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Journal of Intellectual Disability Research
doi: 10.1111/jir.12199
1061 VOLUME
59 PART 11 pp 1061–1070 NOVEMBER 2015
Routinised and compulsive-like behaviours in individuals with Down syndrome S. Glenn,1 C. Cunningham,1 A. Nananidou,1 V. Prasher2 & P. Glenholmes2 1 School of Natural Sciences and Psychology, Liverpool John Moores University, Liverpool, UK 2 The Greenfields, Birmingham, UK
Abstract Background Increased intensities of routinised and compulsive-like behaviours are seen in those with intellectual disabilities and have sometimes been shown to be associated with worries. We used the Childhood Routines Inventory (CRI, Evans et al., 1997) with two samples of children and adults with Down syndrome: (1) to determine whether routinised and compulsive-like behaviours were associated with mental health problems and (2) to determine the factor structure of the CRI. Method Parents or carers completed the CRI for (1) 125 adults with Down syndrome (aged 18–43 years) who had been assessed for mental health problems; worries and fears were also rated by parents/carers and (2) 206 individuals with Down syndrome (aged 4.5–43 years, with verbal mental ages of 2 years and above). Results (1) People with a psychiatric diagnosis had significantly more worries and fears than those without such a diagnosis, but there was no significant difference in CRI scores. Logistic regression indicated that the fear rating was the only significant predictor of a diagnosis. (2) Exploratory and confirmatory analyses showed a
Correspondence: Prof. Sheila Glenn, School of Natural Sciences and Psychology, Faculty of Science, Liverpool John Moores University, Tom Reilly Building, Byrom Street, Liverpool L3 3AF, UK (e-mail: [email protected]).
three-factor model (Just right, Repetitive behaviour and Clothes sensitivity) to be the best solution. Those with psychiatric diagnoses had significantly higher ratings on the Repetitive behaviour factor. Conclusion Increased levels of routinised and compulsive-like behaviours were shown by individuals with Down syndrome of all ages, were not associated with mental health problems, but were associated with worries and fears. Factor analysis found three factors, two of which (Just right and Repetitive behaviours), were similar to those identified in typically developing samples. This suggests that the behaviours have similar adaptive functions in individuals with developmental delays.
Introduction Routinised and compulsive-like behaviours in children and adults with Down syndrome have much in common with behaviours seen in the typical development of young children (e.g Gesell et al., 1974; Evans et al., 1997, Glenn et al., 2012, Ҫevikaslan et al., 2013). Examples include insistence on the same household routines, wanting aspects of the environment or actions to be ‘just right’ and repeatedly carrying out the same play activities. Leonard et al. (1990) argued that routinised and
© 2015 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
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compulsive-like behaviours are important for reducing anxiety (e.g. in bedtime routines). Piaget (1953) also emphasised the importance of repetition and ‘just right’ behaviours in developing classification skills and learning how to interact with the environment. In other words, routines (and familiarity) for young children increase feelings of competence, reduce anxiety and free up cognitive capacity, all of which foster the likelihood that the individual will explore and learn new ideas and skills. Hurley (1996) argued that in adults with intellectual disabilities (IDs), behaviours usually seen in typical children may be a normal developmental phenomena and should not, by themselves, be interpreted as an abnormal behaviour. Routinised and compulsive-like behaviours could have the same functions for older individuals with limited cognitive capacities as are hypothesised for typically developing children, that is, they are developmentally appropriate rather than pathological. Evans et al. (1997) developed the Childhood Routines Inventory (CRI) to study routinised and compulsive-like behaviours in children. The Inventory, completed by parents or carers, was designed to reflect DSM-IV symptoms for compulsivity, but using normal childhood behaviour. The CRI has been used in many subsequent studies with typically developing children and also a growing number of studies with individuals with developmental delays. Evans and Gray (2000) matched typically developing children for mental age (MA) with individuals with Down syndrome. They found that by a mental age of 6 years, those with Down syndrome showed similar declines in CRI scores to typically developing children. The number of CRI items endorsed was similar, suggesting developmental typicality, but the behaviours were rated as significantly more intense for the children with Down syndrome. Glenn and Cunningham (2007) replicated the study, also including adults with DS. However, they found no decreases with age up to 6 years either in typically developing children or in those with Down syndrome up to verbal mental age (VMA) of 6 years. Nor were there decreases with increasing VMA in adults with Down syndrome. For children with Down syndrome with VMAs greater than 5 years and all adults with
Down syndrome, CRI scores were associated with behaviour problems (as measured by the Child Behavior Checklist (CBCL), Achenbach, 1991). However, Cunningham and Glenn (2008) reanalysed this data and concluded that the raised rating scores seen on the CBCL were predominantly caused by items ‘related to ID’ (p.657), for example, ‘acts young for age’. The CBCL is not standardised for those with ID and gave a misleading impression if only total scores are used. CRI scores were only significantly associated with the worry item of the CBCL, supporting the suggestion that one of the functions of routinised and compulsive-like behaviours may be to reduce anxiety. There have also been some inconsistent results with typically developing children about the age at which CRI scores decline. Evans et al. (1997) demonstrated a significant drop between the ages of 5 and 6 years. Glenn and Cunningham (2007) found no decline with increasing VMA or chronological age (CA) in children with mean VMAs up to 7 years and Tregay et al. (2009) found no significant decline in children aged 3 to 9 years. Zohar and Bruno (1997) examined the level of obsessive–compulsive behaviours (similar in some respects to routinised and compulsive-like behaviours) in children aged 8 to 14 years using the Maudsley Obsessive Compulsive Inventory (Hodgson & Rachman, 1977), and found a significant decrease in such behaviours from 8 to 14 years. Glenn et al. (2012) tested 1137 typically developing children aged 2 to 11 years. There was a significant linear decline in CRI scores with age, with some suggestion of a more significant drop between 7 and 8 years; a minority of children continued to have high levels up to 11 years of age. Anxious behaviours reported by parents were associated with higher ratings on CRI intensity. In addition, studies on typically developing populations (Evans et al., 1997; Glenn et al., 2012; Zohar and Felz, 2001; Ҫevikaslan et al., 2013) have found different factor solutions for the CRI. Thus, we investigated the factor structure of the CRI with individuals with Down syndrome in the present study. There have also been studies with children with other conditions associated with ID. For example, Wigren and Hansen (2003) found significantly more intense repetitive behaviour on the CRI for
© 2015 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
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50 individuals with Prader–Willi syndrome (PWS, aged 5 to 18 years), compared with 50 typically developing children aged 4 years, although there was no matching for MA. Greaves et al. (2006) matched children with PWS and children with autism for age and found similar high scores. Glenn & Egen (2010) found significantly higher intensity CRI scores in primary school children with autism compared with age matched children with Down syndrome. Restrictive and repetitive behaviours are a core feature in autism; the review by Leekham et al. (2011) noted little evidence on change with age in restricted and repetitive behaviours and highlighted the need to study the adaptive functions of such behaviours for children with autism. In relation to possible functions of routinised and compulsive-like behaviours, Rodgers et al. (2012) demonstrated a positive association between anxiety and levels of repetitive behaviours in children with autism. In summary (and in relation to the issue of pathology), the literature reviewed earlier shows: (1) some inconsistency in the factor structure of the CRI and the age at which routinised and compulsive-like behaviours decline in typically developing children, with a minority continuing with high levels up to 11 years of age; (2) no decline with age or MA in individuals with Down syndrome; (3) some evidence of high levels of routinised and compulsive-like behaviours in other diagnostic groups; and (4) positive associations with anxiety in typically developing children and those with developmental disorders. A recent study on mental health carried out with 125 adults with Down syndrome aged from 20 to 40 years Prasher et al. (2012) is pertinent to this question, as participants were rated on the CRI. In Study 1, we report CRI scores for those with and without a psychiatric diagnosis. In Study 2, in order to allow for factor analysis of the CRI in a larger sample of individuals with Down syndrome, we combined study 1 CRI data with those from Glenn & Cunningham (2007), together with unpublished data. Hence, the following questions were addressed: Study 1 •
Are CRI ratings significantly higher in adults with Down syndrome with a psychiatric diagnosis?
