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Assessment of the TGF-beta Pathway and Micro-RNA in Pediatric Pulmonary Arterial Hypertension

Conditions
Pulmonary Arterial Hypertension
Interventions
Diagnostic Test: Protein Elisa analysis, microRNA analysis, whole exome sequencing
Registration Number
NCT04489251
Lead Sponsor
Medical College of Wisconsin
Brief Summary

This is a prospective pilot study to assess the plasma levels of particular proteins involved in the transforming growth factor beta (TGF-β) pathway and its down stream regulators, CHIP, as well as micro RNA molecules in subjects with pulmonary arterial hypertension (PAH) and compare them to control subjects without PAH to see if they can be used as a diagnostic or prognostic marker of PAH and how this compares to other diagnostic biomarkers N-terminal pro-natriuretic peptide (NT Pro-BNP) and C-reactive protein (CRP).

Detailed Description

Aim 1: This study will correlate proteins in the TGF- β signaling pathway and micro RNA levels with invasive (catheterization) and non-invasive (echocardiography) measurements of pulmonary artery pressures to assess for the presence and severity of PAH and compare these measurements to the established biomarkers of NT Pro BNP and CRP levels.

Hypothesis 1: Plasma levels of proteins of the TGF-β pathway; bone morphogenic protein (BMP) 2, 4, 6, 7, 9 and 10 along with activin A and TGF-β1 protein as well as CHIP (carboxyl-terminus of Hsp70-intracting protein), an enzyme that regulates the activations and exports of TGF- β to the nucleus will be significantly different in subjects with PH over control subjects.

Hypothesis 2: Plasma levels of proteins in the TGF- β pathway; BMP 2, 4, 6, 7, 9 and 10 along with activin A and TGF-β1 protein as well as CHIP will show better correlation with the presence of PAH and its severity than NT-Pro BNP and CRP levels.

Hypothesis 3: The micro-RNA profiles in plasma will be significantly different in subjects with PAHPH over control subjects.

Aim 2: To correlate protein/micro-RNA levels with clinical status in PAH subjects as assessed by functional status, exercise testing, and PAH drug regimen to determine if they can correlate with disease severity.

Hypothesis 1: Clinical findings in PAH patients will correlate with disease severity and study proteins and micro-RNA levels better than established biomarkers.

Aim 3: To correlate evidence of genetic abnormalities through whole exome sequencing especially in regions known or suspected to cause PAH (e.g. BMPR2, ENG, and ALK1 mutations), within the TGF-β pathway or lung development with the tested protein/micro-RNA levels.

Hypothesis 1: Genetic evaluation of patients with PAH will show abnormalities within the TGF-β pathway or lung development.

Recruitment & Eligibility

Status
UNKNOWN
Sex
All
Target Recruitment
40
Inclusion Criteria
  • Pediatric subjects ages 2-17 years
  • Subjects undergoing a clinically indicated cardiac catheterization.
  • Subjects with proven or being evaluated for pulmonary hypertension in WHO classification group 1 or 3†
  • Subjects will be categorized as PAH subjects if they meet the hemodynamic criteria: pulmonary artery pressure >20mmHg, pulmonary vascular resistance index >3 Woods units*m2, and wedge pressures <15mmHg.
  • Subjects can be categorized as control subjects if they do not have PH on catheterization and do not meet any exclusion criteria.
Exclusion Criteria

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Arm && Interventions
GroupInterventionDescription
Control subjectsProtein Elisa analysis, microRNA analysis, whole exome sequencing* Pediatric subjects ages 2-17 years * Subjects undergoing a clinically indicated cardiac catheterization. * Subjects can be categorized as control subjects if they do not have PH on catheterization and do not meet any exclusion criteria.
PH subjectsProtein Elisa analysis, microRNA analysis, whole exome sequencing* Pediatric subjects ages 2-17 years * Subjects undergoing a clinically indicated cardiac catheterization. * Subjects with proven or being evaluated for pulmonary hypertension in WHO classification group 1 or 3† * Subjects will be categorized as PAH subjects if they meet the hemodynamic criteria: pulmonary artery pressure \>20mmHg, pulmonary vascular resistance index \>3 Woods units\*m2, and wedge pressures \<15mmHg.
Primary Outcome Measures
NameTimeMethod
Plasma levels of BMP proteins of the TGF-β pathwayOne day

bone morphogenic protein (BMP) 2, 4, 6, 7, 9 and 10 levels will be obtained in PH subjects and will be compared to control subjects in effort to describe how those levels are different in subjects with PH over control subjects. All measured amounts of proteins will be in pg/ml.

Plasma CHIP levels assessment in PH patients and control subjects.One day

CHIP (carboxyl-protein terminus of Hsp70-intracting protein) plasma levels will be obtained in PH subjects and compared to control subjects in effort to describe how those levels are different in subjects with PH over control subjects. All measured levels will be in pg/ml

Plasma Activin A and TGF-β1 level of the TGF-β pathwayOne day

Activin A and TGF-β1 plasma levels will be obtained in PH subjects and compared to control subjects in effort to describe how those levels are different in subjects with PH over control subjects. All measured amounts will be will be in pg/ml.

Secondary Outcome Measures
NameTimeMethod
Whole exome sequence analysis in PH patients and protein/microRNA levelsOne day

To correlate evidence of genetic abnormalities through whole exome sequencing especially in regions known or suspected to cause PAH (e.g. BMPR2, ENG, and ALK1 mutations), within the TGF-β pathway or lung development with the tested protein/micro-RNA levels.

Plasma levels of proteins in the TGF- β pathway and PH disease severityOne day

BMP 2, 4, 6, 7, 9 and 10 along with activin A and TGF-β1 protein as well as CHIP levels will be correlated with the presence of PAH and its severity and how that compares to the levels of NT-Pro BNP and CRP levels.

MicroRNA levels in PH vs control subjectsOne day

The micro-RNA profiles in plasma will be compared between in subjects with PAH and control subjects.

Clinical findings correlation with study proteins/microRNAOne day

Clinical findings in PAH patients will be correlated with disease severity and study protein and micro-RNA levels.

Trial Locations

Locations (1)

Children's Hospital of Wisconsin

🇺🇸

Milwaukee, Wisconsin, United States

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