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Clinical Study to Evaluate the Effects of Macitentan on Exercise Capacity in Subjects With Eisenmenger Syndrome

Phase 3
Completed
Conditions
Pulmonary Arterial Hypertension
Interventions
Registration Number
NCT01743001
Lead Sponsor
Actelion
Brief Summary

Clinical study to assess the efficacy, safety, and tolerability of macitentan in subjects with Eisenmenger Syndrome.

Detailed Description

Not available

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
226
Inclusion Criteria
  • Subjects:

    • not participating in the hemodynamic sub-study: males or females ≥ 12 years of age.
    • participating in the hemodynamic sub-study: males or females ≥ 18 years of age.
  • Subjects (including those with Down Syndrome [DS]) with confirmed Eisenmenger Syndrome [ES] (European Society of Cardiology [ESC] and the European Respiratory Society [ERS] guidelines):

    1. Established by echocardiography as:

      • Large congenital shunting defect at atrial, ventricular or arterial level*
      • and right to left shunt or bi-directional shunt with prevalent right to left direction.
    2. Resting peripheral oxygen saturation (SpO2) ≤ 90% and > 70% (pulse oximetry, room air).

The lower limit is 65% if a subject is living at an altitude greater than 2500 m above sea level.

*Subjects with any of the following open defects are eligible for the study either as an isolated defect or in combination:

  • atrial septal defect (ASD)
  • ventricular septal defect (VSD)
  • partial or complete atrioventricular septal defect (AVSD)
  • patent ductus arteriosus (PDA)
  • aortopulmonary window (AP window)
  • total or partial anomalous pulmonary venous return (TAPVR, PAPVR) The defects may be either unoperated or previously palliated surgically (provided significant residual defect remains).

The Steering Committee will review the echocardiography data of all subjects (main study and sub study) to confirm eligibility prior to Randomization.

  • Subjects with the following findings at cardiac catheterization:

    • Mean resting pulmonary arterial pressure (mPAP) > 25 mmHg
    • Pulmonary capillary wedge pressure (PCWP) or mean left atrial pressure (LAP) or left ventricular end diastolic pressure (LVED) ≤ 15 mmHg
    • Pulmonary vascular resistance (PVR) ≥ 800 dyn∙s/cm5 or ≥ 10 Wood units
  • Subjects with WHO functional class ≥ II.

  • Subjects able to reliably perform the the 6-minute walk test (6MWT) with a minimum distance of 50 m and a maximum distance of 450 m.

Exclusion Criteria
  • Main study and hemodynamic sub-study: Any of the following conditions previously known or identified via cardiac catheterization or echocardiography:
  • Pulmonary arterial or venous stenosis > 25% size of native pulmonary artery (PA) or pulmonary vein
  • Severe tricuspid regurgitation in the setting of left to right shunt at the ventricular or atrial level
  • Greater than mild tricuspid stenosis
  • Intracavitary RV outflow obstruction
  • Greater than mild mitral stenosis
  • Intracavitary LV outflow obstruction
  • Subvalvular or supravalvular aortic stenosis
  • Aortic coarctation
  • Greater than moderate mitral regurgitation
  • Recognized extracardiac systemic venous collaterals to the pulmonary venous circulation
  • Recognized hepatic wedge pressure-inferior vena cava pressure gradient >12 mm Hg
  • PCWP "v" waves >20 mmHg
  • Tetralogy of Fallot
  • Truncus arteriosus
  • Interrupted aortic arch
  • Transposition of great arteries
  • Single ventricle defects: absent AV connection (mitral or tricuspid atresia), double inlet AV connections left or right ventricle, functional univentricular heart (unbalanced AVSD, hypoplastic RV, double outlet RV), hypoplastic left heart syndrome
  • Ebstein's anomaly
  • Severe aortic regurgitation
  • Pulmonary atresia
  • PAPVR or TAPVR, ONLY if there is lung hypoplasia or if documentation confirming the absence of lung hypoplasia does not exist.

