Evidence map›Paper›PMID 37501103›Full record

ArticleActa neuropathologica communications2023

Mitochondrial DNA mutations in Medulloblastoma.

Viktoria L E Funke, Sarah Sandmann, Viktoria Melcher, Jochen Seggewiss, Judit Horvath, Natalie Jäger, Marcel Kool, David T W Jones, Stefan M Pfister, Till Milde and 11 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in Acta neuropathologica communications, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.6field-weighted citation impact, top 30% of its field
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

2 citing papers in PubMed, 4 citations in OpenAlex.

  1. Review
  2. Review
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

21 authors at 4 institutions in 2 countries.

Viktoria L E FunkeDepartment of Pediatric Hematology and Oncology, University Children's Hospital Münster, Albert-Schweitzer-Campus 1, 48149, Münster, Germany.
Sarah SandmannInstitute of Medical Informatics, University of Münster, 48149, Münster, Germany.
Viktoria MelcherDepartment of Pediatric Hematology and Oncology, University Children's Hospital Münster, Albert-Schweitzer-Campus 1, 48149, Münster, Germany.
Jochen SeggewissInstitute of Human Genetics, University Hospital Münster, Münster, Germany.
Judit HorvathInstitute of Human Genetics, University Hospital Münster, Münster, Germany.
Natalie JägerHopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Marcel KoolHopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
David T W JonesHopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Stefan M PfisterHopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Till MildeHopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Stefan RutkowskiDepartment of Pediatric Hematology and Oncology, University Medical Center Hamburg-Eppendorf, 20251, Hamburg, Germany.
Martin MynarekDepartment of Pediatric Hematology and Oncology, University Medical Center Hamburg-Eppendorf, 20251, Hamburg, Germany.
Julian VargheseInstitute of Medical Informatics, University of Münster, 48149, Münster, Germany.
Ronald SträterDepartment of Pediatric Hematology and Oncology, University Children's Hospital Münster, Albert-Schweitzer-Campus 1, 48149, Münster, Germany.
Stephan RustDepartment of General Pediatrics, Metabolic Diseases, University Children's Hospital Münster, 48149, Münster, Germany.
Anja SeelhöferDepartment of General Pediatrics, Metabolic Diseases, University Children's Hospital Münster, 48149, Münster, Germany.
Janine ReunertDepartment of General Pediatrics, Metabolic Diseases, University Children's Hospital Münster, 48149, Münster, Germany.
Barbara FiedlerDepartment of Neuropediatrics, University Children's Hospital, Münster, Germany.
Ulrich SchüllerDepartment of Pediatric Hematology and Oncology, University Medical Center Hamburg-Eppendorf, 20251, Hamburg, Germany.
Thorsten MarquardtDepartment of General Pediatrics, Metabolic Diseases, University Children's Hospital Münster, 48149, Münster, Germany.
Kornelius KerlDepartment of Pediatric Hematology and Oncology, University Children's Hospital Münster, Albert-Schweitzer-Campus 1, 48149, Münster, Germany. kornelius.kerl@ukmuenster.de.ORCID 0000-0002-5676-8102
University Hospital Münster · DEGerman Cancer Research Center · DEUniversität Hamburg · DEUniversity of Münster · DE

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

To date, several studies on genomic events underlying medulloblastoma (MB) biology have expanded our understanding of this tumour entity and led to its division into four groups-WNT, SHH, group 3 (G3) and group 4 (G4). However, there is little information about the relevance of pathogenic mitochondrial DNA (mtDNA) mutations and their consequences across these. In this report, we describe the case of a female patient with MB and a mitochondriopathy, followed by a study of mtDNA variants in MB groups. After being diagnosed with G4 MB, the index patient was treated in line with the HIT 2000 protocol with no indications of relapse after five years. Long-term side effects of treatment were complemented by additional neurological symptoms and elevated lactate levels ten years later, resulting in suspected mitochondrial disease. This was confirmed by identifying a mutation in the MT-TS1 gene which appeared homoplasmic in patient tissue and heteroplasmic in the patient's mother. Motivated by this case, we explored mtDNA mutations across 444 patients from ICGC and HIT cohorts. While there was no statistically significant enrichment of mutations in one MB group, both cohorts encompassed a small group of patients harbouring potentially deleterious mtDNA variants. The case presented here highlights the possible similarities between sequelae caused by MB treatment and neurological symptoms of mitochondrial dysfunction, which may apply to patients across all MB groups. In the context of the current advances in characterising and interpreting mtDNA aberrations, recognising affected patients could enhance our future knowledge regarding the mutations' impact on carcinogenesis and cancer treatment.

Indexed as

Cerebellar NeoplasmsMedulloblastomaMitochondrial DiseasesDNA, MitochondrialFemaleHumansMutationDNA, MitochondrialDNA, MitochondrialDNA mutational analysisMedulloblastomaMitochondrial diseases

Identifiers

PMID37501103
PMCPMC10373251
OpenAlexW4385330674

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

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.