NCT01907295

Brief Summary

Pulmonary arterial hypertension (PAH), or high blood pressure in the lungs, is a rare condition that can shorten life. Although the cause of this disease is usually unknown, in about 70% of heritable and 15-20% of idiopathic cases there is a change in a gene (a mutation) that controls how blood vessels grow and function. The gene is called bone morphogenetic protein type receptor 2 (BMPR2). Although mutations in BMPR2 are a risk factor for PAH, not everyone with a mutation gets the disease. Additional genetic and environmental factors are likely to contribute. The investigators suspect that mutations in other genes are responsible for some cases of PAH. In this study the investigators aim to recruit all patients with PAH and some of their relatives and follow them up for several years. The investigators hope to discover new mutations for this disease and to determine what factors lead to poor outcome, and to understand what triggers disease in patients with mutations. Who can participate? Adults with PAH, their relatives and controls (one off blood sample)

Trial Health

43
At Risk

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Trial has exceeded expected completion date
Enrollment
3,600

participants targeted

Target at P75+ for all trials

Timeline
Completed

Started Feb 2014

Longer than P75 for all trials

Geographic Reach
1 country

8 active sites

Status
unknown

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

First Submitted

Initial submission to the registry

July 8, 2013

Completed
16 days until next milestone

First Posted

Study publicly available on registry

July 24, 2013

Completed
6 months until next milestone

Study Start

First participant enrolled

February 1, 2014

Completed
8.8 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

December 1, 2022

Completed
Same day until next milestone

Study Completion

Last participant's last visit for all outcomes

December 1, 2022

Completed
Last Updated

May 20, 2022

Status Verified

May 1, 2022

Enrollment Period

8.8 years

First QC Date

July 8, 2013

Last Update Submit

May 19, 2022

Conditions

Keywords

idiopathicheritableanorexigenPCH/PVOD

Outcome Measures

Primary Outcomes (1)

  • To recruit a national cohort (1000 subjects) of heritable, idiopathic PAH and PVOD/PCH cases.

    The purpose of this study is to set up a national cohort of heritable, idiopathic PAH cases, PVOD/PCH and their relatives, to study the genetic and environmental contributions to disease. Setting up of this cohort of patients and relatives will provide the best resource for understanding what causes or triggers the disease, how to predict risk of death and response to therapy in individual patients, and to provide new ways of preventing and treating pulmonary arterial hypertension. The study will enable a better understanding for the first time the natural history of PAH, whether inherited or not. National outcomes to be measured will include survival, progression of the disease, changes in 6 minute walk distance, admissions to hospital for PAH and cause of death. Incidence of new cases of PAH will be measured in relatives as well.

    8 years

Secondary Outcomes (1)

  • To recruit PAH patients (1000) and family members to a Biorepository for serum/plasma and urine to identify biomarkers of disease onset, progression and response to treatment.

    8 years

Other Outcomes (2)

  • longitudinal clinical evaluation and sampling of HPAH family members

    8 years

  • Elucidation of the underlying genetic architecture of idiopathic and heritable PAH

    8 years

Study Arms (2)

Patients

Patients diagnosed with idiopathic, anorexigen-induced, heritable PAH and PVOD/PCH

Relatives and controls

Relative has a family member diagnosed with idiopathic, anorexigen-induced, heritable PAH and PVOD/PCH Self declared healthy individuals

Eligibility Criteria

Sexall
Healthy VolunteersYes
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)
Sampling MethodNon-Probability Sample
Study Population

Patients diagnosed with idiopathic, anorexigen-induced, heritable PAH or PVOD. Relative who has a family member diagnosed with idiopathic, anorexigen-induced, heritable PAH or PVOD/PCH

You may qualify if:

  • Participant is willing and able to give informed consent for participation in the study.
  • Male or Female, any age
  • Participant is willing and able to give informed consent for participation in the study.
  • Male or Female, any age
  • Has a family member diagnosed with idiopathic, anorexigen-induced, PVOD/PCH or heritable PAH

You may not qualify if:

  • The participant may not enter the study if ANY of the following apply:
  • Patient is unable to give informed consent.
  • Not suffering from idiopathic, anorexigen-induced, PVOD/PCH or heritable PAH
  • The participant may not enter the study if ANY of the following apply:
  • Patient is unable to give informed consent.
  • Participant is willing and able to give informed consent for participation in the study.
  • Self-reported to be healthy
  • Age range up to 75 years

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (8)

Royal United Hospitals Bath

Bath, BA1 3NG, United Kingdom

RECRUITING

Royal Papworth Hospital NHS Trust

Cambridge, United Kingdom

RECRUITING

Golden Jubilee National Hospital

Glasgow, United Kingdom

RECRUITING

Imperial Hospital

London, United Kingdom

RECRUITING

Royal Brompton Hospital

London, United Kingdom

RECRUITING

Royal Free Hospital

London, United Kingdom

RECRUITING

Freeman Hospital

Newcastle, United Kingdom

RECRUITING

Sheffield Hospital

Sheffield, United Kingdom

RECRUITING

Related Links

Biospecimen

Retention: SAMPLES WITH DNA

All subjects will have a sample of blood taken for next generation genetic sequencing (up to their entire genome). Samples will be sequenced to identify novel genetic mutations associated with PAH. This blood sample will be taken once during the study

MeSH Terms

Conditions

Pulmonary Arterial Hypertension

Condition Hierarchy (Ancestors)

Hypertension, PulmonaryLung DiseasesRespiratory Tract Diseases

Study Officials

  • Nicholas Morrell

    University of Cambridge

    PRINCIPAL INVESTIGATOR

Central Study Contacts

Study Design

Study Type
observational
Observational Model
COHORT
Time Perspective
PROSPECTIVE
Target Duration
3 Years
Sponsor Type
OTHER
Responsible Party
PRINCIPAL INVESTIGATOR
PI Title
BHF Professor of Cardiopulmonary Medicine

Study Record Dates

First Submitted

July 8, 2013

First Posted

July 24, 2013

Study Start

February 1, 2014

Primary Completion

December 1, 2022

Study Completion

December 1, 2022

Last Updated

May 20, 2022

Record last verified: 2022-05

Data Sharing

IPD Sharing
Will not share

Consent for sharing of non identifiable study data for regulatory authorities, third parties including commercial companies outside the UK and NHS trusts where it is relevant to taking part in this research.

Locations