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Open Access 01-06-2011 | Article

Is theory of mind related to social dysfunction and emotional problems in 22q11.2 deletion syndrome (velo-cardio-facial syndrome)?

Authors: Linda E. Campbell, Angela F. Stevens, Kathryn McCabe, Lynne Cruickshank, Robin G. Morris, Declan G. M. Murphy, Kieran C. Murphy

Published in: Journal of Neurodevelopmental Disorders | Issue 2/2011

Abstract

Social dysfunction is intrinsically involved in severe psychiatric disorders such as depression and psychosis and linked with poor theory of mind. Children with 22q11.2 deletion syndrome (22q11DS, or velo-cardio-facial syndrome) have poor social competence and are also at a particularly high risk of developing mood (40%) and psychotic (up to 30%) disorders in adolescence and young adulthood. However, it is unknown if these problems are associated with theory of mind skills, including underlying social-cognitive and social-perceptual mechanisms. The present cross-sectional study included classic social-cognitive false-belief and mentalising tasks and social-perceptual face processing tasks. The performance of 50 children with 22q11DS was compared with 31 age-matched typically developing sibling controls. Key findings indicated that, while younger children with 22q11DS showed impaired acquisition of social-cognitive skills, older children with 22q11DS were not significantly impaired compared with sibling controls. However, children with 22q11DS were found to have social-perceptual deficits, as demonstrated by difficulties in matching faces on the basis of identity, emotion, facial speech and gaze compared with sibling controls. Furthermore, performance on the tasks was associated with age, language ability and parentally rated social competence and emotional problems. These results are discussed in relation to the importance of a better delineation of social competence in this population.

Andersson F, Glaser B, Spiridon M, Debbané M, Vuilleumier P, Eliez S. Impaired activation of face processing networks revealed by functional magnetic resonance imaging in 22q11.2 deletion syndrome. Biol Psychiatry. 2008;63(1):49–57.CrossRefPubMed

Baron-Cohen S, Leslie A, Frith U. Does the autistic child have a “theory of mind”? Cognition. 1985;21(1):37–46.CrossRefPubMed

Bassett A, Caluseriu O, Weksberg R, Young D, Chow E. Catechol-o-methyl transferase and expression of schizophrenia in 73 adults with 22q11 deletion syndrome. Biol Psychiatry. 2007;61:1135–40.CrossRefPubMedCentralPubMed

Bishop DVM. The test for reception of grammar (TROG). 2nd edition. Published by the author and available from Age and Cognitive Performance Research Centre, University of Manchester, M13 9PL, UK; 1983.

Bruce V, Young A. Understanding face recognition. Br J Psychol. 1986;77:305–27.CrossRefPubMed

Bruce V, Campbell RN, Doherty-Sneddon G, Import A, Langton S, McAuley S, et al. Testing face processing skills in children. Br J Dev Psychol. 2000;18:319–33.CrossRef

Campbell L. Brain, behaviour and cognition in 22q11.2 deletion syndrome (22qDS). London: University of London, King’s College London; 2006.

Campbell L, Daly E, Toal F, Stevens A, Azuma R, Catani M, et al. Brain and behaviour in children with 22q11.2 deletion syndrome: a volumetric and voxel-based morphometry MRI study. Brain. 2006;129(5):1218–28.CrossRefPubMed

Campbell L, Stevens A, Daly E, Toal F, Azuma R, Karmiloff-Smith A, et al. A comparative study of cognition and brain anatomy between two neurodevelopmental disorders: 22q11.2 deletion syndrome and Williams syndrome. Neuropsychologia. 2009;47(4):1034–44.CrossRefPubMed

Campbell L, McCabe K, Leadbeater K, Schall U, Loughland C, Rich D. Visual scanning of faces in 22q11.2 deletion syndrome: attention to the mouth or the eyes? Psychiatry Res. 2010a;177(1–2):211–5.CrossRefPubMed

Campbell L, McCabe K, Leadbeater K, Schall U, Loughland C, Rich D. Visual scanning of faces in 22q11.2 deletion syndrome: Attention to the mouth or the eyes? Psychiatry Research. 2010;177(1–2):211–5. doi:10.1016/j.psychres.2009.06.007.

