Lymphatic DiseasesNCT05731141
A Prospective Natural History Study of Lymphatic Anomalies
An observational study of Lymphatic Diseases and Lymphatic Abnormalities, run by Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD) at 2 sites in United States.
Trial summary
Background: The lymphatic system is a network of vessels that carry a clear fluid called lymph through the body. Problems in the lymphatic system can cause pain, fluid buildup, and issues with immunity. There is much researchers do not understand about lymphatic anomalies. Read more about this study
- Who is it for?
- Ages 1 day to 100 yearsHealthy volunteers can take part
- Conditions the study is for
- Lymphatic Diseases, Lymphatic Abnormalities
- How many people
- 1,200 participants
- Where
- 2 sitesUnited States
- Study dates
- 20 Mar 2023to 31 Dec 2028
- Phase
- Observational
- Are there results?
- Not yet: the study is under way
Key requirements
Who can take part, as listed by the sponsor. Only the study team can confirm whether you are eligible, so take this list to your doctor.
- Ages 1 day to 100 years
- All sexes
- Healthy volunteers accepted
- Child, Adult, Older adult
Study population: Patients with lymphatic anomalies and their parents.
Who may be able to join
- Affected (Proband)
- In order to be eligible to participate in this study, an individual must meet one of the following criteria as determined after review of medical history:
- Current or history of lymphatic anomaly or symptoms suggestive of a lymphatic disorder Or
- An ill-defined vascular anomaly that is suspected to have an abnormal lymphatic component Or
- A pathogenic, likely pathogenic, or VUS in a genetic disorder with a known lymphatic component Or
- Clinical diagnosis of a syndrome with a known lymphatic component
Show all 9Show fewer
- Unaffected (First Degree Relatives: Parents and Siblings)
- Genetic variants underlying complex lymphatic anomalies can be passed down through parents or be new in a child (de novo). Inclusion of first-degree relatives will assist in genetic analysis to delineate whether the variant is inherited or de novo.
- To be eligible to participate as a first degree relative in this study, an individual must be a first-degree family member of an affected participants
Who cannot join
- Affected Proband
- An individual who meets any of the following criteria will be excluded from participation in this study after review of medical history, concomitant medication and allergy review, anthropometrics, and performance status:
- -Any condition, in the opinion of the investigator, that would increase risk of participation or impair their ability to comply with protocol requirements.
- Lymphatic anomalies that are definitively determined to be secondary by the principal investigator will be excluded from this study. For example, participants who develop a lymphedema after breast cancer surgery.
- Unaffected (First Degree Relatives)
- -Any condition, in the opinion of the investigator, that would increase risk of participation or impair their ability to comply with protocol requirements.
Trial locations and contacts
2 sites in 1 country. Contact the study team or a site near you to ask about taking part.
United States2 sites, 2 recruiting
- Recruiting
National Institutes of Health Clinical Center
Bethesda, Maryland, 20892
NIH Clinical Center Office of Patient Recruitment (OPR), 800-411-1222 ext. TTY dial 711, ccopr@nih.gov
- Not yet recruiting
Children's Hospital of Philadelphia
Philadelphia, Pennsylvania, 19104
Christopher Smith, 215-590-2482, SmithC12@chop.edu
About this study
Background:
The lymphatic system is a network of vessels that carry a clear fluid called lymph through the body. Problems in the lymphatic system can cause pain, fluid buildup, and issues with immunity. There is much researchers do not understand about lymphatic anomalies. In this natural history study, they will collect data from a lot of people over a long time.
Read more
Objective:
To better understand why lymphatic anomalies develop. The goal is to improve future treatments.
Eligibility:
People aged 0 days and older with a suspected or confirmed lymphatic anomaly. Their unaffected parents or siblings aged 7 years or older are also needed.
Design:
Participants may remain in the study indefinitely. Affected participants may be evaluated every 10 months to 2 years. Some participants will be seen over telemedicine. Others will be seen at the NIH Clinical Center for 2-5 days.
