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Functioning and disability in autism spectrum disorder: A worldwide survey of experts

Functioning and disability in autism spectrum disorder: A worldwide survey of experts RESEARCH ARTICLE Functioning and Disability in Autism Spectrum Disorder: A Worldwide Survey of Experts Elles de Schipper,† Soheil Mahdi,† Petrus de Vries, Mats Granlund, Martin Holtmann, Sunil Karande, Omar Almodayfer, Cory Shulman, Bruce Tonge, Virginia V.C.N. Wong, Lonnie Zwaigenbaum, and Sven Bolte Objective: This study is the second of four to prepare International Classification of Functioning, Disability and Health (ICF; and Children and Youth version, ICF(-CY)) Core Sets for Autism Spectrum Disorder (ASD).The objective of this study was to survey the opinions and experiences of international experts on functioning and disability in ASD. Methods: Using a protocol stipulated by the World Health Organization (WHO) and monitored by the ICF Research Branch, an email-based questionnaire was circulated worldwide among ASD experts, and meaningful functional ability and disability concepts were extracted from their responses. These concepts were then linked to the ICF(-CY) by two independent researchers using a standardized linking procedure. Results: N5 225 experts from 10 different disciplines and all six WHO-regions completed the survey. Meaningful concepts from the responses were linked to 210 ICF(-CY) categories. Of these, 103 categories were considered most relevant to ASD (i.e., identified by at least 5% of the experts), of which 37 were related to Activities and Participation,35 to Body functions,22to Environmental factors, and 9 to Body structures. A variety of personal characteristics and ASD-related functioning skills were provided by experts, including honesty, loyalty, attention to detail and creative talents. Reported gender differences in ASD comprised more externaliz- ing behaviors among males and more internalizing behaviors in females. Conclusion: The ICF(-CY) categories derived from international expert opinions indicate that the impact of ASD on functioning extends far beyond core symptom V C domains. Autism Res 2016, 9: 959–969. 2016 The Authors Autism Research published by Wiley Periodicals, Inc. on behalf of International Society for Autism Research Keywords: autism; neurodevelopmental disorder; assessment; child psychiatry; heterogeneity; quality of life Introduction comorbid conditions (e.g., ADHD, epilepsy, and gener- alized anxiety disorder), in combination with factors A multitude of research provides information on the pertaining to the environment like access to interven- impact of Autism Spectrum Disorder (ASD) on function- tion programs and availability of support services ing in everyday life. Poor outcomes are reported in accounting for the largest part of the variability [Brund- terms of education, employment, social relationships, son & Happe, 2014; Levy & Perry, 2011]. There is also independent living, mental health, and quality of life evidence that ASD is associated with certain gender dif- [Bolte & Poustka, 2002; Howlin & Moss, 2012; Lai & ferences [Bolte, € Dukretis, Poustka, & Holtmann, 2011; Lombardo, Baron-Cohen, 2014]. Studies also reveal sub- € Halladay et al., 2015; Holtmann, Bolte, & Poustka, stantial interindividual differences in outcomes and lev- 2007] and strengths [Happe & Frith, 2009; Mottron, els of functioning for individuals with ASD, with factors Dawson, & Soulie `res, 2009] such as visual spatial, ana- such as intelligence quotient, language abilities, and lytical or savant skills that may positively impact on an From the Department of Women’s and Children’s Health, Center of Neurodevelopmental Disorders (KIND), Pediatric Neuropsychiatry Unit, Karolin- ska Institutet, Stockholm, Sweden (E.S., S.M., S.B.,); Child and Adolescent Psychiatry, Center of Psychiatry Research, Stockholm County Council, Stockholm, Sweden (E.S., S.M., S.B.,); Division of Child & Adolescent Psychiatry, University of Cape Town, Cape Town, South Africa (P.V.,); CHILD, SIDR, Jonk € oping € University, Jonk € oping, € Sweden (M.G.,); LWL-University Hospital for Child and Adolescent Psychiatry, Ruhr-University Bochum, Hamm, Germany (M.H.,); Learning Disability Clinic, Department of Pediatrics, Seth G.S. Medical College & K.E.M. Hospital, Mumbai, India (S.K.,); Psychiatry Section, King Abdulaziz Medical City, College of Medicine, Riyadh, Saudi-Arabia (O.A.,); Paul Baerwald School of Social Work and Social Welfare, Hebrew University of Jerusalem, Jerusalem, Israel (C.S.); Centre for Developmental Psychiatry and Psychology, Monash University, Melbourne, Victoria, Australia (B.T.,); Department of Paediatrics and Adolescent Medicine, The University of Hong Kong, Hong Kong SAR, China (V.C.N.W.,); Department of Pediatrics, University of Alberta, Edmonton, Alberta, Canada (L.Z.) E.S. and S.M. contributed equally to this work. Received August 19, 2015; accepted for publication November 27, 2015 This is an open access article under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made. Address for correspondence and reprints: Sven Bolte, € Department of Women’s and Children’s Health, Center of Neurodevelopmental Disorders (KIND), Pediatric Neuropsychiatry Unit, Karolinska Institutet, Stockholm, Sweden. E-mail: sven.bolte@ki.se Published online 08 January 2016 in Wiley Online Library (wileyonlinelibrary.com) DOI: 10.1002/aur.1592 V 2016 The Authors Autism Research published by Wiley Periodicals, Inc. on behalf of International Society for Autism Research Autism Research 9: 959–969, 2016 959 INSAR individual’s level of functioning and quality of life. mostly because in its current full form it appears too Although the fifth edition of the Diagnostic and Statis- generic for certain health conditions and impractical for tical Manual of Mental Disorders (DSM-5) [American daily use. To address this issue the development of ICF Psychiatric Association, 2013] provides novel options Core Sets was initiated. ICF Core Sets are shortlists of ICF(- for individualizing diagnosis in ASD through specifiers CY) categories that are considered most relevant to indi- and severity/need of support ratings, it does neither viduals with a certain health condition in clinical contexts. provide explicit definitions for disability nor for adaptive These ICF Core Sets are developed following a rigorous functioning. The International Classification of Func- qualitative and quantitative, scientific protocol stipulated tioning (ICF; [WHO, 2001]) complements the Interna- by the WHO and ICF Research Branch [Kostanjsek et al., tional Classification of Diseases-Tenth Revision [ICD-10; 2011]. Its main objective is to use existing research data, World Health Organization (WHO), 1992], conceptualiz- involve a large and multi-professional selection of ing functioning and disability as being separate from researchers and clinicians, as well as service users/patients pathology. It applies a biopsychosocial perspective to and their caregivers from all over the world to ensure uni- operationalize an individual’s level of functioning as the versal applicability of the ICF Core Sets. The protocol con- interplay of abilities and disabilities that emerge in the sists of a systematic literature review (“research contexts of physical constitution, social participation, perspective”), an expert survey (current study, “opinion personal as well as environmental resources [WHO, leader perspective”), focus groups (“client and caregiver 2001]. Moreover, the ICF facilitates standardized assess- perspective”), and a cross-sectional clinical study ment of functioning and health by providing detailed (“clinical perspective”). Thus, the present study is embed- classifications in the areas of Body functions (i.e., physio- ded into the process of a larger research and development logical functions of body systems), Body structures (i.e., project, aimed at developing standardized ICF Core Sets anatomical parts of the body), Activities (i.e., execution for ASD. ICF Core Sets for ADHD are being developed as of tasks), Participation (i.e., involvement in life situa- well in this project, but the results are reported in sepa- tions), and Environmental factors (i.e., physical, social and € rate publications [Bolte et al., 2014a; De Schipper et al., attitudinal environment). The bio-psycho-social model 2015a]. In order to accomplish ICF Core Sets that cover also includes Personal factors, but these have not yet been functioning as well as disabilities in ASD across the life classified in the ICF given how these are grounded in span, the development will be based on the ICF(-CY), the social and cultural context. To capture the particular sit- extended version of the ICF, that captures not only func- uation of a developing individual, the ICF Children and tioning in adulthood but also childhood and youth. A Youth version [ICF(-CY); WHO, 2007] was derived from complete description of the overall ASD ICF Core Set the ICF by adding development-relevant categories and development process has been published in a previous expanding on the descriptions of existing ICF categories. issue of this journal [Bolte € et al., 2014b]. The ICF and ICF(-CY) (hereafter, “ICF(-CY)”) consist The objective of this study was to capture the experi- of hierarchically structured categories describing aspects ences and opinions of experts in the assessment and of functioning in the three key components of the bio- treatment of ASD. Together with the other three prepar- psycho-social model described above (i.e., Body Func- atory studies mentioned above, this expert survey gen- tions and Structures, Activities & Participation, Environ- erates content for an international ICF Core Sets mental factors) in up to four levels of increasing detail. Consensus Conference, during which a group of ASD The first level categories are known as chapters, and pro- experts from all WHO regions will follow a formal vide a general overview of the areas of functioning that decision-making process to arrive at ICF(-CY) categories are covered in the ICF(-CY). The chapters consist of sec- to be included in the ICF Core Sets for ASD. ond, third and fourth level categories. The following example of an ASD-relevant classification from the Body Methods functions component shows the hierarchical structure of the ICF(-CY): Design Level 1 chapter: b1 Mental functions In an email-based survey, professionals from various Level 2 category: b167 Mental functions of language disciplines worldwide provided their perspectives on Level 3 category: b1671 Expression of language which features of ability, disability and context are to Level 4 category: b16710 Expression of spoken language be considered essential for functioning in everyday life of individuals with ASD. Consisting of over 1600 categories, the ICF(-CY) pro- vides a comprehensive framework for the classification of Recruitment Procedure an individual’s functioning. However, despite its potential value to health care and research [Bolte, 2009; Escorpizo An internet search was performed to identify contact et al., 2013], the ICF(-CY)’s use is still rather limited, information for internationally known ASD experts and 960 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR Figure 1. An overview of the recruitment process. for centers (academic and community-based), clinics and were then contacted via email and received information university departments in all WHO-regions regularly about the study along with a request to participate in involved in the assessment and management of individ- the survey. If they agreed to participate, they were asked uals with ASD. Identified organizations were contacted to fill in a reply sheet with information about their work via email with information about the study and a experience to confirm their eligibility. After confirma- request to provide contact information of eligible tion, the experts received the survey as a Word or PDF experts. Contact information of experts was also pro- file via email, which they were requested to fill in and vided by the project Steering Committee (see acknowl- return within one month. Experts who did not submit edgement), a group of key opinion leaders in ASD from the survey on time received up to three reminders to all WHO-regions providing guidance on the project, and return their survey response and, if requested, they by the authors’ personal professional networks. Finally, received extended time to respond. Data collection took snowball sampling was applied, as all contacted experts place between September 2013 and October 2014. were requested to recommend additional experts to be recruited for the survey. To be considered an “expert” Participating Experts for the purposes of this study, potential participants Experts were approached via email with an invitation