Evidence map›Paper›PMID 34743752›Full record

ArticleOrphanet journal of rare diseases2021

Williams syndrome: on the role of intellectual abilities in anxiety.

Charlotte Willfors, Deborah M Riby, Marcus van der Poll, Katja Ekholm, Hanna Avdic Björlin, Johan Lundin Kleberg, Ann Nordgren

Open access · goldAbstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed
0.6field-weighted citation impact, top 35% 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

7 citing papers in PubMed, 9 citations in OpenAlex.

  1. Article
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  3. In-depth profile analysis and developmental trends in Wechsler performance among individuals with Williams syndrome.Child neuropsychology : a journal on normal and abnormal development in childhood and adolescence · 2026
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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

7 authors at 3 institutions in 3 countries.

Charlotte WillforsRare Diseases Research Group, Department of Molecular Medicine and Surgery, Karolinska Institutet, BioClinicum, J10:20, Visionsgatan 4, 171 64, Stockholm, Sweden. charlotte.willfors@ki.se.ORCID 0000-0001-8775-6537
Deborah M RibyDepartment of Psychology, Durham University, South Road, Durham, DH1 3LE, UK.
Marcus van der PollRare Diseases Research Group, Department of Molecular Medicine and Surgery, Karolinska Institutet, BioClinicum, J10:20, Visionsgatan 4, 171 64, Stockholm, Sweden.
Katja EkholmRare Diseases Research Group, Department of Molecular Medicine and Surgery, Karolinska Institutet, BioClinicum, J10:20, Visionsgatan 4, 171 64, Stockholm, Sweden.
Hanna Avdic BjörlinCentre for Psychiatry Research, Department of Clinical Neuroscience, Karolinska Institutet, Stockholm Health Care Services, Gävlegatan 22, 113 30, Stockholm, Sweden.
Johan Lundin Kleberg *Rare Diseases Research Group, Department of Molecular Medicine and Surgery, Karolinska Institutet, BioClinicum, J10:20, Visionsgatan 4, 171 64, Stockholm, Sweden.
Ann Nordgren *Rare Diseases Research Group, Department of Molecular Medicine and Surgery, Karolinska Institutet, BioClinicum, J10:20, Visionsgatan 4, 171 64, Stockholm, Sweden.
Karolinska Institutet · SEKarolinska University Hospital · SEDurham University · GB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundIndividuals with Williams syndrome (WS) have an elevated risk for anxiety disorders throughout the life span, making it a research priority to identify the individual factors associated with anxiety. Most of the existing literature is based on questionnaire data and suggests that impaired executive functions (EF) increase the risk for anxiety in WS. The aim of this study was to use direct measures by trained clinicians to investigate the effects of general intelligence, inhibition, sustained attention, and working memory on anxiety in WS, to further elucidate potential underlying mechanisms.

methodTwenty-four individuals with WS participated in the study (mean age: 29 years, range: 9-53 years), together with at least one of their parents. The MINI international neuropsychiatric interview for DSM-5 was completed to establish clinical diagnosis of anxiety, and the Clinical Global Impression Scale - Severity was used for an expert rating of symptom severity. Intellectual abilities were measured using the Wechsler scales, and attention and inhibition using the Conner's Continuous Performance Test. In addition, a parent-report questionnaire measuring EF, learning and memory was collected.

resultsIn contrast to the apriori hypothesis, there was no significant association between anxiety and core elements of EF such as working memory, sustained attention, and inhibition (i.e. the process of restraining one's impulses or behaviour). Using ordinal logistic regression analyses, we showed that decreasing intelligence quotient (IQ) and age are associated with elevated anxiety. We confirmed these results in between-groups analyses (anxiety disorder vs no current anxiety disorder), and low IQ was associated with higher risk of having an anxiety diagnosis. In addition, Bayesian statistics gave substantial evidence for no significant association between anxiety and inhibition.

conclusionBy using direct measures of psychological pathology and functioning, the current results provide a deeper characterisation of the WS phenotype and provide novel insights into the potential mechanisms underpinning anxiety.

Indexed as

Williams SyndromeAdultAnxietyAnxiety DisordersBayes TheoremHumansIntelligence

Identifiers

PMID34743752
PMCPMC8573929
OpenAlexW3214697869

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

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.