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The sensory-reactivity PROM set: identification of a parent reported outcome measure set for autism spectrum disorder

Journal of Patient-Reported Outcomes volume 5, Article number: 123 (2021) Cite this article

Abstract

Background

Most children with autism spectrum disorder (ASD) suffer from aberrant responses to sensory stimuli that significantly impact the quality of life. To develop sensory interventions, individually tailored outcome measures are crucially needed for the domain of sensory reactivity problems. Here, we describe the identification of relevant sensory themes according to caregivers of children with ASD according to the guidelines for developing a (parent proxy) patient-reported outcome measure set. Subsequently, we identify parallels between these themes and a well-validated and supported PROMIS® portal to facilitate implementation. Interviews with clinicians and focus groups and interviews with parents of children with ASD were used in the initial phase for concept elicitation. Codes and themes were generated by qualitative thematic data analysis on the transcripts and cognitive interviews with different parents were used for revisions. The resulting themes were compared to existing generic PROMIS-item banks and other existing questionnaires.

Results

A total of 11 parent-reported outcomes were identified that could be either classified as directly or indirectly related to sensory reactivity. Directly related themes comprised of: (1) sensory stimulation tolerance and (2) sensitivity to sensory stimuli. Indirectly related themes were: (3) irritable behavior (4) anxiety problems (5) mood problems (6) sleep problems (7) fatigue (8) physical complaints (9) daily functioning and participation (10) routines, structure and dealing with change and (11) problems in social interaction and communication. Seven out of 11 themes could be measured with generic PROMIS item banks. The four remaining outcomes (sensory stimulation tolerance; irritable behaviour; routines, structure and dealing with change; and sensitivity to sensory stimuli) were found suitable to be inventoried by existing PROMs.

Conclusion

The majority of parent-reported problems seemed related to indirect consequences of sensory reactivity, which are suitable to be measured with generic item banks. In sum, we identified a sensory-reactivity PROM (parent-proxy) set consisting of PROMIS® item banks and additional domains that together form a comprehensive and readily available outcome set for sensory reactivity problems in children with ASD.

Background

The majority of children with autism spectrum disorder (ASD) display aberrant responses to sensory experiences compared to their typically developing peers [1]. These responses are commonly referred to as sensory reactivity problems (SRPs) and are estimated to occur in 69–95% of patients with ASD [2, 3]. SRPs often hamper opportunities to participate in daily activities that promote learning [4, 5] and have a significant impact on quality of life for both children and caregivers [6, 7]. Intensive behavioral therapies such as the Early Start Denver Model [8] and Pivotal Response Treatment [9] partly rely on improving SRPs. In addition, novel mechanism-based medications are being developed to ameliorate SRPs through effects on neuronal activity [10,11,12]. Overall, interventions for SRPs are seen as a top priority in the ASD community [13].

To study the effect of existing and future interventions for SRPs, reliable and relevant outcome measures are mandatory, preferably from the patient or parent perspective to assess meaningful effects in daily life. Several existing instruments characterize sensory sensitivity behaviors such as the Sensory Profile [14] and the Evaluation of Sensory Processing [15] that have great utility for diagnostic profiling especially in typically developing populations. For repeated outcome measurements in clinical intervention trials, such in ASD, the questionnaires are rather lengthy and not well suited to detect change [16]. In addition, these questionnaires were mostly developed for typically developing populations and may be less suitable to address specific SRPs in clinical populations. Indeed, sensory reactivity problems in these populations can extend into problematic behavior or affective dysregulation that in turn may lead to some of the core symptoms of ASD [16]. In sum, there is a great need for appropriate outcome measures for treatments targeting SRPs in ASD.

To address this need, we set out to identify a comprehensive set of sensory reactivity related outcomes relevant to ASD. We first followed the steps of developing a (parent proxy) patient reported outcome measure (PROM) to be used as endpoint to establish efficacy in clinical trials [17, 18]. Our aim was to understand concepts that are relevant for patients and their caregivers. Our questions of interest therefore focused on what caregivers find relevant about sensory reactivity difficulties; which aspects have the most impact on their child’s and their own life; and how they would notice improvement when an intervention targeting sensory reactivity would be successful. To inventory these issues, we initiated focus groups and interviews with caregivers of children with ASD and sensory reactivity problems. We then assessed face validity of these concepts against currently existing instruments, which would facilitate their implementation in clinical trials. Therefore, we chose to compare our patient/caregiver relevant concepts to the items from the child and parent-proxy item banks from the Patient-Reported Outcomes Measurement Information System® (PROMIS) [19]. The PROMIS was initiated by the “NIH Roadmap Initiative” and has developed large item banks, based on Item Response Theory (IRT) with the possibility to use Computerized Adaptive Testing (CAT) [20] to develop meaningful, reliable and precise outcome measures which can be used internationally and across disorders. These properties enable easy and direct implementation of PROMIS CATs in clinical trials.

