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Skin Picking in People with Prader–Willi Syndrome: Phenomenology and Management

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Abstract

Skin picking is highly prevalent in people with Prader–Willi syndrome (PWS). This study addressed the temporal (frequency, duration) and wider characteristics (e.g. type of skin picked, apparent motivations, or management strategies) of skin picking to inform intervention strategies. Nineteen parents/carers who observe skin picking shown by the person they care for completed a semi-structured interview. Results were consistent with previous research but advanced the field by finding that most participants picked skin with an imperfection and that parents/carers most commonly use distraction as a management strategy. Interventions that are behavioural, support emotion regulation and/ or are used in the typically developing population are therefore likely to be beneficial for future research.

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References

  • Akefeldt, A., & Gillberg, C. (1999). Behavior and personality characteristics of children and young adults with Prader–Willi syndrome: A controlled study. Journal of the American Academy of Child & Adolescent Psychiatry, 38(6), 761–769.

    Google Scholar 

  • American Psychiatric Association. (2013). Diagnostic and statistical manual of mental disorders (5th ed.). Washington, DC: American Psychiatric Association.

    Google Scholar 

  • Banga, A., & Connor, D. F. (2012). Effectiveness of naltrexone for treating pathologic skin picking behavior in an adolescent with Prader–Willi syndrome. Journal of Child and Adolescent Psychopharmacology, 22(5), 396–398.

    PubMed  Google Scholar 

  • Beck, A. T. (1967). Depression causes and treatment. Philadelphia, PA: University of Pennsylvania Press.

    Google Scholar 

  • Benjamin, E., & Buot-Smith, T. (1993). Naltrexone and fluoxetine in Prader–Willi syndrome. Journal of the American Academy of Child & Adolescent Psychiatry, 32(4), 870–873.

    Google Scholar 

  • Boer, H., & Clarke, D. (1999). Development and behaviour in genetic syndromes: Prader-Willi syndrome. Journal of Applied Research in Intellectual Disabilities, 12, 294–301.

    Google Scholar 

  • Bull, L. E. (2014). Understanding and changing behaviour in Prader-Willi syndrome. Unpublished PhD thesis. University of Birmingham, UK.

  • Bull, L. E. (2015). Self-injurious behaviour in people with Prader-Willi syndrome. Unpublished thesis. University of Birmingham, UK.

  • Bull, L. E., Oliver, C., Tunnicliffe, P. L., & Woodcock, K. A. (2015). An informant report behavior diary for measuring temper outbursts in an intervention setting. Journal of Developmental and Physical Disabilities, 27, 1–16.

    Google Scholar 

  • Buono, S., Palmigiano, M., Scannella, F., Occhipinti, P., & Greco, D. (2005). Self-injury and Präder–Willi syndrome. Journal of Policy and Practice in Intellectual Disabilities, 2, 256–259.

    Google Scholar 

  • Buono, S., Scannella, F., & Palmigiano, M. B. (2010). Self-injurious behavior: A comparison between Prader–Willi syndrome, down syndrome and autism. Life Span and Disability, 13(2), 187–201.

    Google Scholar 

  • Cassidy, S. B., & Driscoll, D. J. (2009). Prader–Willi syndrome. European Journal of Human Genetics, 17, 3–13.

    PubMed  Google Scholar 

  • Cassidy, S. B., Forsythe, M., Heeger, S., Nicholls, R. D., Schork, N., Benn, P., et al. (1997). Comparison of phenotype between patients with Prader–Willi syndrome due to deletion 15q and uniparental disomy 15. American Journal of Medical Genetics, 68, 433–440.

    PubMed  Google Scholar 

  • Didden, R., Korzilius, H., & Curfs, L. M. G. (2007). Skin-picking in individuals with Prader–Willi syndrome: Prevalence, functional assessment, and its comorbidity with compulsive and self-injurious behaviours. Journal of Applied Research in Intellectual Disabilities, 20, 409–419.

