Rigidity in routines and the development of resistance to change in individuals with Prader-Willi syndrome

E.L. Haig, K.A. Woodcock

Research output: Contribution to journalArticle

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Abstract

Background
Individuals with Prader-Willi syndrome (PWS) commonly show debilitating resistance to change, which has been linked to cognitive deficits in task switching. Anecdotal reports suggest that exposure to flexibility in routines during development may be beneficial for limiting subsequent resistance to change in people with PWS; which is consistent with a beneficial role of such exposure on the development of task switching, highlighted in typical children. Here we aim to investigate the development of resistance to change in individuals with PWS and hypothesise that exposure to increased rigidity in routines will be associated with increased subsequent resistance to change.

Method
An author compiled informant report interview and two previously validated questionnaires were administered to the caregivers of 10 individuals with PWS (5-23 years). The interview examined rigidity in routines and resistance to change across life stages defined by easily distinguishable events (before school; during primary school; during secondary school; after school; currently), using open ended and structured yes/no and five-point Likert questions. Open ended data were coded using an author compiled system. Responses from two additional informants; and data from the questionnaires were used to assess inter-informant reliability and concurrent validity of the structured questions.

Results
The validity of the interview was supported by acceptable inter-rater reliability of the open ended coding system; and inter-informant reliability, internal consistency and concurrent validity of structured questions. Descriptive analyses of ratings of behaviour change showed a pattern of increasing resistance to change over the life course for the four oldest individuals, who had all been exposed to substantial rigidity in routines before and during primary school. Furthermore, only one individual – currently in primary school – was exposed to very little rigidity in routines before and during primary school, and he had showed a decrease in resistance to change after entering primary school. Open ended data showed that more individuals currently evidencing little resistance to change had been exposed to parent or self-imposed flexibility in routines, than those currently evidencing substantial resistance to change. However, correlational analyses on rigidity and resistance to change ratings, highlighted the possibility that rigidity during primary school is most relevant for developing resistance to change. Finally, open ended data emphasised an important beneficial role of rigidity in routines for limiting the current challenging behaviour of individuals with high resistance to change.

Conclusion
Since task switching appears to evidence a period of high developmental sensitivity during early primary school years, we propose that this period may represent a critical time when increasing flexibility in the routines of children with PWS could limit the development of resistance to change. However, a careful balance would need to be struck, given the apparent benefit of rigid routines on current behaviour. Further work in this area is much needed.
Original languageEnglish
Pages (from-to)488-500
JournalJournal of intellectual disability research : JIDR
Volume61
Issue number5
Early online date06 Mar 2017
DOIs
Publication statusPublished - May 2017

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Prader-Willi Syndrome
Interviews
Rigidity
Syndrome
Reproducibility of Results
Caregivers
Primary School

Keywords

  • Resistance to change
  • Behavioural flexibility
  • Autism spctrum disorder
  • Prader-Willi syndrome
  • Preference for routine
  • Task switching
  • Executive function

Cite this

@article{286af15056b34cdcbe6c0806818637ac,
title = "Rigidity in routines and the development of resistance to change in individuals with Prader-Willi syndrome",
abstract = "Background Individuals with Prader-Willi syndrome (PWS) commonly show debilitating resistance to change, which has been linked to cognitive deficits in task switching. Anecdotal reports suggest that exposure to flexibility in routines during development may be beneficial for limiting subsequent resistance to change in people with PWS; which is consistent with a beneficial role of such exposure on the development of task switching, highlighted in typical children. Here we aim to investigate the development of resistance to change in individuals with PWS and hypothesise that exposure to increased rigidity in routines will be associated with increased subsequent resistance to change.Method An author compiled informant report interview and two previously validated questionnaires were administered to the caregivers of 10 individuals with PWS (5-23 years). The interview examined rigidity in routines and resistance to change across life stages defined by easily distinguishable events (before school; during primary school; during secondary school; after school; currently), using open ended and structured yes/no and five-point Likert questions. Open ended data were coded using an author compiled system. Responses from two additional informants; and data from the questionnaires were used to assess inter-informant reliability and concurrent validity of the structured questions. Results The validity of the interview was supported by acceptable inter-rater reliability of the open ended coding system; and inter-informant reliability, internal consistency and concurrent validity of structured questions. Descriptive analyses of ratings of behaviour change showed a pattern of increasing resistance to change over the life course for the four oldest individuals, who had all been exposed to substantial rigidity in routines before and during primary school. Furthermore, only one individual – currently in primary school – was exposed to very little rigidity in routines before and during primary school, and he had showed a decrease in resistance to change after entering primary school. Open ended data showed that more individuals currently evidencing little resistance to change had been exposed to parent or self-imposed flexibility in routines, than those currently evidencing substantial resistance to change. However, correlational analyses on rigidity and resistance to change ratings, highlighted the possibility that rigidity during primary school is most relevant for developing resistance to change. Finally, open ended data emphasised an important beneficial role of rigidity in routines for limiting the current challenging behaviour of individuals with high resistance to change.ConclusionSince task switching appears to evidence a period of high developmental sensitivity during early primary school years, we propose that this period may represent a critical time when increasing flexibility in the routines of children with PWS could limit the development of resistance to change. However, a careful balance would need to be struck, given the apparent benefit of rigid routines on current behaviour. Further work in this area is much needed.",
keywords = "Resistance to change, Behavioural flexibility, Autism spctrum disorder, Prader-Willi syndrome, Preference for routine, Task switching, Executive function",
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Rigidity in routines and the development of resistance to change in individuals with Prader-Willi syndrome. / Haig, E.L.; Woodcock, K.A.