For subjects participating in the hemodynamic sub-study the following will also be considered exclusion criteria:

  • SVC stenosis >25% size of native vessel

  • PDA, AP window, TAPVR, PAPVR, or ASD sinus venosus with anomalous pulmonary veins

  • Down Syndrome

    • Subjects with deterioration of their clinical status within 3 months prior to Screening or during the Screening period.
    • Known moderate-to-severe restrictive (i.e., total lung capacity [TLC] < 60% of predicted value) or obstructive lung disease (i.e., forced expiratory volume in one second [FEV1] < 80 % of predicted value, and with FEV1 / forced vital capacity [FVC] < 70%)
    • Treatment with prostanoids within 1 month prior to Randomization
    • Subjects who initiated a PDE-5 inhibitor within 1 month prior to Randomization or those on a PDE-5 inhibitor for whom the dose has not been stable within 1 month prior to Randomization
    • Treatment with endothelin receptor antagonists (ERAs) within 1 month prior to Randomization
    • Subjects who initiated diuretics within 1 week prior to Randomization or subjects whose diuretic treatment has not been stable for at least 1 week prior to Randomization
    • Subjects being considered for an organ transplant

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
MacitentanMacitentan 10 mgSubjects receive macitentan 10 mg oral tablet once daily
PlaceboPlaceboSubjects receive macitentan-matching placebo oral tablet once daily
Primary Outcome Measures
NameTimeMethod
Change From Baseline to Week 16 in Exercise Capacity, as Measured by 6-minute Walk Distance (6MWD)From baseline to Week 16

The purpose of the six minute walk is to test exercise tolerance and capacity. The test measures the distance an individual is able to walk over a total of six minutes on a hard, flat surface. The goal is for the individual to walk as far as possible in six minutes.

Secondary Outcome Measures
NameTimeMethod
Change From Baseline to Week 16 in Quality of Life (QoL), Assessed by the Short Form-36 (SF-36) QuestionnaireFrom baseline to Week 16

The SF-36 is a multi-purpose, short-form health survey with 36 questions. It yields an 8-scale profile of the functional health and well-being scores (i.e., physical functioning, role physical, bodily pain, general health, vitality, social functioning, role emotional and mental health), as well as psychometrically based physical and mental health summary measures and a preference-based health utility (health rated as much better now than one year ago to much worse now than one year ago). It is a generic measure, as opposed to one that targets a specific age, disease, or treatment group.

For each of the domains and scores that the SF36 measures an aggregate percentage score is produced. The percentage scores range from 0% (lowest or worst possible level of functioning) to 100% (highest or best possible level of functioning). A higher score for the individual domains and summary component scores indicates a better condition of the subject.

Change From Baseline to Week 16 in Dyspnea, Assessed by the Borg Dyspnea IndexFrom baseline to Week 16

This outcome measures the difference in the Borg dyspnea index collected at the end of the 6-minute walk test (6MWT) at Week 16 compared to baseline. The Borg dyspnea index rates the severity of dyspnea (difficult or labored breathing) on a scale from 0 ('Nothing at all') to 10 ('Very, very severe - maximal'). A decrease in the Borg dyspnea index indicates an improvement.

Change From Baseline to Week 16 in WHO Functional ClassFrom baseline to Week 16

A shift in WHO functional classes is considered an 'improvement' when shifting to a lower class (e.g. from class III to class II) or a 'worsening' when shifting to a higher class (e.g. from class III to class IV). Definition of functional classes as follows - Class I: no symptoms with exercise or at rest. Class II: No symptoms at rest but uncomfortable and short of breath with normal activity such as climbing a flight of stairs, grocery shopping, or making the bed. Class III: May not have symptoms at rest but activities greatly limited by shortness of breath, fatigue, or near fainting (e.g. doing normal chores around the house, have to take breaks while doing activities of daily living). Class IV: Symptoms at rest and severe symptoms with any activity. Most patients also have edema in the feet and ankles as result of right heart failure.

Trial Locations

Locations (55)

Omu Pediatry

🇹🇷

Samsun, Turkey

Wu Han Asia Heart Hosp

🇨🇳

Wuhan, Hubei, China

Texas Children'S Hosp - Dept of Cardiology

🇺🇸

Houston, Texas, United States

Institut Jantung Negara

🇲🇾

Kuala Lumpur, Malaysia

Unidad de Investigacion Clin En Med, Sc (Udicem)

