Evidence map›Paper›PMID 42625355›Full record

ArticleJournal of inherited metabolic disease2026

An Optimized Diagnostic Approach for Adults With Suspected Inherited Metabolic Disorders.

Machteld M Oud, Elise A Ferreira, Clara D M van Karnebeek, Robin Wijngaard, Nicole I Wolf, Marc Engelen, Mareen Datema, Sacha Ferdinandusse, André B P van Kuilenburg, Laura Steinbusch and 3 more

Abstract read
In one paragraph

Article in Journal of inherited metabolic disease, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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

1 citing paper in PubMed.

  1. 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

13 authors.

Machteld M OudDepartment of Human Genetics, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, the Netherlands.ORCID https://orcid.org/0000-0003-4864-9346
Elise A FerreiraUnited for Metabolic Diseases, the Netherlands.ORCID https://orcid.org/0000-0001-9397-942X
Clara D M van KarnebeekUnited for Metabolic Diseases, the Netherlands.
Robin WijngaardDepartment of Human Genetics, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, the Netherlands.
Nicole I WolfAmsterdam Leukodystrophy Center, Department of Child Neurology, Emma Children's Hospital and Amsterdam Neuroscience, Amsterdam University Medical Center, Vrije Universiteit Amsterdam, Amsterdam, the Netherlands.ORCID https://orcid.org/0000-0003-1721-0728
Marc EngelenDepartment of Neurology and Pediatric Neurology, Emma Children's Hospital, Amsterdam Leukodystrophy Center, Amsterdam University Medical Center, Amsterdam, the Netherlands.
Mareen DatemaDepartment of Endocrinology and Metabolism, Amsterdam UMC, University of Amsterdam, Amsterdam, the Netherlands.
Sacha FerdinandusseDepartment of Laboratory Medicine, Laboratory Genetic Metabolic Diseases, Amsterdam UMC Location University of Amsterdam, Amsterdam Gastroenterology Endocrinology Metabolism, Amsterdam, the Netherlands.ORCID https://orcid.org/0000-0002-7738-5709
André B P van KuilenburgDepartment of Genetic Metabolic Diseases, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam Gastroenterology Endocrinology Metabolism, Amsterdam, the Netherlands.ORCID https://orcid.org/0000-0002-7989-4910
Laura SteinbuschDepartment of Clinical Genetics and Genome Services Maastricht, Maastricht University Medical Centre+, Maastricht, the Netherlands.ORCID https://orcid.org/0000-0001-7076-8662
Saskia N van der CrabbenUnited for Metabolic Diseases, the Netherlands.ORCID https://orcid.org/0000-0001-8619-1511
Mirjam LangeveldUnited for Metabolic Diseases, the Netherlands.ORCID https://orcid.org/0000-0002-9934-6831
ZOEMBA Study Group

Funding

Stichting Metakids UMD-ZOE-2022-012
6 · The paper itself

Abstract

Inherited metabolic disorders (IMDs) arise from defects in metabolic pathways essential for normal biochemical function. Although the pathogenic genetic variants are present at birth, clinical manifestations may not emerge until adolescence or adulthood. Symptoms may be more subtle compared to those of childhood onset IMDs and may have overlap with signs and symptoms of acquired disorders. This makes diagnosing IMDs in adulthood challenging. To identify how routine diagnostic care for adults suspected of having an IMD could be improved, we conducted a reanalysis study in a cohort of 31 adult participants. The participants, who remained undiagnosed after standard diagnostic care, were seen by a clinical geneticist, adult metabolic specialist and neurologist for deep phenotyping and data collection. Subsequently, exome sequencing data were reanalyzed and possible disease-causing variants were prioritized for further evaluation by a multidisciplinary team including both clinicians and geneticists (biochemical and molecular). This approach led to a (partial) diagnosis for nine participants from six families, giving a 25% diagnostic yield. We could distinguish four possible reasons why the identified disease-associated variants were not detected during routine diagnostic care. Beyond the expected impact of technical advancements and emerging knowledge of disease genes, we found that the most impactful factor was of organizational nature. Our findings underscore the value of reviewing all strong genetic variants within a multidisciplinary team setting, facilitating a bidirectional exchange of expertise. This approach enhances diagnostic accuracy, particularly when the underlying cause of the disease is not immediately apparent.

Indexed as

Metabolic DiseasesMetabolism, Inborn ErrorsAdultCohort StudiesExome SequencingFemaleGenetic TestingHumansMaleMiddle AgedPhenotypeYoung Adultadultsdiagnosticsgeneticsinherited metabolic disorders

Identifiers

PMID42625355
PMCPMC13494450

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.