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A Study of Tadalafil in Pediatric Participants With Pulmonary Arterial Hypertension (PAH)

Phase 3
Completed
Conditions
Hypertension, Pulmonary
Interventions
Drug: Placebo
Drug: ERA as specific PAH treatment
Registration Number
NCT01824290
Lead Sponsor
Eli Lilly and Company
Brief Summary

The main purpose of this study is to evaluate the safety and efficacy of tadalafil in pediatric participants with pulmonary arterial hypertension. Participants will receive study treatment for 6 months in the double-blind period (Period 1), and then will be eligible to enroll into an open-label 2 year extension period (Period 2) during which participants will receive tadalafil.

Detailed Description

Not available

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
35
Inclusion Criteria
  • ≥6 months to <18 years of age at screening

  • Currently have a diagnosis of PAH that is either:

    • idiopathic, including hereditary
    • related to connective tissue disease
    • related to anorexigen use
    • associated with surgical repair of at least 6-month duration of congenital systemic to pulmonary shunt (eg, atrial septal defect, ventricular septal defect, patent ductus arteriosus)
  • Have a history of a diagnosis of PAH established by a resting mean pulmonary artery pressure (mPAP) ≥25 millimeter of mercury (mm Hg), pulmonary artery wedge pressure ≤15 mm Hg, and a pulmonary vascular resistance (PVR) ≥3 Wood units via right heart catheterization (RHC). In the event that a pulmonary artery wedge pressure cannot be obtained during RHC, participants with a left ventricular end diastolic pressure (LVEDP) <15 mm Hg, with normal left heart function, and absence of mitral stenosis on echocardiography can be eligible for enrollment

  • Have a World Health Organization (WHO) functional class value of II or III at the time of screening

  • All participants must be receiving an endothelin receptor antagonist (ERA) (such as bosentan or ambrisentan) and must be on a maintenance dose with no change in dose (other than weight-based adjustments) for at least 12 weeks prior to screening and have a screening aspartate transaminase (AST)/alanine transaminase (ALT) <3 times the upper limit of normal (ULN)

  • If on conventional PAH medication, including but not restricted to, anticoagulants, diuretics, digoxin, and oxygen therapy, the participant must be on stable doses with no changes (other than weight-based adjustments) for at least 4 weeks before screening

  • Female participants of childbearing potential must test negative for pregnancy during screening. Furthermore, female participants must agree to abstain from sexual activity or to use two different reliable methods of birth control as determined by the Investigator during the study. Examples of reliable birth control methods include true abstinence as a lifestyle choice (periodic sexual abstinence method is not acceptable); the use of oral contraceptives; a reliable barrier method of birth control (diaphragms with contraceptive jelly; cervical caps with contraceptive jelly; condoms with contraceptive foam; intrauterine devices)

  • Written informed consent from parents (and written assent from appropriately aged participants) will be obtained prior to any study procedure being performed

Exclusion Criteria
  • Have pulmonary hypertension related to conditions other than specified above, including but not limited to chronic thromboembolic disease, portal pulmonary hypertension, left-sided heart disease or lung disease and hypoxia

  • History of left-sided heart disease, including any of the following:

    • clinically significant [pulmonary artery occlusion pressure (PAOP) 15-18 mm Hg] aortic or mitral valve disease (ie, aortic stenosis, aortic insufficiency, mitral stenosis, moderate or greater mitral regurgitation)
    • pericardial constriction
    • restrictive or congestive cardiomyopathy
    • left ventricular ejection fraction <40% by multigated radionucleotide angiogram (MUGA), angiography, or echocardiography
    • left ventricular shortening fraction <22% by echocardiography
    • life-threatening cardiac arrhythmias
    • symptomatic coronary artery disease within 5 years of study entry
  • Unrepaired congenital heart disease

  • Have a history of angina pectoris or other condition that was treated with long- or short-acting nitrates within 12 weeks before administration of study drug

  • Have severe hepatic impairment, Child-Pugh Grade C

  • Have severe renal insufficiency, defined as receiving renal dialysis or having a measured or estimated creatinine clearance (CC) <30 millimeter per minute (mL/min) (Schwartz Formula)

  • Diagnosed with a retinal disorder (eg, hereditary retinal disorders, retinopathy of the preterm participant and other retinal disorders)

  • Have severe hypotension or uncontrolled hypertension as determined by the Investigator

