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Safety and PK-PD Study of Oral L-CIT in Preterm Infants with BPD±PH and NEC

Not Applicable
Recruiting
Conditions
BPD - Bronchopulmonary Dysplasia
Pulmonary Hypertension
NEC
Interventions
Dietary Supplement: L-Citrulline
Registration Number
NCT05636397
Lead Sponsor
The Hospital for Sick Children
Brief Summary

The purpose of this study is to evaluate the safety and explore the PK/PD of L-CIT supplementation in preterm infants to prevent the development of inflammatory pathways initiated by low levels of plasma CIT, specifically in preterm infants with post surgical NEC and BPD±PH.

Detailed Description

Preterm infants are born with underdeveloped organs and immune systems, placing them at great risk for morbidity. They are more susceptible to inflammatory injury, particularly from conditions of prematurity mediated by inflammatory pathways such as bronchopulmonary dysplasia (BPD) and necrotizing enterocolitis (NEC).

L-CIT, an amino acid, is the first intermediate in the urea cycle as well as a precursor to arginine and nitric oxide (NO), which promotes blood flow. It is made in the intestine and has been shown to exert vasoprotective and anti-inflammatory effects. BPD-PH and NEC are two specific inflammatory diseases of prematurity involving CIT, arginine or NO deficiencies.

Evaluation of the safety and PK/PD of L-CIT supplementation for diseases involving CIT, arginine or NO deficiencies in preterm infants is important. Therefore, in this trial the investigator would like to evaluate the safety and pharmacokinetics/pharmacodynamics (PD) of L-CIT supplementation in preterm infants post surgical NEC and BPD-PH.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
60
Inclusion Criteria
  • Born ≤ 30 weeks at birth
  • Post-menstrual age (PMA) ≥ 34 weeks
  • Echocardiographic evidence of PH for infants with BPD+PH
  • On invasive or non-invasive ventilation with RSS >2.0 for >12hours/day for at least 48 hours
  • Informed written consent (parents/substitute decision maker)
Exclusion Criteria
  • Congenital Heart Disease [Exceptions: small atrial septal defect (ASD), small ventricular septal defect (VSD), small patent ductus arteriosus (PDA)]
  • Infants with pulmonary vein stenosis
  • Concurrent sepsis with hemodynamic instability
  • Infants considered likely to die within next 7 days
  • Any other condition that, in the opinion of the investigator, may adversely affect the infant's ability to complete the study or its measures or pose significant risk to the infant.

Arm 2: surgical NEC

Inclusion Criteria

  • Born ≤ 30 weeks at birth
  • Recovering from Stage IIIb NEC as per modified Bell's staging (pneumoperitoneum requiring surgery)
  • Tolerating 30 ml/kg/day of enteral feeds
  • On invasive or non-invasive ventilation (NIPPV/nCPAP) with RSS >2.0 for > 12hours/day for at least 48 hours, 10-14 days post surgery
  • Informed written consent (parents/substitute decision maker)
  • Considered medically stable by clinical team

Exclusion Criteria:

  • Congenital heart disease (except small ASD, small VSD and non hsPDA)
  • Pulmonary vein stenosis
  • Concurrent sepsis with hemodynamic instability
  • Likely to die within next 7 days
  • Other condition significantly affecting pulmonary function independent of prematurity or NEC

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
BPD±PHL-CitrullineArm 1: BPD±PH Total of 12-24 infants; 6-12 at each dose level of oral dosage form of L-Citrulline. (300 or 500 mg/kg/day divided q6 hours). Dose Level 1 = 300 mg/kg/day Dose Level 2 = 500 mg/kg/day
Surgical NECL-CitrullineArm 2: sNEC A total of 18-36 infants with Stage III NEC; 6-12 at each dose level of oral dosage form of L-Citrulline. (75, 150 or 225 mg/kg/day divided q6 hours) Dose Level 1 = 75 mg/kg/day Dose Level 2 = 150 mg/kg/day Dose Level 3 = 225 mg/kg/day
Primary Outcome Measures
NameTimeMethod
Safety of oral L-Citrulline administration5 years

The number of patients with adverse events (AE) as a measure of safety and tolerability

Secondary Outcome Measures
NameTimeMethod
Association of blood pressure as one of the PD outcomes with maximum L-CIT concentration (Cmax)5 years

Blood pressure of the study participants will be associated with PK measures of L-CIT exposure i.e. Cmax using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen. Future larger studies will then be used to examine these relationships with adequately powered studies.

Association of stoma or nasogastric output as one of the PD outcomes with maximum L-CIT concentration (Cmax)5 years

Stoma or nasogastric output of the study participants will be associated with PK measures of L-CIT exposure i.e. Cmax using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen. Future larger studies will then be used to examine these relationships with adequately powered studies.