In this report we describe the identification of patient/caregiver relevant concepts and the development of a Sensory Reactivity-PROM set using PROMIS as a first milestone in establishing ecologically valid outcome measures for sensory reactivity.

Methods

Selection of PRO by concept elicitation

Ethical approval was granted by the Medical Ethical Committee of the University Medical Center (UMC) Utrecht. We selected parents/caregivers as respondents to be able to assess children from the age of 5 years of age. Parents of children with ASD were invited via advertisement on the Dutch ASD parent association (NVA) website to attend a focus group or interview over the phone. The inclusion criteria were parents of patients (boys and girls) with a confirmed ASD diagnosis (based on the DSM-IV or DSM-5) varying between 5 and 17 years of age. To obtain a heterogeneous and representative sample, no exclusion criterion based on intellectual functioning of patients and levels of parental education were selected. During the first steps of the qualitative phase, the aims and logistics of the PROM were developed to aid concept elicitation. Concept elicitation is a process by which concepts deemed important to patients and parents (i.e. symptoms as well as the impact of symptoms) emerge spontaneously through open-ended questions in interview settings, for instance in focus groups. First, symptoms of and impact of symptoms on children with ASD and their parents visiting the outpatient Psychiatry department for consultation or participating in scientific research at the ‘Care and Research program Sensory Processing’ between September 2016 and March 2019 (n = 200) were reviewed (case files). Second, structured expert brainstorms with 5 child psychiatrists from the department of Psychiatry of the UMC Utrecht were conducted to gather information from clinicians that worked with children with ASD. Lastly, a focus group (n = 8) and interviews over the phone (n = 10) with caregivers of children with ASD were conducted by following a structured interview guide. These conversations were transcribed and entered into Nvivo once the phone interviews reached data saturation (i.e. the point where no new themes or topics were obtained from further interviews).

A qualitative thematic data analysis [21], based on the method of Boeije [22] was performed on the transcription of phone interviews. A total of 181 codes were generated by DA and scored by an independent second rater (GT) and these were conceptually grouped into 29 (sub)themes. As a result, eleven overarching themes were extracted. Subsequently, these themes were presented to 21 new participants (i.e., other parents of children with ASD) to evaluate whether themes were missing or irrelevant. The eleven themes were confirmed and therefore maintained.

Results

The study population that was interviewed and used for concept elicitation consisted of 38 caregivers of 37 children (age M = 11.5; SD = 3.0) with an ASD diagnosis. The population was balanced with regard to older and younger children (age 5–11: n = 19; age 12–17: n = 18) and boys (n = 19) and girls (n = 18). Intellectual functioning ranged from total intelligence quotient (TIQ) 50 to 145 (M = 98.9; SD = 28.2) and was proportionally divided into children with below average (TIQ 50–84: n = 10), average (TIQ 85–115: n = 10), and above average (TIQ 116–145: n = 8) intellectual functioning. Caregivers were most likely to have followed higher education: 56% completed higher professional education and 11% completed research-oriented education. Vocational education was completed by 30% and 4% had followed pre-vocational education.

The following eleven concepts were identified through concept elicitation: (1) Sensory stimulation tolerance (2) Sensitivity to sensory stimuli (3) Irritable behavior (4) Anxiety (5) Mood problems (6) Sleep problems (7) Fatigue (8) Physical complaints (9) Daily functioning and participation (10) Routines, structure and dealing with change and (11) Problems in social interaction and communication. Thus, the majority of these concepts reflected what we would refer to as ‘indirect’ consequences of altered sensory reactivity. For instance, many caregivers regarded their children’s fatigue and lack of energy to be closely related to aberrant sensory reactivity and to have a profound impact on the quality of life. Another example of indirect consequences were irritable behaviours, especially when their child experienced “sensory overload” leading to anger, temper tantrums, crying, yelling or short-temperedness. Only two themes—Sensory stimulation tolerance and Sensitivity to sensory stimuli—entailed direct behavioral responses to sensory stimuli, e.g., covering their ears, adjusting their daily routines or contacts to avoid sensory stimuli, or finding it difficult to differentiate between relevant and irrelevant stimuli.

