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  • 1.
    Frölander, Hans Erik
    et al.
    Linköping University, Department of Behavioural Sciences and Learning. Linköping University, Faculty of Arts and Sciences. University of Örebro, Sweden; Örebro University Hospital, Sweden.
    Moller, Claes
    Linköping University, The Swedish Institute for Disability Research. University of Örebro, Sweden; Örebro University Hospital, Sweden; Örebro University Hospital, Sweden.
    Rudner, Mary
    Linköping University, Department of Behavioural Sciences and Learning, Disability Research. Linköping University, Faculty of Arts and Sciences. Linköping University, The Swedish Institute for Disability Research.
    Mishra, Sushmit
    Utkal University, India.
    Marshall, Jan D.
    Jackson Lab, ME 04609 USA; Alstrom Syndrome Int, ME USA.
    Piacentini, Heather
    Alstrom Syndrome Int, ME USA.
    Lyxell, Björn
    Linköping University, Department of Behavioural Sciences and Learning, Disability Research. Linköping University, Faculty of Arts and Sciences. Linköping University, The Swedish Institute for Disability Research.
    Theory-of-mind in individuals with Alstrom syndrome is related to executive functions, and verbal ability2015In: Frontiers in Psychology, ISSN 1664-1078, E-ISSN 1664-1078, Vol. 6, no 1426, p. 1-11Article in journal (Refereed)
    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 Alstrom 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 Happes 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.

  • 2.
    Frölander, Hans Erik
    et al.
    Linköping University, The Swedish Institute for Disability Research. Örebro University; Örebro University Hospital, Sweden.
    Möller, Claes
    Linköping University, The Swedish Institute for Disability Research. Örebro University; Örebro University Hospital, Sweden.
    Marshall, Jan D.
    Jackson laboratory, Bar Harbor, ME, USA.
    Sundqvist, Anett
    Linköping University, Department of Behavioural Sciences and Learning, Psychology. Linköping University, Faculty of Arts and Sciences. Linköping University, The Swedish Institute for Disability Research.
    Rönnåsen, Berit
    Linköping University, The Swedish Institute for Disability Research. Örebro University; Örebro University Hospital, Sweden.
    Falkensson, Lil
    National Resource Centre, Lund, Sweden.
    Lyxell, Björn
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning, Disability Research. Linköping University, Faculty of Arts and Sciences. Östergötlands Läns Landsting, Anaesthetics, Operations and Specialty Surgery Center, Department of Otorhinolaryngology in Linköping.
    Theory-of-mind in adolescents and young adults with Alström syndrome2014In: International Journal of Pediatric Otorhinolaryngology, ISSN 0165-5876, E-ISSN 1872-8464, Vol. 78, no 3, p. 530-536Article in journal (Refereed)
    Abstract [en]

    OBJECTIVE:

    The study focuses on theory-of-mind in adolescents and young adults with Alström syndrome (ALMS). ALMS, an autosomal recessive syndrome causes juvenile blindness, sensorineural hearing loss, cardiomyopathy, endocrinological disorders and metabolic dysfunction. Theory-of-mind (ToM) refers to the ability to impute mental states to one self and to others. Clinical observations have revealed an increased occurrence of deviances in mental state understanding in ALMS. In the present study ToM will be examined and related to working memory (WM), verbal ability and sensory loss.

    METHODS:

    Twelve young individuals (16-37 years) with ALMS and 24 nondisabled individuals matched on age, gender and educational level participated. ToM was assessed by means of a multiple task that taxes the ability to understand thoughts and feelings of story characters'. WM was examined by means of a reading span task and verbal ability by means of a vocabulary test.

    RESULTS:

    The ALMS group performed at significantly lower levels in ToM tasks and displayed a higher variability in performance than the control group. Individuals with ALMS and a relatively poor level performance provided fewer correct mental state inferences in ToM tasks than ALMS individuals with relatively higher performance levels. ALMS individuals with relatively high performance levels made as many correct inferences in ToM tasks as the control group, but their inferences were more often incomplete. Vocabulary skills and educational level, but not WM-capacity predicted ToM performance. Degree of deafblindness did not have an impact on ToM. Age of onset of visual loss but not hearing loss related to ToM.

    CONCLUSIONS:

    The individuals with ALMS display a high degree of heterogeneity in terms of ToM, where some individuals reached performance levels comparable to nondisabled individuals. The results are discussed with respect to how cognitive and verbal abilities and factors related to the disability affect ToM.

