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C1 Esterase Inhibitor (C1INH-nf) for the Treatment of Acute Hereditary Angioedema (HAE) Attacks

Phase 3
Completed
Conditions
Hereditary Angioedema
Interventions
Drug: Placebo (saline)
Biological: C1 esterase inhibitor [human] (C1INH-nf)
Registration Number
NCT00289211
Lead Sponsor
Shire
Brief Summary

The study objective was to determine the safety and efficacy of C1INH-nf for the treatment of acute HAE attacks.

Detailed Description

Randomized subjects treated for a qualifying attack were eligible to receive rescue dosing with 1,000 U of C1INH-nf if they did not achieve beginning of substantial relief of the defining symptom within 4 hours after initial treatment with blinded study drug, or if at any time the attack progressed to include airway compromise. A second 1,000 U rescue dose was permitted 60 minutes after the initial rescue dose, if necessary.

The study design also allowed for administration of open-label C1INH-nf for laryngeal angioedema attacks, which were non-randomizable events due to the presence of or potential for airway compromise (immediate 1,000 U dose of C1INH-nf, repeated after 60 minutes, if necessary). In addition, subjects were eligible to receive open-label C1INH-nf (1,000 U single dose) prior to emergency surgical (non-cosmetic) procedures.

A total of 83 subjects were enrolled in the study. Seventy-one (71) subjects experienced qualifying attacks and were randomized to blinded study drug (36 C1INH-nf, 35 placebo); only the 71 randomized subjects were analyzed for efficacy. An additional 12 subjects were never randomized but received open-label C1INH-nf for treatment of laryngeal angioedema and/or prior to emergency surgical procedures. Of the 35 subjects randomized to placebo, 23 also received C1INH-nf (eg, rescue, open-label). In total, 83 subjects received at least 1 dose of study drug and were analyzed for safety; 71 subjects were exposed to C1INH-nf (59 randomized, 12 open-label only) and 12 subjects were exposed only to placebo.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
83
Inclusion Criteria
  • Documented HAE
  • Normal C1q level
Exclusion Criteria
  • Low C1q level
  • B-cell malignancy
  • Presence of anti-C1INH autoantibody
  • History of allergic reaction to C1INH or other blood products
  • Narcotic addiction
  • Current participation in any other investigational drug study or within the past 30 days
  • Participation in a C1 esterase inhibitor trial, or received blood or a blood product in the past 90 days
  • Pregnancy or lactation
  • Any clinically significant medical condition, such as renal failure, that in the opinion of the investigator would interfere with the subject's ability to participate in the study

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
PlaceboPlacebo (saline)Matching placebo (saline) administered IV. If there was no response to treatment 60 minutes after the first dose, a second placebo (saline) dose could be administered.
C1INH-nfC1 esterase inhibitor [human] (C1INH-nf)1,000 Units (U) of C1INH-nf administered intravenously (IV). If there was no response to treatment 60 minutes after the first dose, a second 1,000 U dose could be administered.
Primary Outcome Measures
NameTimeMethod
Time to Beginning of Substantial Relief of the Defining SymptomWithin 4 hours after initial treatment

Randomized subjects assessed their symptoms every 15 minutes up to 4 hours after the initial dose of blinded study drug or until substantial relief of the defining symptom was achieved. Substantial relief was defined as 3 consecutive assessments of improvement of the defining symptom. Beginning of substantial relief was considered the first of the 3 consecutive assessments.

Secondary Outcome Measures
NameTimeMethod
Number of Subjects With Beginning of Substantial Relief of the Defining SymptomWithin 4 hours after initial treatment

Randomized subjects assessed their symptoms every 15 minutes up to 4 hours after the initial dose of blinded study drug or until substantial relief of the defining symptom was achieved. Substantial relief was defined as 3 consecutive assessments of improvement of the defining symptom. Beginning of substantial relief was considered the first of the 3 consecutive assessments.

Antigenic C1 Inhibitor (C1INH) Serum LevelsPre-infusion to 1-, 2-, 4-, and 12 hours post-infusion

Change in antigenic C1INH serum levels from pre-infusion to 1-, 2-, 4-, and 12 hours after the initial dose of blinded study drug.