PROM selection

Next, we identified which reliable existing PROMs can be used to represent the identified sensory concepts. We chose to use PROMIS item banks, measured by CATs. Seven out of eleven concepts were found to be covered by PROMIS item banks (see Table 1). Importantly, each of these PROMIS item banks are available as parent proxies and pediatric self-report versions. Four concepts were not measurable by PROMIS: Sensory Stimulation Tolerance; Irritable Behaviour; Routines, Structure and Dealing with Change and Sensitivity to Sensory Stimuli. The domain Daily Functioning and Participation was only partly covered by the PROMIS items bank ‘Cognitive function’ and therefore an additional questionnaire would be needed to cover all codes that parents reported. The identification of relevant sensory reactivity PROs and PROMs resulted in a Sensory Reactivity-PROM set.

Table 1 Final sensory reactivity-PROM set
Full size table

The sensory reactivity-PROM set

To measure Sensory stimulation tolerance and Sensitivity to sensory stimuli, we advise to use the following two questionnaires:

Short sensory profile (SSP)

The SSP is a shortened form of Dunn’s SP caregiver questionnaire [14] and contains 38 items, arranged into 7 subscales, aimed at measuring abnormal responses to sensory stimuli [23]. The normative group consists of 697 children from the United States. Both the SP full version as well as the SSP have shown good content validity [24]. It has a reliability of 0.90 and discriminate validity > 95% to identify children with and without sensory processing difficulties [25]. Although the total score is reliable for youth with ASD (α = 0.89), the structural validity of the SSP subscales shows poor fit [26, 27] and some researchers have recommended against the use of the SSP total score due to the measure’s multidimensionality [27]. The raw scores of both subscales and total scores are classified in three groups: typical performance, probable difference, and definite difference. A total score between 38 to 141 is classified as “definite difference” and indicates significant problems with processing sensory stimuli and difficulties in daily life performance which can be viewed as a clinically meaningful threshold.

Sensory Experiences Questionnaire Version 3.0 (SEQ-3.0)

Unlike other instruments that are often used in sensory processing research (e.g., SP-NL [14] and Sensory Processing measure [28], the SEQ-3.0 [29] was developed and standardized in ASD populations. Earlier versions of the SEQ have demonstrated its reliability and validity (version 1: Internal consistency α = 0.80; test–retest reliability total score ICC = 0.92 [30]). Studies about the content validity of the SEQ-3.0 have not been published although the developmental process is known (i.e., the items included were developed from reviews of ASD sensory literature and through a consensus process with a team of experts) [31]. Higher scores indicate greater symptoms, but no clinically meaningful threshold is established yet.

To measure Irritable behavior, we advise to use the following subscale:

Aberrant behavior checklist-irritability (ABC-I)

The ABC measures problematic behavior and contains 58 items organized into 5 subscales [32]. The ABC is developed as a scale to assess treatment effects in people with developmental disabilities (primarily in residential facilities) [32] and has very good internal consistency (α = ranging from low 0.80 s to the middle 0.90 s) and test–retest reliability (mid-0.60 s to highs in the 0.90 s) [33, 34]. Studies have further given psychometric support for the use of the ABC in ASD [35]. The Irritability subscale consists of 15 items and measures agitated/irritable behavior. This subscale has been a primary outcome to measure treatment response in large ASD trials [36,37,38,39] and psychosocial intervention studies [40, 41]. Higher scores indicate greater symptoms. The ABC does not have a clinically meaningful cut-off score, although pharmacological trials that included the Irritability subscale often required a baseline score of ≥ 18 [36, 42, 43].

To measure Routines, structure and dealing with change, we advise to use the following subscale:

Repetitive behavior scale-revised (RBS-R)

The RBS-R is a measure of the presence and severity of restricted and repetitive behaviors and contains 43 items organized into 6 subscales [44]. This refined version of the RBS was initiated after feedback from parents and clinicians and was developed to more appropriately capture the variety of repetitive behaviors in ASD. The subscales Ritualistic Behavior (i.e., performing activities of daily living in a similar manner) and Sameness Behavior (i.e., resistance to change, insisting things stay the same) have been selected. Internal consistency of these subscales in an ASD sample was α = 0.71 and 0.88, respectively [45]. Higher scores indicate more severe problems, but no clinically meaningful threshold has been established.

To measure Daily Functioning and Participation, the following two questionnaires can be used:

Child and adolescent scale of participation (CASP)

The CASP is a 20-items caregiver questionnaire measuring the extent of participation and restriction of children (3–22 years) in home, school and community life situations and activities [46]. The scale was initially developed in children with acquired brain injuries. The questionnaire has shown high internal consistency (Cronbach’s α = 0.98), test–retest reliability (ICC = 0.94) and construct validity [47, 48], and good to excellent content validity [49]. Lower scores indicate a lower extent of participation but a clinically meaningful threshold for ASD has not been established as a study on responsiveness is only available in children with traumatic brain injury [50]. A study with the German CASP version did report reference values for disability-free children, with recommended cut-off scores of < 95 for mildly and < 92 for severely impaired social participation [49].

