Safety of Sildenafil in Premature Infants With Severe Bronchopulmonary Dysplasia

NCT ID: NCT04447989

Last Updated: 2026-01-26

Study Results

Results available

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Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

PHASE2

Total Enrollment

125 participants

Study Classification

INTERVENTIONAL

Study Start Date

2021-05-27

Study Completion Date

2025-01-15

Brief Summary

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This is a multicenter, randomized, placebo-controlled, sequential dose-escalating, double-masked, safety study of sildenafil in premature infants (inpatient in Neonatal Intensive Care Units (NICUs)) with severe bronchopulmonary dysplasia (BPD).

Detailed Description

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Screening/Baseline

Research staff will document informed consent from the parent/guardian for all participants who satisfy eligibility criteria. The following information will be recorded in the case report form (eCRF) from the clinical medical record:

1. Participant demographics, including birth weight and gestational age at birth
2. Maternal race/ethnicity
3. Medical history
4. Physical examination, including actual weight
5. All mean arterial pressure (MAP) obtained in the 24 hours before the first dose
6. Concomitant medications (within 24 hours prior to start of study drug)
7. Respiratory assessment
8. Laboratory evaluations
9. Echocardiogram: If performed per local standard of care \< 14 days prior to start of study drug, a study-specific echocardiogram need not be repeated. If not performed per local standard of care \< 14 days prior to start of study drug, an echocardiogram will be required to confirm eligibility.
10. Cardiac catheterization reports, if performed per local standard of care \< 14 days prior to start of study drug.
11. Adverse events following initial study-specific procedure

Treatment Period

The treatment period will include Days 1-28 or last day of study drug if early withdrawal of study drug. The following information will be collected and recorded while the participant is on study drug:

1. Actual weight on study Days 7 (± 1 day), 14 (± 1 day), 21 (± 1 day), and 28 (± 1 day) of study drug administration
2. Date, time, amount, and route of study drug dose
3. All concomitant medications
4. MAP

A. All MAP values obtained 24 hours after the first dose of study drug regardless of administration route.

B. MAP values will be obtained at a minimum at the following time points i. Prior to the first dose of study drug or dose escalation: 2 hours (± 5 minutes), 1 hour (± 5 minutes), and 15 minutes (± 5 minutes) ii. If administration route is IV:
1. During and following the first dose of study drug or dose escalation: MAP at start of infusion, every 15 minutes (± 5 minutes) during infusion, at end of infusion (inclusive of flush) (± 5 minutes), at 15 and 30 minutes (± 5 minutes) after end of infusion, hourly (± 15 minutes) for 4 hours, and once in the remaining 2 hours prior to the next dose.
2. For subsequent IV doses, the lowest valid MAP value should be recorded daily while on study drug.

iii. If the administration route is enteral:
1. During and following the first dose of study drug or dose escalation: MAP at start of enteral administration, then every 15 minutes (± 5 minutes) for 90 minutes (1.5 hours), then every 30 minutes (± 5 minutes) for 60 minutes (1 hour), then hourly (± 15 minutes) for 4 hours, then once in the remaining 2 hours prior to the next dose.
2. For subsequent enteral doses, the lowest valid MAP value should be recorded daily while on study drug.
5. Respiratory assessment, weekly
6. Laboratory evaluations, at least every other week
7. Echocardiograms and cardiac catheterization reports, if performed per local standard of care
8. Pharmacokinetic (PK) sampling (after Day 7)
9. Adverse events

Weaning Period (Cohorts 2 and 3)

The weaning period will begin following Day 28 of study drug or, following the last day of study drug if participant was withdrawn from study drug prior to Day 28 and the dose escalated to ≥ 0.5 mg/kg IV or ≥ 1 mg/kg enteral.

The following information will be collected and recorded while the participant is weaning from study drug:

1. Date, time, amount and route of study drug dose
2. MAP (the lowest MAP value on last day of wean should be recorded).
3. Respiratory assessment on last day of wean
4. Echocardiogram and cardiac catheterization reports, if performed per local standard of care
5. Adverse events

Follow-up Period

The follow-up period will include Days 1-28 after the last study drug dose; last study drug dose may occur prior to Day 28 for those participants who withdraw from study drug early; on Day 28 for those participants who complete the full treatment period; or after last weaning dose for those participants who require weaning. The following information will be reported in electronic data capture system (EDC) at Day 1 (+ 2 days) and 14 (± 2days) of the follow-up period (or days closest to and after Day 1 and 14, if \>1 assessment is available), except for MAP, adverse events (AEs), and serious adverse events (SAEs) (which will be reported from Days 1-28 post last study drug dose) and standard of care echocardiograms or cardiac catheterization reports:

1. Physical examination, including actual weight
2. MAP (the lowest valid MAP value on follow-up Day 1, 14, 21, and 28 should be recorded).
3. Respiratory assessment
4. Laboratory evaluations
5. Echocardiogram on follow-up Day 1 (+2 days). If performed per local standard of care, a study-specific echocardiogram need not be repeated. If not performed per local standard of care on Day 1 (+2 days) of the follow-up period, an echocardiogram will need to be performed.
6. Echocardiograms and cardiac catheterization reports, if performed per local standard of care (during follow-up Days 1-28)
7. Adverse events and SAEs (during follow-up Days 1-28)

Final Study Assessment

Final study assessment will occur at the time of discharge or transfer. The following information will be collected:

1. Physical examination, including actual weight
2. Respiratory assessment
3. Echocardiogram and cardiac catheterization reports, if performed per local standard of care on the day of discharge or transfer or up to 2 days prior.
4. Global rank
5. Discharge information A. Discharge or transfer B. Death C. Duration of hospitalization
6. Record if treatment for retinopathy of prematurity (ROP) was required

Conditions

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Bronchopulmonary Dysplasia of Newborn

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

PARALLEL

Premature infants with severe BPD (inpatient in neonatal intensive care units) will be randomized in a dose escalating approach 3:1 (sildenafil: placebo) sequentially, into each of 3 cohorts. There will be approximately 40 randomized and dosed participants in each cohort for a total of up to 120 participants.
Primary Study Purpose

TREATMENT

Blinding Strategy

QUADRUPLE

Participants Caregivers Investigators Outcome Assessors
Study drug (sildenafil or placebo) will be prepared in the pharmacy by the unblinded pharmacist. Treatment cohort will be randomly assigned electronically and communicated to the pharmacist via the study portal. All other study staff and the patients/parents will be blinded to the treatment assignment.

Study Groups

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Cohort 1, sildenafil

Sildenafil (0.5 mg/kg IV or 1 mg/kg enteral) every 8 hours for 28 days

Group Type ACTIVE_COMPARATOR

Sildenafil

Intervention Type DRUG

Sildenafil citrate injection or powder for suspension

Cohort 1, placebo

Placebo (IV or enteral) every 8 hours for 28 days

Group Type PLACEBO_COMPARATOR

Placebo

Intervention Type DRUG

dextrose 5%

Cohort 2, sildenafil

Sildenafil (1 mg/kg IV or 2 mg/kg enteral) every 8 hours for 28 days

Group Type ACTIVE_COMPARATOR

Sildenafil

Intervention Type DRUG

Sildenafil citrate injection or powder for suspension

Cohort 2, placebo

Placebo (IV or enteral) every 8 hours for 28 days

Group Type PLACEBO_COMPARATOR

Placebo

Intervention Type DRUG

dextrose 5%

Cohort 3, sildenafil

Sildenafil (2 mg/kg IV or 4 mg/kg enteral) every 8 hours for 28 days

Group Type ACTIVE_COMPARATOR

Sildenafil

Intervention Type DRUG

Sildenafil citrate injection or powder for suspension

Cohort 3, placebo

Placebo (IV or enteral) every 8 hours for 28 days

Group Type PLACEBO_COMPARATOR

Placebo

Intervention Type DRUG

dextrose 5%

Interventions

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Sildenafil

Sildenafil citrate injection or powder for suspension

Intervention Type DRUG

Placebo

dextrose 5%

Intervention Type DRUG

Other Intervention Names

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Revatio Dextrose 5%

Eligibility Criteria

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Inclusion Criteria

1. Documented informed consent from parent or guardian, prior to study procedures
2. \< 29 weeks gestational age at birth
3. 32-44 weeks postmenstrual age
4. Receiving respiratory support at enrollment:

* If 32 0/7-35 6/7 weeks postmenstrual age: mechanical ventilation (high frequency or conventional)
* If 36 0/7-44 6/7 weeks postmenstrual age: mechanical ventilation (high frequency or conventional) OR continuous positive airway pressure (CPAP)

Note:

* Criteria 3 and 4 define severe BPD for the purposes of this study
* CPAP is defined as any of the following:

* Nasal cannula \> 2 liters per minute (LPM)
* Nasal continuous positive airway pressure (NCPAP)
* Nasal intermittent positive pressure ventilation (NIPPV)
* Noninvasive neurally adjusted ventilatory assist (NAVA)
* Any other device designed to provide positive pressure through a nasal device (e.g., RAM cannula, etc.)