•
Are ratings of worries and fears significantly higher in adults with Down syndrome with a psychiatric diagnosis? Study 2
• •
Is there a decline of CRI scores with increasing verbal mental age? What is the factor structure of the CRI with a large sample of individuals with Down syndrome aged from 2 to 11+ years mental age, and how does this compare to previous findings with typically developing populations??
Method Study 1 Ethical scrutiny and approval was obtained from the local National Health Service and University Committees. Details of recruitment of the sample and procedure can be obtained from Prasher et al. (2014). We asked all the General practitioners within the study area to identify individuals with Down syndrome aged 18 to 45 years; of the 167 individuals and their families/carers contacted, 130 agreed to participate in the study (77.8%).
Participants One hundred and twenty five participants (62 men and 63 women) had ratings on the CRI. Their mean age was 30 years 5 months (range 18 to 43 years, SD = 7.0 years). Socio-economic status (SES) was assessed by calculating a multiple deprivation index from post codes Noble et al. (2007) and was shown to be representative of the catchment area, as was ethnicity (80% were Caucasian, 17% Asian and 3% Black).
Measures British vocabulary scale Dunn et al. (1997) The British Picture Vocabulary Scale (BPVS) was used to assess VMA. Thirty-four participants refused to participate in this assessment. The mean VMA level (N = 91) was 4 years 5 months (range 12 months to 13 years 6 months).
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Psychiatric assessment
Study 1
The diagnosis was based on the ICD-10 system of classification (Organisation, 1992) and assessed the present mental health of the person. Thirty-one individuals had a concurrent psychiatric diagnosis.
•
The childhood routines inventory (Evans et al., 1997) The CRI consists of 19 behaviours that the parent rates from 1 (not present) to 5 (very common). Evans et al. reported a Cronbach’s 003B1 of 0.89 and identified two factors: ‘just right’ behaviours (6 items), for example, ‘prefers to have things done in a particular order or certain way’ and ‘repetitive behaviours’ (5 items), for example, ‘repeats certain actions over and over’. The CRI produces two scores: the total number of items (rated between 2 and 5) and the mean intensity of the items. The instructions on the inventory were changed from ‘child’ to ‘person’; items were otherwise identical to the original CRI.
Strengths and difficulties questionnaire (Goodman, 1997) The Strengths and Difficulties Questionnaire (SDQ) has 25 items divided into 5 domains including emotional symptoms; the emotional items ‘Many worries, often appears worried’ and ‘Many fears, easily scared’ were used as measures of worries and fears. Scores used were as follows: 1 (not true), 2 (somewhat true) and 3 (certainly true).
Procedure All participants received a psychiatric assessment from a psychiatrist, a specialist in ID as well as Down syndrome. Independent of the psychiatric assessment, other assessments (including interviews on lifestyle with people with Down syndrome) were carried out in the home by a research nurse.
Are CRI levels higher in adults with Down syndrome with a psychiatric diagnosis?
Two out of three adults who had received a formal diagnosis of Obsessive Compulsive Disorder (OCD) had CRI assessments; they had total intensity scores of 78 and 66. Given the similarity between OCD and CRI items, their data were omitted from the comparison of those with and without a psychiatric diagnosis. There were no significant differences between those with (n = 29) and without (n = 94) a current psychiatric diagnosis for total intensity and total number of CRI items (means 57.1, SD 18.2 and 51.6 SD 17.9, t121 = 1.45, P = .149, respectively for CRI total; means 12.1 SD 4.3 and 10.6 SD 5.2, t121 = 1.34, P = .182 for number of items). •
Are ratings of worries and fears significantly higher in adults with Down syndrome with a psychiatric diagnosis?
Those with a psychiatric diagnosis had significantly higher ratings on worries and fears than those without (t121 = 2.41, P = .017, and t121 = 3.50, P = .001, respectively). Fear and worries were significantly related (r = 0.33, P = .000). Logistic Binary Regression showed that it was the fear score that was the significant predictor of mental health problem diagnosis (P = .010), not worries or CRI intensity. There were no changes with CA; the correlation between total CRI and CA was 0.04 and between number and CA was 0.021. There were no changes with VMA (n = 91); the correlation between total CRI and VMA was 0.065 and between number and VMA was 0.01.
Study 2 properties of the childhood routine inventory with the larger sample Two analyses were carried out: first, looking at change with VMA on CRI scores and second, a factor analysis of the CRI.
Results Childhood routines inventory data satisfied the assumptions for parametric tests (Kolmogorov–Smirnov statistics were as follows: 0.060, P = .20 Study 1 and .055, P = .20 Study 2).
Participants Participants from Study 1 with CRI ratings were included, if they had VMAs of 2 years and over, in order to compare to previous studies with typically
© 2015 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
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developing children. An additional 20 participants were included who had refused to be tested on the BPVS in Study 1, but who had participated in an interview about their lifestyle, and were therefore judged to have a VMA of at least 2 years. This gave a total of 97 participants from Study 1. A further 109 participants with Down syndrome were included for whom there were data on both VMA and the CRI; 85 of these were from Glenn & Cunningham (2007) and 24 were previously unpublished. This provided a total of 206 participants. •
Is there a decline of CRI scores with increasing VMA?