Participation and environment measure for children and youth (PEM-CY)

The PEM-CY is a 25-items caregiver questionnaire that measures participation across life situations at home, school and community settings in children (5–17 years) with and without disabilities [51]. It has been developed in caregivers of both children with diverse disabilities (with a large proportion diagnosed with ASD) and without disabilities [52]. The questionnaire has both moderate to good internal consistency (Cronbach’s α = 0.59 and above) and test–retest reliability (0.58 and above) [52]. No data on clinically meaningful thresholds is available.

Discussion

This study aimed to derive a valid outcome measure set for sensory reactivity targeting interventions in children with ASD elicited by parent interviews. The parent interviews and focus groups revealed a total of eleven concepts relating to SRPs. We can classify the most frequently mentioned problems as indirect consequences of sensory reactivity, such as fatigue or behavioral irritability. Interestingly, these kinds of symptoms are often recognized as comorbid features, but were here identified by parents as indirect consequences of altered sensory processing functions associated with ASD [16, 53]. In contrast, other behavioral responses were noted that were directly related to the sensory environment such as immediate distress or an exaggerated avoidance to sensory stimuli. In all, we classified nine out of 11 concepts as indirect versus two direct consequences of sensory reactivity. Such a distinction has not yet been explicated in the field [16, 53], but may be important to fully appreciate the effect of SRP targeting treatments.

The majority of the identified concepts (7 out of 11) can be measured with generic item banks provided by PROMIS. It would be recommended to validate these item banks in the target population (children with ASD). This set can be administered by CATs with both parent proxy reports (ages 5–17) and pediatric self-reports (ages 8–17) available. These CATs are an important method to reduce the time to complete questionnaires and to render them more individually tailored. CATs follow decision trees by each time choosing tailored selections based upon previous answers of the respondent, e.g. if a patient is unable to walk then all questions on mobility are deemed irrelevant. CAT algorithms hereby maximize the efficiency of number of questioned items (usually 4 to 12 items in PROMIS) and reduces the burden for respondents whilst allowing more domains to be measured. Another advantage of CAT in clinical trials is that smaller sample sizes are needed to achieve the same statistical power in comparison to conventional instruments. Lastly, there is less floor or ceiling effect with T-scores and different domains can be compared on the same scale. Thus, using PROMIS item banks for ASD is an important way forward since it is more reliable and reduces administration time for respondents and researchers. Hence, they are suitable to be implemented in clinical trials where multiple and repeated assessments are often desirable. In addition, using these generic item banks allow for comparisons between different (rare) disorders, typically developing children as well as comparisons across different countries.

A number of other concepts are not covered by existing PROMIS item banks at present: tolerance and sensitivity to sensory stimuli, behavioral irritability and problems with structure and dealing with change. To assemble an all-encompassing outcome measure set, these outcomes need to be added and (for the time-being) to be measured with other validated PROMs. To this end, we propose to add six additional, existing PROMs to cover the above-mentioned missing outcomes: the ABC-I subscale to assess irritable behavior, the SSP or SEQ-3.0 to assess items in the missing sensory-specific outcomes, the RBS-R Ritualistic Behavior and Sameness Behavior-subscales to address dealing with routine and change and the CASP or PEM-CY to cover the daily functioning and participation outcome. We do acknowledge that some of these measures have been developed more than 40 years ago and may not be in line with regulatory qualification demands in order to be implemented (immediately) in registrational clinical trials. Unfortunately, there are currently only few measures that have been validated and cover the identified relevant concepts. Indeed, a limitation of this study is the extensive variety and number of instruments that are needed to cover all relevant concepts identified in this study, which poses a significant burden on respondents. These results therefore highlight the need for the development of PRO instruments in the ASD population, that capture relevant concepts and have established clinically meaningful thresholds. Ultimately, the goal would be to fully rely on PROMs administered with CAT to comprise all identified concepts in the sensory reactivity domain.

Conclusion

In conclusion, we bring forward a sensory reactivity-PROM set that addresses the main concepts relevant to parents of children with ASD. Through parent concept elicitation, we emphasize the need to measure both direct and indirect consequences of altered sensory reactivity. We hope that this report inspires ASD researchers to implement relevant outcome measures in their clinical trials with instruments that are user-friendly, less time-consuming and measure patient-relevant outcomes relating to sensory reactivity. In the short term, we suggest clinical trials in ASD focusing on SRPs to include this hybrid PROM set of PROMIS items banks and existing questionnaires. For the longer term, we propose to complete the PROM set by transforming conventional scales to reliable PROMs administered by CATs.