In: Journal of intellectual disability research : JIDR, Vol. 61, No. 5, 05.2017, p. 488-500.

Research output: Contribution to journalArticle

TY - JOUR

T1 - Rigidity in routines and the development of resistance to change in individuals with Prader-Willi syndrome

AU - Haig, E.L.

AU - Woodcock, K.A.

PY - 2017/5

Y1 - 2017/5

N2 - Background Individuals with Prader-Willi syndrome (PWS) commonly show debilitating resistance to change, which has been linked to cognitive deficits in task switching. Anecdotal reports suggest that exposure to flexibility in routines during development may be beneficial for limiting subsequent resistance to change in people with PWS; which is consistent with a beneficial role of such exposure on the development of task switching, highlighted in typical children. Here we aim to investigate the development of resistance to change in individuals with PWS and hypothesise that exposure to increased rigidity in routines will be associated with increased subsequent resistance to change.Method An author compiled informant report interview and two previously validated questionnaires were administered to the caregivers of 10 individuals with PWS (5-23 years). The interview examined rigidity in routines and resistance to change across life stages defined by easily distinguishable events (before school; during primary school; during secondary school; after school; currently), using open ended and structured yes/no and five-point Likert questions. Open ended data were coded using an author compiled system. Responses from two additional informants; and data from the questionnaires were used to assess inter-informant reliability and concurrent validity of the structured questions. Results The validity of the interview was supported by acceptable inter-rater reliability of the open ended coding system; and inter-informant reliability, internal consistency and concurrent validity of structured questions. Descriptive analyses of ratings of behaviour change showed a pattern of increasing resistance to change over the life course for the four oldest individuals, who had all been exposed to substantial rigidity in routines before and during primary school. Furthermore, only one individual – currently in primary school – was exposed to very little rigidity in routines before and during primary school, and he had showed a decrease in resistance to change after entering primary school. Open ended data showed that more individuals currently evidencing little resistance to change had been exposed to parent or self-imposed flexibility in routines, than those currently evidencing substantial resistance to change. However, correlational analyses on rigidity and resistance to change ratings, highlighted the possibility that rigidity during primary school is most relevant for developing resistance to change. Finally, open ended data emphasised an important beneficial role of rigidity in routines for limiting the current challenging behaviour of individuals with high resistance to change.ConclusionSince task switching appears to evidence a period of high developmental sensitivity during early primary school years, we propose that this period may represent a critical time when increasing flexibility in the routines of children with PWS could limit the development of resistance to change. However, a careful balance would need to be struck, given the apparent benefit of rigid routines on current behaviour. Further work in this area is much needed.

AB - Background Individuals with Prader-Willi syndrome (PWS) commonly show debilitating resistance to change, which has been linked to cognitive deficits in task switching. Anecdotal reports suggest that exposure to flexibility in routines during development may be beneficial for limiting subsequent resistance to change in people with PWS; which is consistent with a beneficial role of such exposure on the development of task switching, highlighted in typical children. Here we aim to investigate the development of resistance to change in individuals with PWS and hypothesise that exposure to increased rigidity in routines will be associated with increased subsequent resistance to change.Method An author compiled informant report interview and two previously validated questionnaires were administered to the caregivers of 10 individuals with PWS (5-23 years). The interview examined rigidity in routines and resistance to change across life stages defined by easily distinguishable events (before school; during primary school; during secondary school; after school; currently), using open ended and structured yes/no and five-point Likert questions. Open ended data were coded using an author compiled system. Responses from two additional informants; and data from the questionnaires were used to assess inter-informant reliability and concurrent validity of the structured questions. Results The validity of the interview was supported by acceptable inter-rater reliability of the open ended coding system; and inter-informant reliability, internal consistency and concurrent validity of structured questions. Descriptive analyses of ratings of behaviour change showed a pattern of increasing resistance to change over the life course for the four oldest individuals, who had all been exposed to substantial rigidity in routines before and during primary school. Furthermore, only one individual – currently in primary school – was exposed to very little rigidity in routines before and during primary school, and he had showed a decrease in resistance to change after entering primary school. Open ended data showed that more individuals currently evidencing little resistance to change had been exposed to parent or self-imposed flexibility in routines, than those currently evidencing substantial resistance to change. However, correlational analyses on rigidity and resistance to change ratings, highlighted the possibility that rigidity during primary school is most relevant for developing resistance to change. Finally, open ended data emphasised an important beneficial role of rigidity in routines for limiting the current challenging behaviour of individuals with high resistance to change.ConclusionSince task switching appears to evidence a period of high developmental sensitivity during early primary school years, we propose that this period may represent a critical time when increasing flexibility in the routines of children with PWS could limit the development of resistance to change. However, a careful balance would need to be struck, given the apparent benefit of rigid routines on current behaviour. Further work in this area is much needed.

KW - Resistance to change

KW - Behavioural flexibility

KW - Autism spctrum disorder

KW - Prader-Willi syndrome

KW - Preference for routine

KW - Task switching

KW - Executive function

U2 - 10.1111/jir.12368

DO - 10.1111/jir.12368

M3 - Article

VL - 61

SP - 488

EP - 500

JO - Journal of intellectual disability research : JIDR

JF - Journal of intellectual disability research : JIDR

SN - 0964-2633

IS - 5

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