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Impact of Child Factors on Parenting Stress of Mothers of Children with Autism Spectrum Disorder and Intellectual Disability: A UK School-Based Study.

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Impact of Child Factors on Parenting Stress of Mothers of Children with Autism Spectrum Disorder and Intellectual Disability: A UK School-Based Study.

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Tsermentseli, S., & Kouklari, E.-C. (2021). Impact of Child Factors on Parenting Stress of Mothers of Children with Autism Spectrum Disorder and Intellectual Disability: A UK School-Based Study. Early Child Development and Care, 191(10), 1555–1566.

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Cultural Differences of Raising Children with ASD

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Cultural Differences of Raising Children with ASD

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Perceptions of Family Problems and Pessimism among Latina and Non-Latina White Mothers Raising Children with Autism Spectrum Disorder

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Perceptions of Family Problems and Pessimism among Latina and Non-Latina White Mothers Raising Children with Autism Spectrum Disorder

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Lopez, Kristina, and Sandy Magaña. “Perceptions of Family Problems and Pessimism among Latina and Non-Latina White Mothers Raising Children with Autism Spectrum Disorder.” Journal of Autism and Developmental Disorders, vol. 50, no. 6, 8 June 2018, 10.1007/s10803-018-3640-8.

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“Longitudinal Study of Perceived Negative Impact in African American and Caucasian Mothers of Children with Autism Spectrum Disorder.

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“Longitudinal Study of Perceived Negative Impact in African American and Caucasian Mothers of Children with Autism Spectrum Disorder.

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Carr, Themba, and Catherine Lord. “Longitudinal Study of Perceived Negative Impact in African American and Caucasian Mothers of Children with Autism Spectrum Disorder.” Autism, vol. 17, no. 4, 2 July 2012, pp. 405–417, 10.1177/1362361311435155.

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'You Don't Look Autistic': A Qualitative Exploration of Women's Experiences of Being the 'Autistic Other'

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'You Don't Look Autistic': A Qualitative Exploration of Women's Experiences of Being the 'Autistic Other'

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Seers, Kate, and Rachel C Hogg. ““You Don’t Look Autistic”: A Qualitative Exploration of Women’s Experiences of Being the “Autistic Other.”” Autism, vol. 25, no. 6, 15 Mar. 2021, p. 136236132199372, 10.1177/1362361321993722.

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Parental Suggestions for Facilitating Acceptance and Understanding of Autism

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Parental Suggestions for Facilitating Acceptance and Understanding of Autism

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Inclusion, Acceptance, Shame and Isolation: Attitudes to Autism in Aboriginal and Torres Strait Islander Communities in Australia.

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Inclusion, Acceptance, Shame and Isolation: Attitudes to Autism in Aboriginal and Torres Strait Islander Communities in Australia.

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Lilley, R., Sedgwick, M., & Pellicano, E. (2020). Inclusion, Acceptance, Shame and Isolation: Attitudes to Autism in Aboriginal and Torres Strait Islander Communities in Australia. Autism: The International Journal of Research and Practice, 24(7), 1860–1873.

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Perceptions of Arab American Mothers of Children with Autism Spectrum Disorder: An Exploratory Study

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Perceptions of Arab American Mothers of Children with Autism Spectrum Disorder: An Exploratory Study

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Alsayyari, H. I. (2018). Perceptions of arab american mothers of children with autism spectrum disorder: An exploratory study Available from ERIC. (2101587651; ED584531). https://ezproxy.bethel.edu/login?url=https://www.proquest.com/dissertations-theses/perceptions-arab-american-mothers-children-with/docview/2101587651/se-2

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Hayes, J., Ford, T., McCabe, R., & Russell, G. (2021). Autism diagnosis as a social process. Autism, 26(2), 488–498. https://doi.org/10.1177/13623613211030392

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A Preliminary Investigation of the Spence Children’s Anxiety Parent Scale as a Screening Tool for Anxiety in Young People with Autism Spectrum Disorders

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A Preliminary Investigation of the Spence Children’s Anxiety Parent Scale as a Screening Tool for Anxiety in Young People with Autism Spectrum Disorders

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Zainal, H., Magiati, I., Tan, J. W.-L., Sung, M., Fung, D. S., & Howlin, P. (2014). A preliminary investigation of the Spence Children’s anxiety parent scale as a screening tool for anxiety in young people with autism spectrum disorders. Journal of Autism and Developmental Disorders, 44(8), 1982–1994. https://doi.org/10.1007/s10803-014-2075-0

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Abstract

Despite high rates of clinically elevated anxiety difficulties in children and adolescents with autism spectrum disorders (ASDs), very few studies have systematically examined the usefulness of commonly used caregiver report anxiety screening tools with this population. This study investigated the use of the Spence Children's Anxiety Scale-Parent version (SCAS-P) as a screening tool for anxiety disorders when compared to a standardized DSM-IV-TR-based clinical interview, the Kiddie-Schedule for Schizophrenia and Affective Disorders-Present and Lifetime version (K-SADS-PL). Thirty-two caregivers of youth with a clinical diagnosis of ASD (mean age 10.3 years) attending a specialist autism school participated in this study. They first completed the SCAS-P, a measure of adaptive functioning and a checklist of other emotional and behavioral difficulties. They were then interviewed with the K-SADS-PL. Internal consistency for the SCAS Total score was .88, but Cronbach's alphas were <.70 in three of the six SCAS-P subscales. The sensitivity, specificity, positive predictive value (PPV) and negative predictive value (NPV) of the SCAS-P against K-SADS diagnosis were .75, .71, .27, and .95, respectively. All values were >.70, except for the PPV. Evidence of convergent validity between the SCAS-P, K-SADS-PL and DBC anxiety subscale was also found. The high false positive rates notwithstanding, the preliminary data of acceptable to excellent sensitivity, specificity and NPV values tentatively suggest that the SCAS-P may be useful for screening non-help seeking young people with ASD for elevated anxiety symptoms. Further replication in larger studies is needed and ways in which the SCAS-P could be further developed and investigated for use with youth with ASD are discussed.