Availability of data and materials

The transcripts generated and analyzed during the current study are not publicly available due to privacy reasons of participants but are available from the corresponding author on reasonable request.

Abbreviations

ASD:

Autism spectrum disorder

CAT:

Computerized adaptive testing

DSM-5:

Diagnostic and statistical manual of mental disorders 5th edition

IRT:

Item response theory

PROM:

Patient-reported outcome measure

PROMIS:

Patient-reported outcomes measurement information system

SRPs:

Sensory reactivity problems

References

  1. Little LM, Dean E, Tomchek S, Dunn W (2018) Sensory processing patterns in autism, attention deficit hyperactivity disorder, and typical development. Phys Occup Ther Pediatr 38(3):243–254. https://doi.org/10.1080/01942638.2017.1390809

    Article  PubMed  Google Scholar 

  2. Baranek GT, David FJ, Poe MD, Stone WL, Watson LR (2006) Sensory Experiences Questionnaire: discriminating sensory features in young children with autism, developmental delays, and typical development. J Child Psychol Psychiatry 47(6):591–601

    Article  Google Scholar 

  3. Tomchek SD, Dunn W (2007) Sensory processing in children with and without autism: a comparative study using the short sensory profile. Am J Occup Ther 61(2):190–200. https://doi.org/10.5014/ajot.61.2.190

    Article  PubMed  Google Scholar 

  4. Ben-Sasson A, Cermak SA, Orsmond GI, Tager-Flusberg H, Carter AS, Kadlec MB et al (2007) Extreme sensory modulation behaviors in toddlers with autism spectrum disorders. Am J Occup Ther 61(5):584–592. https://doi.org/10.5014/ajot.61.5.584

    Article  PubMed  Google Scholar 

  5. Dunn W (2007) Supporting children to participate successfully in everyday life by using sensory processing knowledge. Infants Young Child 20(2):84–101

    Article  Google Scholar 

  6. Hazen EP, Stornelli JL, O’Rourke JA, Koesterer K, McDougle CJ (2014) Sensory symptoms in autism spectrum disorders. Harv Rev Psychiatry 22(2):112–124. https://doi.org/10.1097/01.Hrp.0000445143.08773.58

    Article  PubMed  Google Scholar 

  7. Hochhauser M, Engel-Yeger B (2010) Sensory processing abilities and their relation to participation in leisure activities among children with high-functioning autism spectrum disorder (HFASD). Res Autism Spectrum Disord 4(4):746–754. https://doi.org/10.1016/j.rasd.2010.01.015

    Article  Google Scholar 

  8. Dawson G, Rogers S, Munson J, Smith M, Winter J, Greenson J et al (2010) Randomized, controlled trial of an intervention for toddlers with autism: the early start denver model. Pediatrics 125(1):e17-23. https://doi.org/10.1542/peds.2009-0958

    Article  PubMed  Google Scholar 

  9. Koegel RL, Koegel LK (2006) Pivotal response treatments for autism: communication, social, & academic development. Paul H Brookes Publishing, Baltimore

    Google Scholar 

  10. Ajram LA, Horder J, Mendez MA, Galanopoulos A, Brennan LP, Wichers RH et al (2017) Shifting brain inhibitory balance and connectivity of the prefrontal cortex of adults with autism spectrum disorder. Transl Psychiatry 7(5):e1137. https://doi.org/10.1038/tp.2017.104

    CAS  Article  PubMed  PubMed Central  Google Scholar 

  11. Sprengers JJ, van Andel DM, Zuithoff NPA, Keijzer-Veen MG, Schulp AJA, Scheepers FE et al (2020) Bumetanide for core symptoms of autism spectrum disorder (BAMBI): a single center, double-blinded, participant-randomized, placebo-controlled, phase-2 superiority trial. J Am Acad Child Adolesc Psychiatry. https://doi.org/10.1016/j.jaac.2020.07.888

    Article  PubMed  Google Scholar 

  12. Veenstra-VanderWeele J, Cook EH, King BH, Zarevics P, Cherubini M, Walton-Bowen K et al (2017) Arbaclofen in children and adolescents with autism spectrum disorder: a randomized, controlled, phase 2 trial. Neuropsychopharmacology 42(7):1390–1398. https://doi.org/10.1038/npp.2016.237

    CAS  Article  PubMed  Google Scholar 

  13. Autistica (2016) Your questions: shaping future autism research. https://www.jla.nihr.ac.uk/priority-setting-partnerships/autism/downloads/Autism-PSP-final-report.pdf. Accessed 28 Jan 2021