Castelli F, Frith C, Happe F, Frith U. Autism, Asperger syndrome and brain mechanisms for the attribution of mental states to animated shapes. Brain. 2002;125:1839–49.CrossRefPubMed

Chow E, Watson M, Young D, Bassett A. Neurocognitive profile in 22q11 deletion syndrome and schizophrenia. Schizophr Res. 2006;87:270–8.CrossRefPubMedCentralPubMed

Dunn LM, Whetton C, Burley J. The British picture vocabulary scale (2nd edition). NFER-Nelson Publishing company Ltd, UK; 1982.

Fine S, Weissman A, Gerdes M, Pinto-Martin J, Zackai E, McDonald-McGinn D, et al. Autism spectrum disorders and symptoms in children with molecularly confirmed 22q11.2 deletion syndrome. J Autism Dev Disord. 2005;35(4):461–70.CrossRefPubMedCentralPubMed

Golding-Kushner K, Weller G, Shprintzen R. Velo-cardio-facial syndrome: language and psychological profiles. J Craniofac Genet Dev Biol. 1985;5(3):259–66.PubMed

Goodman R, Ford T, Simmons H, Gatward R, Meltzer H. Using the strengths and difficulties questionnaire (SDQ) to screen for child psychiatric disorders in a community sample. Br J Psychiatry. 2000;177:534–9.CrossRefPubMed

Gopnik A, Astington J. Children’s understanding of representational change and its relation to the understanding of false belief and the appearance reality distinction. Child Dev. 1988;59:26–37.CrossRefPubMed

Gothelf D, Presburger G, Levy D, Nahmani A, Burg M, Berant M, et al. Genetic, developmental, and physical factors associated with attention deficit hyperactivity disorder in patients with velocardiofacial syndrome. Am J Med Genet B Neuropsychiatr Genet. 2004;126B(1):116–21.CrossRefPubMed

Happé F. Autism, an introduction to psychological theory. London: UCL Press Ltd; 1994.

Haxby J, Hoffman E, Gobbini I. The distributed human neural system for face perception. Trends Cogn Sci. 2000;4:223–33.CrossRefPubMed

Heineman-de Boer J, Van Haelst M, Cordia-de Haan M, Beemer F. Behavior problems and personality aspects of 40 children with velo-cardio-facial syndrome. Genet Couns. 1999;1:1089–93.

Henry J, van Amelsvoort T, Morris R, Owen M, Murphy D, Murphy K. An investigation of the neuropsychological profile in adults with velo-cardio-facial syndrome (VCFS). Neuropsychologia. 2002;40(5):471–8.CrossRefPubMed

Hughes C, Adlam A, Happe F, Jackson J, Taylor A, Caspi A. Good test-retest reliability for standard and advanced false-belief tasks across a wide range of abilities. J Child Psychol Psychiatry. 2000;41:483–90.CrossRefPubMed

Iarocci G, Yager J, Elfers T. What gene-environment interactions can tell us about social competence in typical and atypical populations. Brain Cogn. 2007;65:112–27.CrossRefPubMed

Jablensky A. Epidemiology of schizophrenia: the global burden of disease and disability. Eur Arch Psychiatry Clin Neurosci. 2000;250(6):274–85.CrossRefPubMed

Jolliffe T, Baron-Cohen S. The Strange Stories test: a replication with high-functioning adults with autism or Asperger syndrome. J Autism Dev Disord. 1999;29(5):395–406.CrossRefPubMed

Kiley-Brabeck K, Sobin C. Social skills and executive function deficits in children with the 22q11 deletion syndrome. Appl Neuropsychol. 2006;13:258–68.CrossRefPubMedCentralPubMed

Lajiness-O’Neill R, Beaulieu I, Titus J, Asamoah A, Bigler E, Bawle E. Memory and learning in children with 22q11.2 deletion syndrome: evidence for ventral and dorsal stream disruption? Child Neuropsychology. 2005;11(1):55–71.CrossRefPubMed

Murphy K. Schizophrenia and velo-cardio-facial syndrome. Lancet. 2002;359(9304):426–30.CrossRefPubMed

Murphy K. Annotation: velo-cardio-facial syndrome. J Child Psychol Psychiatry. 2005;45(6):563–71.CrossRef

Niklasson L, Rasmussen P, Oskarsdottir S, Gillberg C. Chromosome 22q11 deletion syndrome (CATCH 22): neuropsychiatric and neuropsychological aspects. Dev Med Child Neurol. 2002;44:44–50.CrossRefPubMed

Perner J, Wimmer H. John thinks that mary thinks that—attribution of 2nd-order beliefs by 5-Year-Old to 10-Year-Old Children. J Exp Child Psychol. 1985;39(3):437–71.CrossRef