Mental age difference analysis For those with VMA scores, there were 186 participants with Down syndrome – mean VMA was 4.8 years, range 2 to 13.5 years, SD 2.3 years. •
•
A 2-way ANOVA (n = 186) on intensity of CRI with two factors of VMA (divided into seven 1-year categories) and gender found no significant main or interaction effects (categories: F6,172 = 1.03, P = .403; gender: F1,172 = .244, P = .622; interaction: F6,172 = .715, P = .638). Inspection of Table 1 shows no clear linear change with VMA. The lowest intensities were seen at 5 to 6 and 8+ years. What is the factor structure of the CRI with a large sample (n = 206) of individuals with Down syndrome from 2 to 11+ years MA, and how does this
Table 1 Mean intensity and number of childhood routine inventory (CRI) across seven verbal mental age (VMA) categories
VMA categories 24–36 months 37–48 months 49–60 months 61–72 months 73–84 months 85–96 months 97+ months Total
N 36 30 32 27 22 22 17 186
Total intensity (SD) CRI
Number (SD) CRI
54.8 (18.04) 55.5 (16.86) 51.19 (15.39) 48.19 (16.97) 55.09 (19.27) 52.5 (18.63) 47.65 (12.13) 52.44 (17.00)
12.4 (5.01) 13.4 (4.75) 11.9 (4.06) 11.0 (4.81) 13.0 (5.4) 12.4 (4.8) 11.6 (4.36) 12.3 (4.7)
compare to previous findings with typically developing populations? First, the factor models produced by Evans et al. (1997; 11 items)1 and Glenn et al. (2012; 16 items)2 were tested in the present dataset. As recommended, a 1-factor model was also tested. Confirmatory Factor Analysis (CFA) with Amos Maximum Likelihood procedure (Arbuckle, 2006) was used. As seen on Table 2, none of the three models had acceptable fit indices. To identify the factor structure for the current sample, exploratory Principal Component Analysis (PCA) with both Varimax and Oblique (delta = 0) rotation was run for the 19 items of the CRI and the 206 participants, satisfying the 5:1 case to ratio requirement (Bryant & Yarnold, 1995) . Hayton et al. (2004) and Lance et al. (2006) reviewed evidence on accuracy of factor retention criteria, and concluded that Monte Carlo parallel analysis is the most accurate criterion compared with the rest. As seen in Table 3, parallel analysis suggests the extraction of the three factors. Principal component analysis with Varimax and Direct Oblimin rotation were conducted specifying three factors. As Factor 1 and Factor 2 were moderately correlated (0.456), the oblique solution was more appropriate (Tabachnick & Fidell, 2007). Items that loaded .5; 16 items)
152 43 101 118
465.8*** 148.4*** 295.48*** 303.97***
Parsimony adjusted GFI CFI NFI measures
RMSEA
.799 .877 .842 .856
.100 .109 .097 .087
.764 .862 .809 .843
.690 .818 .740 .770
PNFI = .613PCFI = .679 PNFI = .640PCFI = .674 PNFI = .623PCFI = .681 PNFI = .668PCFI = .732
101 177.78*** .904 .927 .849 PNFI = .714PCFI = .681 .061
Note: ***P < .001 GFI, goodness-of-fit index; CFI, comparative fix index; NFI, normed fit index; PNFI, parsimony normed fix index; RMSEA, root mean square of error approximation; PCA, principal component analysis.
Table 3 Monte Carlo parallel analysis
95th Mean Percentile Component eigenvalue eigenvalues
Current dataset extracted eigenvalues
1 2 3 4
6.577 1.573 1.382 1.119
1.561549 1.459520 1.380883 1.303721
1.651184 1.535199 1.449821 1.353944
index (PCFI)] fit indices showed improved fit and were within the recommended thresholds. As the chi-square statistic did not show a good fit, several other indices, less dependent on sample size, were used. Good to excellent fit was suggested by the Parsimony Adjusted Measures (PNFI = .714 and PCFI = .681; both over .6), the root mean square of error approximation (RMSEA = .061; excellent fit as it is 02264.06), CMIN/DF (=1.77; good fit if between 1 and 2). The normed fit index (NFI) tends to underestimate fit for samples close to 200 cases (Bentler, 1990 and Mulaik et al., 1989), which may explain why it does not meet the .9 threshold. In contrast, comparative fix index (CFI), which takes into account sample size and works well with smaller samples (Tabachnick & Fidell, 2007), was over the 9 threshold. Good fit is suggested by goodness-of-fit index (GFI = .9; meeting the recommended >.9 cut off). A schematic representation of the CFA, with the regression weights, is shown in Fig. 1.
We, therefore, retained the 3-factor solution for further analysis (Table 4). The composite scores on the three factors (Just right: JR – 8 items; Repetitive: Rep – 6 items, and Clothes sensitivity: CS – 2 items) were then compared. There were no significant differences between the factors (F2,410 = 1.59, P = .205), nor were there any significant changes with VMA. However, those with diagnosed mental health problems had significantly higher intensity repetitive scores than those without a diagnosis (means 2.9, SD 1.1, 2.4, SD 1.0, respectively; t121 = 2.4, P = .018). Just Right and Clothes Sensitivity were not significantly different (t121 = .198, P = .843, and t121 = 1.37, P = .173).
Discussion Study 1 As expected, the intensity of routinised and compulsive–like behaviours was higher in individuals with Down syndrome, than those reported in studies of typically developing children (e.g. Glenn & Cunningham, 2007; Glenn et al., 2012). We were interested in whether the levels were significantly higher in adults with Down syndrome with a psychiatric diagnosis. Although higher, CRI scores were not significantly different. The reason for the slightly higher CRI scores was probably the significantly higher levels of worries and fears found in the psychiatric group. A logistic regression indicated that the fear score was the only significant predictor of
© 2015 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd
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Figure 1 Confirmatory factor analysis 3-factor model with standardised regression weights and factor correlations.
