Anxiety Disorders in Williams Syndrome Contrasted with Intellectual Disability and the General Population: A Systematic Review and Meta-Analysis

R Royston, P Howlin, J Waite, C Oliver, R Royston, P Howlin, J Waite, C Oliver

Abstract

Individuals with specific genetic syndromes associated with intellectual disability (ID), such as Williams syndrome (WS), are at increased risk for developing anxiety disorders. A systematic literature review identified sixteen WS papers that could generate pooled prevalence estimates of anxiety disorders for WS. A meta-analysis compared these estimates with prevalence estimates for the heterogeneous ID population and the general population. Estimated rates of anxiety disorders in WS were high. WS individuals were four times more likely to experience anxiety than individuals with ID, and the risk was also heightened compared to the general population. The results provide further evidence of an unusual profile of high anxiety in WS.

Keywords: Anxiety disorders; Genetic syndromes; Intellectual disability; Meta-analysis; Systematic review; Williams syndrome.

Conflict of interest statement

Conflict of Interest

The authors have no conflict of interest to declare.

Research Involving Human Participants and/or Animals

This article does not contain any studies with human participants or animals performed by any of the authors.

Informed Consent

Not applicable.

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References

    1. American Psychiatric Association . Diagnostic and statistical manual of mental disorders: DSM-III. 3. Washington, DC: American Psychiatric Association; 1980.
    1. American Psychiatric Association . Diagnostic and statistical manual of mental disorders: DSM-III-R. 3. Washington, DC: American Psychiatric Association; 1987.
    1. American Psychiatric Association . Diagnostic and statistical manual of mental disorders: DSM-IV. 4. Washington, DC: American Psychiatric Association; 1994.
    1. American Psychiatric Association . Diagnostic and statistical manual of mental disorders. 5. Arlington, VA: American Psychiatric Publishing; 2013.
    1. Avery S., Thornton-Wells T., Blackford J. Altered white matter integrity of prefrontal-amygdala pathways in Williams syndrome. Biological Psychiatry. 2011;1:255S.
    1. Barendregt, J. J., & Doi, S. A. (2011). Meta XL user guide: Version 2.0.
    1. Baxter A. J., Scott K. M., Vos T., Whiteford H. A. Global prevalence of anxiety disorders: A systematic review and meta-regression. Psychological Medicine. 2013;43(05):897–910.
    1. Bellugi U., Bihrle A., Jernigan T., Trauner D., Doherty S. Neuropsychological, neurological, and neuroanatomical profile of Williams syndrome. American Journal of Medical Genetics. 1990;37(S6):115–125.
    1. Bellugi U., Lichtenberger L., Jones W., Lai Z., St. George M. The neurocognitive profile of Williams syndrome: A complex pattern of strengths and weaknesses. Journal of Cognitive Neuroscience: Linking Cognitive Neuroscience and Molecular Genetics: New Perspectives from Williams Syndrome. 2000;12(Supp 1):7–29.
    1. Borenstein M., Hedges L. V., Higgins J. P. T., Rothstein H. R. A basic introduction to fixed-effect and random-effects models for meta-analysis. Research Synthesis Methods. 2010;1(2):97–111.
    1. Boyle M. H., Offord D. R., Racine Y., Sanford M., Szatmari P., Fleming J. E., Price-Munn N. Evaluation of the diagnostic interview for children and adolescents for use in general population samples. Journal of Abnormal Child Psychology. 1993;21(6):663–681.
    1. Brock J. Language abilities in Williams syndrome: A critical review. Development and Psychopathology. 2007;19(01):97–127.