Posamentier M, Abdi H. Processing faces and facial expressions. Neuropsychol Rev. 2003;13(3):113–43.CrossRefPubMed

Premack D, Woodruff G. Does the chimpanzee have a theory of mind? Behav Brain Sci. 1978;1:515–26.CrossRef

Sands JR, Harrow M. Vulnerability to psychosis in unipolar major depression: is premorbid functioning involved? Am J Psychiatry. 1995;152(7):1009–15. 1995 July 1.CrossRefPubMed

Shprintzen R. Velo-cardio-facial syndrome: a distinctive behavioural phenotype. Ment Retard Dev Disabil Res Rev. 2000;6(2):142–7.CrossRefPubMed

Shprintzen R. Velo-cardio-facial syndrome. Prog Pediatr Cardiol. 2005;20:187–93.CrossRef

Siegal M, Varley R. Neural systems involved in “theory of mind”. Nature Review Neuroscience. 2002;3(6):463–71.

Swillen A, Devriendt K, Legius E, Eyskens B, Dumoulin M, Gewillig M, et al. Intelligence and psychosocial adjustment in velocardiofacial syndrome: a study of 37 children and adolescents with VCFS. J Med Genet. 1997;34(6):453–8.CrossRefPubMedCentralPubMed

Swillen A, Devriendt K, Legius E, Prinzie P, Vogels A, Ghesquiere P. The behavioural phenotype in velo-cardio-facial syndrome (VCFS): from infancy to adolescence. Genet Couns. 1999;10(1):79–88.PubMed

Swillen A, Vogels A, Devriendt K, Fryns JP. Chromosome 22q11 deletion syndrome: update and review of the clinical features, cognitive-behavioral spectrum, and psychiatric complications. Am J Med Genet. 2000;97:128–35.CrossRefPubMed

Tager-Flusberg H, Sullivan K. A componential view of theory of mind: evidence from Williams syndrome. Cognition. 2000;76:59–89.CrossRefPubMed

Tager-Flusberg H, Plesa-Skwerer D, Faja S, Joseph R. People with Williams syndrome process faces holistically. Cognition. 2003;89:11–24.CrossRefPubMed

van Amelsvoort T, Daly E, Robertson D, Suckling J, Ng V, Critchley H, Owen M, Henry J, Murphy KC, Murphy DGM. Structural brain abnormalities associated with deletion at chromosome 22q11: quantitative neuroimaging study of adults with velo-cardio-facial syndrome. Br J Psych. 2001;178:412–419.

van Amelsvoort T, Schmitz N, Daly E, Deeley Q, Critchley H, Henry J, et al. Processing facial emotions in adults with velo-cardio-facial syndrome: functional magnetic resonance imaging. Br J Psychiatry. 2006;189:560–1.CrossRefPubMed

Vorstman J, Morcus M, Duijff S, Klaassen P, Heineman-de Boer J, Beemer F. The 22q11.2 deletion in children: high rate of autistic disorders and early onset of psychotic symptoms. J Am Acad Child Adolesc Psychiatry. 2006;45(9):1104–13.CrossRefPubMed

Wang Y-G, Wang Y-Q, Chen S-L, Zhu C-Y. Theory of mind disability in major depression with or without psychotic symptoms: a componential view. Psychiatry Res. 2008;161(2):153–61.

Wechsler D. Wechsler Intelligence Scales for Children—third edition. San Antonio, Texas: The Psychological Corporation; 1991.

Title: Is theory of mind related to social dysfunction and emotional problems in 22q11.2 deletion syndrome (velo-cardio-facial syndrome)?
Authors: Linda E. Campbell
Angela F. Stevens
Kathryn McCabe
Lynne Cruickshank
Robin G. Morris
Declan G. M. Murphy
Kieran C. Murphy
Publication date: 01-06-2011
Publisher: BioMed Central
Published in: Journal of Neurodevelopmental Disorders / Issue 2/2011
Print ISSN: 1866-1947
Electronic ISSN: 1866-1955
DOI: https://doi.org/10.1007/s11689-011-9082-7

At a glance: The STEP trials

Springer Medicine

Is theory of mind related to social dysfunction and emotional problems in 22q11.2 deletion syndrome (velo-cardio-facial syndrome)?

Abstract

At a glance: The STEP trials

Springer Medicine

Abstract

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