Table 4 Pattern matrix of exploratory principal component analysis with direct oblimin rotation
Factors
Just right
Repetitive
Clothes sensitivity
Variance explained 1 Prefer to have things done in a particular way/perfectionistorder or certain way, i.e. Is he/she a ‘perfectionist’ 3 Seem very concerned with dirt, cleanliness or neatness. 4 Arrange objects, or perform certain behaviour until they seem ‘just right’ tm/he 6 Line up objects in straight lines or symmetrical patterns 16 Seem very aware of certain details at home 9 Insist on having certain belongings around the house ‘in their place’ 19 Prepare for bedtime by engaging in a special activity or routine 17 Strongly prefer to stick to one game or activity rather than change to new one 15 Collect or store objects 7 Prefer the same household schedule or routine every day 2 Seem very attached to one favourite object 10 Repeat actions over and over 8 Act out the same thing over and over in pretend play 11 Have strong preferences for certain foods 18 Make requests/excuses that would enable him/her to postpone bedtime 5 Have persistent habits 12 Like to eat food in a particular way 14 Have a strong preference for wearing or not wearing certain clothes 13 Seem very aware of, or sensitive to, how certain clothes feel Cronbach’s alpha
34.6% 842
8.28%
7.28%
.792 .749 .682 .630 .568 .550 .518
.757 .678 .606 .606 .606 .566
.84
.75
.734 .707 .72
Factor loadings .5 on any factors.
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a psychiatric diagnosis, and not the CRI scores. This suggests that routinised and compulsive-like behaviours, as measured by the CRI, are not pathological, but rather have some adaptive functions in the lives of these adults. We then combined results from two studies in order to carry out a factor analysis of the CRI with a larger sample of 206 individuals with Down syndrome, aged 2–43 years. The best solution was a 3factor solution, with two factors that could be labelled ‘just right’ (items3 1, 3, 4, 6, 9, 16 and 19) and ‘repetitive’ (items 5, 8, 10, 11, 12 and 18), similar but not identical to Evans’ original factor structure. A third factor labelled ‘clothes sensitivity’ consisted of 2 items (13 and 14). All previous studies have found slightly different factor solutions. One might expect that this might be the case when comparing the factor structure of scales for individuals with developmental disabilities (e.g. Haynes et al., 2013). However, the studies on typically developing populations (Evans et al., 1997; Zohar & Felz, 2001; Glenn et al., 2012; Ҫevikaslan et al. 2013) have also found different factor solutions. Some of the differences may be related to the sample sizes as both the present study, and the Zohar and Feltz study, had small sample sizes compared with the others. Furthermore, the inclusion of older age groups including adults in the present study might also have affected the results, although we found no trends with CA. In spite of some item differences however, all research produced factors could be labelled ‘just right’ and ‘repetitive’. Thus, the present study confirms the validity of these concepts for individuals with Down syndrome as well as for typically developing children.
Comparison of the three factors There were no significant differences in the intensities or numbers endorsed of the items composing the three factors, nor a change with VMA. Routinised and compulsive-like behaviours did not change in intensity with CA or MA, and this suggests that they continue to be functional well into adult life for individuals with Down syndrome.
3
Items identical to Evans et al. (1997 in italics)
However, those with a psychiatric diagnosis were rated to have significantly higher scores on the repetitive factor than those without, whereas scores for the just right and clothes sensitivity factors were not significantly different.
Functions of routinized and compulsive-like behaviours (RCB) The lack of a significant difference in total CRI intensity and number between those with and without mental health problems confirms previous suggestions that routinised and compulsive-like behaviours are not pathological in children and adults with Down syndrome, rather they are part of typical developmental processes. The significantly higher ratings for the repetitive factor in those with mental health problems probably relates to the significantly higher levels of worries and fears found in that group. The fact that the just right and sensitive factors were not associated indicates that it is repetitive behaviour that may be used in an attempt to reduce fear. Reduction of anxiety levels (produced by fear of situations) is a hypothesised function of routinised and compulsive-like behaviours (as is also found in OCDs) and is reported in other developmental disorders. For example, Crespi (2013) argued that much of behaviour that characterises autism spectrum disorders can be seen as longer retention of typical developmental behaviours: ‘To the extent that restricted interests and repetitive behavior exhibit normative adaptive behavioral functions, such as reducing anxiety, decreasing arousal, simplifying complex situations, and fostering a sense of control, extension of their age-dependent expression may reflect extension of the conditions favoring such behavior. ’(P.3). Joosten et al. (2012) investigating contextual influences on repetitive behaviours with children with autism found that the children were more likely to display such behaviours during times of transition where a task was finishing or the child was being instructed in a new task. They also showed more signs of anxiety during transitions. High ratings on the ‘just right’ factor indicate that order and feeling control over the environment are still important for individuals with Down syndrome irrespective of age. This insistence on ‘sameness’ may have a positive function by compensating for ID. For
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people who have some difficulties making sense of the world and thinking quickly and flexibly, it takes less effort and cognitive capacity if things stay much the same. Before attempting to change routinised and compulsive-like behaviours in older children and adults with Down syndrome, it is important to consider their developmental appropriateness. The behaviours are on a continuum from appropriate to pathological (for example, as seen in stereotyped behaviour), depending on the context, frequency and intensity. If they are interfering with everyday functioning, assessment of antecedents is needed, and intervention directed towards possible causes. One limitation of the study is that it was based on parental reports rather than direct and independent observation. This raises the possibility of bias, as parents may view behaviour as problematic in older children or adults, although acceptable in younger children. Thus, some caution is needed in forming conclusions. Further research could employ the methods of Joosten et al. (2012) to observe individuals in real life settings.
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repetitive behaviour in children diagnosed with intellectual disability with and without autism. Journal of Applied Research in Intellectual Disabilities 25, 262–70. Lance C. E., Butts M. M., & Michels L. C. (2006). The sources of four commonly reported cutoff criteria: what did they really say? Organizational Research Methods 9, 202–20. Leekham S.R., Prior M.R., & Uljarevic M. (2011). Restrictive and repetitive behaviours in autism spectrum disorders: a review of research in the last decade. Psychological Bulletin 137, 562–93. Leonard H. L., Goldberger B. A., Rapoport J. L., Cheslow D. L., & Swedo S. E. (1990) Childhood rituals: normal development or obsessive-compulsive symptoms?. Journal of the American Academy of Child and Adolescent Psychiatry 29, 17–23. Mulaik S.A., James L.R., Van Alstine J., Bennet N., Lind S., & Stilwell C.D. (1989) Evaluation of goodness-of-fit indices for structural equation models. Psychological Bulletin 105, 430–45. Noble M., McLennan D., Wilkinson K., Whitworth A., Exley S. & Barnes H. (2007) The English Indices of Deprivation 2007. Department of Communities and Local Government, London: HMSO. Piaget J. (1953) The Origins of Intelligence in Children. London: Routledge & Kegan Paul. Prasher V., Glenn S., Cunningham C. & Glenholmes P. (2012) Survey of Well Being and Access to Services in 18 to 43 Year old Adults with Down Syndrome. Final report to the Down’s Syndrome Association, UK.