    1. Cherniske E. M., Carpenter T. O., Klaiman C., Young E., Bregman J., Insogna K., et al. Multisystem study of 20 older adults with Williams syndrome. American Journal of Medical Genetics Part A. 2004;131(3):255–264.
    1. Cooper S. A., Melville C. A., Einfeld S. L. Psychiatric diagnosis, intellectual disabilities and diagnostic criteria for psychiatric disorders for use with adults with learning disabilities/mental retardation (DC-LD) Journal of Intellectual Disability Research: JIDR. 2003;47:3–15.
    1. Davies M., Udwin O., Howlin P. Adults with Williams syndrome-preliminary study of social, emotional and behavioural difficulties. The British Journal of Psychiatry: The Journal of Mental Science. 1998;172:273–276.
    1. Deb S., Thomas M., Bright C. Mental disorder in adults with intellectual disability. I: Prevalence of functional psychiatric illness among a community-based population aged between 16 and 64 years. Journal of Intellectual Disability Research: JIDR. 2001;45:495–505.
    1. Dekker M. C., Koot H. M., van der Ende J., Verhulst F. C. Emotional and behavioral problems in children and adolescents with and without intellectual disability. Journal of Child Psychology and Psychiatry, and Allied Disciplines. 2002;43(8):1087–1098.
    1. Dodd H. F., Porter M. A. Psychopathology in Williams syndrome: The effect of individual differences across the life span. Journal of Mental Health Research in Intellectual Disabilities. 2009;2(2):89–109.
    1. Dodd, H. F., & Porter, M. A. (2011a). Interpretation of ambiguous situations: evidence for a dissociation between social and physical threat in Williams syndrome. Journal of Autism and Developmental Disorders, 41(3), 266–274.
    1. Dodd, H. F., & Porter, M. A. (2011b). There’s that scary picture: Attention bias to threatening scenes in Williams syndrome. Neuropsychologia, 49(2), 247–253.
    1. Dodd H. F., Schniering C. A., Porter M. A. Beyond behaviour: Is social anxiety low in Williams syndrome? Journal of Autism and Developmental Disorders. 2009;39(12):1673–1681.
    1. Dykens E. M. Annotation: Psychopathology in children with intellectual disability. Journal of Child Psychology and Psychiatry, and Allied Disciplines. 2000;41(4):407–417.
    1. Dykens E. M. Anxiety, fears, and phobias in persons with Williams syndrome. Developmental Neuropsychology. 2003;23(1–2):291–316.
    1. Dykens E. M., Rosner B. A., Ly T., Sagun J. Music and anxiety in Williams syndrome: A harmonious or discordant relationship? American Journal of Mental Retardation: AJMR. 2005;110(5):346–358.
    1. Einfeld S. L., Tonge B. J. Population prevalence of psychopathology in children and adolescents with intellectual disability: II epidemiological findings. Journal of Intellectual Disability Research: JIDR. 1996;40(2):99–109.
    1. Einfeld S. L., Tonge B. J., Florio T. Behavioral and emotional disturbance in individuals with Williams syndrome. American Journal of Mental Retardation: AJMR. 1997;102(1):45–53.
    1. Einfeld S. L., Tonge B. J., Rees V. W. Longitudinal course of behavioral and emotional problems in Williams syndrome. American Journal of Mental Retardation: AJMR. 2001;106(1):73–81.
    1. Emerson E. Prevalence of psychiatric disorders in children and adolescents with and without intellectual disability. Journal of Intellectual Disability Research: JIDR. 2003;47(1):51–58.
    1. Ewart A. K., Morris C. A., Atkinson D., Jin W. S., Sternes K., Spallone P., et al. Hemizygosity at the elastin locus in a developmental disorder, Williams syndrome. Nature Genetics. 1993;5(1):11–16.
