Lymphedema, Low-grade Inflammation and the Vasculature in Turner Syndrome

NCT ID: NCT06325618

Last Updated: 2024-03-22

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

RECRUITING

Total Enrollment

150 participants

Study Classification

OBSERVATIONAL

Study Start Date

2024-01-08

Study Completion Date

2027-08-31

Brief Summary

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100 women with karyotype verified TS, previously examined at 4 study visits during a 19-year period will be asked to participate in a 5th study visit. Healthy age-matched females will be included as controls in a ratio 2:1.

The aim is to examine and quantify the cardiovascular and lymphatic system in women with TS. The investigators will study a possible causal mechanism between the known pathologic phenotype and alterations in these systems to understand, prevent or treat the life-threatening complications in TS.

Detailed Description

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Background:

Patients with Turner syndrome (TS) are at risk of many complications during childhood, adolescence, and adulthood, including reduced final height, estrogen deficiency, infertility, lymphedema, ischemic heart disease, aortic dilation and dissection, congenital heart defect, hypertension, stroke, and autoimmune diseases in general.

The aim of this study is to evaluate the lymphatic and cardiovascular system in a cohort of adult women with TS to elucidate any defects, abnormalities or dysfunctions that may explain the myriad of complications related to TS.

Hypotheses:

1. Both the lymphatic and cardiovascular system are affected in TS with alterations in both anatomy, flow, and function.
2. Changes in the lymphatic system are more prevalent than previously assumed, affecting both central and peripheral lymphatic vessels.
3. Changes in lymphatic transportation and development of cardiovascular malformations occur simultaneously in early fetal life, thus disease in either system may interplay with malfunctions in the other.
4. The vascular structure in both lymph- and cardiovascular tissue is affected throughout the body and low-grade inflammation could be a possible factor in this pathogenesis in TS.
5. Changes in the vascular structure affects flow through the systems resulting in increased stress point on vessel walls leading to a possible entry site for a dissection which can be detected through flow analyses.
6. Genomic examination of multiple tissues will help in understanding the underlying genomic background for the congenital malformations seen in TS

Design:

100 women with karyotype verified TS, previously examined at 4 study visits during a 19-year period will be asked to participate in a 5th study visit. Healthy age-matched females will be included as controls in a ratio 2:1.

The lymphatic system will be examined using three different techniques to assess changes in both anatomy and function.

1. The investigators will use Near infrared light to evaluate the lymphatic system and grade dysfunction and detect subclinical lymphedema. Indocyanine Green will be injected subcutaneously in the foot and hand and used to assess the flow 5-60 minutes after injection.
2. A novel MRI technique to illustrate slow moving fluids, thus visualizing lymphatic fluid, will be used to display the contrast between fluids and tissue to view the thoracic duct and more peripheral lymphatic vessels and quantify their function, abnormalities and morphology.
3. A DEXA scan will be performed to calculate and differentiate between subdermal fat and lymphatic drainage or edema.

To evaluate the cardiovascular system, the investigators will use MRI of the heart and aorta to asses both function and morphology to calculate, among others, mean cardiac output, stroke volume, ejection fraction and asses myocardial fibrosis. A novel technique is to use a PET-CT, as this has been associated with atherosclerosis, because it can visualize macrophage rich atherosclerotic plaques and thus detect otherwise undetectable low-grade inflammation and disease in the cardiovascular system which may contribute to the development of cardiovascular complications.

The investigators will also evaluate the flow in the aorta using 4D-flow measurements based on customized MRI protocols to evaluate stress on the aortic wall and analyze the risk of aortic wall rupture and dissection. The potential of this analysis is the possibility to identify patients at high risk and need for medical or surgical intervention at an early stage and thus prevent or minimize this acute life-threatening complication to TS.

Genomic studies: Samples will be taken from multiple tissues (Blood, fat, muscle, skin, buccal swaps, urine) and DNA and RNA will be isolated using standard methodology.

Conditions

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Turner Syndrome Cardiovascular Diseases Lymphedema Inflammation

Study Design

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Observational Model Type

CASE_CONTROL

Study Time Perspective

CROSS_SECTIONAL

Study Groups

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Turner Syndrome

Women with karyotype verified Turner Syndrome n=100

No intervention other than obtaining biopsies

Intervention Type OTHER

Skin, fat, and muscle biopsies will be obtained

Indocyanine green (ICG)

Intervention Type DRUG

Indocyanine green will be injected to evaluate the lymphatic system.