Prasher V, Glenn S, Cunningham C, Glenholmes P, Arshad H & Kirby A (2014). Health morbidity and access to services by young adults with Down syndrome. International Journal of Developmental Disabilities 60, 26–34. Rodgers J., Riby D.M., Janes E., Connolly B. & McConachie H. (2012) Anxiety and repetitive behaviours in autism spectrum disorders and Williams syndrome: a cross-syndrome comparison. Journal of Autism and Developmental Disorders 42, 175–180. Tabachnick B.G. & Fidell L.S. (2007) Using Multivariate th Statistics 5 Edition. Pearson Education, Inc. Tregay J., Gilmour J. & Charman T. (2009). Childhood rituals and executive functions. British Journal of Developmental Psychology 27, 283–96. World Health Organisation (1992) The ICD-10 Classification of Mental and Behavioural Disorders: clinical descriptions and diagnostic guidelines.Geneva. Wigren M., & Hansen S. (2003) Rituals and compulsivity in Prader–Willi syndrome: profile and stability. Journal of Intellectual Disability Research 47, 428–38. Zohar A. H., & Bruno R. (1997) Normative and pathological obsessive-compulsive behavior and ideation in childhood: a question of timing. Journal of Child Psychology and Psychiatry 38, 993–9. Zohar A. H., & Felz L. (2001) Ritualistic behavior in young children. Journal of Abnormal Child Psychology 29, 121–28.
Accepted 14 April 2015
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Routinised and compulsive-like behaviours in individuals with Down syndrome S. Glenn,1 C. Cunningham,1 A. Nananidou,1 V. Prasher2 & P. Glenholmes2 1 School of Natural Sciences and Psychology, Liverpool John Moores University, Liverpool, UK 2 The Greenfields, Birmingham, UK
Abstract Background Increased intensities of routinised and compulsive-like behaviours are seen in those with intellectual disabilities and have sometimes been shown to be associated with worries. We used the Childhood Routines Inventory (CRI, Evans et al., 1997) with two samples of children and adults with Down syndrome: (1) to determine whether routinised and compulsive-like behaviours were associated with mental health problems and (2) to determine the factor structure of the CRI. Method Parents or carers completed the CRI for (1) 125 adults with Down syndrome (aged 18–43 years) who had been assessed for mental health problems; worries and fears were also rated by parents/carers and (2) 206 individuals with Down syndrome (aged 4.5–43 years, with verbal mental ages of 2 years and above). Results (1) People with a psychiatric diagnosis had significantly more worries and fears than those without such a diagnosis, but there was no significant difference in CRI scores. Logistic regression indicated that the fear rating was the only significant predictor of a diagnosis. (2) Exploratory and confirmatory analyses showed a
Correspondence: Prof. Sheila Glenn, School of Natural Sciences and Psychology, Faculty of Science, Liverpool John Moores University, Tom Reilly Building, Byrom Street, Liverpool L3 3AF, UK (e-mail: [email protected]).
three-factor model (Just right, Repetitive behaviour and Clothes sensitivity) to be the best solution. Those with psychiatric diagnoses had significantly higher ratings on the Repetitive behaviour factor. Conclusion Increased levels of routinised and compulsive-like behaviours were shown by individuals with Down syndrome of all ages, were not associated with mental health problems, but were associated with worries and fears. Factor analysis found three factors, two of which (Just right and Repetitive behaviours), were similar to those identified in typically developing samples. This suggests that the behaviours have similar adaptive functions in individuals with developmental delays.
Introduction Routinised and compulsive-like behaviours in children and adults with Down syndrome have much in common with behaviours seen in the typical development of young children (e.g Gesell et al., 1974; Evans et al., 1997, Glenn et al., 2012, Ҫevikaslan et al., 2013). Examples include insistence on the same household routines, wanting aspects of the environment or actions to be ‘just right’ and repeatedly carrying out the same play activities. Leonard et al. (1990) argued that routinised and
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compulsive-like behaviours are important for reducing anxiety (e.g. in bedtime routines). Piaget (1953) also emphasised the importance of repetition and ‘just right’ behaviours in developing classification skills and learning how to interact with the environment. In other words, routines (and familiarity) for young children increase feelings of competence, reduce anxiety and free up cognitive capacity, all of which foster the likelihood that the individual will explore and learn new ideas and skills. Hurley (1996) argued that in adults with intellectual disabilities (IDs), behaviours usually seen in typical children may be a normal developmental phenomena and should not, by themselves, be interpreted as an abnormal behaviour. Routinised and compulsive-like behaviours could have the same functions for older individuals with limited cognitive capacities as are hypothesised for typically developing children, that is, they are developmentally appropriate rather than pathological. Evans et al. (1997) developed the Childhood Routines Inventory (CRI) to study routinised and compulsive-like behaviours in children. The Inventory, completed by parents or carers, was designed to reflect DSM-IV symptoms for compulsivity, but using normal childhood behaviour. The CRI has been used in many subsequent studies with typically developing children and also a growing number of studies with individuals with developmental delays. Evans and Gray (2000) matched typically developing children for mental age (MA) with individuals with Down syndrome. They found that by a mental age of 6 years, those with Down syndrome showed similar declines in CRI scores to typically developing children. The number of CRI items endorsed was similar, suggesting developmental typicality, but the behaviours were rated as significantly more intense for the children with Down syndrome. Glenn and Cunningham (2007) replicated the study, also including adults with DS. However, they found no decreases with age up to 6 years either in typically developing children or in those with Down syndrome up to verbal mental age (VMA) of 6 years. Nor were there decreases with increasing VMA in adults with Down syndrome. For children with Down syndrome with VMAs greater than 5 years and all adults with