    1. Ezpeleta L., Osa N., Domenech J. M., Navarro J. B., Losilla J. M., Judez J. Diagnostic agreement between clinicians and the diagnostic interview for children and adolescents-DICA-R-in an outpatient sample. Journal of Child Psychology and Psychiatry. 1997;38(4):431–440.
    1. Gagliardi C., Martelli S., Tavano A., Borgatti R. Behavioural features of Italian infants and young adults with Williams–Beuren syndrome. Journal of Intellectual Disability Research. 2011;55(2):121–131.
    1. Goodman R., Ford T., Richards H., Gatward R., Meltzer H. The development and well-being assessment: Description and initial validation of an integrated assessment of child and adolescent psychopathology. Journal of Child Psychology and Psychiatry. 2000;41(05):645–655.
    1. Gothelf D., Farber N., Raveh E., Apter A., Attias J. Hyperacusis in Williams syndrome characteristics and associated neuroaudiologic abnormalities. Neurology. 2006;66(3):390–395.
    1. Green H., McGinnity Á., Meltzer H., Ford T., Goodman R. Mental health of children and young people in Great Britain, 2004. Basingstoke: Palgrace Macmillan; 2005.
    1. Green T., Avda S., Dotan I., Zarchi O., Basel-Vanagaite L., Zalsman G., et al. Phenotypic psychiatric characterization of children with Williams syndrome and response of those with ADHD to methylphenidate treatment. American Journal of Medical Genetics, Part B-Neuropsychiatric Genetics. 2011;159B(1):13–20.
    1. Hermans H., van der Pas F. H., Evenhuis H. M. Instruments assessing anxiety in adults with intellectual disabilities: A systematic review. Research in Developmental Disabilities. 2011;32(3):861–870.
    1. Jabbi M., Chen Q., Turner N., Kohn P., White M., Kippenhan J. S., et al. Variation in the Williams syndrome GTF2I gene and anxiety proneness interactively affect prefrontal cortical response to aversive stimuli. Translational Psychiatry. 2015;5:e622–e622.
    1. Jabbi M., Kippenhan J., Kohn P., Marenco S., Mervis C. B., Morris C. A., et al. The Williams syndrome chromosome 7q11.23 hemideletion confers hypersocial, anxious personality coupled with altered insula structure and function. Proceedings of the National Academy of Sciences of the United States of America. 2012;109(14):E860–E866.
    1. Jarvinen A., Korenberg J. R., Bellugi U. The social phenotype of Williams syndrome. Current Opinion in Neurobiology. 2013;23(3):414–422.
    1. Jones W., Bellugi U., Lai Z., Chiles M., Reilly J., Lincoln A., Adolphs R. II. hypersociability in Williams syndrome. Journal of Cognitive Neuroscience. 2000;12(Supplement 1):30–46.
    1. Kaufman J., Birmaher B., Brent D., Rao U., Flynn C., Moreci P., et al. Schedule for affective disorders and schizophrenia for school-age children present and lifetime version (K-SADS-PL): Initial reliability and validity data. Journal of the American Academy of Child and Adolescent Psychiatry. 1997;36(7):980–988.
    1. Kennedy J. C., Kaye D. L., Sadler L. S. Psychiatric diagnoses in patients with Williams syndrome and their families. Jefferson Journal of Psychiatry. 2006;20(1):4.
    1. Khreim I., Mikkelsen E. Anxiety disorders in adults with mental retardation. Psychiatric Annals. 1997;27(3):175–181.
    1. Klein A. J., Armstrong B. L., Greer M. K., Brown F. R. Hyperacusis and otitis media in individuals with Williams syndrome. Journal of Speech and Hearing Disorders. 1990;55(2):339–344.
    1. Leyfer O., John A. E., Woodruff-Borden J., Mervis C. B. Factor structure of the children’s behavior questionnaire in children with Williams syndrome. Journal of Autism and Developmental Disorders. 2012;42(11):2346–2353.
    1. Leyfer O., Woodruff-Borden J., Mervis C. B. Anxiety disorders in children with Williams syndrome, their mothers, and their siblings: Implications for the etiology of anxiety disorders. Journal of Neurodevelopmental Disorders. 2009;1(1):4.