    Google Scholar 

  • Didden, R., Proot, I., Lancioni, G. E., van Os, R., & Curfs, L. M. G. (2008). Individuals with Prader–Willi syndrome and their perceptions of skin-picking behaviour. British Journal of Developmental Disabilities, 54, 123–130.

    Google Scholar 

  • Dykens, E. M., Cassidy, S. B., & King, B. H. (1999). Maladaptive behavior differences in Prader–Willi syndrome due to paternal deletion versus maternal uniparental disomy. American Journal on Mental Retardation, 104, 67–77.

    PubMed  Google Scholar 

  • Dykens, E. M., Hodapp, R. M., Walsh, K., & Nash, L. J. (1992). Adaptive and maladaptive behavior in Prader–Willi syndrome. Journal of the American Academy of Child & Adolescent Psychiatry, 31(6), 1131–1136.

    Google Scholar 

  • Eccleston, C., & Crombez, G. (1999). Pain demands attention: A cognitive–affective model of the interruptive function of pain. Psychological Bulletin, 125(3), 356.

    PubMed  Google Scholar 

  • Feurer, I. D., Dimitropoulos, A., Stone, W. L., Roof, E., Butler, M. G. & Thompson, T. (1998). The latent variable structure of the Compulsive Behaviour Checklist in people with Prader-Willi syndrome. Journal on Intellectual Disability Research, 42(6), 472–480.

    Google Scholar 

  • Gelinas, B. L., & Gagnon, M. M. (2013). Pharmacological and psychological treatments of pathological skin-picking: A preliminary meta-analysis. Journal of Obsessive-Compulsive and Related Disorders, 2(2), 167–175.

    Google Scholar 

  • Grant, J. E., Odlaug, B. L., & Kim, S. W. (2010). A clinical comparison of pathological skin picking and obsessive compulsive disorder. Comprehensive Psychiatry, 51(4), 347–352.

    PubMed  Google Scholar 

  • Grant, J. E., Stein, D. J., Woods, D. W., & Keuthen, N. J. (2012). Trichotillomania, skin picking & other body-focused repetitive behaviors. Arlington, VA: American Psychiatric Publishing.

    Google Scholar 

  • Hall, S. S., Hammond, J. L., & Hustyi, K. M. (2013). Examining the relationship between heart rate and problem behavior: A case study of severe skin picking in Prader–Willi syndrome. American Journal on Intellectual and Developmental Disabilities, 118(6), 460–474.

    PubMed  Google Scholar 

  • Hartley, S. L., MacLean, W. E., Butler, M. G., Zarcone, J., & Thompson, T. (2005). Maladaptive behaviors and risk factors among the genetic subtypes of Prader–Willi syndrome. American Journal of Medical Genetics Part A, 136(2), 140–145.

    PubMed  PubMed Central  Google Scholar 

  • Holland, A. J., Whittington, J. E., Butler, J., Webb, T., Boer, H., & Clarke, D. (2003). Behavioural phenotypes associated with specific genetic disorders: Evidence from a population-based study of people with Prader–Willi syndrome. Psychological Medicine, 33(1), 141–153.

    PubMed  Google Scholar 

  • Hustyi, K. M., Hammond, J. L., Rezvani, A. B., & Hall, S. S. (2013). An analysis of the topography, severity, potential sources of reinforcement, and treatments utilized for skin picking in Prader–Willi syndrome. Research in Developmental Disabilities, 34(9), 2890–2899.

    PubMed  Google Scholar 

  • Kushlick, A., Blunden, R., & Cox, G. (1973). A method of rating behaviour characteristics for use in large scale surveys of mental handicap. Psychological Medicine, 3(4), 466–478.

    PubMed  Google Scholar 

  • McAllister, C. J., Whittingtin, J. E., & Holland, A. J. (2011). Development of the eating behaviour in Prader–Willi syndrome: Advances in our understanding. International Journal of Obesity, 35, 188–197.

    PubMed  Google Scholar 

  • Miller, J. L., & Angulo, M. (2014). An open-label pilot study of N-acetylcysteine for skin-picking in Prader–Willi syndrome. American Journal of Medical Genetics Part A, 164(2), 421–424.