All participants will have a physical exam. They may provide specimens including blood, saliva, hair follicles, stool, skin, and other tissues. Samples may be used for genetic testing.
Participants may undergo other tests depending on their medical conditions. The NIH Clinical Center visit may include:
Heart tests include placing stickers on the chest to measure electrical activity and using sound waves to capture pictures of the heart.
A lung test measures the muscle strength in the chest. Participants will blow into a tube.
Photographs may be taken of participants faces and other features.
Imaging scans will take pictures of the inside of the body. One scan will measure bone density.
One type of scan tracks how lymph fluid moves through the body. Participants will be under anesthesia, and they will be injected with a dye.
Read the detailed description
Study Description:
A natural history study for lymphatic anomalies to systematically evaluate the disease phenotypes and long-term outcomes to provide improved prognostication to families, establish screening/monitoring guidelines, determine best practices for genetic diagnosis, explore family opinions, and explore fertility for those on long term medication management. This study will allow us to identify novel end points for future clinical trials.
Objectives:
Primary objectives:
- To establish a longitudinal cohort of participants with lymphatic anomalies
- To longitudinally determine the age at presentation and incidence of clinical features
Secondary objectives:
- To establish a longitudinal biospecimen repository
- To determine the best practices for genetic diagnosis based on phenotype.
- To determine the malignant potential of anomalies longitudinally
Endpoints:
Primary endpoints:
- The number of participants with lymphatic anomalies
- For each clinical feature or symptoms, the range of ages at the development of that feature/symptom and fraction of participants with that feature
- Quantification and identification of novel features associated with disease.
Secondary endpoints:
- The number of specimens collected
- Diagnostic yields by phenotype and genetic test methodology
- Number of malignancies related to the primary lesion that have developed
- Conditions
- Lymphatic Diseases, Lymphatic Abnormalities
- Keywords
- Protein Losing Enteropathy, Lymphedema, Lymphangiectasia, Kaposiform Lymphangiomatosis, Gorham Stout Disease, Generalized Lymphatic Anomaly, Complex Lymphatic Anomaly, Chylous Effusion, Chylous Ascites, Central Conducting Lymphatic Anomaly
- Medical subject headings
- Lymphatic Diseases, Lymphatic Abnormalities, Protein-Losing Enteropathies, Lymphedema, Lymphangiectasis, Osteolysis, Essential, Chylous Ascites
What participants receive
What participants receive. In a randomised study each person is assigned to one group by chance.
| Study group | Role | What they receive |
|---|---|---|
| First Degree Relatives | Siblings or parents of patients. | |
| Patients | Patients with lymphatic anomalies. |
What is being measured
The primary outcome is the main question the study is designed to answer.
To establish a longitudinal cohort of participants with lymphatic anomalies
We plan to enroll a group of participants willing to participate in the study over time.
Measured: 12/31/2028
To longitudinally determine the age at presentation and incidence of clinical features
For each clinical feature or symptoms, the range of ages at the development of that feature/symptom and fraction of participants with that feature
Measured: 12/31/2028
Secondary outcomes (3)
To establish a longitudinal biospecimen repository
We plan to collect biospecimens including, but not limited to blood and stool from participants over time.
Measured: 12/31/2028
To determine the best practices for genetic diagnosis based on phenotype
We will analyze diagnostic yields by phenotype (how many participants are able to have a genetic diagnosis in proportion to the number of participants who receive genetic testing) and genetic test methodology (to determine which genetic test is most helpful in diagnosing lymphatic anomalies)
Measured: 12/31/2028
To determine the malignant potential of anomalies longitudinally
We will track the number of malignancies related to the primary lesion that have developed
Measured: 12/31/2028
Timeline
From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.
- First posted16 Feb 2023
- Study start20 Mar 2023
- Primary completion31 Dec 2028 (estimated)
- Study completion31 Dec 2028 (estimated)
Resources
- Official record on ClinicalTrials.govThe registry entry this page is built from, with its full change history.
- Publications (3)Papers the sponsor has linked to this study.
- Other trials for Lymphatic DiseasesStudies of the same condition, open and under way.