to were required to (a) practice in one or more of the fol- participate in the survey. The majority of them accepted lowing professions: coach, counselor, nurse, occu- the invitation. There were also some who did not pational therapist, physician (e.g., psychiatrists, respond to the invitation or declined the invitation. neurologists, etc.), physiotherapist, psychologist, psycho- Reasons for declining the invitation were mostly a lack therapist, social worker, special educator, or speech and of time, but there were also some who felt their exper- language therapist; (b) have at least five years of experi- ence in the assessment and/or treatment of individuals tise was more theoretical than practical. Moreover, there with ASD; (c) fluency in English. These identified experts were also experts who were ineligible to participate in de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 961 INSAR Figure 2. Representation of professions per WHO region (n5 245). (43 different countries). See Figure 2 for an overview of Table 1. Representation of participants per professional group (some experts have been duplicated due to their the representation of professions per WHO region and multiprofessional background) Table 1 for participants per professional group. Participating experts ranged in age between 26 and 74 Profession N (%) years, with a mean age of 47 years (SD510.8). They had Physician 54 (22%) on average 16 years of experience in working with indi- Occupational therapist 50 (20%) viduals with ASD (SD58.0), ranging between 5 and 43 Psychologist 36 (15%) Special educator 27 (11%) years. The majority of participating experts were females Physiotherapist 23 (9%) (76%). At the time of the survey, the participants spent Speech & language therapist 23 (9%) the majority of their time working in clinical depart- Psychotherapist 14 (6%) ments (45%), education (16%), or research (11%). A Social worker 8 (4%) Nurse 7 (3%) small group of experts worked mainly in management, Coach 3 (1%) or in other fields, such as training or supervision of other professionals (5%). The remaining experts divided their work time among more than one field (19%), mostly A profession in which an individual supports clients in achieving combining their work in the clinic with education or different goals in life, for example, improving school grades, finding a job, and so forth. research. A proportion of 39% of experts worked with children and adolescents exclusively, 38% with children only, and 12% with ASD across the lifespan. A minority the study (<5 years of experience). Participating experts of the participants (2%) worked exclusively with adoles- received up to two reminders to return their survey cents and 4% with adults only, or a combination of these response. An overview of the recruitment process is pro- two age groups (4%). Three experts (1%) did not specify vided in Figure 1. the age group of clients they encountered the most. Thus, complete survey responses were obtained from 225 experts. A small minority of participating experts Expert Survey stated that they were practicing more than one profes- The expert survey consisted of three parts (see Support- sion, for example, worked both as a psychologist and as a psychotherapist. Participating experts represented 10 ing Information Appendix S1). Part one contained ques- different professions and all six of the WHO-regions tions about the participating expert’s demography. Part 962 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR two consisted of six questions regarding functioning and 85% at the level of ICF(-CY) chapters. Cohen’s and disability in individuals with ASD. The questions kappa and confidence intervals were calculated to examine the extent to which the agreements exceed were specifically formulated to cover each component chance. The kappa value was j50.81 (SE50.004) with a of the bio-psycho-social of the ICF(-CY), using a similar 95% confidence interval of 0.80–0.81 for the second- approach as in previous ICF core set developments level categories. At the level of ICF(-CY) chapters the [Coenen et al., 2011]. Part two of the survey included Kappa value was j50.84 (SE50.004) with a confidence two further questions, one concerning the possible interval of 0.83–0.84. These Kappa values indicate functional strengths in ASD and the second possible excellent interrater agreement. gender differences in functioning and disability. Part three of the survey contained information regarding Data Analysis privacy and terms of agreement for taking part in the Frequency analysis was used to analyze the responses to expert survey. the six ICF(-CY) based questions of the survey. The Identification of Meaningful Concepts and linking to the absolute number of expert survey responses in which ICF(-CY) each of the ICF(-CY) categories were identified, along with the corresponding percentages relative to the total “Meaningful Concepts” are concise descriptions of spe- number of responses received, was examined. ICF(-CY) cific behaviors, skills or other aspects of functioning categories are presented at the second-level. If a concept that are to be linked to ICF(-CY) categories. These con- was linked to a third- or fourth-level ICF(-CY) category, cepts were extracted from the experts’ survey responses the corresponding second-level category was reported. and then linked to ICF(-CY) categories following for- Because the ICF(-CY) is organized hierarchically, aspects malized linking rules and procedures determined by the of the more specific third- and fourth-level categories WHO ICF Research Branch [Cieza et al., 2002; Cieza were included in the less specific second-level catego- et al., 2005]. The linking rules provide guidance on ries. Following the ICF Core Sets development guide- how to link concepts to ICF(-CY) categories, and what lines [Selb et al., 2014], a second-level ICF(-CY) category to do when that is not possible. Specific codes assigned that was identified repeatedly in the same expert survey to these concepts in the latter situation are (a) Personal response was counted only once. Consistent with WHO factor, if the concept is not contained in the ICF(-CY), and previous core set development conventions [Escor- but is clearly a Personal factor as defined in the ICF(- pizo et al., 2011; Granberg et al., 2014], only ICF(-CY) CY); (b) non- covered, if the concept is not contained in categories that were identified in at least 5% of the the ICF(-CY) and also is not a Personal factor; (c) non- expert survey responses were included in the list of definable, when the information provided in the con- potential categories to be included in the Core Set for cept is not sufficient to assign it to a specific ICF(-CY) ASD. This is done to ensure that only those categories category; and (d) health condition, if the concept refers that are most relevant to a certain condition, in this to a diagnosis or health condition. Given that there are case ASD, are included. Additional frequency analysis many different ways to describe the same aspect of was used to explore the possible relation between functioning, it is possible for different meaningful con- experts’ self-identified expertise with respect to stage of cepts to be assigned to the same ICF(-CY) category. development and the ICF(-CY) categories identified in To assure the quality and consistency of results, both their survey responses. The last questions regarding spe- the identification of meaningful concepts (e.g., difficul- cific abilities related to ASD and possible gender differ- ties in decision making) and the linking to ICF(-CY) ences in functioning were not linked due to the fact categories (e.g., d177 Making decisions) was conducted that responses were inconsistent and heterogeneous, independently by two researchers (ES and SM), who and therefore it was impossible to define meaningful had received extensive linking training by the ICF concepts for linking. Instead, the answers were carefully Research Branch prior to the project. Linking results of reviewed independently by two researchers (ES and SM) each of the researchers were compared and consensus before recurring themes or patterns of answers were discussions were used to resolve disagreements. If con- summarized. sensus could not be reached, the coordinator of the ICF Research Branch (MS) was available to make final deci- Results sions. However, this option was never used because dis- agreements were resolved by discussion between the Linking Results two researchers. The overall percentage of agreement between the two researchers (prior to consensus in case In total, 8792 meaningful concepts were extracted from of disagreement) for the linking process was calculated. 225 survey responses. These concepts were linked to This was 81% for the second-level ICF(-CY) categories 210 second-level ICF(-CY) categories, 191 Personal de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 963 INSAR Table 2. Absolute and relative frequencies of ICF(-CY) Table 3. Absolute and relative frequencies of ICF(-CY) categories from the Activities and Participation component categories from the Body Functions component Second level category N (%) Second level category N (%) d720 complex interpersonal interactions 101 (45%) b760 control of voluntary movement functions 156 (69%) d440 fine hand use 89 (40%) b164 higher-level cognitive functions 151 (67%) d710 basic interpersonal interactions 85 (38%) b147 psychomotor functions 117 (52%) d446 fine foot use 70 (31%) b156 perceptual functions 108 (48%) d230 carrying out daily routine 68 (30%) b152 emotional functions 105 (47%) d310 communicating with—receiving—spoken messages 62 (28%) b125 dispositions and intra-personal functions 97 (43%) d335 producing nonverbal messages 59 (26%) b167 mental functions of language 82 (36%) d570 looking after one’s health 59 (26%) b765 involuntary movement functions 81 (36%) d820 school education 59 (26%) b140 attention functions 80 (36%) d920 recreation and leisure 56 (25%) b265 touch function 75 (33%) d750 informal social relationships 55 (24%) b230 hearing functions 71 (32%) d177 making decisions 47 (21%) b117 intellectual functions 62 (28%) d250 managing one’s own behavior 47 (21%) b260 proprioceptive function 61 (27%) d330 speaking 47 (21%) b210 seeing functions 60 (27%) d315 communicating with—receiving—nonverbal messages 46 (20%) b255 smell function 52 (23%) d240 handling stress and other psychological demands 31 (14%) b270 sensory functions related to temperature 52 (23%) d530 toileting 30 (13%) and other stimuli d830 higher education 27 (12%) b130 energy and drive functions 49 (22%) d510 washing oneself 26 (12%) b250 taste function 49 (22%) d350 conversation 25 (11%) b163 basic cognitive functions 48 (21%) d550 eating 24 (11%) b235 vestibular functions 45 (20%) d880 engagement in play 24 (11%) b735 muscle tone functions 45 (20%) d130 copying 23 (10%) b122 global psychosocial functions 41 (18%) d760 family relationships 23 (10%) b180 experience of self and time functions 37 (16%) d825 vocational training 23 (10%) b160 thought functions 35 (16%) d845 acquiring, keeping and terminating a job 21 (9%) b134 sleep functions 34 (15%) d220 undertaking multiple tasks 20 (9%) b770 gait pattern functions 34 (15%) d520 caring for body parts 20 (9%) b126 temperament and personality functions 31 (14%) d540 dressing 20 (9%) b176 mental function of sequencing complex 24 (11%) d571 looking after one’s safety 19 (8%) movements d163 thinking 18 (8%) b144 memory functions 22 (10%) d740 formal relationships 16 (7%) b515 digestive functions 19 (8%) d210 undertaking a single task 15 (7%) b330 fluency and rhythm of speech functions 18 (8%) d850 remunerative employment 15 (7%) b114 orientation functions 17 (8%) d455 moving around 14 (6%) b280 sensation of pain 17 (8%) d160 focusing attention 12 (5%) b320 articulation functions 15 (7%) d770 intimate relationships 12 (5%) b310 voice functions 12 (5%) factors (e.g., self-esteem, restricted interests), 611 non- Activities and Participation component are presented in definable codes (e.g., structured environment, behavior Table 2, along with the number and percentage of problems), 208 noncovered codes (e.g., stress, social expert survey responses in which they were identified. rejection), and 41 health condition-codes (e.g., depres- They cover eight of the nine chapters making up the sion, dyspraxia, epilepsy). The nondefinable codes and component, that is, (d5) self-care,(d7) interpersonal inter- noncovered codes were mostly identified in the ques- actions and relationships,(d8) major life areas,(d2) general tions that captured the environmental factors of func- tasks and demands,(d3) communication,(d1) learning and tioning and disability, whereas the health-condition applying knowledge,(d4) mobility, and (d9) community, codes were mostly applied in questions related to Body