    Google Scholar 

  • Morgan, J. R., Storch, E. A., Woods, D. W., Bodzin, D., Lewin, A. B., & Murphy, T. K. (2010). A preliminary analysis of the phenomenology of skin-picking in Prader–Willi syndrome. Child Psychiatry & Human Development, 41, 448–463.

    Google Scholar 

  • Najdowski, A. C., Wallace, M. D., Ellsworth, C. L., MacAleese, A. N., & Cleveland, J. M. (2008). Functional analyses and treatment of precursor behavior. Journal of Applied Behavior Analysis, 41(1), 97–105.

    PubMed  PubMed Central  Google Scholar 

  • Odlaug, B. L., & Grant, J. E. (2008). Clinical characteristics and medical complications of pathological skin picking. General Hospital Psychaitry, 30(1), 61–66.

    Google Scholar 

  • Odlaug, B. L., & Grant, J. E. (2012). Pathological skin picking. In J. E. Grant, D. J. Stein, D. W. Woods, & N. J. Keuthen (Eds.), Trichotillomania, skin picking & other body-focused repetitive behaviors. Arlington, VA: American Psychiatric Publishing.

    Google Scholar 

  • Oliver, C., McClintock, K., Hall, S., Smith, M., Dagnan, D., & Stenfert-Kroese, B. (2003). Assessing the severity of challenging behaviour: Psychometric properties of the challenging behaviour interview. Journal of Applied Research in Intellectual Disabilities, 16, 53–61.

    Google Scholar 

  • Oliver, C., & Richards, C. (2015). Practitioner review: Self-injurious behaviour in children with developmental delay. Journal of Child Psychology and Psychiatry. https://doi.org/10.1111/jcpp.12425.

    Article  PubMed  Google Scholar 

  • Oppenheim, A. N. (2005). Questionnaire design, interviewing and attitude measurement: New edition. New York, NY: Continuum.

    Google Scholar 

  • Prochaska, J. O., & DiClemente, C. C. (1982). Transtheoretical therapy: Toward a more integrative model of change. Psychotherapy Theory, Research & Practice, 19(3), 276.

    Google Scholar 

  • Radstaake, M., Didden, R., Bolio, M., Lang, R., Lancioni, G. E., & Curfs, L. M. (2011). Functional assessment and behavioral treatment of skin picking in a teenage girl with Prader–Willi syndrome. Clinical Case Studies, 10, 67–78.

    Google Scholar 

  • Robb, N., Waller, A., & Woodcock, K. A. (2019). Developing a task switching training game for children with a rare genetic syndrome linked to intellectual disability. Simulation & Gaming, 50(2), 160–179.

    Google Scholar 

  • Roberts, S., O’Connor, K., & Bélanger, C. (2013). Emotion regulation and other psychological models for body-focused repetitive behaviours. Clinical Psychology Review, 33(6), 745–762.

    PubMed  Google Scholar 

  • Rollnick, S., & Miller, W. R. (1995). What is motivational interviewing? Behavioural and Cognitive Psychotherapy, 23(04), 325–334.

    Google Scholar 

  • Selikowitz, M., Sunman, J., Pendergast, A., & Wright, S. (1990). Fenfluramine in Prader–Willi syndrome: A double blind, placebo controlled trial. Archives of Disease in Childhood, 65, 112–114.

    PubMed  PubMed Central  Google Scholar 

  • Shapira, N. A., Lessig, M. C., Lewis, M. H., Goodman, W. K., & Driscoll, D. J. (2004). Effects of topiramate in adults with Prader–Willi syndrome. American Journal on Mental Retardation, 109(4), 301–309.

    PubMed  Google Scholar 

  • Singh, N. N., Lancioni, G. E., Myers, R. E., Karazsia, B. T., Courtney, T. M., & Nugent, K. (2017). A mindfulness-based intervention for self-management of verbal and physical aggression by adolescents with Prader–Willi syndrome. Developmental Neurorehabilitation, 20(5), 253–260.

