

🇺🇸

Atlanta, Georgia, United States

Augusta University

🇺🇸

Augusta, Georgia, United States

John Hopkins Hospital

🇺🇸

Baltimore, Maryland, United States

Wayne State University Harper University Hospital

🇺🇸

Detroit, Michigan, United States

University of Kentucky

🇺🇸

Lexington, Kentucky, United States

Children's Mercy Kansas City

🇺🇸

Kansas City, Missouri, United States

Stanford University Medical Center

🇺🇸

Palo Alto, California, United States

University of Iowa

🇺🇸

Iowa City, Iowa, United States

Children's National Medical Center

🇺🇸

Washington, District of Columbia, United States

Providence Alaska Medical Center

🇺🇸

Anchorage, Alaska, United States

The Minnesota Cystic Fibrosis Center

🇺🇸

Minneapolis, Minnesota, United States

Children's Hospital Colorado

🇺🇸

Aurora, Colorado, United States

University of Florida

🇺🇸

Gainesville, Florida, United States

Saint Francis Medical Center

🇺🇸

Peoria, Illinois, United States

Washington University School of Medicine

🇺🇸

Saint Louis, Missouri, United States

Rutgers Robert Wood Johnson Medical School

🇺🇸

New Brunswick, New Jersey, United States

New York Medical College at Westchester Medical Center

🇺🇸

Valhalla, New York, United States

Children's Hospital of New York

🇺🇸

New York, New York, United States

University of Pennsylvania

🇺🇸

Philadelphia, Pennsylvania, United States

Children's Hospital of Philadelphia

🇺🇸

Philadelphia, Pennsylvania, United States

Helen DeVos Children's Hospital

🇺🇸

Grand Rapids, Michigan, United States

University of Virginia

🇺🇸

Charlottesville, Virginia, United States

Seattle Children's Hospital

🇺🇸

Seattle, Washington, United States

Cohen Children's Medical Center of New York

🇺🇸

Lake Success, New York, United States

The Cystic Fibrosis Center of Western New York

🇺🇸

Buffalo, New York, United States

Cook Children's Medical Center

🇺🇸

Fort Worth, Texas, United States

University of North Carolina at Chapel Hill

🇺🇸

Chapel Hill, North Carolina, United States

Hershey Medical Center Pennsylvania State University

🇺🇸

Hershey, Pennsylvania, United States

Northwell CF Center

🇺🇸

New York, New York, United States

University of Texas Southwestern Medical Center

🇺🇸

Dallas, Texas, United States

Children's Hospital of Pittsburgh of UPMC

🇺🇸

Pittsburgh, Pennsylvania, United States

University of Rochester Medical Center Strong Memorial

🇺🇸

Rochester, New York, United States

Virginia Commonwealth University

🇺🇸

Richmond, Virginia, United States

Children's Hospital Medical Center of Akron

🇺🇸

Akron, Ohio, United States

University of Alabama at Birmingham

🇺🇸

Birmingham, Alabama, United States

National Jewish Health

🇺🇸

Denver, Colorado, United States

University of Nebraska Medical Center

🇺🇸

Omaha, Nebraska, United States

Oregon Health Sciences University

🇺🇸

Portland, Oregon, United States

Children's Hospital of Wisconsin

🇺🇸

Milwaukee, Wisconsin, United States

University of Michigan Health System

🇺🇸

Ann Arbor, Michigan, United States

University of Wisconsin

🇺🇸

Madison, Wisconsin, United States

University of Kansas Medical Center

🇺🇸

Kansas City, Kansas, United States

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