social and civic life. Table 3 presents the absolute and relative frequencies functions and Body structures component. Of the identified ICF(-CY) categories, 103 were found of the second-level categories that were identified in in the responses of at least 5% of the experts (range: 5– the Body functions component. Five out of the eight 87%) and are included in the list of candidate catego- chapters making up this component are represented in ries. Each of the four components of the ICF(-CY) are these categories. The majority of categories were identi- represented in these categories, with 37 categories from fied in the chapter (b1) mental functions. The remainder Activities and Participation, 35 from Body functions,22 of categories was identified in four additional chapters, from Environmental factors, and 9 from Body structures. that is, (b2) sensory functions and pain,(b7) neuromuscu- The second-level categories that were identified in the loskeletal and movement-related functions,(b3) voice and 964 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR Table 4. Absolute and relative frequencies of ICF(-CY) Table 5. Absolute and relative frequencies of ICF(-CY) categories from the Environmental Factors component categories from the Body Structures component Second level category N (%) Second level category N (%) e310 immediate family 100 (44%) s110 structure of brain 195 (87%) e585 education and training services, systems and 100 (44%) s540 structure of intestine 34 (15%) policies s730 structure of upper extremity 18 (8%) e580 health services, systems and policies 84 (37%) s750 structure of lower extremity 16 (7%) e115 products and technology for personal use in 68 (30%) s530 structure of stomach 15 (7%) daily living s520 structure of esophagus 14 (6%) e460 societal attitudes 51 (23%) s260 structure of inner ear 12 (5%) e410 individual attitudes of immediate family members 41 (18%) s320 structure of mouth 11 (5%) e125 products and technology for communication 37 (16%) s770 additional musculoskeletal structures related 11 (5%) e250 sound 37 (16%) to movement e590labor and employment services, systems and policies 34 (15%) e360 other professionals 28 (12%) e575 general social support services, systems and policies 26 (12%) Additional frequency analysis of 10% of the expert sur- e240 light 22 (10%) vey responses was conducted in order to explore a possible e165 assets 19 (8%) relationship between the age group in which the experts e340 personal care providers and personal assistants 17 (8%) are specialized in and the ICF(-CY) categories that were e355 health professionals 17 (8%) identified in their survey responses. The analysis showed e425 individual attitudes of acquaintances, peers, 17 (8%) colleagues, neighbors and community members that a large majority of the categories in the survey e455 individual attitudes of health-related professionals 15 (7%) responses were identified across the lifespan. There were, e465 social norms, practices and ideologies 15 (7%) however, a few categories more often identified for chil- e155 design, construction and building products and 14 (6%) dren as compared to adults: e310 Immediate family, e585 technology of buildings for private use e325 acquaintances, peers, colleagues, neighbors and 12 (5%) Education and training services, systems and policies, d440 community members Fine hand use, d446 Fine foot use and d740 Informal social e315 extended family 11 (5%) relationships. Given that children usually live with their e450 individual attitudes of health professionals 11 (5%) immediate family and attend school on a regular basis, some of the categories (e.g., e310 Immediate family and e585 Education and training services, systems and policies) speech functions, and (b5) functions of the digestive, meta- covered in the experts’ survey responses are not surprising. bolic and endocrine systems. Four out of the five most often identified categories in this component are mental ASD-Related Abilities functions. The most often identified category pertains to A large majority of experts (92%) indicated observed movement-related function. Absolute and relative frequencies of the second-level ASD-related skills, many of which were reported to be categories identified in the Environmental factors com- mainly associated with higher functioning individuals. ponent are presented in Table 4. All five of this compo- The experts further remarked that these skills need to nent’s chapters are represented by these categories, be actively recognized and nurtured in order for the that is, (e3) support and relationships,(e4) attitudes,(e1) individuals to reach their full potential. Skills and abil- products and technology,(e5) services, systems and policies, ities that were often mentioned by the experts are sum- and (e2) natural environment and human-made changes to marized in Table 6. environment. Gender Differences Table 5 shows the second-level categories that were identified in the Body structures component, together Sixty percent of the surveyed experts reported gender- with their absolute and relative frequencies. These rep- related differences in ASD. Of those, more than half resent five out of eight chapters making up this compo- replied that they had observed gender differences in nent. By far the most frequently identified category was functioning and disabilities. Some experts highlighted from chapter (s1) structures of the nervous system, which that these differences were dependent on the age range was mentioned by 87% of the experts, making it the and intellectual capacities, and that observed differen- most often identified of all ICF(-CY) categories in this ces might not necessarily be specific to ASD, but rather, study. Other categories from this component were reflect gender differences in the general population. derived less often, and represent four additional chap- Other experts indicated that males with ASD tend to ters, that is, (s5) structures related to the digestive, meta- show more externalizing behavioral difficulties (hyper- bolic and endocrine systems,(s7) structures related to activity, aggression) than females, making them easier movement,(s2) the eye, ear and related structures, and (s3) to diagnose. On the other hand, females with ASD were structures involved in voice and speech. described to be better socially adjusted, showing more de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 965 INSAR Table 6. Overview of ASD-related skills and abilities men- tion schedules, questionnaire, interviews) will be tioned by the experts derived and standardized for broad use in clinical and research settings. These tools will be ready made and Abilities and skills easily implementable for clinicians. Attention to detail The present study identified a large number of catego- Strong sense of morality (e.g., honesty, lack of judgmental ries covering all four components contained in the ICF(- attitude, etc.) A preference to work on repeated or monotonous tasks CY). It underlines that experts accept ASD to be a com- Expertise in a specific area plex condition involving many physical, environmental Mathematical abilities and personal factors and areas of life. The most fre- Creative talents (e.g., look at the world differently) quently identified category was (s110) structure of brain Artistic skills (e.g., music, drawing, visual arts) from the Body structures, indicating that there is consen- Visual perception Intellectual functions sus among international experts of various professions Technical abilities (computer skills, engineering) that ASD is a brain-based condition. However, as indi- Trustworthiness cated by other derived categories (s2, s3, s5, s7), body Loyalty structures related to voice and speech, movement and Kindness digestive system are also presumed to be closely related Good memory to ASD. Not surprisingly, multiple categories from the Activities and Participation component were derived, such prosocial behaviors, communication skills and friendships as problems with social relationships (d7) and communi- than males, making them easily overlooked. Additional cation (d3), which constitute the core symptoms of ASD experts perceived females and males with ASD to share as described in the DSM-5. Nevertheless, the majority of core abilities and disabilities, but also experienced that identified categories represent other challenges, includ- females invested more efforts in blending in, complying ing the ability to take care of oneself and manage every- with social demands and hiding their difficulties. Experts day life and tasks (d5, d2), participation in school, work concluded that this might not only lead to underdiagnos- and social life (d8, d9), and cognitive and motor skills ing females, but also to higher expectations on females (d1, d4). Categories identified in the Body functions com- with ASD, which in turn results in increased levels of ponent concern a rich variation of mental functions stress and anxiety, and a greater sense of failure, com- (b1), such as language (b167), perception (b156), pared to males with ASD. Males with ASD were described emotion (b152), and attention (b140). Furthermore, cate- as more pronounced in socially unadjusted and unac- gories from other chapters suggest a broader range of cepted behaviors, leading to more social exclusion, fewer ASD-related problems, for example, sensation (b2), prob- friendships, and more bullying. Males as compared to lems with speech (b3), and with digestion (b5). Finally, females with ASD seemed on the other hand less emo- the categories identified from the Environmental factors tionally affected by social exclusion, showing a lower component indicate that the social context, both indi- average desire for social participation. vidual support and common attitudes towards individu- als with ASD (e3, e4), organized support and services Discussion provided by professionals (e5), the physical context and environment (e1, e2) (products designed to be helpful The objective of the present study was to characterize for coping with challenges in everyday life; physical functioning aspects and disabilities that are often asso- environment in general), interact with an individual’s ciated with ASD, based on the perspectives of experts functioning and disabilities in determining their level of who assess and treat the disorder. To this end, an everyday functioning. These findings endorse the useful- email-based survey was conducted among a large sam- ness of the bio-psycho-social model of the ICF(CY), ple of international experts from multiple professions. which describes functioning as a dynamic interaction The experiences and opinions of such a large and between health condition and contextual factors. diverse international group of ASD experts on this topic This study is among the first to explore specific have not been systematically examined previously. The strengths and gender differences in ASD as perceived by list of candidate ICF(-CY) categories derived from the experts from different professions and cultures. Overall, current study will be combined with the identified cate- opinions put forward were too heterogeneous and gories from three additional preparatory studies to form inconsistent to derive meaningful concepts and link the basis for an “ICF Core Sets for ASD” consensus con- ICF categories. Thus, this survey data was analyzed ference. At this conference a group of international qualitatively, in which recurring aspects of functioning ASD-experts will determine which ICF(-CY) categories and abilities mentioned by the experts were summar- should be included in the first official ICF Core Sets for ized without additional frequency analysis. Our survey ASD, on the basis of which diagnostic tools (observa- data should, however, in the future be more 966 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR comprehensively examined using thematic analyzes in composition of experts who were surveyed. Notably, a separate study. Many experts reported both certain there may be certain professions (e.g., psychologists) abilities and positive traits linked to ASD and pheno- whose role is not yet established in the assessment or types related to gender in ASD. Some of the survey find- treatment of individuals with ASD in some countries ings such as an overrepresentation of savant and (e.g., Azerbaijan). Restricting the survey to professionals scientific talents in ASD are supported by research [e.g., proficient in the English language may have also limited Treffert, 2014], while others, such as a strong sense of participation in some WHO-regions. For instance, in justice, loyalty and helpfulness currently have not been regions or countries where English is not commonly systematically investigated. Surprisingly, research find- used, such as Central and South-America (e.g., Mexico ings of gender