    PubMed  Google Scholar 

  • Symons, F. J., Butler, M. G., Sanders, M. D., Feurer, I. D., & Thompson, T. (1999). Self-injurious behavior and Prader–Willi syndrome: Behavioral forms and body locations. American Journal on Mental Retardation, 104(3), 260–269.

    PubMed  Google Scholar 

  • Toogood, S., & Timlin, K. (1996). The functional assessment of challenging behaviour: A comparison of informant-based, experimental and descriptive methods. Journal of Applied Research in Intellectual Disabilities, 9(3), 206–222.

    Google Scholar 

  • Tunnicliffe, P., Woodcock, K., Bull, L., Penhallow, J., & Oliver, C. (2014). Temper outbursts in Prader–Willi syndrome: Causes, behavioural and emotional sequence and responses by carers. Journal of Intellectual Disability Research, 58(2), 134–150.

    PubMed  Google Scholar 

  • Van Ameringen, M., Patterson, B., & Simpson, W. (2014). DSM-5 obsessive-compulsive and related disorders: Clinical implications of new criteria. Depression and Anxiety, 31(6), 487–493.

    PubMed  Google Scholar 

  • Whittington, J. E., Holland, A. J., Webb, T., Butler, J., Clarke, D., & Boer, H. (2004). Cognitive abilities and genotype in a population-based sample of people with Prader–Willi syndrome. Journal of Intellectual Disability Research, 48, 172–187.

    PubMed  Google Scholar 

  • Wigren, M., & Hansen, S. (2005). ADHD symptoms and insistence on sameness in Prader-Willi syndrome. Journal on Intellectual Disability Research, 49, 449–456.

    Google Scholar 

  • Wigren, M., & Heimann, M. (2010). Excessive picking in Prader–Willi syndrome: A pilot study of phenomenological aspects and comorbid symptoms. International Journal of Diasability, Development and Education, 48(2), 129–142.

    Google Scholar 

  • Wilson, D. M., Iwata, B. A., & Bloom, S. E. (2012). Computer-assisted measurement of wound size associated with self-injurious behaviour. Journal of Applied Behavior Analysis, 45(4), 797–808.

    PubMed  PubMed Central  Google Scholar 

  • Woodcock, K., Oliver, C., & Humphreys, G. (2009). Associations between repetitive questioning, resistance to change, temper outbursts and anxiety in Prader–Willi and Fragile-X syndromes. Journal of Intellectual Disability Research, 53, 265–278.

    PubMed  Google Scholar 

  • Ye, L., Bawa, R., & Lippmann, S. (2013). Compulsive skin manipulation treated by topiramate. Journal of child and adolescent psychopharmacology, 23(5), 369–370.

    PubMed  Google Scholar 

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Acknowledgments

Thank you to the families who took part making this research possible. Thank you to Emma Shepherd for supporting the research by coding the interviews.

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Author notes

  1. L. E. Bull

    Present address: Great Ormond Street Hospital for Children, Great Ormond Street, London, WC1N 3JH, UK

  2. K. A. Woodcock

    Present address: Centre for Applied Psychology, School of Psychology & Institute for Mental Health, University of Birmingham, 52 Pritchatts Road, Edgbaston, Birmingham, B15 2SA, UK

Authors and Affiliations

  1. Cerebra Centre for Neurodevelopmental Disorders, School of Psychology, University of Birmingham, 52 Pritchatts Road, Edgbaston, Birmingham, B15 2SA, UK

    L. E. Bull & C. Oliver

  2. School of Psychology, Queen’s University Belfast, Belfast, UK

    K. A. Woodcock

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  1. L. E. Bull
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  2. C. Oliver
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Correspondence to L. E. Bull or K. A. Woodcock.

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Bull, L.E., Oliver, C. & Woodcock, K.A. Skin Picking in People with Prader–Willi Syndrome: Phenomenology and Management. J Autism Dev Disord 51, 286–297 (2021). https://doi.org/10.1007/s10803-020-04504-5

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