oru.sePublikationer
Change search
CiteExportLink to record
Permanent link

Direct link
Cite
Citation style
  • apa
  • harvard1
  • ieee
  • modern-language-association-8th-edition
  • vancouver
  • Other style
More styles
Language
  • de-DE
  • en-GB
  • en-US
  • fi-FI
  • nn-NO
  • nn-NB
  • sv-SE
  • Other locale
More languages
Output format
  • html
  • text
  • asciidoc
  • rtf
Theory-of-mind in individuals with Alström syndrome is related to executive functions, and verbal ability
Örebro University, School of Health and Medical Sciences, Örebro University, Sweden. Audiological Research Centre, Örebro University Hospital, Örebro, Sweden; Swedish Institute for Disability Research, Linköping, Sweden; Linnaeus Centre HEAD, Linköping, Sweden; Research on Hearing and Deafness (HEAD) graduate School, Linköping, Sweden. (Dövblindforskargruppen)ORCID iD: 0000-0003-2172-9792
Örebro University, School of Health and Medical Sciences, Örebro University, Sweden. Orebro University Hospital. Audiological Research Centre, Örebro; Swedish Institute for Disability Research, Linköping, Sweden; Linnaeus Centre HEAD, Linköping, Sweden; Department of Audiology, Örebro University Hospital, Örebro, Sweden.ORCID iD: 0000-0001-6557-6359
Swedish Institute for Disability Research, Linköping, Sweden; Linnaeus Centre HEAD, Linköping, Sweden; Department of Behavioral Sciences and Learning, Linköping University, Linköping, Sweden.
Institute of Health Sciences, Utkal University, Bhubaneswar, India.
Show others and affiliations
2015 (English)In: Frontiers in Psychology, ISSN 1664-1078, E-ISSN 1664-1078, Vol. 6, 1426Article in journal (Refereed) Published
Abstract [en]

Objective: This study focuses on cognitive prerequisites for the development of theory-of-mind (ToM), the ability to impute mental states to self and others in young adults with Alström syndrome (AS). AS is a rare and quite recently described recessively inherited ciliopathic disorder which causes progressive sensorineural hearing loss and juvenile blindness, as well as many other organ dysfunctions. Two cognitive abilities were considered; Phonological working memory (WM) and executive functions (EF), both of importance in speech development.                                                                                              

Methods: Ten individuals (18–37 years) diagnosed with AS, and 20 individuals with no known impairment matched for age, gender, and educational level participated. Sensory functions were measured. Information about motor functions and communicative skills was obtained from responses to a questionnaire. ToM was assessed using Happés strange stories, verbal ability by a vocabulary test, phonological WM by means of an auditory presented non-word serial recall task and EF by tests of updating and inhibition.                                           

Results: The AS group performed at a significantly lower level than the control group in both the ToM task and the EF tasks. A significant correlation was observed between recall of non-words and EF in the AS group. Updating, but not inhibition, correlated significantly with verbal ability, whereas both updating and inhibition were significantly related to the ability to initiate and sustain communication. Poorer performance in the ToM and EF tasks were related to language perseverance and motor mannerisms.                                                     

Conclusion: The AS group displayed a delayed ToM as well as reduced phonological WM, EF, and verbal ability. A significant association between ToM and EF, suggests a compensatory role of EF. This association may reflect the importance of EF to perceive and process input from the social environment when the social interaction is challenged by dual sensory loss. We argue that limitations in EF capacity in individuals with AS, to some extent, may be related to early blindness and progressive hearing loss, but maybe also to gene specific abnormalities.