    1. Leyfer O. T., Woodruff-Borden J., Klein-Tasman B. P., Fricke J. S., Mervis C. B. Prevalence of psychiatric disorders in 4 to 16-year-olds with Williams syndrome. American Journal of Medical Genetics Part B: Neuropsychiatric Genetics. 2006;141(6):615–622.
    1. Mervis C. B., Dida J., Lam E., Crawford-Zelli N. A., Young E. J., Henderson D. R., et al. Duplication of GTF2I results in separation anxiety in mice and humans. The American Journal of Human Genetics. 2012;90(6):1064–1070.
    1. Meyer-Lindenberg A., Hariri A. R., Munoz K. E., Mervis C. B., Mattay V. S., Morris C. A., et al. Neural correlates of genetically abnormal social cognition in Williams syndrome. Nature Neuroscience. 2005;8(8):991–993.
    1. Moher D., Liberati A., Tezlaff J., Altman D. G. Preferred reporting items for systematic reviews and meta-analyses: The PRISMA statement. Annals of Internal Medicine. 2009;151(4):264–269.
    1. Morris C. A. The behavioral phenotype of Williams syndrome: A recognizable pattern of neurodevelopment. American Journal Medical Genetics, Part C: Seminars in Medical Genetics. 2010;154C(4):427–431.
    1. Morris C. A., Mervis C. B. Williams syndrome and related disorders. Annual Review of Genomics and Human Genetics. 2000;1:461–484.
    1. Moss S., Goldberg D., Patel P., Prosser H., Ibbotson B., Simpson N., et al. The psychiatric assessment schedule for adults with developmental disabilities. Manchester: University of Manchester, Hester Adrian Research Centre; 1996.
    1. Moss S., Ibbotson B., Prosser H., Goldberg D., Patel P., Simpson N. Validity of the PAS-ADD for detecting psychiatric symptoms in adults with learning disability (mental retardation) Social Psychiatry and Psychiatric Epidemiology. 1997;32(6):344–354.
    1. Munoz K. E., Meyer-Lindenberg A., Hariri A. R., Mervis C. B., Mattay V. S., Morris C. A., et al. Abnormalities in neural processing of emotional stimuli in Williams syndrome vary according to social vs. non-social content. NeuroImage. 2010;50(1):340–346.
    1. Ng R., Järvinen A., Bellugi U. Characterizing associations and dissociations between anxiety, social, and cognitive phenotypes of Williams syndrome. Research in Developmental Disabilities. 2014;35(10):2403–2415.
    1. Papaeliou C., Polemikos N., Fryssira E., Kodakos A., Kaila M., Yiota X., et al. Behavioural profile and maternal stress in Greek young children with Williams syndrome. Child: Care, Health and Development. 2012;38(6):844–853.
    1. Pegoraro L. F. L., Steiner C. E., Celeri E. H. R. V., Banzato C. E. M., Dalgalarrondo P. Cognitive and behavioral heterogeneity in genetic syndromes. Jornal de Pediatria. 2014;90(2):155–160.
    1. Plissart L., Borghgraef M., Volcke P., Vandenberghe H., Fryns J. P. Adults with Williams–Beuren syndrome-evaluation of the medical, psychological and behavioural aspects. Clinical Genetics. 1994;46(2):161–167.
    1. Polanczyk G. V., Salum G. A., Sugaya L. S., Caye A., Rohde L. A. Annual research review: A meta-analysis of the worldwide prevalence of mental disorders in children and adolescents. Journal of Child Psychology and Psychiatry. 2015;56(3):345–365.
    1. Reardon T. C., Gray K. M., Melvin G. A. Anxiety disorders in children and adolescents with intellectual disability: Prevalence and assessment. Research In Developmental Disabilities. 2015;36:175-190.
    1. Reich W., Shayka J.J., Taibelson C. Diagnostic interview schedule for children and adolescents, parent version. St. Louis, MO: Washington University; 1991.
