Evidence map›Paper›PMID 38907138›Full record

Observational studyFamilial cancer2024

Benign tumors and non-melanoma skin cancers in patients with Fanconi anemia.

Aura Enache, Bia Sajjad, Burak Altintas, Neelam Giri, Lisa J McReynolds, Edward W Cowen

Erratum issued Registry-linked trialAbstract readObservational Study
In one paragraph

Observational study in Familial cancer, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. It is linked to trial NCT00027274 (Etiologic Investigation of Cancer Susceptibility in Inherited Bone Marrow Failure Syndromes), which is not on this map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT00027274 recruitingnot on this map

Etiologic Investigation of Cancer Susceptibility in Inherited Bone Marrow Failure Syndromes: A Natural History Study

TypeobservationalSponsorNational Cancer Institute (NCI)Ran2001Enrolled4,000ConditionsDiamond Blackfan Anemia, Dyskeratosis Congenita, Fanconi Anemia, Shwachman Diamond Syndrome
3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Skin cancer risk in hereditary mixed cancer syndromes.Hereditary cancer in clinical practice · 2025
    Review
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

6 authors.

Aura Enache *Division of Cancer Epidemiology and Genetics, Clinical Genetics Branch, National Cancer Institute, 9609 Medical Center Drive 6E434, Bethesda, MD, 20892, USA.
Bia Sajjad *Division of Cancer Epidemiology and Genetics, Clinical Genetics Branch, National Cancer Institute, 9609 Medical Center Drive 6E434, Bethesda, MD, 20892, USA.
Burak AltintasDivision of Cancer Epidemiology and Genetics, Clinical Genetics Branch, National Cancer Institute, 9609 Medical Center Drive 6E434, Bethesda, MD, 20892, USA.
Neelam GiriDivision of Cancer Epidemiology and Genetics, Clinical Genetics Branch, National Cancer Institute, 9609 Medical Center Drive 6E434, Bethesda, MD, 20892, USA.
Lisa J McReynoldsDivision of Cancer Epidemiology and Genetics, Clinical Genetics Branch, National Cancer Institute, 9609 Medical Center Drive 6E434, Bethesda, MD, 20892, USA. lisa.mcreynolds@nih.gov.
Edward W CowenDivision of Cancer Epidemiology and Genetics, Clinical Genetics Branch, National Cancer Institute, 9609 Medical Center Drive 6E434, Bethesda, MD, 20892, USA.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Fanconi anemia (FA) is an inherited bone marrow failure syndrome (IBMFS) characterized by pathogenic variants in the FA/BRCA DNA repair pathway genes. Individuals with FA have an elevated risk of developing myelodysplastic syndrome, acute myeloid leukemia, and solid tumors. Hematopoietic cell transplantation (HCT) is the most effective treatment for FA related bone marrow failure but can increase the risk of cancer development. Information on benign tumors and NMSC is lacking in patients with FA. Our objective was to characterize patients with FA enrolled in the National Cancer Institute IBMFS Study who have experienced non-melanoma skin cancers (NMSC) and/or benign tumors (BT). A total of 200 patients diagnosed with FA were enrolled in the Institutional Review Board approved study "Etiologic Investigation of Cancer Susceptibility in IBMFS: A Natural History Study" (NCT00027274). Through medical records review, we identified 30 patients with at least one NMSC, either squamous or basal cell carcinoma, or benign tumor. The remaining 170 patients comprised the control group. Out of 200 patients, 12 had NMSC, 25 had benign tumors, with an age range of 11-64 and 0-56 years, respectively. The median age at HCT was 30.5 years for NMSC patients, 9 years for benign tumor patients, and 9.1 years for controls. The most common genotype observed was FANCA, followed by FANCC and FANCI. Benign tumors spanned diverse anatomical locations. Early onset NMSC in patients with FA compared to the general population emphasizes the need for consistent monitoring in patients with FA, while the diverse anatomical locations of benign tumors underscore the importance of comprehensive surveillance for timely interventions in managing symptomatology and heightened cancer risk.

Indexed as

Basal Cell CarcinomaFanconi AnemiaSkin NeoplasmsAdolescentAdultCarcinoma, Squamous CellChildChild, PreschoolFemaleHumansInfantInfant, NewbornMaleMiddle AgedYoung AdultBasal cell carcinomaBenign tumorFanconi anemiaSkin cancerSquamous cell carcinoma

Identifiers

PMID38907138
PMCPMC11512875

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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.