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Unraveling the Mechanisms Underlying Primary Sclerosing Cholangitis Through a Multidisciplinary, Integrative Research Approach

Recruiting
Conditions
Primary Sclerosing Cholangitis
Registration Number
NCT04685200
Lead Sponsor
National Institute of Diabetes and Digestive and Kidney Diseases (NIDDK)
Brief Summary

Background:

Primary sclerosing cholangitis is a rare chronic liver disease. It affects the bile ducts of the

liver. It can result in bile duct infections, cirrhosis, cancer, and end stage liver disease. Researchers want to learn more about this disease.

Objective:

To understand the biological causes of primary sclerosing cholangitis.

Eligibility:

Adults age 18 and older who have primary sclerosing cholangitis.

Design:

Participants will be screened with a medical history, physical exam, and blood tests.

Participants will give blood, saliva, urine, and stool samples. They will have nasal swabs. They will complete surveys.

Participants will get an intravenous (IV) catheter. A plastic tube is inserted into an arm vein.

Participants will have a colonoscopy. A tube with a video camera at the end is inserted into the rectum.

Participants will have an upper endoscopy. A scope with a light and camera at its tip is used to look inside the upper digestive tract.

Participants will have a liver biopsy, entering through the chest wall or a neck vein. Blood is drawn from a blood vessel that carries blood to the liver. A liver tissue sample is taken.

Participants will have magnetic resonance imaging or spectroscopy. They will get a contrast agent through an IV.

Participants may have an optional bone marrow aspiration. A large needle is inserted into the hip to withdraw marrow.

Participants will have a liver ultrasound.

Participants will complete a 3-day food diary. They will have a nutrition assessment.

Participants may give contact details for people who live with them, to also take part in this study.

Participation will last for 12 months.

Detailed Description

Study Description:

We hypothesize that primary sclerosing cholangitis (PSC) develops as a consequence of a genetically driven aberrant immune response to commensal or pathogenic bacteria, and that unique genetic-immunemicrobial associations may underlie development of distinct disease patterns. We intend to conduct a thorough radiologic, endoscopic, histologic and microbiological investigation of patients with PSC in order to determine potential associations.

Objectives:

Primary Objective:

The ultimate goal of this study is to generate understanding of how factors driving pathogenesis in PSC interact by capturing and integrating collated datasets from across relevant biologic systems and interpreting those in the context of phenotypic presentation in one exceptionally well-characterized set of patients.

Secondary Objectives:

1. To collect comprehensive data on distinct patterns of disease expression, through single cell sequencing and microRNA and transcriptome profiling.

2. To conduct extensive phenotypic characterization of cellular and humoral immune responses as well as microbiome signatures at multiple anatomic sites.

3. To evaluate metabolic signatures or biomarkers for PSC diagnosis and prognostication.

4. To generate a humanized mouse model of each subject s disease by obtaining bone marrow aspirate.

Endpoints:

Primary Endpoint:

1. Identification of immune signatures at multiple levels and from different anatomical sites and tissues

2. Characterization of microbiome signatures (taxonomic and functional), as well as identification of specific species.

3. Identification of metabolomic signatures from different anatomical sites and tissues as well as identification of different biomarkers correlating with disease progression.

4. MicroRNA profiling of portal and systemic blood.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
143
Inclusion Criteria

Not provided

Exclusion Criteria

Not provided

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
The ultimate goal of this study is to generate understanding of how factors driving pathogenesis in PSC interact by capturing and integrating collated datasets from across relevant biologic systems and interpreting those in the context of phenot...End of Study

1. Identification of immune signatures at multiple levels and from different anatomical sites and tissues 2. Characterization of microbiome signatures (taxonomic and functional), as well as identification of specific species. 3. Identification of metabolomic signatures from different anatomical sites and tissues as well as identification of different biomarkers correlating with disease progression. 4. MicroRNA profiling of portal and systemic blood.

Secondary Outcome Measures
NameTimeMethod
To evaluate metabolic signatures or biomarkers for PSC diagnosis and prognosticationEnd of Study

High throughput metabolomics screening of portal and systemic blood, liver tissue and bile in search of bile acids and other target metabolites

To generate a humanized mouse model of each subject s disease by obtaining bone marrow aspirateEnd of Study

The study of immune phenotype and function in a host (in this case the mouse) na(SqrRoot) ve to immunosuppressive therapy

To collect comprehensive data on distinct patterns of disease expression, through single cell sequencing and microRNA and transcriptome profiling.End of Study

Single cell sequencing, microRNA and transcriptome profiling

To conduct extensive phenotypic characterization of cellular and humoral immune responses as well as microbiome signatures at multiple anatomic sitesEnd of Study

Immune phenotyping, immune repertoire sequencing and cytokine profiling techniques, as well as next generation sequencing of microbiota in saliva, stool, blood, bile, small intestine, colon and liver tissue

Trial Locations

Locations (1)

National Institutes of Health Clinical Center

🇺🇸

Bethesda, Maryland, United States

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