differences in ASD, such as repetitive and Uruguay) and the Middle East (e.g., Qatar), some behaviors being more pronounced in males than invited experts were unable to participate due to this females with ASD (Szatmari et al., 2011), were not con- restriction. The four preparatory studies combined will sistently reported by experts. On the other hand, differ- however make it possible for one study to fill in the gaps ing comorbidity profiles were often mentioned by the of another study. For example, certain countries (e.g., Bra- experts, despite the fact that research has shown mixed zil) or regions (e.g., the Middle East) may be underrepre- results [Holtmann et al., 2007; Rubenstein, Wiggins, & sented in this study and in the systematic literature Lee, 2014]. In summary, the expert survey data on these review given the requirement for publications to be in topics demonstrate not merely deficits, but also English and more general research opportunity inequal- strengths oriented views on ASD among experts, and a ities, but these will have a stronger representation in the widespread awareness that gender issues might be sig- upcoming qualitative and clinical studies. Language nificant for understanding ASD. Furthermore, they gen- should not be a barrier in these studies, since collabora- erate novel hypotheses based on clinical experience to tions with members from the Steering Committee and be tested in future studies. other international study sites will allow for the use of Certain meaningful concepts in the expert survey local languages. The focus group study will consist of responses which the experts identified could not be group discussions with different stakeholders (clients with translated into ICF(-CY) categories. Indeed, the large ASD, their families, professional caregivers), whereas the number of nondefinable codes (n5611) invites closer clinical study will involve investigation of problems as examination. Nondefinable codes are largely explained experienced by actual patients in real life clinical settings. by the fact that experts were free to answer the questions All in all, the results of the four studies together should in the survey in their own words and expressions. Even provide us with a comprehensive picture of the specific though the experts were instructed to be as concrete as abilities and disabilities that are related to functioning in possible in their survey responses, the answers provided ASD for individuals across different countries, cultures by them were sometimes very brief or ambiguous (e.g., and life situations. “behavior,” “participation”) or formulated in general The use of written survey might also have caused dif- terms (e.g., “social impairment,” “adaptive skills”). Con- ficulties in verifying that the questions were always sequently, this presented a challenge for the linking. interpreted correctly by the experts, as it would have Nevertheless, a closer look at the non-definable codes been possible in a face-to-face interview. Some experts revealed a few recurring themes, such as the level of (n5 17) indicated that they were unsure what was structure and routine in the environment (e.g., intended with a certain question and thus refrained “structured environment,” “daily routine”), the level of from answering. In other cases experts (n5 5) explicitly sensory input from the environment (e.g., “sensory over- stated that they may have misinterpreted the questions. load,” “limited distractions”), and the general adjust- Furthermore there were also technical issues to the ment of the environment to the individual’s needs and study resulting in some of the participating experts abilities (e.g., “autism-friendly environment,” “balance being unable to open or work with several versions of between abilities and demands from environment”). the Word- and PDF-files of the survey. These experts Whilst the current study provides a unique, world- were therefore unable to submit their survey responses, wide, multidisciplinary expert perspective on function- as a result of which their expert opinion was not ing and disability in individuals with ASD, it cannot be included in the final results of this study. However, assumed that it is complete, exhaustive or representative these were just very few participants (n5 3), unlikely to of the totality of experts around the globe. For instance, have affected the study validity. even though experts included in the study represented a It is important to see the findings of the current broad range of different professions and all six of the study in the context of the larger project. This study WHO-regions, some professions were represented by is the second of four studies conducted in the process very few experts or no experts at all in certain regions. of developing ICF Core Sets for ASD. The first, a sys- As such, the identified categories may reflect in part the tematic literature review aimed to capture the research de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 967 INSAR perspective of functioning and disability related to (now renamed FORTE), FORMAS, and VINNOVA (trans- ASD, and was previously reported in this journal [De disciplinary research program on child and youth mental Schipper, et al., 2015). The current study showed that health, grant nr. 259-2012-24). ASD is not only related to disability, but also to strengths and abilities (e.g., honesty, attention to detail, persistence, etc.). These findings are inconsis- Conflict of interest tent with the results that the systematic literature review study yielded, which only provided evidence Elles de Schipper declares that she has no conflict of for disabilities. One reason for the inconsistency might interest related to this work. Soheil Mahdi declares that be related to the fact that in the expert survey a spe- he has no conflict of interest related to this work. Petrus cific question was designed to capture the possible J de Vries declares no conflict of interest related to this strengths and abilities pertaining to ASD, whereas in work. He has served as study steering committee mem- the systematic literature review this option was not ber, advisory board member, and working group mem- available. Moreover, categories from the Body struc- ber for a number of studies on Tuber Sclerosis Complex tures component (e.g., brain, intestine and upper funded or part-funded by Novartis. Mats Granlund extremities) were identified in this study, but not in declares no conflict of interest related to this work. the systematic literature review. These two studies also Martin Holtmann declares no conflict of interest related differed in respect to the categories that were identi- to this work. In the last 5 years he served in an advisory fied in the Body functions component. While both or consultancy role for Lilly and Shire, and received studies found that mental functions were affected in conference attendance support or was paid for public ASD, the expert survey also showed aspects of movement- speaking by Bristol-Myers Squibb, Janssen-Cilag, Lilly, related functioning (e.g., coordination and control of vol- Medice, Neuroconn, Novartis and Shire. Sunil Karande untary movement functions) and mobility (e.g., fine declares no conflict of interest related to this work. hand use) to be very relevant in ASD. Consistent with the Omar Almodayfer declares no conflict of interest related literature review, the core defining features of ASD, social- to this work. Cory Shulman declares no conflict of inter- communication characteristics, were captured by ICF(-CY) est related to this work. Bruce Tonge declares no conflict categories from the interpersonal interaction and commu- of interest related to this work. Virginia C.N. Wong nication chapters in the current study. declares no conflict of interest related to this work. The results acquired from the expert survey demon- Lonnie Zwaigenbaum declares no conflict of interest strate the complexity of ASD. Thus, enhanced communi- related to this work. He has in the last 3 years received cation between different stakeholders is required in grant funding from SynaptDx. Sven B€ olte declares no order to improve functioning and quality of life for indi- conflict of interest related to this work. He has in the viduals with ASD. The ICF Core Sets for ASD can meet last 3 years acted as a consultant or lecturer for Shire, this objective by providing professionals and other stake- Roche, Eli Lilly, Prima Psychiatry, Kompetento, Expo holders with terminology and definitions of functioning Medica and Prophase, and receives royalties from that are universally applicable and understandable irre- Kohlhammer and Hogrefe/Huber publishers. spective of disciplinary, cultural and country borders. Acknowledgments References The development of the ICF Core Sets for ASD is a coop- American Psychiatric Association. (2013). Diagnostic and statis- erative effort of the WHO, the ICF Research Branch, a tical manual of mental disorders (5th ed.). Arlington, VA: partner of the WHO Collaboration Centre for the Family American Psychiatric Publishing. Brundson, V.E.A., & Happ e, F. (2014). Exploring the ’fractiona- of International Classifications in Germany (at DIMDI), tion’ of autism at the cognitive level. Autism, 18, 17–30. the International Society for Autism Research (INSAR), Bolte, S. (2009). The ICF and its meaning for child and adoles- and the Center of Neurodevelopmental Disorders at the cent psychiatry. Zeitschriftfur Kinder-und Jugendpsychiatrie Karolinska Institutet (KIND) in Sweden. Guidance on this und Psychotherapie, 37, 495–497. project is provided by a Steering Committee comprised of Bolte, € S., de Schipper, E., Holtmann, M., Karande, S., de Vries, key opinion leaders in the field of ASD from all six WHO P.J., Selb, M., & Tannock, R. (2014a). Development of ICF regions. This Steering Committee consists of the co- Core Sets to standardize assessment of functioning and authors of this paper and John E. Robison, Melissa Selb, impairment in ADHD: the path ahead. European Child and Nidhi Singhal, Susan Swedo and Bedirhan Ustun. The Adolescent Psychiatry, 23, 1139–1148. development of ICF Core Sets for ASD is supported by the Bolte, € S., de Schipper, E., Robison, J.E., Wong, V.C.N., Selb, M., Swedish Research Council (grant nr. 523-2009-7054), and Singhal, N., de Vries, P.J., & Zwaigenbaum, L. (2014b). Clas- the Swedish Research Council in partnership with FAS sification of functioning and impairment: The 968 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR Development of ICF Core Sets for Autism Spectrum Disor- gender differences in autism spectrum disorder: Summarizing der. Autism Research, 7, 167–172. evidence gaps and identifying emerging areas of priority. Bolte, S., Dukretis, E., Poustka, F., & Holtmann, M. (2011). Sex Molecular Autism, 6, 36. doi:10.1186/s13229-015-0019-y. differences in cognitive domains and their clinical corre- Happ e, F., & Frith, U. (2009). 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Journal of Developmental & Physical Children and Youth version (ICF(-CY)). European Child & Disabilities, 27, 119–139. Adolescent Psychiatry, 24, 859–872. € Selb, M., Escorpizo, R., Kostanjsek, N., Stucki, G., Ustun, € B., De Schipper, E., Lundequist, A., Coghill, D., de Vries, P.J., &Cieza, A. (2014). A guide on how to develop an Interna- Granlund, M., Holtmann, M., Jonsson, U., Karande, S., tional Classification of Functioning, Disability and Health Robison, J.E., Shulman, C., Singhal, N., Tonge, B., Wong, Core Set. European Journal of Physical and Rehabilitation V.C., Zwaigenbaum, L. & Bolte, S. (2015b). Ability and dis- Medicine, 51, 105–117. ability in autism spectrum disorder: A systematic literature Szatmari, P., Xiao-Qing, L., Goldberg, J., Zwaigenbaum, L., review employing the international classification of func- Paterson, A.D., Woodbury-Smith, M., Georgiades, S., Duku, E., tioning, disability and health-children and youth version. & Thompson, A. (2011). 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International classification WHO’s International Classification of Diseases (ICD) and of functioning, disability and health: ICF. Geneva: World International Classification of Functioning, Disability and Health Organization. Health (ICF): Importance and methods to link disease and World Health Organization. (2007). International classification functioning. BMC Public Health, 13, 742. of functioning, disability and health: Children and youth Granberg, S., Swanepoel, D.W., Englund, U., Moller, C., & version: ICF-CY. Geneva: World Health Organization. Danermark, B. (2014).The ICF core sets for hearing loss pro- ject: International expert survey on functioning and disabil- ity of adults with hearing loss using the international Supporting Information classification of functioning, disability and health (ICF). International Journal of Audiology, 53, 497–506. Additional Supporting Information may be found in the Halladay, A.K., Bishop, S., Constantino, J.N., Daniels, A.M., online version of this article at the publisher’s web-site: Koenig, K., Palmer, K., Messinger, D., Pelphrey, K., Sanders, S.J.,Singer, A.T.,Taylor, J.L.,& Szatmari,P.(2015).Sex and Appendix S1. The ASD expert survey de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 969 INSAR http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Autism Research Wiley

