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RecruitingNCT05687149Updated Nov 6, 2026

Defining the Natural History of Squamous Cell Carcinoma in Fanconi Anemia

An observational study in Fanconi Anemia and Inherited Bone Marrow Failure Syndrome, sponsored by National Cancer Institute (NCI). Recruiting at 1 site in United States. Open to participants aged 8 Years to 90 Years. Per ClinicalTrials.gov, last updated 2026-11-06.

Sponsored by National Cancer Institute (NCI) · Observational

From the registry’s dates

  • Started Mar 2023; still recruiting 3 years 7 months later.
Study type
Observational
Model
Cohort
Time perspective
Other
Enrollment
200
Ages
8 Years to 90 Years
Sex
All
01

Study 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....

Read the detailed description

Study Description:

This is a natural history study involving questionnaires, clinical and research evaluations, clinical and research laboratory tests, review of medical records, and cancer surveillance. A prospective cohort of individuals with Fanconi anemia (FA) at very high risk of squamous cell carcinoma (SCC) will be screened and provide new information on oral potentially malignant lesion (OPML) development and robustly quantify the risk of progression of OPML to cancer in FA.

Objectives:

Primary Objectives:

  1. To establish a central program and a team of expert clinicians and scientists at the NIH Clinical Center to conduct a comprehensive longitudinal study of cancer screening in adolescent and young adults (AYA) with FA at high risk of SCC through detailed clinical evaluation and biospecimen collection.
  2. To characterize the clinical and pathological natural history of OPMLs in AYAs with FA using brush biopsies for cytopathologic diagnosis and DNA aneuploidy and correlate those findings with tissue biopsies and genomic analyses of oral epithelial dysplasia (OED) and SCC.
  3. To prospectively screen individuals with FA for early indicators for the development of esophageal and anogenital SCC and hematological malignancy.

Secondary Objectives:

  1. To identify genetic, epigenetic, and immunologic mechanisms underlying tumorigenesis and immune escape in individuals with FA.
  2. To facilitate the enrollment of individuals with FA with high-grade dysplasia or SCC in intra- and extra-mural precision intervention trials.

Endpoints:

Primary Endpoints:

  1. Characterize the natural history of OPMLs in FA, rates of progression, regression, and development of new lesions
  2. Determine the utility of brush biopsy to identify oral dysplasia and SCC in FA
  3. Identify potential precursor states for esophageal and anogenital cancers in FA
  4. Develop screening guidelines for esophageal and anogenital cancer in FA

Secondary Endpoints:

  1. Identify predictive biomarkers of oral SCC development
  2. Characterize genetic and epigenetic changes that lead to SCC development
  3. Facilitate patient enrollment in intervention trials
02

Conditions studied

  • Fanconi Anemia
  • Inherited Bone Marrow Failure Syndrome

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Keywords

  • Hereditary
  • Inherited Bone Marrow Failure Syndrome
  • Oral Potentially Malignant Lesion
  • Surveillance
  • Precancer
03

In context

Fanconi Anemia

95 studies on the registry are indexed under Fanconi Anemia; 20 are open to participants now.

This study's planned enrollment of 200 is above the median of 94 across 24 observational studies indexed under Fanconi Anemia.

Browse Fanconi Anemia studies →

Lead sponsor

National Cancer Institute (NCI) is the lead sponsor of 3,556 studies on the registry; 336 are open to participants now.

Of its 411 completed or terminated interventional studies of FDA-regulated products, 373 (91%) have results posted.

Counted across the registry records on this site, refreshed daily.

04

Who can participate

Ages eligible
8 Years to 90 Years
Sexes eligible
All
Accepts healthy volunteers
No
Sampling method
Non-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.

Go through the criteria one by one — yes, no or not sure — and take the list to your doctor or the study team. Kept in this browser only.

Inclusion criteria

  1. 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.

