Evidence map›Paper›PMID 38839988›Full record

ArticleEuropean journal of human genetics : EJHG2024

Quality assurance for next-generation sequencing diagnostics of rare neurological diseases in the European Reference Network.

Aleš Maver, Katja Lohmann, Fran Borovečki, Nicola Wolstenholme, Rachel L Taylor, Malte Spielmann, Tobias B Haack, Matthias Gerberding, Borut Peterlin, Holm Graessner

Abstract read
In one paragraph

Article in European journal of human genetics : EJHG, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing 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

4 citing papers in PubMed.

  1. Article
  2. Review
  3. Review
  4. Summer reading in EJHG.European journal of human genetics : EJHG · 2024
    Article
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

10 authors.

Aleš MaverClinical Institute of Genomic Medicine, University Medical Centre Ljubljana, Ljubljana, Slovenia.
Katja LohmannInstitute of Neurogenetics, University of Luebeck, Luebeck, Germany.
Fran BorovečkiDepartment of Neurology, University Hospital Centre Zagreb, Zagreb, Croatia.
Nicola WolstenholmeEMQN CIC, Unit 4 Enterprise Hse, Manchester Science Park, Manchester, UK.ORCID 0000-0002-9366-8549
Rachel L TaylorEMQN CIC, Unit 4 Enterprise Hse, Manchester Science Park, Manchester, UK.
Malte SpielmannInstitute of Human Genetics, University of Lübeck, Lübeck, Germany.ORCID 0000-0002-0583-4683
Tobias B HaackInstitute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.ORCID 0000-0001-6033-4836
Matthias GerberdingInstitute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
Borut PeterlinClinical Institute of Genomic Medicine, University Medical Centre Ljubljana, Ljubljana, Slovenia.
Holm GraessnerInstitute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany. Holm.Graessner@med.uni-tuebingen.de.ORCID 0000-0001-9803-7183

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

In the past decade, next-generation sequencing (NGS) has revolutionised genetic diagnostics for rare neurological disorders (RND). However, the lack of standardised technical, interpretative, and reporting standards poses a challenge for ensuring consistent and high-quality diagnostics globally. To address this, the European Reference Network for Rare Neurological Diseases (ERN-RND) collaborated with the European Molecular Genetics Quality Network (EMQN) to establish an external quality assessment scheme for NGS-based diagnostics in RNDs. The scheme, initiated in 2021 with a pilot involving 29 labs and followed by a second round in 2022 with 42 labs, aimed to evaluate the performance of laboratories in genetic testing for RNDs. Each participating lab analysed genetic data from three hypothetical cases, assessing genotyping, interpretation, and clerical accuracy. Despite a majority of labs using exome or genome sequencing, there was considerable variability in gene content, sequencing quality, adherence to standards, and clinical guidance provision. Results showed that while most labs provided correct molecular diagnoses, there was significant variability in reporting technical quality, adherence to interpretation standards, reporting strategies, and clinical commentary. Notably, some labs returned results with the potential for adverse medical outcomes. This underscores the need for further harmonisation, guideline development, and external quality assessment in the evolving landscape of genomic diagnostics for RNDs. Overall, the experience with the scheme highlighted the generally good quality of participating labs but emphasised the imperative for ongoing improvement in data analysis, interpretation, and reporting to enhance patient safety.

Indexed as

Genetic TestingNervous System DiseasesRare DiseasesEuropeHigh-Throughput Nucleotide SequencingHumansQuality Assurance, Health Care

Identifiers

PMID38839988
PMCPMC11292006

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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.