Bayley's scale for infant development5 years

Bayley Scales of Infant and Toddler Development is an extensive formal developmental assessment tool for diagnosing developmental delays in early childhood. BSID is the commonly used abbreviation for Bayley Scales of Infant and Toddler Development. Bayley-III includes a motor score, and fine and gross motor subtest scores. The standardized mean motor score is 100 (SD 15), with scores lower than 85 indicating mild impairment, and lower than 70 indicating moderate or severe impairment.In this particular trial, the investigator would be looking at the correlation between the inflammatory markers (IL-1β, IL-6, IL-8, IL-10, TNFα) and Neurodevelopmental outcomes from Bayley's scale during 18-24M follow up visit in babies received L-Citrulline during their NICU stay.

Association of stool output as one of the PD outcomes with maximum L-CIT concentration (Cmax)5 years

Stool output from the study participants will be associated with PK measures of L-CIT exposure i.e. Cmax using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen. Future larger studies will then be used to examine these relationships with adequately powered studies.

Association of stoma or nasogastric output with the area under the concentration time curve (AUC) for L-CIT5 years

Stoma or nasogastric output of the study participants will be associated with PK measures of L-CIT exposure i.e. Area under Concentration time curve (AUC) using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen.

Association of stool output with the area under the concentration time curve (AUC) for L-CIT5 years

Stool output from the study participants will be associated with PK measures of L-CIT exposure i.e. Area under Concentration time curve (AUC) using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen.

Association of blood pressure with minimum L-CIT concentration (Cmin)5 years

Blood pressure of study participants will be associated with PK measures of L-CIT exposure i.e. Cmin using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen. Future larger studies will then be used to examine these relationships with adequately powered studies.

Oxidative stress5 years

Oxidative stress (measured in tracheal aspirates and blood)

Desaturation index5 years

The oxygen desaturation index (ODI) is commonly used to evaluate the severity of nocturnal hypoxemia. The ODI is defined as the number of episodes of oxygen desaturation per hour of sleep with desaturation events of \>=10sec/ sampled hour.

Changes in Blood Pressure5 years

Changes in diastolic and systolic blood pressure prior to and during CIT treatment.

Ventilation5 years

Days on mechanical ventilation, non-invasive ventilation, and supplementary oxygen

BPD5 years

number of days of survival free of BPD

Association of stoma or nasogastric output with minimum L-CIT concentration (Cmin)5 years

Stoma or nasogastric output from study participants will be associated with PK measures of L-CIT exposure i.e. Cmin using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen. Future larger studies will then be used to examine these relationships with adequately powered studies.

Respiratory Score (RSS)5 years

The respiratory severity score (RSS) is a simplified severity score consisting of the mean airway pressure (MAP) multiplied by the fraction of inspired oxygen (FiO2). This score ranges from 0 to 12, with a higher score indicating more severe lung disease.

Stoma, nasogastric or stool output5 years

Volume of stoma, nasogastric or stool output prior to and during CIT treatment

Postnatal steroid Use5 years

Number of days study participants received postnatal steroids during their NICU stay.

Association of stool output with minimum L-CIT concentration (Cmin)5 years

Stool output from study participants will be associated with PK measures of L-CIT exposure i.e. Cmin using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen. Future larger studies will then be used to examine these relationships with adequately powered studies.

Correlation between CIT and arginine levels5 years

Correlation between changes in CIT and arginine levels with nitrite/nitrate levels

Association of blood pressure with the area under the concentration time curve (AUC) for L-CIT5 years

Blood pressure of the study participants will be associated with PK measures of L-CIT exposure i.e. Area under Concentration time curve (AUC) using univariate correlation approaches. The investigator will then attempt to construct a PK/PD model to link PK and PD measures found to be of significance during the univariate screen.

Biomarkers of inflammation5 years

The levels of IL-1β, IL-6, IL-8, IL-10, TNFα will be measured in tracheal aspirates and blood plasma. The aggregated levels fo these cytokines will reflect the inflammatory status of the study participant.

BPD severity5 years

Moderate to severe BPD based on the different mode of ventilatory support needed at 36 wks PMA.

No BPD = off all oxygen and positive pressure support Moderate BPD = low flow oxygen only Severe BPD= positive pressure support (high flow, CPAP, NIPPV, or ETT)

Pre-discharge mortality5 years

Number of study participants who died during NICU admission.

Trial Locations

Locations (1)

The Hospital For Sick Children

🇨🇦

Toronto, Ontario, Canada

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