Female controls

Healthy, age-matched controls n=50

No intervention other than obtaining biopsies

Intervention Type OTHER

Skin, fat, and muscle biopsies will be obtained

Indocyanine green (ICG)

Intervention Type DRUG

Indocyanine green will be injected to evaluate the lymphatic system.

Interventions

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No intervention other than obtaining biopsies

Skin, fat, and muscle biopsies will be obtained

Intervention Type OTHER

Indocyanine green (ICG)

Indocyanine green will be injected to evaluate the lymphatic system.

Intervention Type DRUG

Eligibility Criteria

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Inclusion Criteria

* Turner Syndrome

Exclusion Criteria

* pregnancy
* contraindications for MRI
Minimum Eligible Age

18 Years

Maximum Eligible Age

100 Years

Eligible Sex

FEMALE

Accepts Healthy Volunteers

Yes

Sponsors

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Aarhus University Hospital

OTHER

Sponsor Role collaborator

University of Aarhus

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Locations

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Aarhus University Hospital

Aarhus N, , Denmark

Site Status RECRUITING

Countries

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Denmark

Central Contacts

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Line Balsby, MD

Role: CONTACT

+4520461203

Facility Contacts

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Line Balsby, MD

Role: primary

+4520461203

Claus Gravholt, MD, Prof.

Role: backup

References

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Mortensen KH, Andersen NH, Gravholt CH. Cardiovascular phenotype in Turner syndrome--integrating cardiology, genetics, and endocrinology. Endocr Rev. 2012 Oct;33(5):677-714. doi: 10.1210/er.2011-1059. Epub 2012 Jun 15.

Reference Type BACKGROUND
PMID: 22707402 (View on PubMed)

Hjerrild BE, Mortensen KH, Sorensen KE, Pedersen EM, Andersen NH, Lundorf E, Hansen KW, Horlyck A, Hager A, Christiansen JS, Gravholt CH. Thoracic aortopathy in Turner syndrome and the influence of bicuspid aortic valves and blood pressure: a CMR study. J Cardiovasc Magn Reson. 2010 Mar 11;12(1):12. doi: 10.1186/1532-429X-12-12.

Reference Type BACKGROUND
PMID: 20222980 (View on PubMed)

Executive Committee. The Diagnosis and Treatment of Peripheral Lymphedema: 2016 Consensus Document of the International Society of Lymphology. Lymphology. 2016 Dec;49(4):170-84.

Reference Type BACKGROUND
PMID: 29908550 (View on PubMed)

Yu DX, Ma XX, Zhang XM, Wang Q, Li CF. Morphological features and clinical feasibility of thoracic duct: detection with nonenhanced magnetic resonance imaging at 3.0 T. J Magn Reson Imaging. 2010 Jul;32(1):94-100. doi: 10.1002/jmri.22128.

Reference Type BACKGROUND
PMID: 20578016 (View on PubMed)

Unno N, Nishiyama M, Suzuki M, Yamamoto N, Inuzuka K, Sagara D, Tanaka H, Konno H. Quantitative lymph imaging for assessment of lymph function using indocyanine green fluorescence lymphography. Eur J Vasc Endovasc Surg. 2008 Aug;36(2):230-236. doi: 10.1016/j.ejvs.2008.04.013. Epub 2008 Jun 4.

Reference Type BACKGROUND
PMID: 18534875 (View on PubMed)

Mohanakumar S, Telinius N, Kelly B, Lauridsen H, Boedtkjer D, Pedersen M, de Leval M, Hjortdal V. Morphology and Function of the Lymphatic Vasculature in Patients With a Fontan Circulation. Circ Cardiovasc Imaging. 2019 Apr;12(4):e008074. doi: 10.1161/CIRCIMAGING.118.008074.

Reference Type BACKGROUND
PMID: 30943769 (View on PubMed)

Other Identifiers

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1-10-72-138-23

Identifier Type: -

Identifier Source: org_study_id

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