Evidence map›Paper›PMID 41542348›Full record

ArticleGenetics in medicine open2025

Clinical and genetic spectrum of Fanconi anemia in Australia and New Zealand.

Hannah Fluhler, Elissah Granger, Michael Sharp, Caitlin Harris, Mark McKinley, Sarbjit Riyat, Christine Krieg, Andrew Deans, Chris Fraser, Siobhan Cross and 13 more

Abstract read
In one paragraph

Article in Genetics in medicine open, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0cells of the map it votes in
0citing papers in PubMed
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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

23 authors.

Hannah FluhlerThe DNA Repair and Recombination Laboratory, St Vincent's Institute of Medical Research, Melbourne, Australia.
Elissah GrangerThe DNA Repair and Recombination Laboratory, St Vincent's Institute of Medical Research, Melbourne, Australia.
Michael SharpThe DNA Repair and Recombination Laboratory, St Vincent's Institute of Medical Research, Melbourne, Australia.
Caitlin HarrisThe DNA Repair and Recombination Laboratory, St Vincent's Institute of Medical Research, Melbourne, Australia.
Mark McKinleyPathology Queensland Central Cytogenetics Laboratory, Brisbane, Queensland, Australia.
Sarbjit RiyatPathology Queensland Central Cytogenetics Laboratory, Brisbane, Queensland, Australia.
Christine KriegDeutsche Fanconi-Anämie-Hilfe e.V., Eschau, Germany.
Andrew DeansThe Faculty of Medicine, Dentistry and Health Science, The University of Melbourne, Parkville, Victoria, Australia.
Chris FraserBlood and Bone Marrow Transplant Program, Queensland Children's Hospital, Brisbane, Queensland, Australia.
Siobhan CrossChildren's Haematology Oncology Centre, Christchurch Hospital, Christchurch, New Zealand.
Tina CarterPerth Children's Hospital, PathWest Haematology and University of WA, Nedlands, WA, Australia.
Lisa WorganClinical Genetics Service, Royal Prince Alfred Hospital, Camperdown NSW Australia.
Adam NelsonSydney Children's Hospital Network, Sydney, NSW, Australia.
Lucy C FoxClinical Haematology Department, Peter MacCallum Cancer Centre and Royal Melbourne Hospital, Melbourne, Australia.
Jillian NichollCytogenomics, Department of Genetics and Molecular Pathology, SA Pathology, Adelaide, South Australia, Australia.
Alison AttwoodCytogenomics, Department of Genetics and Molecular Pathology, SA Pathology, Adelaide, South Australia, Australia.
Lorna McLemanGenome Stability Unit, St Vincent's Institute of Medical Research, Melbourne, Australia.
David HughesRoyal Children's Hospital, Melbourne, Victoria, Australia.
Rachel ConyersRoyal Children's Hospital, Melbourne, Victoria, Australia.
Omar KujanUWA Dental School, The University of Western Australia, Nedlands, WA, Australia.
Eunike Velleuer-CarlbergCenter for Child and Adolescent Health, Germany.
Adayapalam NandiniPathology Queensland Central Cytogenetics Laboratory, Brisbane, Queensland, Australia.
Wayne CrismaniThe DNA Repair and Recombination Laboratory, St Vincent's Institute of Medical Research, Melbourne, Australia.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Purpose: Fanconi anemia (FA) is a rare genetic condition that predisposes to progressive bone marrow failure, a specific spectrum of malignancies, including head and neck squamous cell carcinoma, and an array of other clinical manifestations. The clinical and genetic spectrum of FA in Australia and New Zealand remains relatively undescribed in the literature and is limited to case reports. Methods: In this study, we conducted a comprehensive investigation of FA within this region combining cohort data and case reports, aiming to elucidate its diagnostic patterns, clinical manifestations, and genetic characteristics. Results: Our findings reveal a positive correlation between national testing rates and case detection, across states and territories of Australia, suggesting that targeted testing strategies may enhance the identification of FA cases. Furthermore, our analysis demonstrates that the physical and genetic profiles of people with FA in Australia and New Zealand resemble those observed in other international cohort studies but also exhibit nuanced differences. Conclusion: This study emphasizes the continued significance of cytogenetic testing for FA while stressing the need for heightened awareness among medical professionals. FA, once perceived primarily as a pediatric condition, now demands vigilance across all age groups because of advances in medical care and complementary detection methods, such as high-throughput sequencing. This study also underscores the heightened susceptibility of individuals with FA in Australia and New Zealand to head and neck squamous cell carcinoma, consistent with observations in other regions worldwide.

Indexed as

Chromosome breakageDNA repairFanconi anemiaGenetic testingGenomic instability

Identifiers

PMID41542348
PMCPMC12800855

What OpenQuestion holds

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LicenceCC BY-NC-ND
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Registered trials

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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.