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Abstract

RESEARCH ARTICLE Functioning and Disability in Autism Spectrum Disorder: A Worldwide Survey of Experts Elles de Schipper,† Soheil Mahdi,† Petrus de Vries, Mats Granlund, Martin Holtmann, Sunil Karande, Omar Almodayfer, Cory Shulman, Bruce Tonge, Virginia V.C.N. Wong, Lonnie Zwaigenbaum, and Sven Bolte Objective: This study is the second of four to prepare International Classification of Functioning, Disability and Health (ICF; and Children and Youth version, ICF(-CY)) Core Sets for Autism Spectrum Disorder (ASD).The objective of this study was to survey the opinions and experiences of international experts on functioning and disability in ASD. Methods: Using a protocol stipulated by the World Health Organization (WHO) and monitored by the ICF Research Branch, an email-based questionnaire was circulated worldwide among ASD experts, and meaningful functional ability and disability concepts were extracted from their responses. These concepts were then linked to the ICF(-CY) by two independent researchers using a standardized linking procedure. Results: N5 225 experts from 10 different disciplines and all six WHO-regions completed the survey. Meaningful concepts from the responses were linked to 210 ICF(-CY) categories. Of these, 103 categories were considered most relevant to ASD (i.e., identified by at least 5% of the experts), of which 37 were related to Activities and Participation,35 to Body functions,22to Environmental factors, and 9 to Body structures. A variety of personal characteristics and ASD-related functioning skills were provided by experts, including honesty, loyalty, attention to detail and creative talents. Reported gender differences in ASD comprised more externaliz- ing behaviors among males and more internalizing behaviors in females. Conclusion: The ICF(-CY) categories derived from international expert opinions indicate that the impact of ASD on functioning extends far beyond core symptom V C domains. Autism Res 2016, 9: 959–969. 2016 The Authors Autism Research published by Wiley Periodicals, Inc. on behalf of International Society for Autism Research Keywords: autism; neurodevelopmental disorder; assessment; child psychiatry; heterogeneity; quality of life Introduction comorbid conditions (e.g., ADHD, epilepsy, and gener- alized anxiety disorder), in combination with factors A multitude of research provides information on the pertaining to the environment like access to interven- impact of Autism Spectrum Disorder (ASD) on function- tion programs and availability of support services ing in everyday life. Poor outcomes are reported in accounting for the largest part of the variability [Brund- terms of education, employment, social relationships, son & Happe, 2014; Levy & Perry, 2011]. There is also independent living, mental health, and quality of life evidence that ASD is associated with certain gender dif- [Bolte & Poustka, 2002; Howlin & Moss, 2012; Lai & ferences [Bolte, € Dukretis, Poustka, & Holtmann, 2011; Lombardo, Baron-Cohen, 2014]. Studies also reveal sub- € Halladay et al., 2015; Holtmann, Bolte, & Poustka, stantial interindividual differences in outcomes and lev- 2007] and strengths [Happe & Frith, 2009; Mottron, els of functioning for individuals with ASD, with factors Dawson, & Soulie `res, 2009] such as visual spatial, ana- such as intelligence quotient, language abilities, and lytical or savant skills that may positively impact on an From the Department of Women’s and Children’s Health, Center of Neurodevelopmental Disorders (KIND), Pediatric Neuropsychiatry Unit, Karolin- ska Institutet, Stockholm, Sweden (E.S., S.M., S.B.,); Child and Adolescent Psychiatry, Center of Psychiatry Research, Stockholm County Council, Stockholm, Sweden (E.S., S.M., S.B.,); Division of Child & Adolescent Psychiatry, University of Cape Town, Cape Town, South Africa (P.V.,); CHILD, SIDR, Jonk € oping € University, Jonk € oping, € Sweden (M.G.,); LWL-University Hospital for Child and Adolescent Psychiatry, Ruhr-University Bochum, Hamm, Germany (M.H.,); Learning Disability Clinic, Department of Pediatrics, Seth G.S. Medical College & K.E.M. Hospital, Mumbai, India (S.K.,); Psychiatry Section, King Abdulaziz Medical City, College of Medicine, Riyadh, Saudi-Arabia (O.A.,); Paul Baerwald School of Social Work and Social Welfare, Hebrew University of Jerusalem, Jerusalem, Israel (C.S.); Centre for Developmental Psychiatry and Psychology, Monash University, Melbourne, Victoria, Australia (B.T.,); Department of Paediatrics and Adolescent Medicine, The University of Hong Kong, Hong Kong SAR, China (V.C.N.W.,); Department of Pediatrics, University of Alberta, Edmonton, Alberta, Canada (L.Z.) E.S. and S.M. contributed equally to this work. Received August 19, 2015; accepted for publication November 27, 2015 This is an open access article under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made. Address for correspondence and reprints: Sven Bolte, € Department of Women’s and Children’s Health, Center of Neurodevelopmental Disorders (KIND), Pediatric Neuropsychiatry Unit, Karolinska Institutet, Stockholm, Sweden. E-mail: sven.bolte@ki.se Published online 08 January 2016 in Wiley Online Library (wileyonlinelibrary.com) DOI: 10.1002/aur.1592 V 2016 The Authors Autism Research published by Wiley Periodicals, Inc. on behalf of International Society for Autism Research Autism Research 9: 959–969, 2016 959 INSAR individual’s level of functioning and quality of life. mostly because in its current full form it appears too Although the fifth edition of the Diagnostic and Statis- generic for certain health conditions and impractical for tical Manual of Mental Disorders (DSM-5) [American daily use. To address this issue the development of ICF Psychiatric Association, 2013] provides novel options Core Sets was initiated. ICF Core Sets are shortlists of ICF(- for individualizing diagnosis in ASD through specifiers CY) categories that are considered most relevant to indi- and severity/need of support ratings, it does neither viduals with a certain health condition in clinical contexts. provide explicit definitions for disability nor for adaptive These ICF Core Sets are developed following a rigorous functioning. The International Classification of Func- qualitative and quantitative, scientific protocol stipulated tioning (ICF; [WHO, 2001]) complements the Interna- by the WHO and ICF Research Branch [Kostanjsek et al., tional Classification of Diseases-Tenth Revision [ICD-10; 2011]. Its main objective is to use existing research data, World Health Organization (WHO), 1992], conceptualiz- involve a large and multi-professional selection of ing functioning and disability as being separate from researchers and clinicians, as well as service users/patients pathology. It applies a biopsychosocial perspective to and their caregivers from all over the world to ensure uni- operationalize an individual’s level of functioning as the versal applicability of the ICF Core Sets. The protocol con- interplay of abilities and disabilities that emerge in the sists of a systematic literature review (“research contexts of physical constitution, social participation, perspective”), an expert survey (current study, “opinion personal as well as environmental resources [WHO, leader perspective”), focus groups (“client and caregiver 2001]. Moreover, the ICF facilitates standardized assess- perspective”), and a cross-sectional clinical study ment of functioning and health by providing detailed (“clinical perspective”). Thus, the present study is embed- classifications in the areas of Body functions (i.e., physio- ded into the process of a larger research and development logical functions of body systems), Body structures (i.e., project, aimed at developing standardized ICF Core Sets anatomical parts of the body), Activities (i.e., execution for ASD. ICF Core Sets for ADHD are being developed as of tasks), Participation (i.e., involvement in life situa- well in this project, but the results are reported in sepa- tions), and Environmental factors (i.e., physical, social and € rate publications [Bolte et al., 2014a; De Schipper et al., attitudinal environment). The bio-psycho-social model 2015a]. In order to accomplish ICF Core Sets that cover also includes Personal factors, but these have not yet been functioning as well as disabilities in ASD across the life classified in the ICF given how these are grounded in span, the development will be based on the ICF(-CY), the social and cultural context. To capture the particular sit- extended version of the ICF, that captures not only func- uation of a developing individual, the ICF Children and tioning in adulthood but also childhood and youth. A Youth version [ICF(-CY); WHO, 2007] was derived from complete description of the overall ASD ICF Core Set the ICF by adding development-relevant categories and development process has been published in a previous expanding on the descriptions of existing ICF categories. issue of this journal [Bolte € et al., 2014b]. The ICF and ICF(-CY) (hereafter, “ICF(-CY)”) consist The objective of this study was to capture the experi- of hierarchically structured categories describing aspects ences and opinions of experts in the assessment and of functioning in the three key components of the bio- treatment of ASD. Together with the other three prepar- psycho-social model described above (i.e., Body Func- atory studies mentioned above, this expert survey gen- tions and Structures, Activities & Participation, Environ- erates content for an international ICF Core Sets mental factors) in up to four levels of increasing detail. Consensus Conference, during which a group of ASD The first level categories are known as chapters, and pro- experts from all WHO regions will follow a formal vide a general overview of the areas of functioning that decision-making process to arrive at ICF(-CY) categories are covered in the ICF(-CY). The chapters consist of sec- to be included in the ICF Core Sets for ASD. ond, third and fourth level categories. The following example of an ASD-relevant classification from the Body Methods functions component shows the hierarchical structure of the ICF(-CY): Design Level 1 chapter: b1 Mental functions In an email-based survey, professionals from various Level 2 category: b167 Mental functions of language disciplines worldwide provided their perspectives on Level 3 category: b1671 Expression of language which features of ability, disability and context are to Level 4 category: b16710 Expression of spoken language be considered essential for functioning in everyday life of individuals with ASD. Consisting of over 1600 categories, the ICF(-CY) pro- vides a comprehensive framework for the classification of Recruitment Procedure an individual’s functioning. However, despite its potential value to health care and research [Bolte, 2009; Escorpizo An internet search was performed to identify contact et al., 2013], the ICF(-CY)’s use is still rather limited, information for internationally known ASD experts and 960 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR Figure 1. An overview of the recruitment process. for centers (academic and community-based), clinics and were then contacted via email and received information university departments in all WHO-regions regularly about the study along with a request to participate in involved in the assessment and management of individ- the survey. If they agreed to participate, they were asked uals with ASD. Identified organizations were contacted to fill in a reply sheet with information about their work via email with information about the study and a experience to confirm their eligibility. After confirma- request to provide contact information of eligible tion, the experts received the survey as a Word or PDF experts. Contact information of experts was also pro- file via email, which they were requested to fill in and vided by the project Steering Committee (see acknowl- return within one month. Experts who did not submit edgement), a group of key opinion leaders in ASD from the survey on time received up to three reminders to all WHO-regions providing guidance on the project, and return their survey response and, if requested, they by the authors’ personal professional networks. Finally, received extended time to respond. Data collection took