  14. Dunn W (1999) Sensory profile: user’s manual. Psychological Corporation San Antonio

    Google Scholar 

  15. Johnson-Ecker CL, Parham LD (2000) The evaluation of sensory processing: a validity study using contrasting groups. Am J Occup Ther 54(5):494–503. https://doi.org/10.5014/ajot.54.5.494

    CAS  Article  PubMed  Google Scholar 

  16. Schauder KB, Bennetto L (2016) Toward an interdisciplinary understanding of sensory dysfunction in autism spectrum disorder: an integration of the neural and symptom literatures. Front Neurosci 10:268. https://doi.org/10.3389/fnins.2016.00268

    Article  PubMed  PubMed Central  Google Scholar 

  17. Food and Drug Administration (2009) Guidance for industry: patient-reported outcome measures: use in medical product development to support labelling claims. www.fda.gov/downloads/Drugs/GuidanceComplianceRegulatoryInformation/Guidances/UCM193282.pdf. Accessed 5 Dec 2020

  18. Streiner DL, Norman GR (2008) Health measure scales: a practical guide to their development and use. Oxford University Press, New York

    Book  Google Scholar 

  19. Cella D, Yount S, Rothrock N, Gershon R, Cook K, Reeve B et al (2007) The patient-reported outcomes measurement information system (PROMIS): progress of an NIH roadmap cooperative group during its first two years. Med Care 45(5 Suppl 1):S3–S11. https://doi.org/10.1097/01.mlr.0000258615.42478.55

    Article  PubMed  PubMed Central  Google Scholar 

  20. HealthMeasures Computer Adaptive Tests (CATs) https://www.healthmeasures.net/resource-center/measurement-science/computer-adaptive-tests-cats. Accessed 26 Feb 2021

  21. Braun V, Clarke V (2006) Using thematic analysis in psychology. Qual Res Psychol 3(2):77–101. https://doi.org/10.1191/1478088706qp063oa

    Article  Google Scholar 

  22. Boeije HR (2009) Analysis in qualitative research. SAGE Publications, London

    Google Scholar 

  23. McIntosh DN, Miller LJ, Shyu V (1999) Development and validation of the short sensory profile. The sensory profile: examiner's manual, San Antonio

  24. Licciardi L, Brown T (2021) An overview & critical review of the sensory profile–second edition. Scand J Occup Ther 1–13

  25. McIntosh DN, Miller LJ, Shyu V, Hagerman RJ (1999) Sensory-modulation disruption, electrodermal responses, and functional behaviors. Dev Med Child Neurol 41(9):608–615. https://doi.org/10.1017/s0012162299001267

    CAS  Article  PubMed  Google Scholar 

  26. Tomchek SD, Huebner RA, Dunn W (2014) Patterns of sensory processing in children with an autism spectrum disorder. Res Autism Spectrum Disord 8(9):1214–1224. https://doi.org/10.1016/j.rasd.2014.06.006

    Article  Google Scholar 

  27. Williams ZJ, Failla MD, Gotham KO, Woynaroski TG, Cascio C (2018) Psychometric evaluation of the short sensory profile in youth with autism spectrum disorder. J Autism Dev Disord 48(12):4231–4249. https://doi.org/10.1007/s10803-018-3678-7

    Article  PubMed  PubMed Central  Google Scholar 

  28. Parham LD, Ecker C, Kuhaneck H, Henry DA, Glennon TJ (2007) SPM sensory processing measure. In: Diane Parham L, Ecker C, Miller-Kuhaneck H, Henry DA, Glennon TJ (eds) Western Psychological Services

  29. Baranek GT (2009) Sensory Experiences Questionnaire (Version 3.0, unpublished manuscript). The University of North Carolina at Chapel Hill, Chapel Hill

    Google Scholar 

  30. Little LM, Freuler AC, Houser MB, Guckian L, Carbine K, David FJ et al (2011) Psychometric validation of the sensory experiences questionnaire. Am J Occup Ther 65(2):207–210

    Article  Google Scholar 

  31. Ausderau K, Sideris J, Furlong M, Little LM, Bulluck J, Baranek GT (2014) National survey of sensory features in children with ASD: factor structure of the sensory experience questionnaire (3.0). J Autism Dev Disord 44(4):915–925

    Article  Google Scholar 

  32. Aman MG, Singh NN, Stewart AW, Field CJ (1985) The aberrant behavior checklist: a behavior rating scale for the assessment of treatment effects. Am J Ment Defic 89(5):485–491