Place, publisher, year, edition, pages
2015. Vol. 6, 1426
Keyword [en]
Alström syndrome (AS), ciliopathy, deafblindness, theory-of-mind, verbal ability, executive functions
National Category
Psychology (excluding Applied Psychology)
Research subject
Psychology
Identifiers
URN: urn:nbn:se:oru:diva-46002DOI: 10.3389/fpsyg.2015.01426ISI: 000361813000001PubMedID: 26441796OAI: oai:DiVA.org:oru-46002DiVA: diva2:858637
Funder
NIH (National Institute of Health), HDO36878
Note

Funding Agency:

Linnaeus Centre HEAD

Available from: 2015-10-02 Created: 2015-10-02 Last updated: 2017-03-15Bibliographically approved
In thesis
1. Deafblindness: Theory-of-mind, cognitive functioning and social network in Alström syndrome
Open this publication in new window or tab >>Deafblindness: Theory-of-mind, cognitive functioning and social network in Alström syndrome
2016 (English)Doctoral thesis, comprehensive summary (Other academic)
Abstract [en]

This thesis addresses young adults with Alström syndrome (AS). AS causes acquired deafblindness, a severe, progressive, combined auditory and visual impairment affecting daily life and self-reliance to a degree that full participation depends on help from others and society. AS is an autosomal, recessively inherited single-gene disorder that affects the ALMS1 gene. AS has a multi-systemic pathology including a high incidence of additional multiple endocrine abnormalities, cardiomyopathy, pulmonary fibrosis, restrictive lung disease and progressive hepatic and renal failure leading to reduced life expectancy. The focus in the present thesis is on the development of Theory-of-mind (ToM) and on how ToM relates to the development of certain cognitive skills and the characteristics of the individual social network. ToM refers to the ability to understand the thoughts and feelings of others.

The results reveal that individuals with AS displayed a significantly higher degree of heterogeneity in the performance of ToM tasks, and some individuals with AS performed on an equal level with nondisabled individuals. ToM performance was predicted by verbal ability and executive functioning (EF), whereas working memory capacity (WM) proved to be an indirect predictor. Later onset of visual loss further characterized AS individuals with better ToM. The sizes of the social networks of individuals with AS were smaller relative to those of nondisabled individuals, and many of the acquaintances were professionals working with individuals with AS. The number of friends correlated with ToM performance.

Methods to improve verbal ability and EF, and interventions to enhance social participation in childhood of individuals with AS might prove to be fruitful. In addition assistive technology to establish and maintain friendships in adulthood is required.

Place, publisher, year, edition, pages
Örebro: Örebro university, 2016. 62 p.
Series
Studies from The Swedish Institute for Disability Research, ISSN 1650-1128 ; 78
Keyword
Alström syndrome, Deafblindness, Theory-of-mind, Communication, Verbal ability, Executive functions, Working memory, Health problems, Friendship, Social network
National Category
Other Health Sciences
Research subject
Disability Research
Identifiers
urn:nbn:se:oru:diva-48930 (URN)978-91-7529-132-1 (ISBN)
Public defence
2016-04-22, Långhuset, Hörsal 2, Örebro universitet, Fakultetsgatan 1, Örebro, 09:00 (Swedish)
Opponent
Supervisors
Available from: 2016-03-03 Created: 2016-03-03 Last updated: 2016-12-16Bibliographically approved

Open Access in DiVA

No full text

Other links

Publisher's full textPubMed

Search in DiVA

By author/editor
Frölander, Hans-ErikMöller, Claes
By organisation
School of Health and Medical Sciences, Örebro University, SwedenOrebro University Hospital
In the same journal
Frontiers in Psychology
Psychology (excluding Applied Psychology)

Search outside of DiVA

GoogleGoogle Scholar

Altmetric score

Total: 252 hits
CiteExportLink to record
Permanent link

Direct link
Cite
Citation style
  • apa
  • harvard1
  • ieee
  • modern-language-association-8th-edition
  • vancouver
  • Other style
More styles
Language
  • de-DE
  • en-GB
  • en-US
  • fi-FI
  • nn-NO
  • nn-NB
  • sv-SE
  • Other locale
More languages
Output format
  • html
  • text
  • asciidoc
  • rtf