- What the trial phases meanHow a treatment moves from first tests in people to approval.
More details
For doctors and researchers: how the study is designed, who runs it, and its registry IDs.
Study design
- Study type
- Observational
- Observational model
- Cohort
- Time perspective
- Prospective
- Enrolment
- 1,200 participants (estimated)
Sponsor and investigators
- Lead sponsor
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)
- Sponsor type
- US National Institutes of Health
- Responsible party
- Sponsor
- Investigators
- Sarah E Sheppard, M.D., Principal investigator, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)
IDs, dates and publications
- ClinicalTrials.gov ID
- NCT05731141
- Sponsor's protocol ID
- 10001084
- Other IDs
- 001084-CH
- Status last verified
- 21 Oct 2026
- First submitted
- 15 Feb 2023
- Last updated
- 23 Oct 2026
- Results posted
- No
- FDA-regulated drug
- No
- FDA-regulated device
- No
- Individual data sharing
- Yes: Investigators will share human data generated in this research for future research as follows:@@@@@@De-identified data in an NIH-funded or approved public repository. @@@@@@De-identified data in another public repository. @@@@@@De-identified data in BTRIS (automatic for activities in the Clinical Center)@@@@@@De-identified or identified data with approved outside collaborators under appropriate agreements.
Publications
Makinen T, Boon LM, Vikkula M, Alitalo K. Lymphatic Malformations: Genetics, Mechanisms and Therapeutic Strategies. Circ Res. 2021 Jun 25;129(1):136-154. doi: 10.1161/CIRCRESAHA.121.318142. Epub 2021 Jun 24.
PubMed 34166072Brouillard P, Witte MH, Erickson RP, Damstra RJ, Becker C, Quere I, Vikkula M. Primary lymphoedema. Nat Rev Dis Primers. 2021 Oct 21;7(1):77. doi: 10.1038/s41572-021-00309-7.
PubMed 34675250Liu M, Smith CL, Biko DM, Li D, Pinto E, O'Connor N, Skraban C, Zackai EH, Hakonarson H, Dori Y, Sheppard SE. Genetics etiologies and genotype phenotype correlations in a cohort of individuals with central conducting lymphatic anomaly. Eur J Hum Genet. 2022 Sep;30(9):1022-1028. doi: 10.1038/s41431-022-01123-9. Epub 2022 May 24.
PubMed 35606495
Study at a glance
From the registry record, last updated by the sponsor on 23 Oct 2026. Recruitment status is as the sponsor reported it: confirm with the study team.
- Official title
- A Prospective Natural History Study of Lymphatic Anomalies
- ClinicalTrials.gov ID
- NCT05731141
- Acronym / sponsor ID
- 10001084
- Conditions
- Lymphatic Diseases, Lymphatic Abnormalities
- Keywords
- Protein Losing Enteropathy, Lymphedema, Lymphangiectasia, Kaposiform Lymphangiomatosis, Gorham Stout Disease, Generalized Lymphatic Anomaly, Complex Lymphatic Anomaly, Chylous Effusion
- Study type
- Observational
- Recruitment status
- Recruiting
- Enrolment
- 1,200 participants (estimated)
- Lead sponsor
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)US National Institutes of Health
- Study start
- 20 Mar 2023 (actual)
- Primary completion
- 31 Dec 2028 (estimated)
- Study completion
- 31 Dec 2028 (estimated)
- First posted
- 16 Feb 2023 (actual)
- Results
- Not posted in the source record
Clinical summary
The research question as the sponsor registered it. This describes what the study plans to test, not what it found.
Background:
The lymphatic system is a network of vessels that carry a clear fluid called lymph through the body. Problems in the lymphatic system can cause pain, fluid buildup, and issues with immunity. There is much researchers do not understand about lymphatic anomalies. In this natural history study, they will collect data from a lot of people over a long time.
Objective:
To better understand why lymphatic anomalies develop. The goal is to improve future treatments.
