Evidence map›Paper›PMID 35758105›Full record

ArticleJournal of inherited metabolic disease2022

Eye movement disorders in inborn errors of metabolism: A quantitative analysis of 37 patients.

Lisette H Koens, Inge Tuitert, Hans Blokzijl, Marc Engelen, Femke C C Klouwer, Fiete Lange, Wilhelmina G Leen, Roelineke J Lunsing, Johannes H T M Koelman, Aad Verrips and 2 more

Open access · hybridAbstract read
In one paragraph

Article in Journal of inherited metabolic disease, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed, 1 pooled it
0.5field-weighted citation impact, top 36% 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

4 citing papers in PubMed, 1 synthesis or guideline pooled it, 5 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Review
  4. 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

12 authors at 3 institutions in 2 countries.

Lisette H KoensDepartment of Neurology and Clinical Neurophysiology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.
Inge TuitertDepartment of Neurology and Clinical Neurophysiology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.
Hans BlokzijlDepartment of Gastroenterology and Hepatology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.
Marc EngelenDepartment of Neurology and Clinical Neurophysiology, University of Amsterdam, Amsterdam University Medical Center, Amsterdam, The Netherlands.
Femke C C KlouwerDepartment of Neurology and Clinical Neurophysiology, University of Amsterdam, Amsterdam University Medical Center, Amsterdam, The Netherlands.
Fiete LangeDepartment of Neurology and Clinical Neurophysiology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.
Wilhelmina G LeenDepartment of Neurology and Clinical Neurophysiology, Canisius Wilhelmina Hospital, Nijmegen, The Netherlands.
Roelineke J LunsingDepartment of Neurology and Clinical Neurophysiology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.
Johannes H T M KoelmanDepartment of Neurology and Clinical Neurophysiology, University of Amsterdam, Amsterdam University Medical Center, Amsterdam, The Netherlands.
Aad VerripsDepartment of Neurology and Clinical Neurophysiology, Canisius Wilhelmina Hospital, Nijmegen, The Netherlands.
Tom J de KoningExpertise Center Movement Disorders Groningen, University Medical Center Groningen (UMCG), Groningen, The Netherlands.
Marina A J TijssenDepartment of Neurology and Clinical Neurophysiology, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.ORCID 0000-0001-5783-571X
University Medical Center Groningen · NLAmsterdam Neuroscience · NLCanisius-Wilhelmina Ziekenhuis · NL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Inborn errors of metabolism are genetic disorders that need to be recognized as early as possible because treatment may be available. In late-onset forms, core symptoms are movement disorders, psychiatric symptoms, and cognitive impairment. Eye movement disorders are considered to be frequent too, although specific knowledge is lacking. We describe and analyze eye movements in patients with an inborn error of metabolism, and see whether they can serve as an additional clue in the diagnosis of particularly late-onset inborn errors of metabolism. Demographics, disease characteristics, and treatment data were collected. All patients underwent a standardized videotaped neurological examination and a video-oculography. Videos are included. We included 37 patients with 15 different inborn errors of metabolism, including 18 patients with a late-onset form. With the exception of vertical supranuclear gaze palsy in Niemann-Pick type C and external ophthalmolplegia in Kearns-Sayre syndrome, no relation was found between the type of eye movement disorder and the underlying metabolic disorder. Movement disorders were present in 29 patients (78%), psychiatric symptoms in 14 (38%), and cognitive deficits in 26 patients (70%). In 87% of the patients with late-onset disease, eye movement disorders were combined with one or more of these core symptoms. To conclude, eye movement disorders are present in different types of inborn errors of metabolism, but are often not specific to the underlying disorder. However, the combination of eye movement disorders with movement disorders, psychiatric symptoms, or cognitive deficits can serve as a diagnostic clue for an underlying late-onset inborn error of metabolism.

Indexed as

Mental DisordersMetabolic DiseasesMetabolism, Inborn ErrorsMovement DisordersOcular Motility DisordersHumanseye movement disordersinborn errors of metabolismlate-onsetmovement disorders

Identifiers

PMID35758105
PMCPMC9541348
OpenAlexW4283588141

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.