Down syndrome, CRI scores were associated with behaviour problems (as measured by the Child Behavior Checklist (CBCL), Achenbach, 1991). However, Cunningham and Glenn (2008) reanalysed this data and concluded that the raised rating scores seen on the CBCL were predominantly caused by items ‘related to ID’ (p.657), for example, ‘acts young for age’. The CBCL is not standardised for those with ID and gave a misleading impression if only total scores are used. CRI scores were only significantly associated with the worry item of the CBCL, supporting the suggestion that one of the functions of routinised and compulsive-like behaviours may be to reduce anxiety. There have also been some inconsistent results with typically developing children about the age at which CRI scores decline. Evans et al. (1997) demonstrated a significant drop between the ages of 5 and 6 years. Glenn and Cunningham (2007) found no decline with increasing VMA or chronological age (CA) in children with mean VMAs up to 7 years and Tregay et al. (2009) found no significant decline in children aged 3 to 9 years. Zohar and Bruno (1997) examined the level of obsessive–compulsive behaviours (similar in some respects to routinised and compulsive-like behaviours) in children aged 8 to 14 years using the Maudsley Obsessive Compulsive Inventory (Hodgson & Rachman, 1977), and found a significant decrease in such behaviours from 8 to 14 years. Glenn et al. (2012) tested 1137 typically developing children aged 2 to 11 years. There was a significant linear decline in CRI scores with age, with some suggestion of a more significant drop between 7 and 8 years; a minority of children continued to have high levels up to 11 years of age. Anxious behaviours reported by parents were associated with higher ratings on CRI intensity. In addition, studies on typically developing populations (Evans et al., 1997; Glenn et al., 2012; Zohar and Felz, 2001; Ҫevikaslan et al., 2013) have found different factor solutions for the CRI. Thus, we investigated the factor structure of the CRI with individuals with Down syndrome in the present study. There have also been studies with children with other conditions associated with ID. For example, Wigren and Hansen (2003) found significantly more intense repetitive behaviour on the CRI for
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50 individuals with Prader–Willi syndrome (PWS, aged 5 to 18 years), compared with 50 typically developing children aged 4 years, although there was no matching for MA. Greaves et al. (2006) matched children with PWS and children with autism for age and found similar high scores. Glenn & Egen (2010) found significantly higher intensity CRI scores in primary school children with autism compared with age matched children with Down syndrome. Restrictive and repetitive behaviours are a core feature in autism; the review by Leekham et al. (2011) noted little evidence on change with age in restricted and repetitive behaviours and highlighted the need to study the adaptive functions of such behaviours for children with autism. In relation to possible functions of routinised and compulsive-like behaviours, Rodgers et al. (2012) demonstrated a positive association between anxiety and levels of repetitive behaviours in children with autism. In summary (and in relation to the issue of pathology), the literature reviewed earlier shows: (1) some inconsistency in the factor structure of the CRI and the age at which routinised and compulsive-like behaviours decline in typically developing children, with a minority continuing with high levels up to 11 years of age; (2) no decline with age or MA in individuals with Down syndrome; (3) some evidence of high levels of routinised and compulsive-like behaviours in other diagnostic groups; and (4) positive associations with anxiety in typically developing children and those with developmental disorders. A recent study on mental health carried out with 125 adults with Down syndrome aged from 20 to 40 years Prasher et al. (2012) is pertinent to this question, as participants were rated on the CRI. In Study 1, we report CRI scores for those with and without a psychiatric diagnosis. In Study 2, in order to allow for factor analysis of the CRI in a larger sample of individuals with Down syndrome, we combined study 1 CRI data with those from Glenn & Cunningham (2007), together with unpublished data. Hence, the following questions were addressed: Study 1 •
Are CRI ratings significantly higher in adults with Down syndrome with a psychiatric diagnosis?
•
Are ratings of worries and fears significantly higher in adults with Down syndrome with a psychiatric diagnosis? Study 2
• •
Is there a decline of CRI scores with increasing verbal mental age? What is the factor structure of the CRI with a large sample of individuals with Down syndrome aged from 2 to 11+ years mental age, and how does this compare to previous findings with typically developing populations??
Method Study 1 Ethical scrutiny and approval was obtained from the local National Health Service and University Committees. Details of recruitment of the sample and procedure can be obtained from Prasher et al. (2014). We asked all the General practitioners within the study area to identify individuals with Down syndrome aged 18 to 45 years; of the 167 individuals and their families/carers contacted, 130 agreed to participate in the study (77.8%).
Participants One hundred and twenty five participants (62 men and 63 women) had ratings on the CRI. Their mean age was 30 years 5 months (range 18 to 43 years, SD = 7.0 years). Socio-economic status (SES) was assessed by calculating a multiple deprivation index from post codes Noble et al. (2007) and was shown to be representative of the catchment area, as was ethnicity (80% were Caucasian, 17% Asian and 3% Black).
Measures British vocabulary scale Dunn et al. (1997) The British Picture Vocabulary Scale (BPVS) was used to assess VMA. Thirty-four participants refused to participate in this assessment. The mean VMA level (N = 91) was 4 years 5 months (range 12 months to 13 years 6 months).
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Psychiatric assessment
Study 1
The diagnosis was based on the ICD-10 system of classification (Organisation, 1992) and assessed the present mental health of the person. Thirty-one individuals had a concurrent psychiatric diagnosis.
•
The childhood routines inventory (Evans et al., 1997) The CRI consists of 19 behaviours that the parent rates from 1 (not present) to 5 (very common). Evans et al. reported a Cronbach’s 003B1 of 0.89 and identified two factors: ‘just right’ behaviours (6 items), for example, ‘prefers to have things done in a particular order or certain way’ and ‘repetitive behaviours’ (5 items), for example, ‘repeats certain actions over and over’. The CRI produces two scores: the total number of items (rated between 2 and 5) and the mean intensity of the items. The instructions on the inventory were changed from ‘child’ to ‘person’; items were otherwise identical to the original CRI.
Strengths and difficulties questionnaire (Goodman, 1997) The Strengths and Difficulties Questionnaire (SDQ) has 25 items divided into 5 domains including emotional symptoms; the emotional items ‘Many worries, often appears worried’ and ‘Many fears, easily scared’ were used as measures of worries and fears. Scores used were as follows: 1 (not true), 2 (somewhat true) and 3 (certainly true).
Procedure All participants received a psychiatric assessment from a psychiatrist, a specialist in ID as well as Down syndrome. Independent of the psychiatric assessment, other assessments (including interviews on lifestyle with people with Down syndrome) were carried out in the home by a research nurse.
Are CRI levels higher in adults with Down syndrome with a psychiatric diagnosis?
Two out of three adults who had received a formal diagnosis of Obsessive Compulsive Disorder (OCD) had CRI assessments; they had total intensity scores of 78 and 66. Given the similarity between OCD and CRI items, their data were omitted from the comparison of those with and without a psychiatric diagnosis. There were no significant differences between those with (n = 29) and without (n = 94) a current psychiatric diagnosis for total intensity and total number of CRI items (means 57.1, SD 18.2 and 51.6 SD 17.9, t121 = 1.45, P = .149, respectively for CRI total; means 12.1 SD 4.3 and 10.6 SD 5.2, t121 = 1.34, P = .182 for number of items). •
Are ratings of worries and fears significantly higher in adults with Down syndrome with a psychiatric diagnosis?