    1. Richards C., Jones C., Groves L., Moss J., Oliver C. Prevalence of autism spectrum disorder phenomenology in genetic disorders: A systematic review and meta-analysis. Lancet Psychiatry. 2015;2(10):909–916.
    1. Sakurai T., Dorr N. P., Takahashi N., McInnes L. A., Elder G. A., Buxbaum J. D. Haploinsufficiency of Gtf2i, a gene deleted in Williams syndrome, leads to increases in social interactions. Autism Research. 2011;4(1):28–39.
    1. Schubert C. The genomic basis of the Williams–Beuren syndrome. Cellular and Molecular Life Sciences: CMLS. 2009;66(7):1178–1197.
    1. Shaffer D., Fisher P., Lucas C., Comer J. Scoring manual: Diagnostic interview schedule for children. New York: (DISC-IV) Columbia University; 2000.
    1. Silverman W. K., Albano A. M. Anxiety disorders interview schedule for DSM-IV: Parent interview schedule. Oxford: Oxford University Press; 1996.
    1. Silverman W. K., Saavedra L. M., Pina A. A. Test–retest reliability of anxiety symptoms and diagnoses with the anxiety disorders interview schedule for DSM-IV: Child and parent versions. Journal of the American Academy of Child & Adolescent Psychiatry. 2001;40(8):937–944.
    1. Slade T., Andrews G. DSM-IV and ICD-10 generalized anxiety disorder: Discrepant diagnoses and associated disability. Social Psychiatry and Psychiatric Epidemiology. 2001;36(1):45–51.
    1. Somers J. M., Goldner E. M., Waraich P., Hsu L. Prevalence and incidence studies of anxiety disorders: A systematic review of the literature. The Canadian Journal of Psychiatry. 2006;51(2):100–113.
    1. Stinton C., Elison S., Howlin P. Mental health problems in adults with Williams syndrome. Ajidd-American Journal on Intellectual and Developmental Disabilities. 2010;115(1):3–18.
    1. Stromme P., Bjornstad P. G., Ramstad K. Prevalence estimation of Williams syndrome. Journal of Child Neurology. 2002;17(4):269–271.
    1. Szymanski, L. S. (1994). Mental retardation and mental health: concepts, aetiology and incidence. In N. Bouras, Mental health in mental retardation: Recent advances and practices (pp. 19–33). Cambridge: Cambridge University Press.
    1. Thornton-Wells T. A., Avery S. N., Blackford J. U. Using novel control groups to dissect the amygdala’s role in Williams syndrome. Developmental Cognitive Neuroscience. 2011;1(3):295–304.
    1. Timmreck T. C. An introduction to epidemiology. Burlington, MA: Jones & Bartlett Learning; 2002.
    1. Tyrer P. A comparison of DSM and ICD classifications of mental disorder. Advances in Psychiatric Treatment. 2014;20(4):280–285.
    1. Udwin O., Yule W. A cognitive and behavioural phenotype in Williams syndrome. Journal of Clinical and Experimental Neuropsychology: Official Journal of the International Neuropsychological Society. 1991;13(2):232–244.
    1. Woodruff-Borden J., Kistler D. J., Henderson D. R., Crawford N. A., Mervis C. B. Longitudinal course of anxiety in children and adolescents with Williams syndrome. American Journal of Medical Genetics, Part C-Seminars in Medical Genetics. 2010;154C(2):277–290.
    1. World Health Organisation . ICD-9 classifications of mental and behavioural disorder: Clinical descriptions and diagnostic guidelines. Geneva: World Health Organisation; 1978.
    1. World Health Organisation . ICD-10 classifications of mental and behavioural disorder: Clinical descriptions and diagnostic guidelines. Geneva: World Health Organisation; 1992.
    1. Zarchi O., Diamond A., Weinberger R., Abbott D., Carmel M., Frisch A., et al. A comparative study of the neuropsychiatric and neurocognitive phenotype in two microdeletion syndromes: Velocardiofacial (22q11. 2 deletion) and Williams (7q11. 23 deletion) syndromes. European Psychiatry. 2014;29(4):203–210.

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