Repetitive and ritualistic behaviour in children with Prader–Willi syndrome and children with autism
Abstract
BACKGROUND: Recent research has shown that the range of repetitive behaviour seen in individuals with Prader-Willi syndrome (PWS) extends beyond food-related behaviour. METHODS: The presence and intensity of repetitive, rigid and routinized behaviour in children with PWS was compared with that seen in children with another neurodevelopmental condition in which repetitive behaviour is common: children with autism. Parents completed the Childhood Routines Inventory (CRI). RESULTS: Contrary to our predictions, controlling for developmental level, children with PWS and children with autism showed similar levels of repetitive and ritualistic behaviour overall and on the two CRI factors measuring 'just right' and 'repetitive' behaviour. Indeed, the majority of the sample of parents of children with PWS endorsed most items on the CRI. However there was some specificity at the level of individual items with parents of children with PWS more frequently endorsing an item on 'collecting and storing objects' and parents of children with autism more frequently endorsing 'lining up objects', 'has a strong preference for certain foods' and 'seems aware of detail at home'. CONCLUSIONS: These findings confirm the range of repetitive behaviours that form part of the behavioural phenotype of PWS, including insistence on sameness and 'just right' behaviours, and uncover a surprising overlap with those seen in children with autism. Clinical management for children with PWS should include advice and education regarding management of repetitive and rigid behaviour. Future research should investigate whether the repetitive behaviours that form part of the behavioural phenotype of both PWS and autism are associated with a common neuropsychological, neurotransmitter or genetic origin.
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RRB is also evidenced in Prader-Willi syndrome, although to a lesser degree than in ASD as measured by the Repetitive Behavior Scale-Revised.
“Furthermore, RRB is also evidenced in PWS (Greaves et al., 2006), although to a lesser degree than in ASD as measured by the Repetitive Behavior Scale-Revised (RBS-R; Flores et al., 2011).”
From Oxytocin and vasopressin systems in genetic syndromes and neurodevelopmental disorders (2014) · cites this paper as Greaves et al., 2006
Cites
Diagnostic and Statistical Manual of Mental Disorders
American Psychiatric Association (2022) · doi:10.1176/appi.books.9780890425787 · Cited by 23,476 · see citing works
The Yale-Brown Obsessive Compulsive Scale: I. Development, Use, and Reliability
Goodman, W. K. (1989) · Archives of General Psychiatry · doi:10.1001/archpsyc.1989.01810110048007 · Cited by 7,712 · see citing works
The Yale-Brown Obsessive Compulsive Scale: II. Validity
Goodman, W. K. (1989) · Archives of General Psychiatry · doi:10.1001/archpsyc.1989.01810110054008 · Cited by 2,799 · see citing works
Vineland Adaptive Behavior Scales
Sparrow, S. S. (2010) · Springer eBooks · doi:10.1007/978-0-387-79948-3_1602 · Cited by 1,974 · see citing works
Epidemiological Surveys of Autism and Other Pervasive Developmental Disorders: An Update
Fombonne, E. (2003) · Journal of Autism and Developmental Disorders · doi:10.1023/a:1025054610557 · Cited by 1,505 · see citing works
Prader-Willi Syndrome: Consensus Diagnostic Criteria
Holm, V. A., Cassidy, S. B., Butler, M. G., Hanchett, J. M., Greenswag, L. R., Whitman, B. Y., & Greenberg, F. (1993) · Pediatrics · doi:10.1542/peds.91.2.398 · Cited by 1,278 · see citing works
Pervasive Developmental Disorders in preschool children
Chakrabarti, S. (2001) · JAMA · doi:10.1001/jama.285.24.3093 · Cited by 1,132 · see citing works
Annotation: Repetitive Behaviour in Autism: A Review of Psychological Research
Turner, M. (1999) · Journal of Child Psychology and Psychiatry · doi:10.1111/1469-7610.00502 · Cited by 688 · see citing works
Problem Behavior Interventions for Young Children with Autism: A Research Synthesis
Horner, R. H., Carr, E. G., Strain, P. S., Todd, A. W., & Reed, H. (2002) · Journal of Autism and Developmental Disorders · doi:10.1023/a:1020593922901 · Cited by 617 · see citing works
Gunay‐Aygun, M., Schwartz, S., Heeger, S., O’Riordan, M. A., & Cassidy, S. B. (2001) · PEDIATRICS · doi:10.1542/peds.108.5.e92 · Cited by 476 · see citing works
Evans, D. W., Leckman, J. F., Carter, A. S., Reznick, J. S., Henshaw, D., King, R. A., & Pauls, D. L. (1997) · Child Development · doi:10.1111/j.1467-8624.1997.tb01925.x · Cited by 449 · see citing works
Self-citationFine Mapping of Autistic Disorder to Chromosome 15q11-q13 by Use of Phenotypic Subtypes
Shao, Y., Cuccaro, M. L., Hauser, E. R., Raiford, K. L., Menold, M. M., Wolpert, C. M., Ravan, S. A., Elston, L. A., Decena, K., Donnelly, S. L., et al. (2003) · The American Journal of Human Genetics · doi:10.1086/367846 · Cited by 382 · see citing works
The Vineland Adaptive Behavior Scales: Supplementary Norms for Individuals with Autism
Carter, A. S., Volkmar, F. R., Sparrow, S. S., Wang, J.-J., Lord, C., Dawson, G., Fombonne, E., Loveland, K., Mesibov, G., & Schopler, E. (1998) · Journal of Autism and Developmental Disorders · doi:10.1023/a:1026056518470 · Cited by 367 · see citing works