Those with a psychiatric diagnosis had significantly higher ratings on worries and fears than those without (t121 = 2.41, P = .017, and t121 = 3.50, P = .001, respectively). Fear and worries were significantly related (r = 0.33, P = .000). Logistic Binary Regression showed that it was the fear score that was the significant predictor of mental health problem diagnosis (P = .010), not worries or CRI intensity. There were no changes with CA; the correlation between total CRI and CA was 0.04 and between number and CA was 0.021. There were no changes with VMA (n = 91); the correlation between total CRI and VMA was 0.065 and between number and VMA was 0.01.
Study 2 properties of the childhood routine inventory with the larger sample Two analyses were carried out: first, looking at change with VMA on CRI scores and second, a factor analysis of the CRI.
Results Childhood routines inventory data satisfied the assumptions for parametric tests (Kolmogorov–Smirnov statistics were as follows: 0.060, P = .20 Study 1 and .055, P = .20 Study 2).
Participants Participants from Study 1 with CRI ratings were included, if they had VMAs of 2 years and over, in order to compare to previous studies with typically
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developing children. An additional 20 participants were included who had refused to be tested on the BPVS in Study 1, but who had participated in an interview about their lifestyle, and were therefore judged to have a VMA of at least 2 years. This gave a total of 97 participants from Study 1. A further 109 participants with Down syndrome were included for whom there were data on both VMA and the CRI; 85 of these were from Glenn & Cunningham (2007) and 24 were previously unpublished. This provided a total of 206 participants. •
Is there a decline of CRI scores with increasing VMA?
Mental age difference analysis For those with VMA scores, there were 186 participants with Down syndrome – mean VMA was 4.8 years, range 2 to 13.5 years, SD 2.3 years. •
•
A 2-way ANOVA (n = 186) on intensity of CRI with two factors of VMA (divided into seven 1-year categories) and gender found no significant main or interaction effects (categories: F6,172 = 1.03, P = .403; gender: F1,172 = .244, P = .622; interaction: F6,172 = .715, P = .638). Inspection of Table 1 shows no clear linear change with VMA. The lowest intensities were seen at 5 to 6 and 8+ years. What is the factor structure of the CRI with a large sample (n = 206) of individuals with Down syndrome from 2 to 11+ years MA, and how does this
Table 1 Mean intensity and number of childhood routine inventory (CRI) across seven verbal mental age (VMA) categories
VMA categories 24–36 months 37–48 months 49–60 months 61–72 months 73–84 months 85–96 months 97+ months Total
N 36 30 32 27 22 22 17 186
Total intensity (SD) CRI
Number (SD) CRI
54.8 (18.04) 55.5 (16.86) 51.19 (15.39) 48.19 (16.97) 55.09 (19.27) 52.5 (18.63) 47.65 (12.13) 52.44 (17.00)
12.4 (5.01) 13.4 (4.75) 11.9 (4.06) 11.0 (4.81) 13.0 (5.4) 12.4 (4.8) 11.6 (4.36) 12.3 (4.7)
compare to previous findings with typically developing populations? First, the factor models produced by Evans et al. (1997; 11 items)1 and Glenn et al. (2012; 16 items)2 were tested in the present dataset. As recommended, a 1-factor model was also tested. Confirmatory Factor Analysis (CFA) with Amos Maximum Likelihood procedure (Arbuckle, 2006) was used. As seen on Table 2, none of the three models had acceptable fit indices. To identify the factor structure for the current sample, exploratory Principal Component Analysis (PCA) with both Varimax and Oblique (delta = 0) rotation was run for the 19 items of the CRI and the 206 participants, satisfying the 5:1 case to ratio requirement (Bryant & Yarnold, 1995) . Hayton et al. (2004) and Lance et al. (2006) reviewed evidence on accuracy of factor retention criteria, and concluded that Monte Carlo parallel analysis is the most accurate criterion compared with the rest. As seen in Table 3, parallel analysis suggests the extraction of the three factors. Principal component analysis with Varimax and Direct Oblimin rotation were conducted specifying three factors. As Factor 1 and Factor 2 were moderately correlated (0.456), the oblique solution was more appropriate (Tabachnick & Fidell, 2007). Items that loaded .5; 16 items)
152 43 101 118
465.8*** 148.4*** 295.48*** 303.97***
Parsimony adjusted GFI CFI NFI measures
RMSEA
.799 .877 .842 .856
.100 .109 .097 .087
.764 .862 .809 .843
.690 .818 .740 .770
PNFI = .613PCFI = .679 PNFI = .640PCFI = .674 PNFI = .623PCFI = .681 PNFI = .668PCFI = .732
101 177.78*** .904 .927 .849 PNFI = .714PCFI = .681 .061
Note: ***P < .001 GFI, goodness-of-fit index; CFI, comparative fix index; NFI, normed fit index; PNFI, parsimony normed fix index; RMSEA, root mean square of error approximation; PCA, principal component analysis.
Table 3 Monte Carlo parallel analysis
95th Mean Percentile Component eigenvalue eigenvalues
Current dataset extracted eigenvalues
1 2 3 4
6.577 1.573 1.382 1.119
1.561549 1.459520 1.380883 1.303721
1.651184 1.535199 1.449821 1.353944
index (PCFI)] fit indices showed improved fit and were within the recommended thresholds. As the chi-square statistic did not show a good fit, several other indices, less dependent on sample size, were used. Good to excellent fit was suggested by the Parsimony Adjusted Measures (PNFI = .714 and PCFI = .681; both over .6), the root mean square of error approximation (RMSEA = .061; excellent fit as it is 02264.06), CMIN/DF (=1.77; good fit if between 1 and 2). The normed fit index (NFI) tends to underestimate fit for samples close to 200 cases (Bentler, 1990 and Mulaik et al., 1989), which may explain why it does not meet the .9 threshold. In contrast, comparative fix index (CFI), which takes into account sample size and works well with smaller samples (Tabachnick & Fidell, 2007), was over the 9 threshold. Good fit is suggested by goodness-of-fit index (GFI = .9; meeting the recommended >.9 cut off). A schematic representation of the CFA, with the regression weights, is shown in Fig. 1.
We, therefore, retained the 3-factor solution for further analysis (Table 4). The composite scores on the three factors (Just right: JR – 8 items; Repetitive: Rep – 6 items, and Clothes sensitivity: CS – 2 items) were then compared. There were no significant differences between the factors (F2,410 = 1.59, P = .205), nor were there any significant changes with VMA. However, those with diagnosed mental health problems had significantly higher intensity repetitive scores than those without a diagnosis (means 2.9, SD 1.1, 2.4, SD 1.0, respectively; t121 = 2.4, P = .018). Just Right and Clothes Sensitivity were not significantly different (t121 = .198, P = .843, and t121 = 1.37, P = .173).