    1. Individuals with prior cancer diagnosis are eligible.
    2. Individuals from other countries are eligible provided they can travel to the USA on their own.
    3. Ability to understand and/or the willingness of the individual, parent, LAR, or minor s legal guardian to provide .

Exclusion criteria

EXCLUSION CRITERIA:

  1. Referred individuals for whom reported diagnosis of FA cannot be verified.
  2. Inability of the individual, parent, LAR, or legal guardian to understand and be willing to sign a written document.
Terms in these criteria, in plain language(1)

Underlined terms in the criteria open these too. Explained by clinicaltrials.gg, not part of the registry record. The study team decides who can take part.

Informed consent

Before you join, the study team explains what taking part involves — its purpose, what happens, the risks and possible benefits — and answers your questions, so you can decide freely. Consent is usually recorded on a form (the informed consent form, ICF) signed by you or, where the law allows, by someone legally authorised to decide for you.

You can keep asking questions during the study, you are told of new information that matters, and you can choose to leave at any time.

05

Study design

Observational model
Cohort
Time perspective
Other
Enrollment
200 participants (estimated)

Groups and cohorts

  • Fanconi anemia

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

06

What researchers measure

Primary outcomes

  1. Screen Patients with FA

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

    Time frame: ongoing

  2. 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.

    Time frame: ongoing

  3. Cohort of Patients with FA

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

    Time frame: ongoing

07

Study locations

1 of 1 sites recruiting
  • National Institutes of Health Clinical Center
    Bethesda, Maryland 20892, United States
    Recruiting
    • For more information at the NIH Clinical Center contact National Cancer Institute Referral Office · Contact888-624-1937
08

References and documents

Publications

  • 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. PubMed 32022466 ↗
  • 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. PubMed 29051281 ↗
09

Timeline

2 registry updates since Sep 25, 2026
ElapsedEstimatedEstimated, not yet reached

Started Mar 2023 (actual). Primary completion is estimated for Dec 2035, 153 months after the start.

How these figures are worked out, and which studies are left out of them.

Registered
Jan 14, 2023
First posted
Jan 18, 2023
StartActual
Mar 23, 2023
Primary completionEst.
Dec 31, 2035
CompletionEst.
Dec 31, 2035
Last update
Nov 6, 2026
What these dates mean
Start.
When the first participant enrolled; while estimated, when the study expects to open to recruitment.
Primary completion.
When the last participant was examined or treated to collect data for the study's main question, its primary outcome. A study that stopped early gives the date its data collection stopped.
Completion.
When the last participant's last visit took place, for every outcome, secondary ones and side effects included: the end of the study. Often later than primary completion, sometimes the same day.
Actual and Est.
Actual means the registry says the date has happened. Est. (estimated) is a date the sponsor plans or expects. It can still move, and the sponsor is expected to change it to actual once it has passed.
Minor edits
Nothing that changes what the study is or who can join. Edited: verification date
2 updates, last Nov 6, 2026
Show all 2 updates
  1. Nov 6, 2026
    Minor edits only
    + 1 other change: verification date
  2. Oct 1, 2026
    Minor edits only
    + 1 other change: verification date

From the registry record's own update history. This site started tracking changes on Sep 25, 2026; for anything earlier, see the record history on ClinicalTrials.gov ↗

10

Registry details

Key details

Study ID
NCT05687149
Responsible party
Sponsor
First posted
Jan 18, 2023
Start date
Mar 23, 2023
Primary completion
Dec 31, 2035 (estimated)
Completion
Dec 31, 2035 (estimated)
Last update
Nov 6, 2026

Study contacts

NCI Family Study Referrals
Contact
ncifamilystudyreferrals@mail.nih.gov
(800) 518-8474
Lisa J McReynolds, M.D.
Contact
mcreynoldslj@mail.nih.gov
(240) 276-5047
Lisa J McReynolds, M.D.
principal investigator · National Cancer Institute (NCI)

Oversight

FDA-regulated drug
No
FDA-regulated device
No
View the source record on ClinicalTrials.gov ↗

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