Evidence map›Paper›PMID 42117003›Full record

ReviewCureus2026

Mosaicism in Marfan Syndrome: Clinical Implications and Variant Allele Frequency Interpretation in a Narrative Review.

Kota Arakawa, Hiroaki Ishida, Akiko Takashima, Takanobu Utsumi

Abstract readReview
In one paragraph

Review in Cureus, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing 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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

4 authors.

Kota ArakawaDepartment of Clinical Genetics Center, Toho University Medical Center Sakura Hospital, Sakura, JPN.
Hiroaki IshidaDepartment of Obstetrics and Gynecology, Toho University Medical Center Sakura Hospital, Sakura, JPN.
Akiko TakashimaDepartment of Obstetrics and Gynecology, Toho University Medical Center Sakura Hospital, Sakura, JPN.
Takanobu UtsumiDepartment of Clinical Genetics Center, Toho University Medical Center Sakura Hospital, Sakura, JPN.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Marfan syndrome (MFS) is an autosomal dominant disorder of the connective tissue caused by pathogenic variants in the FBN1 gene, characterized by cardiovascular, ocular, and skeletal involvement. Although some of these cases are considered sporadic, recent advances in next-generation sequencing (NGS) have revealed that certain cases might be attributable to parental mosaicism. Mosaicism, defined as the presence of genetically distinct cell populations arising from postzygotic mutations, retains important implications in diagnosis, phenotypic variability, recurrence risk, and genetic counseling. However, the interpretation of variant allele frequency (VAF) remains challenging and lacks standardization. This narrative review summarizes our current knowledge on mosaicism in MFS, with a focus on VAF interpretation and its clinical implications. We performed a literature search using PubMed, focusing on studies published between 2010 and 2025, including relevant original and review articles addressing mosaicism in MFS. Although VAF serves as a quantitative indicator of mosaicism, its interpretation is influenced by tissue distribution, mutation timing, and sequencing methodology. Peripheral blood VAF does not necessarily reflect variant burden in clinically relevant tissues, limiting its utility in predicting disease severity. Advances in high-sensitivity techniques, including NGS and digital droplet PCR, have improved the detection of low-level mosaicism, although multi-tissue analysis might be required for accurate diagnosis. Mosaicism is an underrecognized but clinically important factor in MFS, particularly in apparently sporadic cases or when genotype-phenotype discordance is observed. Failure to detect mosaic variants may result in underestimation of disease severity, inaccurate risk assessment, and suboptimal genetic counseling. Clinicians should therefore consider mosaicism in atypical presentations, utilize high-sensitivity sequencing methods when appropriate, and assess parental status to refine recurrence risk. However, interpretation of VAF remains challenging due to tissue variability and methodological differences, and standardized frameworks linking VAF to clinical outcomes are lacking.

Indexed as

fbn1 genegenetic counselingmarfan syndromemosaicismvariant allele frequency

Identifiers

PMID42117003
PMCPMC13157862

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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.