Eligibility:
People aged 0 days and older with a suspected or confirmed lymphatic anomaly. Their unaffected parents or siblings aged 7 years or older are also needed.
Design:
Participants may remain in the study indefinitely. Affected participants may be evaluated every 10 months to 2 years. Some participants will be seen over telemedicine. Others will be seen at the NIH Clinical Center for 2-5 days.
All participants will have a physical exam. They may provide specimens including blood, saliva, hair follicles, stool, skin, and other tissues. Samples may be used for genetic testing.
Participants may undergo other tests depending on their medical conditions. The NIH Clinical Center visit may include:
Heart tests include placing stickers on the chest to measure electrical activity and using sound waves to capture pictures of the heart.
A lung test measures the muscle strength in the chest. Participants will blow into a tube.
Photographs may be taken of participants faces and other features.
Imaging scans will take pictures of the inside of the body. One scan will measure bone density.
One type of scan tracks how lymph fluid moves through the body. Participants will be under anesthesia, and they will be injected with a dye.
Detailed description
Study Description:
A natural history study for lymphatic anomalies to systematically evaluate the disease phenotypes and long-term outcomes to provide improved prognostication to families, establish screening/monitoring guidelines, determine best practices for genetic diagnosis, explore family opinions, and explore fertility for those on long term medication management. This study will allow us to identify novel end points for future clinical trials.
Objectives:
Primary objectives:
- To establish a longitudinal cohort of participants with lymphatic anomalies
- To longitudinally determine the age at presentation and incidence of clinical features
Secondary objectives:
- To establish a longitudinal biospecimen repository
- To determine the best practices for genetic diagnosis based on phenotype.
- To determine the malignant potential of anomalies longitudinally
Endpoints:
Primary endpoints:
- The number of participants with lymphatic anomalies
- For each clinical feature or symptoms, the range of ages at the development of that feature/symptom and fraction of participants with that feature
- Quantification and identification of novel features associated with disease.
Secondary endpoints:
- The number of specimens collected
- Diagnostic yields by phenotype and genetic test methodology
- Number of malignancies related to the primary lesion that have developed
Study design
- Observational model
- Cohort
- Time perspective
- Prospective
- Number of arms
- 2
| Arm | Type | Receives |
|---|---|---|
| First Degree Relatives | – | –Siblings or parents of patients. |
| Patients | – | –Patients with lymphatic anomalies. |
Who may qualify
- Ages
- 1 day to 100 years
- Sex
- All
- Healthy volunteers
- Accepted
- Age groups
- Child, Adult, Older adult
- Study population
- Patients with lymphatic anomalies and their parents.
Key inclusion criteria (9 listed)
- Affected (Proband)
- In order to be eligible to participate in this study, an individual must meet one of the following criteria as determined after review of medical history:
- Current or history of lymphatic anomaly or symptoms suggestive of a lymphatic disorder Or
- An ill-defined vascular anomaly that is suspected to have an abnormal lymphatic component Or
- A pathogenic, likely pathogenic, or VUS in a genetic disorder with a known lymphatic component Or
Key exclusion criteria (6 listed)
- Affected Proband
- An individual who meets any of the following criteria will be excluded from participation in this study after review of medical history, concomitant medication and allergy review, anthropometrics, and performance status:
- -Any condition, in the opinion of the investigator, that would increase risk of participation or impair their ability to comply with protocol requirements.
- Lymphatic anomalies that are definitively determined to be secondary by the principal investigator will be excluded from this study. For example, participants who develop a lymphedema after breast cancer surgery.
- Unaffected (First Degree Relatives)
Eligibility is decided by the study team after screening; this overview does not establish that a patient qualifies. Full inclusion and exclusion criteria.
Primary outcome
To establish a longitudinal cohort of participants with lymphatic anomalies
Time frame: 12/31/2028
We plan to enroll a group of participants willing to participate in the study over time.