Exclusion Criteria

1. Previous enrollment and dosing in this study, protocol number (NHLBI-2019-SIL), "Safety of Sildenafil in Premature Infants with Severe Bronchopulmonary Dysplasia (BPD)"
2. Previous exposure to sildenafil within 7 days prior to randomization\*
3. Previous exposure to vasopressors within 24 hours prior to randomization\*
4. Previous exposure to inhaled nitric oxide within 24 hours prior to randomization\*
5. Previous exposure to milrinone within 24 hours prior to randomization\*
6. Evidence of pulmonary hypertension or moderate/large patent ductus arteriosus (PDA) on the most recent echocardiogram performed within 14 days prior to randomization
7. Known major congenital heart defect requiring medical or surgical intervention in the neonatal period
8. Known allergy to sildenafil
9. Known sickle cell disease
10. Aspartate aminotransferase (AST) \> 225 U/L \< 72 hours prior to randomization
11. Alanine aminotransferase (ALT) \> 150 U/L \< 72 hours prior to randomization
12. Any condition that would make the participant, in the opinion of the investigator, unsuitable for the study.

* Participant will be reassessed prior to dosing to reconfirm eligibility criteria.
Maximum Eligible Age

29 Weeks

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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University of North Carolina, Chapel Hill

OTHER

Sponsor Role collaborator

National Heart, Lung, and Blood Institute (NHLBI)

NIH

Sponsor Role collaborator

Christoph Hornik

OTHER

Sponsor Role lead

Responsible Party

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Christoph Hornik

Associate Professor of Pediatrics

Responsibility Role SPONSOR_INVESTIGATOR

Principal Investigators

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Christoph Hornik, MD

Role: PRINCIPAL_INVESTIGATOR

Duke UMC

Matt Laughon, MD

Role: PRINCIPAL_INVESTIGATOR

UNC

Locations

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Arkansas Children's Research Institute

Little Rock, Arkansas, United States

Site Status

University of Arkansas Medical Sciences

Little Rock, Arkansas, United States

Site Status

Rady Children's Hospital and Health Center

San Diego, California, United States

Site Status

Childrens National Medical Center

Washington D.C., District of Columbia, United States

Site Status

University of Florida Jacksonville Shands Medical Center

Jacksonville, Florida, United States

Site Status

Wolfson Children's Hospital

Jacksonville, Florida, United States

Site Status

Emory Children's Center

Atlanta, Georgia, United States

Site Status

Lurie Children's Hospital

Chicago, Illinois, United States

Site Status

University of Illinois at Chicago

Chicago, Illinois, United States

Site Status

University of Kentucky Chandler Medical Center

Lexington, Kentucky, United States

Site Status

University of Louisville School of Medicine

Louisville, Kentucky, United States

Site Status

Ochsner Baptist Medical Center

New Orleans, Louisiana, United States

Site Status

Boston Children's Hospital

Boston, Massachusetts, United States

Site Status

University of Mississippi Medical Center

Jackson, Mississippi, United States

Site Status

Childrens Mercy Hospital

Kansas City, Missouri, United States

Site Status

Children's Hospital of Nevada at University Medical Center

Las Vegas, Nevada, United States

Site Status

University of Rochester School of Medicine Children's Hospital

Rochester, New York, United States

Site Status

Westchester Medical Center - New York Medical College

Valhalla, New York, United States

Site Status

University of NC at Chapel Hill

Chapel Hill, North Carolina, United States

Site Status

East Carolina University

Greenville, North Carolina, United States

Site Status

Wake Forest University Health Sciences

Winston-Salem, North Carolina, United States

Site Status

Rainbow Babies and Childrens Hospital

Cleveland, Ohio, United States

Site Status

University of Tennessee Health Science Center

Memphis, Tennessee, United States

Site Status

University of Texas Health

Austin, Texas, United States

Site Status

Women's Hospital of Texas

Houston, Texas, United States

Site Status

Countries

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United States

References

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Gonzalez D, Laughon MM, Smith PB, Ge S, Ambalavanan N, Atz A, Sokol GM, Hornik CD, Stewart D, Mundakel G, Poindexter BB, Gaedigk R, Mills M, Cohen-Wolkowiez M, Martz K, Hornik CP; Best Pharmaceuticals for Children Act - Pediatric Trials Network Steering Committee. Population pharmacokinetics of sildenafil in extremely premature infants. Br J Clin Pharmacol. 2019 Dec;85(12):2824-2837. doi: 10.1111/bcp.14111. Epub 2019 Dec 15.