snowball sampling was applied, as all contacted experts place between September 2013 and October 2014. were requested to recommend additional experts to be recruited for the survey. To be considered an “expert” Participating Experts for the purposes of this study, potential participants Experts were approached via email with an invitation to were required to (a) practice in one or more of the fol- participate in the survey. The majority of them accepted lowing professions: coach, counselor, nurse, occu- the invitation. There were also some who did not pational therapist, physician (e.g., psychiatrists, respond to the invitation or declined the invitation. neurologists, etc.), physiotherapist, psychologist, psycho- Reasons for declining the invitation were mostly a lack therapist, social worker, special educator, or speech and of time, but there were also some who felt their exper- language therapist; (b) have at least five years of experi- ence in the assessment and/or treatment of individuals tise was more theoretical than practical. Moreover, there with ASD; (c) fluency in English. These identified experts were also experts who were ineligible to participate in de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 961 INSAR Figure 2. Representation of professions per WHO region (n5 245). (43 different countries). See Figure 2 for an overview of Table 1. Representation of participants per professional group (some experts have been duplicated due to their the representation of professions per WHO region and multiprofessional background) Table 1 for participants per professional group. Participating experts ranged in age between 26 and 74 Profession N (%) years, with a mean age of 47 years (SD510.8). They had Physician 54 (22%) on average 16 years of experience in working with indi- Occupational therapist 50 (20%) viduals with ASD (SD58.0), ranging between 5 and 43 Psychologist 36 (15%) Special educator 27 (11%) years. The majority of participating experts were females Physiotherapist 23 (9%) (76%). At the time of the survey, the participants spent Speech & language therapist 23 (9%) the majority of their time working in clinical depart- Psychotherapist 14 (6%) ments (45%), education (16%), or research (11%). A Social worker 8 (4%) Nurse 7 (3%) small group of experts worked mainly in management, Coach 3 (1%) or in other fields, such as training or supervision of other professionals (5%). The remaining experts divided their work time among more than one field (19%), mostly A profession in which an individual supports clients in achieving combining their work in the clinic with education or different goals in life, for example, improving school grades, finding a job, and so forth. research. A proportion of 39% of experts worked with children and adolescents exclusively, 38% with children only, and 12% with ASD across the lifespan. A minority the study (<5 years of experience). Participating experts of the participants (2%) worked exclusively with adoles- received up to two reminders to return their survey cents and 4% with adults only, or a combination of these response. An overview of the recruitment process is pro- two age groups (4%). Three experts (1%) did not specify vided in Figure 1. the age group of clients they encountered the most. Thus, complete survey responses were obtained from 225 experts. A small minority of participating experts Expert Survey stated that they were practicing more than one profes- The expert survey consisted of three parts (see Support- sion, for example, worked both as a psychologist and as a psychotherapist. Participating experts represented 10 ing Information Appendix S1). Part one contained ques- different professions and all six of the WHO-regions tions about the participating expert’s demography. Part 962 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR two consisted of six questions regarding functioning and 85% at the level of ICF(-CY) chapters. Cohen’s and disability in individuals with ASD. The questions kappa and confidence intervals were calculated to examine the extent to which the agreements exceed were specifically formulated to cover each component chance. The kappa value was j50.81 (SE50.004) with a of the bio-psycho-social of the ICF(-CY), using a similar 95% confidence interval of 0.80–0.81 for the second- approach as in previous ICF core set developments level categories. At the level of ICF(-CY) chapters the [Coenen et al., 2011]. Part two of the survey included Kappa value was j50.84 (SE50.004) with a confidence two further questions, one concerning the possible interval of 0.83–0.84. These Kappa values indicate functional strengths in ASD and the second possible excellent interrater agreement. gender differences in functioning and disability. Part three of the survey contained information regarding Data Analysis privacy and terms of agreement for taking part in the Frequency analysis was used to analyze the responses to expert survey. the six ICF(-CY) based questions of the survey. The Identification of Meaningful Concepts and linking to the absolute number of expert survey responses in which ICF(-CY) each of the ICF(-CY) categories were identified, along with the corresponding percentages relative to the total “Meaningful Concepts” are concise descriptions of spe- number of responses received, was examined. ICF(-CY) cific behaviors, skills or other aspects of functioning categories are presented at the second-level. If a concept that are to be linked to ICF(-CY) categories. These con- was linked to a third- or fourth-level ICF(-CY) category, cepts were extracted from the experts’ survey responses the corresponding second-level category was reported. and then linked to ICF(-CY) categories following for- Because the ICF(-CY) is organized hierarchically, aspects malized linking rules and procedures determined by the of the more specific third- and fourth-level categories WHO ICF Research Branch [Cieza et al., 2002; Cieza were included in the less specific second-level catego- et al., 2005]. The linking rules provide guidance on ries. Following the ICF Core Sets development guide- how to link concepts to ICF(-CY) categories, and what lines [Selb et al., 2014], a second-level ICF(-CY) category to do when that is not possible. Specific codes assigned that was identified repeatedly in the same expert survey to these concepts in the latter situation are (a) Personal response was counted only once. Consistent with WHO factor, if the concept is not contained in the ICF(-CY), and previous core set development conventions [Escor- but is clearly a Personal factor as defined in the ICF(- pizo et al., 2011; Granberg et al., 2014], only ICF(-CY) CY); (b) non- covered, if the concept is not contained in categories that were identified in at least 5% of the the ICF(-CY) and also is not a Personal factor; (c) non- expert survey responses were included in the list of definable, when the information provided in the con- potential categories to be included in the Core Set for cept is not sufficient to assign it to a specific ICF(-CY) ASD. This is done to ensure that only those categories category; and (d) health condition, if the concept refers that are most relevant to a certain condition, in this to a diagnosis or health condition. Given that there are case ASD, are included. Additional frequency analysis many different ways to describe the same aspect of was used to explore the possible relation between functioning, it is possible for different meaningful con- experts’ self-identified expertise with respect to stage of cepts to be assigned to the same ICF(-CY) category. development and the ICF(-CY) categories identified in To assure the quality and consistency of results, both their survey responses. The last questions regarding spe- the identification of meaningful concepts (e.g., difficul- cific abilities related to ASD and possible gender differ- ties in decision making) and the linking to ICF(-CY) ences in functioning were not linked due to the fact categories (e.g., d177 Making decisions) was conducted that responses were inconsistent and heterogeneous, independently by two researchers (ES and SM), who and therefore it was impossible to define meaningful had received extensive linking training by the ICF concepts for linking. Instead, the answers were carefully Research Branch prior to the project. Linking results of reviewed independently by two researchers (ES and SM) each of the researchers were compared and consensus before recurring themes or patterns of answers were discussions were used to resolve disagreements. If con- summarized. sensus could not be reached, the coordinator of the ICF Research Branch (MS) was available to make final deci- Results sions. However, this option was never used because dis- agreements were resolved by discussion between the Linking Results two researchers. The overall percentage of agreement between the two researchers (prior to consensus in case In total, 8792 meaningful concepts were extracted from of disagreement) for the linking process was calculated. 225 survey responses. These concepts were linked to This was 81% for the second-level ICF(-CY) categories 210 second-level ICF(-CY) categories, 191 Personal de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 963 INSAR Table 2. Absolute and relative frequencies of ICF(-CY) Table 3. Absolute and relative frequencies of ICF(-CY) categories from the Activities and Participation component categories from the Body Functions component Second level category N (%) Second level category N (%) d720 complex interpersonal interactions 101 (45%) b760 control of voluntary movement functions 156 (69%) d440 fine hand use 89 (40%) b164 higher-level cognitive functions 151 (67%) d710 basic interpersonal interactions 85 (38%) b147 psychomotor functions 117 (52%) d446 fine foot use 70 (31%) b156 perceptual functions 108 (48%) d230 carrying out daily routine 68 (30%) b152 emotional functions 105 (47%) d310 communicating with—receiving—spoken messages 62 (28%) b125 dispositions and intra-personal functions 97 (43%) d335 producing nonverbal messages 59 (26%) b167 mental functions of language 82 (36%) d570 looking after one’s health 59 (26%) b765 involuntary movement functions 81 (36%) d820 school education 59 (26%) b140 attention functions 80 (36%) d920 recreation and leisure 56 (25%) b265 touch function 75 (33%) d750 informal social relationships 55 (24%) b230 hearing functions 71 (32%) d177 making decisions 47 (21%) b117 intellectual functions 62 (28%) d250 managing one’s own behavior 47 (21%) b260 proprioceptive function 61 (27%) d330 speaking 47 (21%) b210 seeing functions 60 (27%) d315 communicating with—receiving—nonverbal messages 46 (20%) b255 smell function 52 (23%) d240 handling stress and other psychological demands 31 (14%) b270 sensory functions related to temperature 52 (23%) d530 toileting 30 (13%) and other stimuli d830 higher education 27 (12%) b130 energy and drive functions 49 (22%) d510 washing oneself 26 (12%) b250 taste function 49 (22%) d350 conversation 25 (11%) b163 basic cognitive functions 48 (21%) d550 eating 24 (11%) b235 vestibular functions 45 (20%) d880 engagement in play 24 (11%) b735 muscle tone functions 45 (20%) d130 copying 23 (10%) b122 global psychosocial functions 41 (18%) d760 family relationships 23 (10%) b180 experience of self and time functions 37 (16%) d825 vocational training 23 (10%) b160 thought functions 35 (16%) d845 acquiring, keeping and terminating a job 21 (9%) b134 sleep functions 34 (15%) d220 undertaking multiple tasks 20 (9%) b770 gait pattern functions 34 (15%) d520 caring for body parts 20 (9%) b126 temperament and personality functions 31 (14%) d540 dressing 20 (9%) b176 mental function of sequencing complex 24 (11%) d571 looking after one’s safety 19 (8%) movements d163 thinking 18 (8%) b144 memory functions 22 (10%) d740 formal relationships 16 (7%) b515 digestive functions 19 (8%) d210 undertaking a single task 15 (7%) b330 fluency and rhythm of speech functions 18 (8%) d850 remunerative employment 15 (7%) b114 orientation functions 17 (8%) d455 moving around 14 (6%) b280 sensation of pain 17 (8%) d160 focusing attention 12 (5%) b320 articulation functions 15 (7%) d770 intimate relationships 12 (5%) b310 voice functions 12 (5%) factors (e.g., self-esteem, restricted interests), 611 non- Activities and Participation component are presented in definable codes (e.g., structured environment, behavior Table 2, along with the number and percentage of problems), 208 noncovered codes (e.g., stress, social expert survey responses in which they were identified. rejection), and 41 health condition-codes (e.g., depres- They cover eight of the nine chapters making up the sion, dyspraxia, epilepsy). The nondefinable codes and component, that is, (d5) self-care,(d7) interpersonal inter- noncovered codes were mostly identified in the