  • Have significant parenchymal lung disease

  • Have bronchopulmonary dysplasia

  • Concurrent phosphodiesterase type 5 (PDE5) inhibitor therapy (sildenafil or vardenafil) or has received PDE5 inhibitor therapy within 12 weeks prior to the first study drug dosing

  • Concurrent therapy with prostacyclin or its analogues within 12 weeks of screening

  • Commenced or discontinued a chronic conventional PAH medication including but not restricted to: diuretics, anti-coagulants, digoxin, and oxygen therapy within 4 weeks of screening

  • Currently receiving treatment with doxazosin, nitrates, or cancer therapy

  • Current treatment with potent Cytochrome P450 3A4 (CYP3A4) inhibitors, such as antiretroviral therapy (protease inhibitor), systemic ketoconazole, or systemic itraconazole, or chronic use of potent CYP3A4 inducers, such as rifampicin

  • Are nursing or pregnant

  • Have previously completed or withdrawn from this study (LVHV), or any other study investigating tadalafil

  • Have received tadalafil therapy within 12 weeks prior to the first study drug dosing or are hypersensitive to tadalafil

  • Have allergy to the excipients, notably lactose

  • Are currently enrolled in, or discontinued within the last 30 days from, a clinical trial involving an investigational product or non-approved use of a drug or device, or concurrently enrolled in any other type of medical research judged not to be scientifically or medically compatible with this study by the Sponsor

  • Unable to take orally administered tablets (without chewing, crushing or breaking) or suspension

  • Are Investigator site personnel directly affiliated with this study or their immediate families. Immediate family is defined as a spouse, parent, child or sibling, whether biological or legally adopted

  • Diagnosis of Down syndrome

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
PlaceboPlaceboPeriod 1: Participants received placebo orally by tablets once a day. Period 2: 20 mg for middle weight and 40 mg for heavy weight administered orally by tablets once a day. Final placebo dose for Period 1 (6-month double-blind) was be assigned after the weight cohort completion from H6D-MC-LVIG (NCT01484431) to maintain blinding depending on body weight cohort. Participants receiving placebo in Period 1 Period 2 (2-year open-label extension) would receive tadalafil in Period 2 at the corresponding tadalafil dose in that participant's weight group.
PlaceboERA as specific PAH treatmentPeriod 1: Participants received placebo orally by tablets once a day. Period 2: 20 mg for middle weight and 40 mg for heavy weight administered orally by tablets once a day. Final placebo dose for Period 1 (6-month double-blind) was be assigned after the weight cohort completion from H6D-MC-LVIG (NCT01484431) to maintain blinding depending on body weight cohort. Participants receiving placebo in Period 1 Period 2 (2-year open-label extension) would receive tadalafil in Period 2 at the corresponding tadalafil dose in that participant's weight group.
TadalafilERA as specific PAH treatmentPeriod 1: 20 mg or 40 mg administered orally by tablets once a day. Period 2: 20 mg for middle weight and 40 mg for heavy weight administered orally by tablets once a day. Final tadalafil doses for Period 1 (6-month double-blind) were assigned after the weight cohort completion from H6D-MC-LVIG (NCT01484431).Tadalafil doses would range from 5 milligram (mg) to 40 mg depending on body weight cohorts. Heavy weight cohort ≥40 kilogram (kg), Middle weight cohort ≥25 kg to \<40 kg: administered orally by tablets once a day. Light weight cohort \<25 kg: administered orally by suspension once a day. Participants receiving tadalafil in Period 1 continued to receive tadalafil during Period 2 (2-year open-label extension).
TadalafilTadalafilPeriod 1: 20 mg or 40 mg administered orally by tablets once a day. Period 2: 20 mg for middle weight and 40 mg for heavy weight administered orally by tablets once a day. Final tadalafil doses for Period 1 (6-month double-blind) were assigned after the weight cohort completion from H6D-MC-LVIG (NCT01484431).Tadalafil doses would range from 5 milligram (mg) to 40 mg depending on body weight cohorts. Heavy weight cohort ≥40 kilogram (kg), Middle weight cohort ≥25 kg to \<40 kg: administered orally by tablets once a day. Light weight cohort \<25 kg: administered orally by suspension once a day. Participants receiving tadalafil in Period 1 continued to receive tadalafil during Period 2 (2-year open-label extension).
Primary Outcome Measures
NameTimeMethod
Period 1: Change From Baseline to Week 24 in a 6 Minute Walk (MW) Distance in MetersBaseline, Week 24

6MWD in meters assessed in a subset of participants who are ≥6 to \<18 years of age who are developmentally capable of performing a 6MW test. Change from baseline was derived using mixed model repeated measures (MMRM) with terms for treatment group, visit, baseline 6MWD, and treatment-by-visit interaction.

