Evidence map›Paper›PMID 37330543›Full record

ArticleHuman genomics2023

Comprehensive genetic screening of early-onset dementia patients in an Austrian cohort-suggesting new disease-contributing genes.

Sara Silvaieh, Theresa König, Raphael Wurm, Tandis Parvizi, Evelyn Berger-Sieczkowski, Stella Goeschl, Christoph Hotzy, Matias Wagner, Riccardo Berutti, Esther Sammler and 2 more

Erratum issuedOpen access · goldAbstract read
In one paragraph

Article in Human genomics, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
1.1field-weighted citation impact, top 24% 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, 6 citations in OpenAlex.

  1. Characterization of a novel inherited splice-site variant inBiochemistry and biophysics reports · 2026
    Article
  2. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

12 authors at 3 institutions in 3 countries.

Sara Silvaieh *Department of Neurology, Medical University of Vienna, Vienna, Austria.
Theresa König *Department of Neurology, Medical University of Vienna, Vienna, Austria.
Raphael WurmDepartment of Neurology, Medical University of Vienna, Vienna, Austria.
Tandis ParviziDepartment of Neurology, Medical University of Vienna, Vienna, Austria.
Evelyn Berger-SieczkowskiDepartment of Neurology, Medical University of Vienna, Vienna, Austria.
Stella GoeschlDepartment of Neurology, Medical University of Vienna, Vienna, Austria.
Christoph HotzyDepartment of Neurology, Medical University of Vienna, Vienna, Austria.
Matias WagnerInstitute of Human Genetics, School of Medicine, Technical University of Munich, Munich, Germany.
Riccardo BeruttiInstitute of Human Genetics, School of Medicine, Technical University of Munich, Munich, Germany.
Esther SammlerMolecular and Clinical Medicine, Ninewells Hospital and Medical School, University of Dundee, Dundee, DD1 9SY, UK.
Elisabeth StögmannDepartment of Neurology, Medical University of Vienna, Vienna, Austria. elisabeth.stoegmann@meduniwien.ac.at.
Alexander ZimprichDepartment of Neurology, Medical University of Vienna, Vienna, Austria.
Medical University of Vienna · ATTechnical University of Munich · DEMRC Protein Phosphorylation and Ubiquitylation Unit · GB

Funding

Austrian Alzheimer Society ÖAG 2019-1
6 · The paper itself

Abstract

Early-onset dementia (EOD), with symptom onset before age 65, has a strong genetic burden. Due to genetic and clinical overlaps between different types of dementia, whole-exome sequencing (WES) has emerged as an appropriate screening method for diagnostic testing and novel gene-finding approaches. We performed WES and C9orf72 repeat testing in 60 well-defined Austrian EOD patients. Seven patients (12%) carried likely disease-causing variants in monogenic genes, PSEN1, MAPT, APP, and GRN. Five patients (8%) were APOE4 homozygote carriers. Definite and possible risk variants were detected in the genes TREM2, SORL1, ABCA7 and TBK1. In an explorative approach, we cross-checked rare gene variants in our cohort with a curated neurodegeneration candidate gene list and identified DCTN1, MAPK8IP3, LRRK2, VPS13C and BACE1 as promising candidate genes. Conclusively, 12 cases (20%) carried variants relevant to patient counseling, comparable to previously reported studies, and can thus be considered genetically resolved. Reduced penetrance, oligogenic inheritance and not yet identified high-risk genes might explain the high number of unresolved cases. To address this issue, we provide complete genetic and phenotypic information (uploaded to the European Genome-phenome Archive), enabling other researchers to cross-check variants. Thereby, we hope to increase the chance of independently finding the same gene/variant-hit in other well-defined EOD patient cohorts, thus confirming new genetic risk variants or variant combinations.

Indexed as

Alzheimer DiseaseAgedAmyloid Precursor Protein SecretasesAspartic Acid EndopeptidasesAustriaGenetic Predisposition to DiseaseGenetic TestingHumansLDL-Receptor Related ProteinsMembrane Transport ProteinsMutationAmyloid Precursor Protein SecretasesAspartic Acid EndopeptidasesLDL-Receptor Related ProteinsMembrane Transport ProteinsSORL1 protein, humanEarly-onset dementiaGenetic variantsWhole-exome sequencing

Identifiers

PMID37330543
PMCPMC10276391
OpenAlexW4381052406

What OpenQuestion holds

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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.