🇲🇽

Monterrey, Nuevo Leon, Mexico

Herzzentrum Berlin, Ped Cardiology

🇩🇪

Berlin, Germany

Hosp Universitario La Fe Dpt Cardiology

🇪🇸

Valencia, Spain

Hosp La Timone - Dept Pediatric Cardiology

🇫🇷

Marseille Cedex 5, France

Hosp Univ Virgen Macarena - Dpt Cardiology

🇪🇸

Sevilla, Spain

Hosp Cardiology Haut Leveque - Dept Congenital Diseases

🇫🇷

Pessac, France

Bristol Univ Hosp Congenital Heart Centre

🇬🇧

Bristol, United Kingdom

Children'S Heart Center Nevada

🇺🇸

Las Vegas, Nevada, United States

Instituto Nacional de Cardiologia (Inc) Ignacio Chavez

🇲🇽

Mexico City, Mexico

Hosp Univ Vall D'Hebron - Dpt Congenital Heart Disease Adult

🇪🇸

Barcelona, Spain

Uni Heidelberg - Kinderkardiologie

🇩🇪

Heidelberg, Germany

Guangdong General Hospital, Cardiology Dpt

🇨🇳

Guangzhou, Guangdong, China

Beijing Anzhen Hospital, Cardiology Dpt

🇨🇳

Beijing, China

Cardiovascular Institute&Fuwai Hospital

🇨🇳

Beijing, China

Shanghai Pulmonary Hospital, Dept of Pulmonary Circulation

🇨🇳

Shanghai, China

Ahmanson/UCLA Heart Disease Center

🇺🇸

Los Angeles, California, United States

Stanford Hospital and Clinic

🇺🇸

Palo Alto, California, United States

Emory University Hospital/the Emory Clinic

🇺🇸

Atlanta, Georgia, United States

Nationwide Children's Hospital

🇺🇸

Columbus, Ohio, United States

Barnes-Jewish Hosp/Wash Univ School of Med

🇺🇸

Saint Louis, Missouri, United States

Hosp Pompidou - Dept Congenital Cardiac Diseases

🇫🇷

Paris Cedex 15, France

The General Hosp of Shenyang Military Region

🇨🇳

Shenyang, Liaoning, China

Mhat Nat Card Hosp - Pediatric Clin / Ped Card Dept

🇧🇬

Sofia, Bulgaria

Universitätsklinikum Giessen - Pediatric Heart Center

🇩🇪

Giessen, Germany

Rabin Medical Centre - Pulmonology

🇮🇱

Petach Tikvah, Israel

Er Inst For Cardvasc Dis "Prof Dr Cc Iliescu" - Card Ii

🇷🇴

Bucuresti, Romania

Hosp Laennec - Dept Cardiology

🇫🇷

Nantes Cedex 1, France

PHC, MAB

🇵🇭

Manila, Philippines

Cardiology Gdańsk Univ

🇵🇱

Gdańsk, Poland

Tam Duc Hospital

🇻🇳

Ho Chi Minh, Vietnam

Gen Hosp Univ Vienna Dept Cardiology

🇦🇹

Vienna, Austria

Children's Hospital, Ho Chi Minh

🇻🇳

Ho Chi Minh, Vietnam

Clinica Tabancura - Cardio Unit

🇨🇱

Santiago, Chile

Cardiology Kraków Univ

🇵🇱

Krakow, Poland

Mhat Nat Card Hosp - Cardiology Clinic

🇧🇬

Sofia, Bulgaria

Mhat Sveta Anna Clin Card

🇧🇬

Sofia, Bulgaria

Hosp Univ Coimbra - Dpt Cardiology

🇵🇹

Coimbra, Portugal

Hosp Sta Marta - Dept Cardiology

🇵🇹

Lisboa, Portugal

Cardio Med Srl

🇷🇴

Targu-Mures, Romania

Inst Nat Torax, Unidad Cardiopatia Congenitas Del Adulto

🇨🇱

Providencia, Chile

Ahepa University General Hospital

🇬🇷

Thessaloniki, Greece

Instituto de Corazon de Querètaro

🇲🇽

Querétaro, Mexico

Cardiology Wrocław

🇵🇱

Wrocław, Poland

Clin Hosp For Inf and Pulm Dis Victor Babes - Ii Pulm

🇷🇴

Timisoara, Romania

Sci Institute Systemic Problems Cardio Diseases Kemerovo

🇷🇺

Kemerovo, Russian Federation

Clin Hosp Ctr Zemun - Cardiology Dept

🇷🇸

Belgrade, Serbia

Russian Cardiology Scientific and Production Complex

🇷🇺

Moscow, Russian Federation

V. A. Almazov Institute of Cardiology

🇷🇺

St Petersburg, Russian Federation

Dedinje Cardiovasc Inst - Cardiovasc Research Ctr

🇷🇸

Belgrade, Serbia

Mother and Child Health Care Inst "Dr Vukan Cupic"

🇷🇸

Belgrade, Serbia

Hanoi Medical University Hospital

🇻🇳

Hanoi, Vietnam

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