Secondary Outcome Measures
NameTimeMethod
Period 1: Time to Adjudicated Clinical Worsening (CW)Baseline through Week 24

Clinical worsening was defined as any of the following: death,lung or heart transplantation,atrial septostomy or Potts' shunt,hospitalization for Pulmonary Arterial Hypertension(PAH) progression,new onset syncope,initiation of new PAH therapy(including increase in the dose of existing PAH specific concomitant therapy,such as endothelin receptor agonist or beraprost medication), or increase of 1 or more in World Health Organization(WHO) Functional Class(except for participants already in Class IV;only for participants unable to perform the 6 minute walk(6MW) test;worsening of WHO functional class by 1 or more for participants who can perform a 6 minute walk(6MW) test and who have a decrease of ≥ 20% in the 6 minute walk distance(for those participants who are ≥6 years of age). Criteria for CW(from Period 1) were adjudicated by an independent,blinded study-specific Clinical Endpoint Committee(CEC).This adjudication was used for data analysis, and was not used to guide subject treatment.

Period 1: Percentage of Participants Who Experience CWBaseline through Week 24

Clinical worsening was defined as any of the following: death,lung or heart transplantation,atrial septostomy or Potts' shunt,hospitalization for Pulmonary Arterial Hypertension(PAH) progression,new onset syncope, initiation of new PAH therapy(including increase in the dose of existing PAH specific concomitant therapy,such as endothelin receptor agonist or beraprost medication),or increase of 1 or more in World Health Organization(WHO) Functional Class(except for participants already in Class IV; only for participants unable to perform the 6 minute walk(6MW) test;worsening of WHO functional class by 1 or more for participants who can perform a 6 minute walk(6MW) test and who have a decrease of ≥ 20% in the 6 minute walk distance(for those participants who are ≥6 years of age).Criteria for CW(from Period 1) were adjudicated by an independent,blinded study-specific Clinical Endpoint Committee(CEC).This adjudication was used for data analysis, and was not used to guide subject treatment.

Period 2: Percentage of Participants Who Experience CWPeriod 2 Baseline through Study Completion (Up to 24 Months)

Clinical worsening was defined as any of the following: death, lung or heart transplantation, atrial septostomy or Potts' shunt, hospitalization for Pulmonary Arterial Hypertension (PAH) progression, new onset syncope, initiation of new PAH therapy (including increase in the dose of existing PAH specific concomitant therapy, such as endothelin receptor agonist or beraprost medication), or increase of 1 or more in World Health Organization(WHO) Functional Class (except for participants already in Class IV; only for participants unable to perform the 6 minute walk (6MW) test; worsening of WHO functional class by 1 or more for participants who can perform a 6 minute walk (6MW) test and who have a decrease of ≥ 20% in the 6 minute walk distance (for those participants who are ≥6 years of age).

Period 1: Pharmacokinetics (PK): Apparent Clearance (CL/F) of Tadalafil at Steady-stateWeek 2, Week 4, Week 16 and Week 24

Period 1: Pharmacokinetics (PK): Apparent Clearance (CL/F) of Tadalafil at steady-state

Period 2: Time to First Occurrence of CWPeriod 2 Baseline through Study Completion (Up to 24 Months)

Clinical worsening was defined as any of the following: death, lung or heart transplantation, atrial septostomy or Potts' shunt, hospitalization for Pulmonary Arterial Hypertension (PAH) progression, new onset syncope, initiation of new PAH therapy (including increase in the dose of existing PAH specific concomitant therapy, such as endothelin receptor agonist or beraprost medication), or increase of 1 or more in World Health Organization(WHO) Functional Class (except for participants already in Class IV; only for participants unable to perform the 6 minute walk (6MW) test; worsening of WHO functional class by 1 or more for participants who can perform a 6 minute walk (6MW) test and who have a decrease of ≥ 20% in the 6 minute walk distance (for those participants who are ≥6 years of age).