    CAS  PubMed  Google Scholar 

  33. Aman MG (2013) Aberrant Behavior Checklist. In: Volkmar FR (ed) Encyclopedia of autism spectrum disorders. Springer, New York, pp 10–17

    Chapter  Google Scholar 

  34. Aman MG, Singh NN, Stewart AW, Field CJ (1985) Psychometric characteristics of the aberrant behavior checklist. Am J Ment Defic 89(5):492–502

    CAS  PubMed  Google Scholar 

  35. Norris M, Aman MG, Mazurek MO, Scherr JF, Butter EM (2019) Psychometric characteristics of the aberrant behavior checklist in a well-defined sample of youth with autism Spectrum disorder. Res Autism Spectrum Disord 62:1–9. https://doi.org/10.1016/j.rasd.2019.02.001

    Article  Google Scholar 

  36. Marcus RN, Owen R, Kamen L, Manos G, McQuade RD, Carson WH et al (2009) A placebo-controlled, fixed-dose study of aripiprazole in children and adolescents with irritability associated with autistic disorder. J Am Acad Child Adolesc Psychiatry 48(11):1110–1119

    Article  Google Scholar 

  37. Owen R, Sikich L, Marcus RN, Corey-Lisle P, Manos G, McQuade RD et al (2009) Aripiprazole in the treatment of irritability in children and adolescents with autistic disorder. Pediatrics 124(6):1533–1540

    Article  Google Scholar 

  38. Research Unit on Pediatric Psychopharmacology (RUPP) Autism Network (2005) Randomized, controlled, crossover trial of methylphenidate in pervasive developmental disorders with hyperactivity. Arch Gen Psychiatry 62(11):1266–1274

    Article  Google Scholar 

  39. Research Unit on Pediatric Psychopharmacology (RUPP) Autism Network (2005) Risperidone treatment of autistic disorder: longer-term benefits and blinded discontinuation after 6 months. Am J Psychiatry 162(7):1361–1369. https://doi.org/10.1176/appi.ajp.162.7.1361

    Article  Google Scholar 

  40. Aman MG, Mcdougle CJ, Scahill L, Handen B, Arnold LE, Johnson C et al (2009) Medication and parent training in children with pervasive developmental disorders and serious behavior problems: results from a randomized clinical trial. J Am Acad Child Adolesc Psychiatry 48(12):1143–1154

    Article  Google Scholar 

  41. Tse J, Strulovitch J, Tagalakis V, Meng L, Fombonne E (2007) Social skills training for adolescents with Asperger syndrome and high-functioning autism. J Autism Dev Disord 37(10):1960–1968

    Article  Google Scholar 

  42. Efron D, Taylor K, Payne JM, Freeman JL, Cranswick N, Mulraney M et al (2020) Does cannabidiol reduce severe behavioural problems in children with intellectual disability? Study protocol for a pilot single-site phase I/II randomised placebo controlled trial. BMJ Open 10(3):e034362

    Article  Google Scholar 

  43. McDougle CJ, Scahill L, Aman MG, McCracken JT, Tierney E, Davies M et al (2005) Risperidone for the core symptom domains of autism: results from the study by the autism network of the research units on pediatric psychopharmacology. Am J Psychiatry 162(6):1142–1148

    Article  Google Scholar 

  44. Bodfish JW, Symons FJ, Parker DE, Lewis MH (2000) Varieties of repetitive behavior in autism: comparisons to mental retardation. J Autism Dev Disord 30(3):237–243. https://doi.org/10.1023/A:1005596502855

    CAS  Article  PubMed  Google Scholar 

  45. Mirenda P, Smith IM, Vaillancourt T, Georgiades S, Duku E, Szatmari P et al (2010) Validating the repetitive behavior scale-revised in young children with autism spectrum disorder. J Autism Dev Disord 40(12):1521–1530. https://doi.org/10.1007/s10803-010-1012-0

    Article  PubMed  Google Scholar 

  46. Bedell G (2009) Further validation of the child and adolescent scale of participation (CASP). Dev Neurorehabil 12(5):342–351. https://doi.org/10.3109/17518420903087277

    Article  PubMed  Google Scholar 

  47. Bedell GM (2004) Developing a follow-up survey focused on participation of children and youth with acquired brain injuries after discharge from inpatient rehabilitation. NeuroRehabilitation 19(3):191–205

    Article  Google Scholar 

  48. Bedell GM, Dumas HM (2004) Social participation of children and youth with acquired brain injuries discharged from inpatient rehabilitation: a follow-up study. Brain Inj 18(1):65–82