Reference Type BACKGROUND
PMID: 31475367 (View on PubMed)

Stoll BJ, Hansen NI, Bell EF, Walsh MC, Carlo WA, Shankaran S, Laptook AR, Sanchez PJ, Van Meurs KP, Wyckoff M, Das A, Hale EC, Ball MB, Newman NS, Schibler K, Poindexter BB, Kennedy KA, Cotten CM, Watterberg KL, D'Angio CT, DeMauro SB, Truog WE, Devaskar U, Higgins RD; Eunice Kennedy Shriver National Institute of Child Health and Human Development Neonatal Research Network. Trends in Care Practices, Morbidity, and Mortality of Extremely Preterm Neonates, 1993-2012. JAMA. 2015 Sep 8;314(10):1039-51. doi: 10.1001/jama.2015.10244.

Reference Type BACKGROUND
PMID: 26348753 (View on PubMed)

Jobe AH, Bancalari E. Bronchopulmonary dysplasia. Am J Respir Crit Care Med. 2001 Jun;163(7):1723-9. doi: 10.1164/ajrccm.163.7.2011060. No abstract available.

Reference Type BACKGROUND
PMID: 11401896 (View on PubMed)

Khemani E, McElhinney DB, Rhein L, Andrade O, Lacro RV, Thomas KC, Mullen MP. Pulmonary artery hypertension in formerly premature infants with bronchopulmonary dysplasia: clinical features and outcomes in the surfactant era. Pediatrics. 2007 Dec;120(6):1260-9. doi: 10.1542/peds.2007-0971.

Reference Type BACKGROUND
PMID: 18055675 (View on PubMed)

Morrow CB, McGrath-Morrow SA, Collaco JM. Predictors of length of stay for initial hospitalization in infants with bronchopulmonary dysplasia. J Perinatol. 2018 Sep;38(9):1258-1265. doi: 10.1038/s41372-018-0142-7. Epub 2018 Jun 8.

Reference Type BACKGROUND
PMID: 29880793 (View on PubMed)

Jackson W, Hornik CP, Messina JA, Guglielmo K, Watwe A, Delancy G, Valdez A, MacArthur T, Peter-Wohl S, Smith PB, Tolia VN, Laughon MM. In-hospital outcomes of premature infants with severe bronchopulmonary dysplasia. J Perinatol. 2017 Jul;37(7):853-856. doi: 10.1038/jp.2017.49. Epub 2017 Apr 6.

Reference Type BACKGROUND
PMID: 28383537 (View on PubMed)

Poets CF, Lorenz L. Prevention of bronchopulmonary dysplasia in extremely low gestational age neonates: current evidence. Arch Dis Child Fetal Neonatal Ed. 2018 May;103(3):F285-F291. doi: 10.1136/archdischild-2017-314264. Epub 2018 Jan 23.

Reference Type BACKGROUND
PMID: 29363502 (View on PubMed)

Tyson JE, Wright LL, Oh W, Kennedy KA, Mele L, Ehrenkranz RA, Stoll BJ, Lemons JA, Stevenson DK, Bauer CR, Korones SB, Fanaroff AA. Vitamin A supplementation for extremely-low-birth-weight infants. National Institute of Child Health and Human Development Neonatal Research Network. N Engl J Med. 1999 Jun 24;340(25):1962-8. doi: 10.1056/NEJM199906243402505.

Reference Type BACKGROUND
PMID: 10379020 (View on PubMed)

Schmidt B, Roberts RS, Davis P, Doyle LW, Barrington KJ, Ohlsson A, Solimano A, Tin W; Caffeine for Apnea of Prematurity Trial Group. Caffeine therapy for apnea of prematurity. N Engl J Med. 2006 May 18;354(20):2112-21. doi: 10.1056/NEJMoa054065.

Reference Type BACKGROUND
PMID: 16707748 (View on PubMed)

Doyle LW, Ehrenkranz RA, Halliday HL. Early (< 8 days) postnatal corticosteroids for preventing chronic lung disease in preterm infants. Cochrane Database Syst Rev. 2014 May 13;(5):CD001146. doi: 10.1002/14651858.CD001146.pub4.

Reference Type BACKGROUND
PMID: 24825456 (View on PubMed)

Schneider S, Bailey M, Spears T, Esther CR Jr, Laughon MM, Hornik CP, Jackson W. Safety of sildenafil in premature infants with severe bronchopulmonary dysplasia (SILDI-SAFE): a multicenter, randomized, placebo-controlled, sequential dose-escalating, double-masked, safety study. BMC Pediatr. 2020 Dec 14;20(1):559. doi: 10.1186/s12887-020-02453-7.

Reference Type DERIVED
PMID: 33317479 (View on PubMed)

Provided Documents

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Document Type: Study Protocol and Statistical Analysis Plan

View Document

Document Type: Informed Consent Form

View Document

Other Identifiers

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1R61HL147833-01

Identifier Type: NIH

Identifier Source: secondary_id

View Link

Pro00104901

Identifier Type: -

Identifier Source: org_study_id

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