McDougle, C. J., Kresch, L. E., Goodman, W. K., NAYLOR, S. T., Volkmar, F. R., Cohen, D. J., & Price, L. H. (1995) · American Journal of Psychiatry · doi:10.1176/ajp.152.5.772 · Cited by 314 · see citing works
Boer, H., Holland, A. J., Whittington, J. E., Butler, J., Webb, T. P., & Clarke, D. (2002) · The Lancet · doi:10.1016/s0140-6736(02)07340-3 · Cited by 302 · see citing works
Obsessions and Compulsions in Prader‐Willi Syndrome
Dykens, E. M., Leckman, J. F., & Cassidy, S. B. (1996) · Journal of Child Psychology and Psychiatry · doi:10.1111/j.1469-7610.1996.tb01496.x · Cited by 275 · see citing works
Fluoxetine Treatment of Children and Adults with Autistic Disorder and Mental Retardation
Cook, E. H., Rowlett, R., JASELSKIS, C. A., & LEVENTHAL, B. L. (1992) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/00004583-199207000-00024 · Cited by 269 · see citing works
Intellectual characteristics of Prader–Willi syndrome: comparison of genetic subtypes
Roof, E., Stone, W. L., MacLean, W. E., Feurer, I. D., Thompson, T., & Butler, M. G. (2000) · Journal of Intellectual Disability Research · doi:10.1046/j.1365-2788.2000.00250.x · Cited by 193 · see citing works
Holland, A. J., Whittington, J. E., Butler, J., WEBB, T., Boer, H., & CLARKE, D. (2002) · Psychological Medicine · doi:10.1017/s0033291702006736 · Cited by 188 · see citing works
Cognitive abilities and genotype in a population‐based sample of people with Prader–Willi syndrome
Whittington, J. E., Holland, A. J., Webb, T., Butler, J., Clarke, D., & Boer, H. (2004) · Journal of Intellectual Disability Research · doi:10.1111/j.1365-2788.2004.00556.x · Cited by 178 · see citing works
Prader–Willi syndrome, compulsive and ritualistic behaviours: the first population-based survey
Clarke, D. J., Boer, H., Whittington, J. E., Holland, A. J., Butler, J., & Webb, T. (2002) · The British Journal of Psychiatry · doi:10.1192/bjp.180.4.358 · Cited by 156 · see citing works
Research in Mental Retardation: Toward an Etiologic Approach
Dykens, E. M., & Hodapp, R. M. (2001) · Journal of Child Psychology and Psychiatry · doi:10.1111/1469-7610.00702 · Cited by 152 · see citing works
Adaptive and Maladaptive Behavior in Prader-Willi Syndrome
Dykens, E. M., Hodapp, R. M., WALSH, K., & NASH, L. J. (1992) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/00004583-199211000-00023 · Cited by 143 · see citing works
Veltman, M. W. M., Thompson, R. J., Roberts, S. E., Thomas, N. S., Whittington, J. E., & Bolton, P. (2004) · European Child & Adolescent Psychiatry · doi:10.1007/s00787-004-0354-6 · Cited by 134 · see citing works
Emotional symptoms in Prader‐Willi syndrome adolescents
Whitman, B. Y., Accardo, P., Opitz, J. M., Reynolds, J. F., & Ledbetter, D. H. (1987) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320280415 · Cited by 115 · see citing works
Evans, D. W., & Gray, F. L. (2000) · Child Development · doi:10.1111/1467-8624.00144 · Cited by 111 · see citing works
Self-citationCorrelates of maladaptive behavior in children and adults with Prader‐Willi syndrome
Dykens, E. M., & Cassidy, S. B. (1995) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320600612 · Cited by 109 · see citing works
Charman, T., Howlin, P., Berry, B., & Prince, E. (2004) · Autism · doi:10.1177/1362361304040641 · Cited by 102 · see citing works
Self-citationCase Study: Autism Associated with Marker Chromosome
Gillberg, C., Steffenburg, S., Wahlström, J., Gillberg, I. C., SJÖSTEDT, A., Martinsson, T., LIEDGREN, S., & Eeg‐Olofsson, O. (1991) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/00004583-199105000-00022 · Cited by 100 · see citing works
Thomas, J. A., Johnson, J. P., Kraai, T. L. P., Wilson, R., Tartaglia, N. R., LeRoux, J., Beischel, L. S., McGavran, L., & Hagerman, R. J. (2003) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.10176 · Cited by 92 · see citing works
Naltrexone and Fluoxetine in Prader-Willi Syndrome
Benjamin, E., & BUOT-SMITH, T. (1993) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/00004583-199307000-00025 · Cited by 82 · see citing works
Handbook of Autism and Pervasive Developmental Disorder
(2022) · Autism and child psychopathology series · doi:10.1007/978-3-030-88538-0 · Cited by 65 · see citing works
Bouras, N., Feurer, I. D., Dimitropoulos, A., et al. (1998) · Journal of Intellectual Disability Research · doi:10.1046/j.1365-2788.1998.4260472.x · Cited by 63 · see citing works
Rituals and compulsivity in Prader–Willi syndrome: profile and stability
Wigren, M., & Hansen, S. (2003) · Journal of Intellectual Disability Research · doi:10.1046/j.1365-2788.2003.00515.x · Cited by 62 · see citing works
Relationship between clinical and genetic diagnosis of Prader-Willi syndrome
Whittington, J. E., Holland, A., Webb, T., Butler, J., Clarke, D., & Boer, H. (2002) · Journal of Medical Genetics · doi:10.1136/jmg.39.12.926 · Cited by 55 · see citing works
Acute tryptophan depletion in autistic disorder: A controlled case study
McDougle, C. J., NAYLOR, S. T., Goodman, W. K., Volkmar, F. R., Cohen, D. J., & Price, L. H. (1993) · Biological Psychiatry · doi:10.1016/0006-3223(93)90011-2 · Cited by 51 · see citing works
Whittington, J. E., & Holland, T. (2004) · Cambridge University Press eBooks · doi:10.1017/cbo9780511543869 · Cited by 38 · see citing works
Molecular screening for proximal 15q abnormalities in a mentally retarded population.
