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Towards Optimal Treatment of Marfan Syndrome

Recruiting
Conditions
Marfan Syndrome
Interventions
Other: Magnetic Resonance imaging (MRI) examination
Registration Number
NCT05944614
Lead Sponsor
Academisch Medisch Centrum - Universiteit van Amsterdam (AMC-UvA)
Brief Summary

The purpose of this study is to determine whether new magnetic resonance imaging techniques can be used as a biomarker of aortic disease severity in patients with Marfan syndrome.

Detailed Description

Not available

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
150
Inclusion Criteria
  • MFS patients with a known FBN1 mutation
  • Between 18-50 years of age
Exclusion Criteria
  • Contraindication for MR imaging
  • Mental retardation
  • Pregnancy, or planned pregnancy during study period

Additional exclusion criteria healthy controls:

  • History of aorta disease

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Arm && Interventions
GroupInterventionDescription
Healthy volunteersMagnetic Resonance imaging (MRI) examinationAge and gender matched healthy control patients without a history of aortic disease.
Marfan Syndrome patientsMagnetic Resonance imaging (MRI) examinationMarfan Syndrome patients age 18-50 with a pathological FBN1 mutation
Primary Outcome Measures
NameTimeMethod
Difference in pulse wave velocity, in meters per second between Marfan patients and healthy volunteers.Baseline

pulse wave velocity expressed in m/s in the thoracic aorta, normal values age dependent: but range +/- 4 - 30 m/s, higher is worse

Difference in oscillatory shear index (dimensionless) expressed in m/s between Marfan patients and healthy volunteersBaseline

oscillatory shear index (dimensionless) in the thoracic aorta, range between 0 - 0.5 (No normal values defined), higher is worse

Change in aortic pulse wave velocity (meters/second), between MFS patient aortas prior to compared to after PEARS procedure or conventional aortic surgery.Change from baseline to 6 months after surgery

pulse wave velocity in the thoracic aorta expressed in meters/second. Higher is worse. normal values for change unknown.

Change in oscillatory shear index (dimensionless) between MFS patient aortas prior to compared to after PEARS procedure or conventional aortic surgery.Change from baseline to 6 months after surgery

oscillatory shear index (dimensionless) in the thoracic aorta. Higher is worse range between 0 -5.

Difference in 3D aortic displacement in millimeters between Marfan patients (MFS) and healthy volunteers.Baseline

aortic displacement in the thoracic aorta. normal values unknown, but single paper: Marfan patients range: 1.77 to 10.03 millimeters, volunteers 7.34 ± 1.69 millimeters. lower is worse.

Change in aortic displacement expressed in millimeters , between MFS patient aortas prior to compared to after PEARS procedure or conventional aortic surgery.Change from baseline to 6 months after surgery

aortic displacement expressed in millimeters in the thoracic aorta. Pre-post change unknown, however operated 3.60 +/- 1.0 millimeter versus native aorta 7.01 +/- 3.09 millimeters. Lower is worse

Secondary Outcome Measures
NameTimeMethod
Difference in pulse wave velocity, expressed in m/s between Marfan patients with an aortic root diameter >4.5 cm versus <4.5 cmBaseline

pulse wave velocity expressed in m/s in the thoracic aorta. Higher is worse. Normal values unknown.

Difference in 3D aortic displacement expressed in mm between Marfan patients based on antihypertensive medication usedBaseline

aortic displacement in the thoracic aorta, Lower is worse. Normal values unknown.

Difference in pulse wave velocity, expressed in meters per second between Marfan patients with and without a native aortaBaseline

pulse wave velocity expressed in m/s in the thoracic aorta, Lower is worse. Normal values unknown. Single study in general aneurysm population: pre surgery: 18.0 +/-4.0 meters per second post surgery 20.6 +/- 6.0 meters per second. higher is worse

Difference in pulse wave velocity, expressed in m/s between Marfan patients based on antihypertensive medication usedBaseline

pulse wave velocity expressed in m/s in the thoracic aorta

Difference in oscillatory shear index (dimensionless) expressed in m/s between Marfan patients with and without a native aortaBaseline

oscillatory shear index (dimensionless) in the thoracic aorta. Higher is worse. Normal values unknown.

Difference in 3D aortic displacement expressed in mm between Marfan patients with and without a native aortaBaseline

aortic displacement in the thoracic aorta, lower is worse. operated 3.60 +/- 1.0 millimeter versus native aorta 7.01 +/- 3.09 millimeters

Difference in 3D aortic displacement expressed in mm between male versus female Marfan patientsBaseline

aortic displacement in the thoracic aorta,Lower is worse. Normal values unknown.

Difference in 3D aortic displacement expressed in mm between Marfan patients with aortic root diameter >4.5cm versus <4.5 cmBaseline

aortic displacement in the thoracic aorta, lower is worse. Normal values unknown.

Difference in 3D aortic displacement expressed in mm between Marfan patients with a haploinsufficient fibrillin 1 mutation versus dominant negative mutationBaseline

aortic displacement in the thoracic aorta. Lower is worse. Normal values unknown.

Difference in pulse wave velocity, expressed in m/s between Marfan patients with a haploinsufficient fibrillin-1 mutation versus a dominant negative mutationBaseline

pulse wave velocity expressed in m/s in the thoracic aorta. Higher is worse. Normal values unknown.

Difference in oscillatory shear index (dimensionless) expressed in m/s between male versus female Marfan patientsBaseline

oscillatory shear index (dimensionless) in the thoracic aorta. Higher is worse. Normal values unknown.

Difference in oscillatory shear index (dimensionless) expressed in m/s between Marfan patients based on antihypertensive medication usedBaseline

oscillatory shear index (dimensionless) in the thoracic aorta. Higher is worse. Normal values unknown.

The correlation between regional wall shear stress and elastic fibre thickness in resected aorta tissue of MFS patients.Baseline MRI with aortic surgery tissue

Correlation of locally MRI measured wall shear stress and histologic findings in the thoracic aorta. Neither is worse. Normal values dependent on location of measurement, exploratory outcome. .

Difference in pulse wave velocity, expressed in m/s between male and female Marfan patientsBaseline

pulse wave velocity expressed in m/s in the thoracic aorta Higher is worse. Normal values unknown.

Difference in oscillatory shear index (dimensionless) expressed in m/s between Marfan patients with aortic root diameter >4.5cm versus <4.5 cmBaseline

oscillatory shear index (dimensionless) in the thoracic aorta. Higher is worse. Normal values unknown.

Difference in oscillatory shear index (dimensionless) expressed in m/s between Marfan patients with a haploinsufficient fibrillin 1 mutation versus dominant negative mutationBaseline

oscillatory shear index (dimensionless) in the thoracic aorta. Higher is worse. Normal values unknown.

Trial Locations

Locations (4)

Amsterdam UMC, location AMC

🇳🇱

Amsterdam, Meibergdreef 9, Netherlands

Radboud University Medical Center

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Nijmegen, Gelderland, Netherlands

University Medical Center Groningen

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Groningen, Netherlands

Leiden University Medical Center

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Leiden, Noord-Holland, Netherlands

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