Discussion Study 1 As expected, the intensity of routinised and compulsive–like behaviours was higher in individuals with Down syndrome, than those reported in studies of typically developing children (e.g. Glenn & Cunningham, 2007; Glenn et al., 2012). We were interested in whether the levels were significantly higher in adults with Down syndrome with a psychiatric diagnosis. Although higher, CRI scores were not significantly different. The reason for the slightly higher CRI scores was probably the significantly higher levels of worries and fears found in the psychiatric group. A logistic regression indicated that the fear score was the only significant predictor of
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Figure 1 Confirmatory factor analysis 3-factor model with standardised regression weights and factor correlations.
Table 4 Pattern matrix of exploratory principal component analysis with direct oblimin rotation
Factors
Just right
Repetitive
Clothes sensitivity
Variance explained 1 Prefer to have things done in a particular way/perfectionistorder or certain way, i.e. Is he/she a ‘perfectionist’ 3 Seem very concerned with dirt, cleanliness or neatness. 4 Arrange objects, or perform certain behaviour until they seem ‘just right’ tm/he 6 Line up objects in straight lines or symmetrical patterns 16 Seem very aware of certain details at home 9 Insist on having certain belongings around the house ‘in their place’ 19 Prepare for bedtime by engaging in a special activity or routine 17 Strongly prefer to stick to one game or activity rather than change to new one 15 Collect or store objects 7 Prefer the same household schedule or routine every day 2 Seem very attached to one favourite object 10 Repeat actions over and over 8 Act out the same thing over and over in pretend play 11 Have strong preferences for certain foods 18 Make requests/excuses that would enable him/her to postpone bedtime 5 Have persistent habits 12 Like to eat food in a particular way 14 Have a strong preference for wearing or not wearing certain clothes 13 Seem very aware of, or sensitive to, how certain clothes feel Cronbach’s alpha
34.6% 842
8.28%
7.28%
.792 .749 .682 .630 .568 .550 .518
.757 .678 .606 .606 .606 .566
.84
.75
.734 .707 .72
Factor loadings .5 on any factors.
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a psychiatric diagnosis, and not the CRI scores. This suggests that routinised and compulsive-like behaviours, as measured by the CRI, are not pathological, but rather have some adaptive functions in the lives of these adults. We then combined results from two studies in order to carry out a factor analysis of the CRI with a larger sample of 206 individuals with Down syndrome, aged 2–43 years. The best solution was a 3factor solution, with two factors that could be labelled ‘just right’ (items3 1, 3, 4, 6, 9, 16 and 19) and ‘repetitive’ (items 5, 8, 10, 11, 12 and 18), similar but not identical to Evans’ original factor structure. A third factor labelled ‘clothes sensitivity’ consisted of 2 items (13 and 14). All previous studies have found slightly different factor solutions. One might expect that this might be the case when comparing the factor structure of scales for individuals with developmental disabilities (e.g. Haynes et al., 2013). However, the studies on typically developing populations (Evans et al., 1997; Zohar & Felz, 2001; Glenn et al., 2012; Ҫevikaslan et al. 2013) have also found different factor solutions. Some of the differences may be related to the sample sizes as both the present study, and the Zohar and Feltz study, had small sample sizes compared with the others. Furthermore, the inclusion of older age groups including adults in the present study might also have affected the results, although we found no trends with CA. In spite of some item differences however, all research produced factors could be labelled ‘just right’ and ‘repetitive’. Thus, the present study confirms the validity of these concepts for individuals with Down syndrome as well as for typically developing children.
Comparison of the three factors There were no significant differences in the intensities or numbers endorsed of the items composing the three factors, nor a change with VMA. Routinised and compulsive-like behaviours did not change in intensity with CA or MA, and this suggests that they continue to be functional well into adult life for individuals with Down syndrome.
3
Items identical to Evans et al. (1997 in italics)
However, those with a psychiatric diagnosis were rated to have significantly higher scores on the repetitive factor than those without, whereas scores for the just right and clothes sensitivity factors were not significantly different.
Functions of routinized and compulsive-like behaviours (RCB) The lack of a significant difference in total CRI intensity and number between those with and without mental health problems confirms previous suggestions that routinised and compulsive-like behaviours are not pathological in children and adults with Down syndrome, rather they are part of typical developmental processes. The significantly higher ratings for the repetitive factor in those with mental health problems probably relates to the significantly higher levels of worries and fears found in that group. The fact that the just right and sensitive factors were not associated indicates that it is repetitive behaviour that may be used in an attempt to reduce fear. Reduction of anxiety levels (produced by fear of situations) is a hypothesised function of routinised and compulsive-like behaviours (as is also found in OCDs) and is reported in other developmental disorders. For example, Crespi (2013) argued that much of behaviour that characterises autism spectrum disorders can be seen as longer retention of typical developmental behaviours: ‘To the extent that restricted interests and repetitive behavior exhibit normative adaptive behavioral functions, such as reducing anxiety, decreasing arousal, simplifying complex situations, and fostering a sense of control, extension of their age-dependent expression may reflect extension of the conditions favoring such behavior. ’(P.3). Joosten et al. (2012) investigating contextual influences on repetitive behaviours with children with autism found that the children were more likely to display such behaviours during times of transition where a task was finishing or the child was being instructed in a new task. They also showed more signs of anxiety during transitions. High ratings on the ‘just right’ factor indicate that order and feeling control over the environment are still important for individuals with Down syndrome irrespective of age. This insistence on ‘sameness’ may have a positive function by compensating for ID. For
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people who have some difficulties making sense of the world and thinking quickly and flexibly, it takes less effort and cognitive capacity if things stay much the same. Before attempting to change routinised and compulsive-like behaviours in older children and adults with Down syndrome, it is important to consider their developmental appropriateness. The behaviours are on a continuum from appropriate to pathological (for example, as seen in stereotyped behaviour), depending on the context, frequency and intensity. If they are interfering with everyday functioning, assessment of antecedents is needed, and intervention directed towards possible causes. One limitation of the study is that it was based on parental reports rather than direct and independent observation. This raises the possibility of bias, as parents may view behaviour as problematic in older children or adults, although acceptable in younger children. Thus, some caution is needed in forming conclusions. Further research could employ the methods of Joosten et al. (2012) to observe individuals in real life settings.
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Accepted 14 April 2015
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