Trial Locations

Locations (43)

Irmandade da Santa Casa de Misericordia de Porto Alegre

🇧🇷

Porto Alegre, Rio Grande Do Sul, Brazil

Uniwersytecki Szpital Dzieciecy w Krakowie-Prokocimiu

🇵🇱

Krakow, Poland

Universitätsklinikum Heidelberg

🇩🇪

Heidelberg, Baden-Württemberg, Germany

Tokyo Metropolitan Children's Medical Center

🇯🇵

Fuchu, Tokyo, Japan

Wojewódzki Szpital Specjalistyczny we Wrocławiu

🇵🇱

Wroclaw, Poland

Primary Childrens Medical Center

🇺🇸

Salt Lake City, Utah, United States

Asahikawa Medical College Hospital

🇯🇵

Asahikawa, Hokkaido, Japan

Okinawa Prefectural Nanbu Medical Center & Children's Med Ct

🇯🇵

Haebaru-cho, Shimajiri-gun, Okinawa, Japan

Uniwersyteckie Centrum Kliniczne

🇵🇱

Gdansk, Poland

Hacettepe University Faculty of Medicine

🇹🇷

Ankara, Turkey

Gazi University Medical Faculty

🇹🇷

Besevler/Ankara, Turkey

Gunma Children's Medical Center

🇯🇵

Shibukawa, Gunma, Japan

Instituto Nacional de Cardiologia Ignacio Chavez

🇲🇽

Ciudad de Mexico, DF, Mexico

Instytut Pomnik-Centrum Zdrowia Dziecka

🇵🇱

Warszawa, Woj Mazowieckie, Poland

Vanderbilt Univeristy School of Medicine

🇺🇸

Nashville, Tennessee, United States

Childrens Hospital of Michigan

🇺🇸

Detroit, Michigan, United States

Universitätsklinikum Ulm

🇩🇪

Ulm, Baden-Württemberg, Germany

AKH

🇦🇹

Wien, Austria

Universitaire Ziekenhuizen Leuven - Campus Gasthuisberg

🇧🇪

Leuven, Belgium

Texas Childrens Hospital

🇺🇸

Houston, Texas, United States

Nationwide Children's Hosp

🇺🇸

Columbus, Ohio, United States

Children's Heathcare of Atlanta, Inc. at Egleston

🇺🇸

Atlanta, Georgia, United States

Cincinnati Childrens Hospital Medical Center

🇺🇸

Cincinnati, Ohio, United States

Pronto Socorro Cardiologico de Pernambuco-PROCAPE

🇧🇷

Recife, PE, Brazil

Instituto Dante Pazzanese de Cardiologia

🇧🇷

Sao Paulo, SP, Brazil

CHU Hopital d'enfants de la Timone

🇫🇷

Marseille Cedex 05, France

GH Necker - Enfants Malades

🇫🇷

Paris Cedex 15, France

UNIFESP - Escola Paulista de Medicina

🇧🇷

Sao Paulo, SP, Brazil

Hopital Haut Leveque - Group hospitalier Sud

🇫🇷

Pessac, France

Chu de Toulouse - Hopital des Enfants

🇫🇷

Toulouse Cedex 3, France

Sheba Medical Center

🇮🇱

Tel Hashomer, Ramat Gan, Israel

Schneider Medical Center

🇮🇱

Petah Tiqva, Israel

Istituto Giannina Gaslini Ospedale Pediatrico I.R.C.C.S.

🇮🇹

Genova, GE, Italy

Ospedale V. Monaldi

🇮🇹

Napoli, Italy

Ospedale Bambino Gesu

🇮🇹

Roma, Italy

Mie University Hospital

🇯🇵

Tsu, Mie, Japan

Toho University Omori Medical Center

🇯🇵

Ohta-Ku, Tokyo, Japan

National Center For Child Health And Development

🇯🇵

Setagaya-ku, Tokyo, Japan

Shizuoka Prefectural Children's Hospital

🇯🇵

Shizuoka, Japan

Universitair Medisch Centrum Groningen

🇳🇱

Groningen, Netherlands

Hospital Universitari Vall d'Hebron

🇪🇸

Barcelona, Spain

Hospital Universitario Ramon y Cajal

🇪🇸

Madrid, Spain

Hospital Universitario 12 de Octubre

🇪🇸

Madrid, Spain

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