ques- actions and relationships,(d8) major life areas,(d2) general tions that captured the environmental factors of func- tasks and demands,(d3) communication,(d1) learning and tioning and disability, whereas the health-condition applying knowledge,(d4) mobility, and (d9) community, codes were mostly applied in questions related to Body social and civic life. Table 3 presents the absolute and relative frequencies functions and Body structures component. Of the identified ICF(-CY) categories, 103 were found of the second-level categories that were identified in in the responses of at least 5% of the experts (range: 5– the Body functions component. Five out of the eight 87%) and are included in the list of candidate catego- chapters making up this component are represented in ries. Each of the four components of the ICF(-CY) are these categories. The majority of categories were identi- represented in these categories, with 37 categories from fied in the chapter (b1) mental functions. The remainder Activities and Participation, 35 from Body functions,22 of categories was identified in four additional chapters, from Environmental factors, and 9 from Body structures. that is, (b2) sensory functions and pain,(b7) neuromuscu- The second-level categories that were identified in the loskeletal and movement-related functions,(b3) voice and 964 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR Table 4. Absolute and relative frequencies of ICF(-CY) Table 5. Absolute and relative frequencies of ICF(-CY) categories from the Environmental Factors component categories from the Body Structures component Second level category N (%) Second level category N (%) e310 immediate family 100 (44%) s110 structure of brain 195 (87%) e585 education and training services, systems and 100 (44%) s540 structure of intestine 34 (15%) policies s730 structure of upper extremity 18 (8%) e580 health services, systems and policies 84 (37%) s750 structure of lower extremity 16 (7%) e115 products and technology for personal use in 68 (30%) s530 structure of stomach 15 (7%) daily living s520 structure of esophagus 14 (6%) e460 societal attitudes 51 (23%) s260 structure of inner ear 12 (5%) e410 individual attitudes of immediate family members 41 (18%) s320 structure of mouth 11 (5%) e125 products and technology for communication 37 (16%) s770 additional musculoskeletal structures related 11 (5%) e250 sound 37 (16%) to movement e590labor and employment services, systems and policies 34 (15%) e360 other professionals 28 (12%) e575 general social support services, systems and policies 26 (12%) Additional frequency analysis of 10% of the expert sur- e240 light 22 (10%) vey responses was conducted in order to explore a possible e165 assets 19 (8%) relationship between the age group in which the experts e340 personal care providers and personal assistants 17 (8%) are specialized in and the ICF(-CY) categories that were e355 health professionals 17 (8%) identified in their survey responses. The analysis showed e425 individual attitudes of acquaintances, peers, 17 (8%) colleagues, neighbors and community members that a large majority of the categories in the survey e455 individual attitudes of health-related professionals 15 (7%) responses were identified across the lifespan. There were, e465 social norms, practices and ideologies 15 (7%) however, a few categories more often identified for chil- e155 design, construction and building products and 14 (6%) dren as compared to adults: e310 Immediate family, e585 technology of buildings for private use e325 acquaintances, peers, colleagues, neighbors and 12 (5%) Education and training services, systems and policies, d440 community members Fine hand use, d446 Fine foot use and d740 Informal social e315 extended family 11 (5%) relationships. Given that children usually live with their e450 individual attitudes of health professionals 11 (5%) immediate family and attend school on a regular basis, some of the categories (e.g., e310 Immediate family and e585 Education and training services, systems and policies) speech functions, and (b5) functions of the digestive, meta- covered in the experts’ survey responses are not surprising. bolic and endocrine systems. Four out of the five most often identified categories in this component are mental ASD-Related Abilities functions. The most often identified category pertains to A large majority of experts (92%) indicated observed movement-related function. Absolute and relative frequencies of the second-level ASD-related skills, many of which were reported to be categories identified in the Environmental factors com- mainly associated with higher functioning individuals. ponent are presented in Table 4. All five of this compo- The experts further remarked that these skills need to nent’s chapters are represented by these categories, be actively recognized and nurtured in order for the that is, (e3) support and relationships,(e4) attitudes,(e1) individuals to reach their full potential. Skills and abil- products and technology,(e5) services, systems and policies, ities that were often mentioned by the experts are sum- and (e2) natural environment and human-made changes to marized in Table 6. environment. Gender Differences Table 5 shows the second-level categories that were identified in the Body structures component, together Sixty percent of the surveyed experts reported gender- with their absolute and relative frequencies. These rep- related differences in ASD. Of those, more than half resent five out of eight chapters making up this compo- replied that they had observed gender differences in nent. By far the most frequently identified category was functioning and disabilities. Some experts highlighted from chapter (s1) structures of the nervous system, which that these differences were dependent on the age range was mentioned by 87% of the experts, making it the and intellectual capacities, and that observed differen- most often identified of all ICF(-CY) categories in this ces might not necessarily be specific to ASD, but rather, study. Other categories from this component were reflect gender differences in the general population. derived less often, and represent four additional chap- Other experts indicated that males with ASD tend to ters, that is, (s5) structures related to the digestive, meta- show more externalizing behavioral difficulties (hyper- bolic and endocrine systems,(s7) structures related to activity, aggression) than females, making them easier movement,(s2) the eye, ear and related structures, and (s3) to diagnose. On the other hand, females with ASD were structures involved in voice and speech. described to be better socially adjusted, showing more de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 965 INSAR Table 6. Overview of ASD-related skills and abilities men- tion schedules, questionnaire, interviews) will be tioned by the experts derived and standardized for broad use in clinical and research settings. These tools will be ready made and Abilities and skills easily implementable for clinicians. Attention to detail The present study identified a large number of catego- Strong sense of morality (e.g., honesty, lack of judgmental ries covering all four components contained in the ICF(- attitude, etc.) A preference to work on repeated or monotonous tasks CY). It underlines that experts accept ASD to be a com- Expertise in a specific area plex condition involving many physical, environmental Mathematical abilities and personal factors and areas of life. The most fre- Creative talents (e.g., look at the world differently) quently identified category was (s110) structure of brain Artistic skills (e.g., music, drawing, visual arts) from the Body structures, indicating that there is consen- Visual perception Intellectual functions sus among international experts of various professions Technical abilities (computer skills, engineering) that ASD is a brain-based condition. However, as indi- Trustworthiness cated by other derived categories (s2, s3, s5, s7), body Loyalty structures related to voice and speech, movement and Kindness digestive system are also presumed to be closely related Good memory to ASD. Not surprisingly, multiple categories from the Activities and Participation component were derived, such prosocial behaviors, communication skills and friendships as problems with social relationships (d7) and communi- than males, making them easily overlooked. Additional cation (d3), which constitute the core symptoms of ASD experts perceived females and males with ASD to share as described in the DSM-5. Nevertheless, the majority of core abilities and disabilities, but also experienced that identified categories represent other challenges, includ- females invested more efforts in blending in, complying ing the ability to take care of oneself and manage every- with social demands and hiding their difficulties. Experts day life and tasks (d5, d2), participation in school, work concluded that this might not only lead to underdiagnos- and social life (d8, d9), and cognitive and motor skills ing females, but also to higher expectations on females (d1, d4). Categories identified in the Body functions com- with ASD, which in turn results in increased levels of ponent concern a rich variation of mental functions stress and anxiety, and a greater sense of failure, com- (b1), such as language (b167), perception (b156), pared to males with ASD. Males with ASD were described emotion (b152), and attention (b140). Furthermore, cate- as more pronounced in socially unadjusted and unac- gories from other chapters suggest a broader range of cepted behaviors, leading to more social exclusion, fewer ASD-related problems, for example, sensation (b2), prob- friendships, and more bullying. Males as compared to lems with speech (b3), and with digestion (b5). Finally, females with ASD seemed on the other hand less emo- the categories identified from the Environmental factors tionally affected by social exclusion, showing a lower component indicate that the social context, both indi- average desire for social participation. vidual support and common attitudes towards individu- als with ASD (e3, e4), organized support and services Discussion provided by professionals (e5), the physical context and environment (e1, e2) (products designed to be helpful The objective of the present study was to characterize for coping with challenges in everyday life; physical functioning aspects and disabilities that are often asso- environment in general), interact with an individual’s ciated with ASD, based on the perspectives of experts functioning and disabilities in determining their level of who assess and treat the disorder. To this end, an everyday functioning. These findings endorse the useful- email-based survey was conducted among a large sam- ness of the bio-psycho-social model of the ICF(CY), ple of international experts from multiple professions. which describes functioning as a dynamic interaction The experiences and opinions of such a large and between health condition and contextual factors. diverse international group of ASD experts on this topic This study is among the first to explore specific have not been systematically examined previously. The strengths and gender differences in ASD as perceived by list of candidate ICF(-CY) categories derived from the experts from different professions and cultures. Overall, current study will be combined with the identified cate- opinions put forward were too heterogeneous and gories from three additional preparatory studies to form inconsistent to derive meaningful concepts and link the basis for an “ICF Core Sets for ASD” consensus con- ICF categories. Thus, this survey data was analyzed ference. At this conference a group of international qualitatively, in which recurring aspects of functioning ASD-experts will determine which ICF(-CY) categories and abilities mentioned by the experts were summar- should be included in the first official ICF Core Sets for ized without additional frequency analysis. Our survey ASD, on the basis of which diagnostic tools (observa- data should, however, in the future be more 966 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR comprehensively examined using thematic analyzes in composition of experts who were surveyed. Notably, a separate study. Many experts reported both certain there may be certain professions (e.g., psychologists) abilities and positive traits linked to ASD and pheno- whose role is not yet established in the assessment or types related to gender in ASD. Some of the survey find- treatment of individuals with ASD in some countries ings such as an overrepresentation of savant and (e.g., Azerbaijan). Restricting the survey to professionals scientific talents in ASD are supported by research [e.g., proficient in the English language may have also limited Treffert, 