Evidence map›Paper›PMID 38863011›Full record

ArticleOrphanet journal of rare diseases2024

Cognitive potential of children and adolescents with CHARGE syndrome and deafblindness.

Lynn Skei, Sigmund Skei, Timothy Hartshorne, Nils Inge Landrø

Abstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0citing papers in PubMed
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1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

4 authors.

Lynn SkeiSigno Resource Centre, Sandefjord, Norway. lynn.skei@signo.no.ORCID 0009-0000-0839-3935
Sigmund SkeiDepartment of Health and Care, County Governor Vestfold & Telemark, Tønsberg, Norway.ORCID 0009-0008-7354-4523
Timothy HartshorneDepartment of Psychology, Central Michigan University, Mt Pleasant, MI, USA.ORCID 0000-0002-3626-5268
Nils Inge LandrøDepartment of Psychology, University of Oslo, Oslo, Norway.ORCID 0000-0003-0354-5148

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundThe present study aimed to test the hypothesis stating that the cognitive potential of individuals with deafblindness is equal to those without a deafblind condition, an assumption that until now has been empirically unsubstantiated within the field of deafblindness.

methodsTo explore the assumption, 15 children and adolescents with CHARGE underwent cognitive assessment with WISC-V using a sequential two-level assessment design. The 1st level involved standardized test conditions. The 2nd level was designed as a continuation of the performances obtained from the 1st level and involved accommodations to compensate for sensory motor impairment. Statistical procedures involved the sample as a whole and when divided into two subgroups: (i) participants with CHARGE without deafblindness; (ii) participants with CHARGE and deafblindness using the 1st level scores as base line.

resultsAlthough results showed significantly lower scores in the deafblind subgroup with standardized procedures, they approximated the others after accommodating for their sensory deficits. This positive increase proved significant.

conclusionFindings supported the assumption of equal cognitive potential of individuals with and without deafblindness. Results indicated that the children and adolescents with deafblindness had most effect of the accommodations, enabling them to approximate the results of the subgroup without deafblindness. These gains were attributed enhanced accessibility endorsed by the accommodations and represented the participants latent cognitive dispositions only realized under certain circumstances.

Indexed as

CHARGE SyndromeCognitionDeaf-Blind DisordersAdolescentChildFemaleHumansMaleAtypical developmentCHARGECognitive assessmentCognitive potentialCongenital deafblindness

Identifiers

PMID38863011
PMCPMC11165843

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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.