NCT05687149

Brief Summary

Background: Fanconi anemia (FA) is an inherited disorder. People with FA are more likely to get certain cancers, especially squamous cell carcinoma (SCC). These cancers usually appear first in the mouth, esophagus, and genital and anal areas. Early detection of SCCs may help improve survival rates for people with FA. Objective: This natural history study will regularly screen people with FA for SCC. Eligibility: People aged 12 years and older with FA or a prior cancer diagnosis. Children aged 8 to 11 years with FA may also be eligible. Design: Participants will receive a comprehensive screening for cancer or early signs of cancer. Participants will have a physical exam. They will provide blood and saliva samples. Cells will be collected by rubbing a swab on the inside of the cheeks. A skin sample may be removed from the back, buttocks, or inside of the upper arm. Participants will have pictures taken of their mouth. Any mouth sores will be mapped. Cells will be collected from the sores with a small brush. Specialists will examine the participant s ears, nose, throat, teeth, and skin. Adult participants may have a gastrointestinal exam or pelvic exam. Participants may have an endoscopy. A long tube with a camera and a light will be inserted through the mouth and down into the stomach. Participants may have a liver ultrasound. A wand will be pressed against their belly to get pictures of the organs inside the body. Participants will have screenings every year for up to 10 years. Each visit will last up to 3 days. They will have remote follow-up visits every 6 - 8 months....

Trial Health

77
On Track

Trial Health Score

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

Enrollment
200

participants targeted

Target at P75+ for all trials

Timeline
118mo left

Started Mar 2023

Longer than P75 for all trials

Geographic Reach
1 country

1 active site

Status
recruiting

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

Study Progress24%
Mar 2023Dec 2035

First Submitted

Initial submission to the registry

January 14, 2023

Completed
4 days until next milestone

First Posted

Study publicly available on registry

January 18, 2023

Completed
2 months until next milestone

Study Start

First participant enrolled

March 23, 2023

Completed
12.8 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

December 31, 2035

Expected
Same day until next milestone

Study Completion

Last participant's last visit for all outcomes

December 31, 2035

Last Updated

March 17, 2026

Status Verified

March 13, 2026

Enrollment Period

12.8 years

First QC Date

January 14, 2023

Last Update Submit

March 14, 2026

Conditions

Keywords

HereditaryInherited Bone Marrow Failure SyndromeOral Potentially Malignant LesionSurveillancePrecancer

Outcome Measures

Primary Outcomes (3)

  • Screen Patients with FA

    Prospectively screen individuals with FA for early indicators for the development of esophageal and anogenital SCC.

    ongoing

  • Clinical and Pathological Natural History of Oral Potentially Malignant Lesion

    Characterize the clinical and pathological natural history of OPMLs in AYAs with FA using brush biopsies for cytopathologic diagnosis and aneuploidy and correlate those findings with tissue biopsies and genomic analyses of oral epithelial dysplasia (OED) and SCC.

    ongoing

  • Cohort of Patients with FA

    A prospective cohort of individuals with Fanconi anemia (FA) at very high risk of squamous cell carcinoma (SCC)

    ongoing

Study Arms (1)

Fanconi anemia

A prospective cohort of individuals with Fanconi anemia (FA) at very high risk of squamous cell carcinoma (SCC)

Eligibility Criteria

Age8 Years - 90 Years
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)
Sampling MethodNon-Probability Sample
Study Population

Persons \>=12 years with FA primarily from North America will be included in the study. Persons \< 12 years can also be included if they have a history of persistent oral potentially malignant lesions, dysphagia, or other concerning symptoms. Persons with FA from other countries are eligible provided they can travel to USA on their own.

You may qualify if:

  • On referral, persons \>= 12 years with FA primarily from North America will be included. An individual with FA who is 8 - 11 years can also be included if they have a history of persistent OPMLs, dysphagia, or other concerning symptoms.
  • Individuals with prior cancer diagnosis are eligible.
  • Individuals from other countries are eligible provided they can travel to the USA on their own.
  • Ability to understand and/or the willingness of the individual, parent, LAR, or minor s legal guardian to provide informed consent.

You may not qualify if:

  • Referred individuals for whom reported diagnosis of FA cannot be verified.
  • Inability of the individual, parent, LAR, or legal guardian to understand and be willing to sign a written informed consent document.

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (1)

National Institutes of Health Clinical Center

Bethesda, Maryland, 20892, United States

RECRUITING

Related Publications (2)

  • Velleuer E, Dietrich R, Pomjanski N, de Santana Almeida Araujo IK, Silva de Araujo BE, Sroka I, Biesterfeld S, Bocking A, Schramm M. Diagnostic accuracy of brush biopsy-based cytology for the early detection of oral cancer and precursors in Fanconi anemia. Cancer Cytopathol. 2020 Jun;128(6):403-413. doi: 10.1002/cncy.22249. Epub 2020 Feb 5.

    PMID: 32022466BACKGROUND
  • Alter BP, Giri N, Savage SA, Rosenberg PS. Cancer in the National Cancer Institute inherited bone marrow failure syndrome cohort after fifteen years of follow-up. Haematologica. 2018 Jan;103(1):30-39. doi: 10.3324/haematol.2017.178111. Epub 2017 Oct 19.

    PMID: 29051281BACKGROUND

Related Links

MeSH Terms

Conditions

Fanconi AnemiaCongenital Bone Marrow Failure Syndromes

Condition Hierarchy (Ancestors)

Anemia, Hypoplastic, CongenitalAnemia, AplasticAnemiaHematologic DiseasesHemic and Lymphatic DiseasesBone Marrow Failure DisordersBone Marrow DiseasesGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and AbnormalitiesDNA Repair-Deficiency DisordersMetabolic DiseasesNutritional and Metabolic DiseasesInfant, Newborn, Diseases

Study Officials

  • Lisa J McReynolds, M.D.

    National Cancer Institute (NCI)

    PRINCIPAL INVESTIGATOR

Central Study Contacts

Lisa J McReynolds, M.D.

CONTACT

Study Design

Study Type
observational
Observational Model
COHORT
Time Perspective
OTHER
Sponsor Type
NIH
Responsible Party
SPONSOR

Study Record Dates

First Submitted

January 14, 2023

First Posted

January 18, 2023

Study Start

March 23, 2023

Primary Completion (Estimated)

December 31, 2035

Study Completion (Estimated)

December 31, 2035

Last Updated

March 17, 2026

Record last verified: 2026-03-13

Locations