To longitudinally determine the age at presentation and incidence of clinical features
Time frame: 12/31/2028
For each clinical feature or symptoms, the range of ages at the development of that feature/symptom and fraction of participants with that feature
3 secondary outcomes
To establish a longitudinal biospecimen repository
Time frame: 12/31/2028
To determine the best practices for genetic diagnosis based on phenotype
Time frame: 12/31/2028
To determine the malignant potential of anomalies longitudinally
Time frame: 12/31/2028
Results and safety
This study is still under way. Results are not available in the source record yet.
Research links
- Official record on ClinicalTrials.govLast updated by the sponsor 23 Oct 2026.
- Background: PubMed 34166072Makinen T, Boon LM, Vikkula M, Alitalo K. Lymphatic Malformations: Genetics, Mechanisms and Therapeutic Strategies. Circ Res. 2021 Jun 25;129(1):136-154. doi: 10.1161/CIRCRESAHA.121.318142. Epub 2021 Jun 24.
- Background: PubMed 34675250Brouillard P, Witte MH, Erickson RP, Damstra RJ, Becker C, Quere I, Vikkula M. Primary lymphoedema. Nat Rev Dis Primers. 2021 Oct 21;7(1):77. doi: 10.1038/s41572-021-00309-7.
- Background: PubMed 35606495Liu M, Smith CL, Biko DM, Li D, Pinto E, O'Connor N, Skraban C, Zackai EH, Hakonarson H, Dori Y, Sheppard SE. Genetics etiologies and genotype phenotype correlations in a cohort of individuals with central conducting lymphatic anomaly. Eur J Hum Genet. 2022 Sep;30(9):1022-1028. doi: 10.1038/s41431-022-01123-9. Epub 2022 May 24.
- NIH Clinical Center Detailed Web PageLink provided by the sponsor.
- Individual participant dataSharing planned: Yes. Investigators will share human data generated in this research for future research as follows:@@@@@@De-identified data in an NIH-funded or approved public repository. @@@@@@De-identified data in another public repository. @@@@@@De-identified data in BTRIS (automatic for activities in the Clinical Center)@@@@@@De-identified or identified data with approved outside collaborators under appropriate agreements.
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Referral and contact
AzTrials does not enrol participants or take referrals. Contact the study team or a site listed below; they decide who can take part.
Sarah E Sheppard, M.D. principal investigator
Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD)
2 sites in 1 country: see every site and its status.
Questions about this trial
What is this clinical trial studying?
An observational study of Lymphatic Diseases and Lymphatic Abnormalities, run by Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD) at 2 sites in United States.
The full registered description is in the About this study section and the official record.
Who may be eligible to take part?
The sponsor lists 9 inclusion and 6 exclusion criteria. Eligibility is decided by the study team after screening, so read the full criteria and contact a listed site.
Is this clinical trial currently recruiting?
The record says: looking for participants now, as of the sponsor's last update on 23 Oct 2026. Status can change between updates, so confirm with the study contact before making plans.
Where is the study taking place?
The record lists 2 sites in United States. Whether a site is taking part right now should be confirmed with that site. See every site.
What are the possible benefits and risks?
They depend on the study and the treatment, and no study can promise a benefit. The study team explains the known risks during informed consent, before anyone joins; the record itself does not list them.
Are results available for this study?
This study is still under way. Results are not available in the source record yet.
How can a doctor refer a patient or contact the research team?
Contact Andrea I Bowling, C.R.N.P. ((301) 451-3824, nichd_lymphaticanoma@mail.nih.gov), or a site near the patient. AzTrials does not enrol participants or take referrals.
What does the primary outcome measure?
To establish a longitudinal cohort of participants with lymphatic anomalies (time frame: 12/31/2028). The study lists 2 primary outcomes in all. It is what the study was designed to measure, not a finding.
Where can I read the original study information?
On the ClinicalTrials.gov record for NCT05731141, which also lists 3 linked publications.
Does a listing mean the treatment is approved or proven to work?
No. A listing means the study is registered. It does not show that the treatment is safe, works, or is approved by a regulator.
How often is this information updated?
AzTrials copies changed records from ClinicalTrials.gov every night and reloads the whole registry every week. The sponsor last updated this record on 23 Oct 2026.