    Article  Google Scholar 

  49. De Bock F, Bosle C, Graef C, Oepen J, Philippi H, Urschitz MS (2019) Measuring social participation in children with chronic health conditions: validation and reference values of the child and adolescent scale of participation (CASP) in the German context. BMC Pediatr 19(1):1–13

    Article  Google Scholar 

  50. Golos A, Bedell G (2018) Responsiveness and discriminant validity of the child and adolescent scale of participation across three years for children and youth with traumatic brain injury. Dev Neurorehabil 21(7):431–438

    Article  Google Scholar 

  51. Coster W, Law M, Bedell G, Khetani M, Cousins M, Teplicky R (2012) Development of the participation and environment measure for children and youth: conceptual basis. Disabil Rehabil 34(3):238–246. https://doi.org/10.3109/09638288.2011.603017

    Article  PubMed  Google Scholar 

  52. Coster W, Bedell G, Law M, Khetani MA, Teplicky R, Liljenquist K et al (2011) Psychometric evaluation of the participation and environment measure for children and youth. Dev Med Child Neurol 53(11):1030–1037. https://doi.org/10.1111/j.1469-8749.2011.04094.x

    Article  PubMed  Google Scholar 

  53. Wigham S, Rodgers J, South M, McConachie H, Freeston M (2015) The interplay between sensory processing abnormalities, intolerance of uncertainty, anxiety and restricted and repetitive behaviours in autism spectrum disorder. J Autism Dev Disord 45(4):943–952. https://doi.org/10.1007/s10803-014-2248-x

    Article  PubMed  Google Scholar 

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Acknowledgements

We sincerely thank all the parents who participated in this study. We also thank research assistants Gisela Timmer and Lisanne Geurts for their assistance in organizing the focus groups and carrying out interviews and transcriptions. In memory of Henk van Stel.

Funding

This study was supported by a grant from Dutch Brain Foundation (Hersenstichting). This foundation had no role in the design of the study and collection, analysis, and interpretation of data and in writing the manuscript.

Author information

Author notes

  1. Henk F. van Stel

    Present address: Department of Healthcare Innovation and Evaluation, Julius Center for Health Sciences and Primary Care, University Medical Center Utrecht, Utrecht, The Netherlands

Affiliations

  1. Department of Psychiatry, UMC Utrecht Brain Center, University Medical Center Utrecht, Utrecht, The Netherlands

    Dorinde M. van Andel, Floortje E. Scheepers & Hilgo Bruining

  2. Child and Adolescent Psychiatry and Psychosocial Care, Emma Children’s Hospital, Amsterdam UMC, Amsterdam Reproduction and Development, Vrije Universiteit Amsterdam, Amsterdam, The Netherlands

    Kim J. Oostrom

  3. Child and Adolescent Psychiatry and Psychosocial Care, Emma Children’s Hospital, Amsterdam UMC, Vrije Universiteit Amsterdam, N=You centre, Levvel, Amsterdam Neuroscience, Amsterdam Reproduction and Development, Vrije Universiteit Amsterdam, Meibergdreef 9, 1105 AZ, Amsterdam, The Netherlands

    Hilgo Bruining

  4. Child and Adolescent Psychiatry and Psychosocial Care, Emma Children’s Hospital, Amsterdam UMC, Amsterdam Public Health, University of Amsterdam, Amsterdam, The Netherlands

    Lotte Haverman

Authors
  1. Dorinde M. van Andel
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  2. Henk F. van Stel
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  3. Floortje E. Scheepers
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  4. Kim J. Oostrom
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  5. Lotte Haverman
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  6. Hilgo Bruining
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Contributions

HB, HS and DA designed the study. DA collected, transcribed and analyzed the data. DA and HB interpreted the data and wrote the initial manuscript. All authors contributed to paper revisions and approved the final manuscript.

Corresponding author

Correspondence to Hilgo Bruining.

Ethics declarations

Ethics approval and consent to participate

The medical ethical committee (METC) of the UMC Utrecht approved the protocol. Written informed consent was waived.

Consent for publication

Not applicable.

Competing interests

D.M. van Andel, F.E. Scheepers, K.J. Oostrom and H. Bruining declare that they have no competing interests. L. Haverman is board member of the Dutch–Flemish PROMIS National Center.

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Deceased: Henk F. van Stel

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van Andel, D.M., van Stel, H.F., Scheepers, F.E. et al. The sensory-reactivity PROM set: identification of a parent reported outcome measure set for autism spectrum disorder. J Patient Rep Outcomes 5, 123 (2021). https://doi.org/10.1186/s41687-021-00396-w

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Keywords

  • ASD
  • Sensory reactivity problems
  • PROM
  • PROMIS
  • Trials