  • 3.
    Frölander, Hans-Erik
    et al.
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning. Linköping University, Faculty of Arts and Sciences.
    Rönnås, Berit
    Institutionen för hälsovetenskap och medicin, Örebro University.
    Möller, Claes
    Örebro Universitetssjukhus, audiologiskt forskningscentrum, Örebro Universitet.
    Lyxell, Björn
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning, Disability Research. Linköping University, Faculty of Arts and Sciences.
    Developmental, Cognitive and behavioral issues in Alströms syndrome2010Conference paper (Refereed)
  • 4.
    Frölander, Hans-Erik
    et al.
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning. Linköping University, Faculty of Arts and Sciences.
    Rönnås, Berit
    Institutionen för hälsovetenskap och medicin, Örebro University.
    Möller, Claes
    Örebro Universitetssjukhus, audiologiskt forskningscentrum, Örebro Universitet.
    Lyxell, Björn
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning, Disability Research. Linköping University, Faculty of Arts and Sciences.
    The Theory-of-Mind - perspective: How the psysical manifestations of ALMS may shape relationships and interactions with others2013Conference paper (Refereed)
  • 5.
    Frölander, Hans-Erik
    et al.
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning. Linköping University, Faculty of Arts and Sciences.
    Rönnås, Berit
    Institutionen för hälsovetenskap och medicin, Örebro University.
    Möller, Claes
    Örebro Universitetssjukhus, audiologiskt forskningscentrum, Örebro Universitet.
    Lyxell, Björn
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning. Linköping University, Faculty of Arts and Sciences.
    Theory-of-Mind in young adults with Alström - a deafblind syndrome2011Conference paper (Refereed)
  • 6.
    Henricson, Cecilia
    et al.
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning, Disability Research. Linköping University, Faculty of Arts and Sciences.
    Frölander, Hans Erik
    Linköping University, Department of Behavioural Sciences and Learning. Linköping University, Faculty of Arts and Sciences. Institutionen för hälsovetenskap och medicin, Örebro universitet.
    Möller, Claes
    Örebro Universitetssjukhus, audiologiskt forskningscentrum, Örebro Universitet.
    Lyxell, Björn
    Linköping University, The Swedish Institute for Disability Research. Linköping University, Department of Behavioural Sciences and Learning. Linköping University, Faculty of Arts and Sciences.
    Theory-of-mind and cognitive function in adults with Usher and Alström syndromes2016In: Journal of Visual Impairment & Blindness, ISSN 0145-482X, E-ISSN 1559-1476, Vol. 110, no 5, p. 349-366Article in journal (Refereed)
    Abstract [en]

    Objective: Theory-of-Mind (ToM) refers to the ability to impute mental states to one self and to others. ToM was investigated in adults with Usher syndrome type II (USH2) and Alström syndrome (AS) - two syndromes causing acquired deafblindness. The syndromes differ with regard to onset and degree of sensory loss. Individuals with AS in contrast to individuals with USH2 display a high incidence of additional physical diseases. Cognitive shortcomings are generally not observed in USH2 or in AS, but cognitive delay and a delay in receptive language have been reported in AS. The results were compared to adults with normal hearing and vision (NHV).

    Methods: Thirteen persons with USH2, 12 persons with AS, and 33 persons with NHV participated. All participants performed a test of working memory capacity and verbal ability. ToM was tested with Happe´s Strange Stories test, taxing the ability to understand the emotions and actions of story characters, comprising a mental condition. The test also include a section of matched stories, tapping verbal problem solving ability in a physical condition, and a set of tasks tapping the ability to recall verbal material.

    Results: There were no differences between the three groups in the ability to recall verbal material. Significant differences were however established on working memory, and on verbal problem solving in a physical condition, with higher results for the NHV group. The two groups with deafblindness also displayed poorer ToM performance than the NHV group, by producing fewer correct mental references. The two groups with deafblindness differed from each other also in the ability to produce mental inferences as such, where the USH group outperformed the AS group. Intra-group variability in this ability was also observed within the two syndromal groups. Differences were related to verbal ability, complex working memory capacity, visual status, and to a minor extent auditory capacity. The prevalence and severity of additional physical diseases in AS was not related to ToM performance.

    Conclusions: A limited access to information as a function of sensory loss could influence degree of experience of the physical world, but also of social situations and of communication, affecting ToM development negatively. Early loss of visual field and visual acuity was related to ToM performance in individuals with USH2 and AS. Access to information also requires processing skills promoted by effective cognitive skills. Working memory capacity was related to ToM in USH. This relation also points to the contribution of hearing in development of ToM. Differences between the two groups could be a function of genetic conditions, where the gene causing USH2 only affects the ear and the eye, while AS in addition has a multi-systemic pathology with varying onset and degree. Differences in ToM performance in the AS group could however not be directly attributed to health conditions.

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