2014], while others, such as a strong sense of participation in some WHO-regions. For instance, in justice, loyalty and helpfulness currently have not been regions or countries where English is not commonly systematically investigated. Surprisingly, research find- used, such as Central and South-America (e.g., Mexico ings of gender differences in ASD, such as repetitive and Uruguay) and the Middle East (e.g., Qatar), some behaviors being more pronounced in males than invited experts were unable to participate due to this females with ASD (Szatmari et al., 2011), were not con- restriction. The four preparatory studies combined will sistently reported by experts. On the other hand, differ- however make it possible for one study to fill in the gaps ing comorbidity profiles were often mentioned by the of another study. For example, certain countries (e.g., Bra- experts, despite the fact that research has shown mixed zil) or regions (e.g., the Middle East) may be underrepre- results [Holtmann et al., 2007; Rubenstein, Wiggins, & sented in this study and in the systematic literature Lee, 2014]. In summary, the expert survey data on these review given the requirement for publications to be in topics demonstrate not merely deficits, but also English and more general research opportunity inequal- strengths oriented views on ASD among experts, and a ities, but these will have a stronger representation in the widespread awareness that gender issues might be sig- upcoming qualitative and clinical studies. Language nificant for understanding ASD. Furthermore, they gen- should not be a barrier in these studies, since collabora- erate novel hypotheses based on clinical experience to tions with members from the Steering Committee and be tested in future studies. other international study sites will allow for the use of Certain meaningful concepts in the expert survey local languages. The focus group study will consist of responses which the experts identified could not be group discussions with different stakeholders (clients with translated into ICF(-CY) categories. Indeed, the large ASD, their families, professional caregivers), whereas the number of nondefinable codes (n5611) invites closer clinical study will involve investigation of problems as examination. Nondefinable codes are largely explained experienced by actual patients in real life clinical settings. by the fact that experts were free to answer the questions All in all, the results of the four studies together should in the survey in their own words and expressions. Even provide us with a comprehensive picture of the specific though the experts were instructed to be as concrete as abilities and disabilities that are related to functioning in possible in their survey responses, the answers provided ASD for individuals across different countries, cultures by them were sometimes very brief or ambiguous (e.g., and life situations. “behavior,” “participation”) or formulated in general The use of written survey might also have caused dif- terms (e.g., “social impairment,” “adaptive skills”). Con- ficulties in verifying that the questions were always sequently, this presented a challenge for the linking. interpreted correctly by the experts, as it would have Nevertheless, a closer look at the non-definable codes been possible in a face-to-face interview. Some experts revealed a few recurring themes, such as the level of (n5 17) indicated that they were unsure what was structure and routine in the environment (e.g., intended with a certain question and thus refrained “structured environment,” “daily routine”), the level of from answering. In other cases experts (n5 5) explicitly sensory input from the environment (e.g., “sensory over- stated that they may have misinterpreted the questions. load,” “limited distractions”), and the general adjust- Furthermore there were also technical issues to the ment of the environment to the individual’s needs and study resulting in some of the participating experts abilities (e.g., “autism-friendly environment,” “balance being unable to open or work with several versions of between abilities and demands from environment”). the Word- and PDF-files of the survey. These experts Whilst the current study provides a unique, world- were therefore unable to submit their survey responses, wide, multidisciplinary expert perspective on function- as a result of which their expert opinion was not ing and disability in individuals with ASD, it cannot be included in the final results of this study. However, assumed that it is complete, exhaustive or representative these were just very few participants (n5 3), unlikely to of the totality of experts around the globe. For instance, have affected the study validity. even though experts included in the study represented a It is important to see the findings of the current broad range of different professions and all six of the study in the context of the larger project. This study WHO-regions, some professions were represented by is the second of four studies conducted in the process very few experts or no experts at all in certain regions. of developing ICF Core Sets for ASD. The first, a sys- As such, the identified categories may reflect in part the tematic literature review aimed to capture the research de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 967 INSAR perspective of functioning and disability related to (now renamed FORTE), FORMAS, and VINNOVA (trans- ASD, and was previously reported in this journal [De disciplinary research program on child and youth mental Schipper, et al., 2015). The current study showed that health, grant nr. 259-2012-24). ASD is not only related to disability, but also to strengths and abilities (e.g., honesty, attention to detail, persistence, etc.). These findings are inconsis- Conflict of interest tent with the results that the systematic literature review study yielded, which only provided evidence Elles de Schipper declares that she has no conflict of for disabilities. One reason for the inconsistency might interest related to this work. Soheil Mahdi declares that be related to the fact that in the expert survey a spe- he has no conflict of interest related to this work. Petrus cific question was designed to capture the possible J de Vries declares no conflict of interest related to this strengths and abilities pertaining to ASD, whereas in work. He has served as study steering committee mem- the systematic literature review this option was not ber, advisory board member, and working group mem- available. Moreover, categories from the Body struc- ber for a number of studies on Tuber Sclerosis Complex tures component (e.g., brain, intestine and upper funded or part-funded by Novartis. Mats Granlund extremities) were identified in this study, but not in declares no conflict of interest related to this work. the systematic literature review. These two studies also Martin Holtmann declares no conflict of interest related differed in respect to the categories that were identi- to this work. In the last 5 years he served in an advisory fied in the Body functions component. While both or consultancy role for Lilly and Shire, and received studies found that mental functions were affected in conference attendance support or was paid for public ASD, the expert survey also showed aspects of movement- speaking by Bristol-Myers Squibb, Janssen-Cilag, Lilly, related functioning (e.g., coordination and control of vol- Medice, Neuroconn, Novartis and Shire. Sunil Karande untary movement functions) and mobility (e.g., fine declares no conflict of interest related to this work. hand use) to be very relevant in ASD. Consistent with the Omar Almodayfer declares no conflict of interest related literature review, the core defining features of ASD, social- to this work. Cory Shulman declares no conflict of inter- communication characteristics, were captured by ICF(-CY) est related to this work. Bruce Tonge declares no conflict categories from the interpersonal interaction and commu- of interest related to this work. Virginia C.N. Wong nication chapters in the current study. declares no conflict of interest related to this work. The results acquired from the expert survey demon- Lonnie Zwaigenbaum declares no conflict of interest strate the complexity of ASD. Thus, enhanced communi- related to this work. He has in the last 3 years received cation between different stakeholders is required in grant funding from SynaptDx. Sven B€ olte declares no order to improve functioning and quality of life for indi- conflict of interest related to this work. He has in the viduals with ASD. The ICF Core Sets for ASD can meet last 3 years acted as a consultant or lecturer for Shire, this objective by providing professionals and other stake- Roche, Eli Lilly, Prima Psychiatry, Kompetento, Expo holders with terminology and definitions of functioning Medica and Prophase, and receives royalties from that are universally applicable and understandable irre- Kohlhammer and Hogrefe/Huber publishers. spective of disciplinary, cultural and country borders. Acknowledgments References The development of the ICF Core Sets for ASD is a coop- American Psychiatric Association. (2013). Diagnostic and statis- erative effort of the WHO, the ICF Research Branch, a tical manual of mental disorders (5th ed.). Arlington, VA: partner of the WHO Collaboration Centre for the Family American Psychiatric Publishing. Brundson, V.E.A., & Happ e, F. (2014). Exploring the ’fractiona- of International Classifications in Germany (at DIMDI), tion’ of autism at the cognitive level. Autism, 18, 17–30. the International Society for Autism Research (INSAR), Bolte, S. (2009). The ICF and its meaning for child and adoles- and the Center of Neurodevelopmental Disorders at the cent psychiatry. Zeitschriftfur Kinder-und Jugendpsychiatrie Karolinska Institutet (KIND) in Sweden. Guidance on this und Psychotherapie, 37, 495–497. project is provided by a Steering Committee comprised of Bolte, € S., de Schipper, E., Holtmann, M., Karande, S., de Vries, key opinion leaders in the field of ASD from all six WHO P.J., Selb, M., & Tannock, R. (2014a). Development of ICF regions. This Steering Committee consists of the co- Core Sets to standardize assessment of functioning and authors of this paper and John E. Robison, Melissa Selb, impairment in ADHD: the path ahead. European Child and Nidhi Singhal, Susan Swedo and Bedirhan Ustun. The Adolescent Psychiatry, 23, 1139–1148. development of ICF Core Sets for ASD is supported by the Bolte, € S., de Schipper, E., Robison, J.E., Wong, V.C.N., Selb, M., Swedish Research Council (grant nr. 523-2009-7054), and Singhal, N., de Vries, P.J., & Zwaigenbaum, L. (2014b). Clas- the Swedish Research Council in partnership with FAS sification of functioning and impairment: The 968 de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective INSAR Development of ICF Core Sets for Autism Spectrum Disor- gender differences in autism spectrum disorder: Summarizing der. Autism Research, 7, 167–172. evidence gaps and identifying emerging areas of priority. Bolte, S., Dukretis, E., Poustka, F., & Holtmann, M. (2011). Sex Molecular Autism, 6, 36. doi:10.1186/s13229-015-0019-y. differences in cognitive domains and their clinical corre- Happ e, F., & Frith, U. (2009). 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Journal of Developmental & Physical Children and Youth version (ICF(-CY)). European Child & Disabilities, 27, 119–139. Adolescent Psychiatry, 24, 859–872. € Selb, M., Escorpizo, R., Kostanjsek, N., Stucki, G., Ustun, € B., De Schipper, E., Lundequist, A., Coghill, D., de Vries, P.J., &Cieza, A. (2014). A guide on how to develop an Interna- Granlund, M., Holtmann, M., Jonsson, U., Karande, S., tional Classification of Functioning, Disability and Health Robison, J.E., Shulman, C., Singhal, N., Tonge, B., Wong, Core Set. European Journal of Physical and Rehabilitation V.C., Zwaigenbaum, L. & Bolte, S. (2015b). Ability and dis- Medicine, 51, 105–117. ability in autism spectrum disorder: A systematic literature Szatmari, P., Xiao-Qing, L., Goldberg, J., Zwaigenbaum, L., review employing the international classification of func- Paterson, A.D., Woodbury-Smith, M., Georgiades, S., Duku, E., tioning, disability and health-children and youth version. & Thompson, A. (2011). 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Additional Supporting Information may be found in the Halladay, A.K., Bishop, S., Constantino, J.N., Daniels, A.M., online version of this article at the publisher’s web-site: Koenig, K., Palmer, K., Messinger, D., Pelphrey, K., Sanders, S.J.,Singer, A.T.,Taylor, J.L.,& Szatmari,P.(2015).Sex and Appendix S1. The ASD expert survey de Schipper et al./Functioning and disability in autism spectrum disorder -the expert perspective 969 INSAR

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