Jacobsen, J., King, B. H., LEVENTHAL, B. L., Christian, S. L., Ledbetter, D. H., & Cook, E. H. (1998) · Journal of Medical Genetics · doi:10.1136/jmg.35.7.534 · Cited by 25 · see citing works
Burger (1998) · doi:10.1002/(sici)1098-2779(1998)4:2
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CARTER, C. (2007) · Behavioural Brain Research · doi:10.1016/j.bbr.2006.08.025 · Cited by 432 · see citing works
Zucker, N. L., Losh, M., Bulik, C. M., LaBar, K. S., Piven, J., & Pelphrey, K. A. (2007) · Psychological Bulletin · doi:10.1037/0033-2909.133.6.976 · Cited by 284 · see citing works
South, M., Ozonoff, S., & Mcmahon, W. M. (2007) · Autism · doi:10.1177/1362361307079606 · Cited by 260 · see citing works
McLennan, Y., Polussa, J., Tassone, F., & Hagerman, R. (2011) · Current Genomics · doi:10.2174/138920211795677886 · Cited by 160 · see citing works
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Tacer, K. F., & Potts, P. R. (2017) · Biochemical Journal · doi:10.1042/bcj20160616 · Cited by 81 · see citing works
Dimitropoulos, A., Ho, A., & Feldman, B. (2013) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-012-1547-3 · Cited by 77 · see citing works
Dimitropoulos, A., Ho, A. Y., Klaiman, C., Koenig, K., & Schultz, R. T. (2009) · Journal of Mental Health Research in Intellectual Disabilities · doi:10.1080/19315860903052204 · Cited by 31 · see citing works
Flores, C. G., Valcante, G., Guter, S., Zaytoun, A., Wray, E., Bell, L., Jacob, S., Lewis, M. H., Driscoll, D. J., Cook, E. H., et al. (2011) · Journal of Neurodevelopmental Disorders · doi:10.1007/s11689-011-9094-3 · Cited by 30 · see citing works
- Authors
- Greaves, N., Prince, E., Evans, D. W., & Charman, T.
- Affiliations
- London Ambulance Service NHS Trust (GB)
- University of London (GB)
- Bucknell University (US)
- Corresponding author
- Charman, T.
- Venue
- Journal of Intellectual Disability Research
- Publisher
- Wiley
- Published
- 2005-07-22
- Volume
- 50
- Issue
- 2
- Pages
- 92-100
- ISSN
- 0964-2633 (ISSN-L)
- 1365-2788
- OpenAlex
- W2058335997
- Cited by
- 107
- References
- 41
- Citations by year
- 2025: 1 · 2024: 3 · 2023: 2 · 2022: 1 · 2021: 4 · 2020: 3 · 2019: 5 · 2018: 4 · 2017: 10 · 2016: 6 · 2015: 10 · 2014: 11 · 2013: 3 · 2012: 7
- FWCI
- 2.89
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Journal of Intellectual Disability Research (publishedVersion)
- PubMed (publishedVersion)
- UCL Discovery (University College London) (submittedVersion)
- Research Portal (King's College London) (submittedVersion)
- Funders
- Great Ormond Street Hospital for Children
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic Syndromes and Imprinting
- Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Keywords
- Prader-Willi syndrome, autism spectrum disorder, repetitive behavior, ritual behavior, insistence on sameness, behavioral phenotypes, food-related behaviour
- MeSH terms
- Adolescent, Autistic Disorder, Ceremonial Behavior, Child, Child, Preschool, Female, Humans, Male, Personality Assessment, Prader-Willi Syndrome, Stereotyped Behavior
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