Time to Complete Resolution of the HAE Attack72 hours

Randomized subjects were contacted 72-96 hours (3-4 days) after discharge from the study site to determine when complete resolution of the HAE attack occurred.

Functional C1INH Serum LevelsPre-infusion to 1-, 2-, 4-, and 12 hours post-infusion

Percent change in functional C1INH serum levels from pre-infusion to 1-, 2-, 4-, and 12 hours after the initial dose of blinded study drug. Functional C1INH serum levels are expressed as a percent of total detectable C1INH (ie, functional C1INH/total detectable C1INH).

Complement C4 Serum LevelsPre-infusion to 1-, 2-, 4-, and 12 hours post-infusion

Change in complement C4 serum levels from pre-infusion to 1-, 2-, 4-, and 12 hours after the initial dose of blinded study drug.

Trial Locations

Locations (37)

Allergy and Asthma Center

🇺🇸

Fort Lauderdale, Florida, United States

Allergy Partners of the Upstate

🇺🇸

Greenville, South Carolina, United States

St. Louis University School of Medicine

🇺🇸

Saint Louis, Missouri, United States

UCLA-David Geffen School of Medicine

🇺🇸

Los Angeles, California, United States

Optimed Research

🇺🇸

Columbus, Ohio, United States

The Baton Rouge Clinic, AMC

🇺🇸

Baton Rouge, Louisiana, United States

Allergy Clinic of Tulsa

🇺🇸

Tulsa, Oklahoma, United States

Allergy and Asthma Clinical Research, Inc

🇺🇸

Walnut Creek, California, United States

Winthrop University Hospital

🇺🇸

Mineola, New York, United States

Grand Traverse Allergy

🇺🇸

Traverse City, Michigan, United States

Mount Sinai School of Medicine

🇺🇸

New York, New York, United States

Penn State University

🇺🇸

Hershey, Pennsylvania, United States

Institute for Asthma and Allergy

🇺🇸

Wheaton, Maryland, United States

Family Allergy and Asthma Center

🇺🇸

Atlanta, Georgia, United States

Puget Sound Allergy, Asthma and Immunology

🇺🇸

Tacoma, Washington, United States

Welborn Clinic Allergy and Immunology

🇺🇸

Evansville, Indiana, United States

Baylor College of Medicine

🇺🇸

Houston, Texas, United States

University of California, San Diego

🇺🇸

San Diego, California, United States

Duke University Medical Center

🇺🇸

Durham, North Carolina, United States

Allergy and Asthma Research Center

🇺🇸

San Antonio, Texas, United States

Allergy and Immunology Associates

🇺🇸

Scottsdale, Arizona, United States

Clinical Research Consultants, Inc

🇺🇸

Hoover, Alabama, United States

University of Iowa Hospital and Clinic

🇺🇸

Iowa City, Iowa, United States

University of Massachusetts Medical School

🇺🇸

Worcester, Massachusetts, United States

MeritCare Clinical Research

🇺🇸

Fargo, North Dakota, United States

Allergy Asthma and Dermatology Research Center

🇺🇸

Lake Oswego, Oregon, United States

Marycliff Allergy Specialists

🇺🇸

Spokane, Washington, United States

AARA Research Center

🇺🇸

Dallas, Texas, United States

Bernstein Clinical Research

🇺🇸

Cincinnati, Ohio, United States

University of Texas Medical Branch

🇺🇸

Galveston, Texas, United States

UMDNJ Asthma and Allergy Research Center

🇺🇸

Newark, New Jersey, United States

Nevada Access to Research and Education Society

🇺🇸

Las Vegas, Nevada, United States

Orlando Regional Healthcare

🇺🇸

Orlando, Florida, United States

Virginia Adult and Pediatric Allergy and Asthma

🇺🇸

Richmond, Virginia, United States

Allergy, Asthma and Pulmonary Clinical Research

🇺🇸

Madison, Wisconsin, United States

Allergy Asthma and Immunology

🇺🇸

Falmouth, Massachusetts, United States

Montefiore Medical Center

🇺🇸

Bronx, New York, United States

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