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Acknowledgments

This research was supported by the Grant from NUS Department of Psychology Start-up Fund awarded to the second author. We would like to acknowledge Ms. Jingyi Chan and Ms. Clarissa Ong for their assistance in the clinical interviews. We are grateful to the caregivers of the youth with ASD who participated in this study and to the specialist school and teachers who facilitated recruitment.

Conflict of interest

The authors report no conflicts of interest.

Author information

Authors and Affiliations

1. Department of Psychology, National University of Singapore (NUS), AS4, 9 Arts Link, Singapore, 117570, Singapore

Hani Zainal, Iliana Magiati & Julianne Wen-Li Tan

2. Department of Child and Adolescent Psychiatry, Institute of Mental Health, Buangkok, Singapore

Min Sung & Daniel S. S. Fung

3. Faculty of Health Sciences, Institute of Psychiatry, King's College London, London, UK

Patricia Howlin

4. University of Sydney, Sydney, NSW, Australia

Patricia Howlin

Corresponding author

Correspondence to Iliana Magiati.

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Cite this article

Zainal, H., Magiati, I., Tan, J.WL. et al. A Preliminary Investigation of the Spence Children's Anxiety Parent Scale as a Screening Tool for Anxiety in Young People with Autism Spectrum Disorders. J Autism Dev Disord 44, 1982-1994 (2014). https://doi.org/10.1007/s10803-014-2075-0
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# data[].attributes.attachment_link.title
A Preliminary Investigation of the Spence Children's Anxiety Parent Scale as a Screening Tool for Anxiety in Young People with Autism Spectrum Disorders - Journal of Autism and Developmental Disorders

# data[].attributes.attachment_link.description
Despite high rates of clinically elevated anxiety difficulties in children and adolescents with autism spectrum disorders (ASDs), very few studies have systematically examined the usefulness of commonly used caregiver report anxiety screening tools with this population.

# data[].attributes.headline
Cross-cultural perspectives on the meaning of family quality of life: comparing Korean immigrant families and Canadian families of children with autism spectrum disorder

# data[].attributes.subject
Cross-cultural perspectives on the meaning of family quality of life: comparing Korean immigrant families and Canadian families of children with autism spectrum disorder

# data[].attributes.body
Fong, V. C., Gardiner, E., & Iarocci, G. (2021). Cross-Cultural Perspectives on the Meaning of Family Quality of Life: Comparing Korean Immigrant Families and Canadian Families of Children with Autism Spectrum Disorder. Autism: The International Journal of Research and Practice, 25(5), 1335–1348.

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Utility of the Taiwan version of the screening tool for autism in two-year-olds to detect autism in children aged three years

# data[].attributes.subject
Utility of the Taiwan version of the screening tool for autism in two-year-olds to detect autism in children aged three years

# data[].attributes.body
Wu, C., Chiang, C., Hou, Y., Chu, C., & Liu, J. (2019). Utility of the taiwan version of the screening tool for autism in two-year-olds to detect autism in children aged three years. Journal of Intellectual & Developmental Disability, 44(3), 337-345. https://doi.org/10.3109/13668250.2017.1413078

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ABSTRACT
Background: The Taiwan version of the Screening Tool for Autism in Two-Year-Olds (T-STAT) had been developed for identifying autism disorder (AD) in children aged 24-35 months. Two studies were performed to elucidate its utility for AD in children aged 36-48 months.

Method: In Study 1, the receiver operating characteristics were utilised to select a cutoff score for the T-STAT with 34 children with AD and with developmental delay (DD). In study 2, 147 children with AD, DD, and pervasive developmental disorder-not otherwise specified (PDD-NOS) were participated for validation.

Results: In study 1, the results indicated that a cutoff of 1.25 would yield high sensitivity and specificity. In study 2, the concurrent agreements between T-STAT risk and clinical diagnosis were high for children with AD and DD, but not PDD-NOS.

Conclusions: The results showed that T-STAT was a promising Level 2 screening tool for AD in children aged three years.

Disclosure statement

No potential conflict of interest was reported by the authors.

Additional information

Funding

This work was supported by Ministry of Science and Technology in Taiwan [grant numbers MOST 103-2628-H-037-001-MY2 (1/2), NSC 101-2410-H-037-008, 102-2410-H-004-044-MY3, and NSC 102-2410-H-037-002].

# data[].attributes.attachment_link.title
Utility of the Taiwan version of the Screening Tool for Autism in Two-Year-Olds to detect autism in children aged three years

# data[].attributes.attachment_link.description
ABSTRACT Background: The Taiwan version of the Screening Tool for Autism in Two-Year-Olds (T-STAT) had been developed for identifying autism disorder (AD) in children aged 24-35 months. Two studies were performed to elucidate its utility for AD in children aged 36-48 months.

# data[].attributes.headline
Identifying students with autism spectrum disorders: a review of selected screening tools

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Identifying students with autism spectrum disorders: a review of selected screening tools

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Wilkinson, L. A. (2011). Identifying students with autism spectrum disorders: A review of selected screening tools. National Association of School Psychologists.

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A Review of Screening Tools for the Identification of Autism Spectrum Disorders and Developmental Delay in Infants and Young Children: Recommendations for Use in Low- and Middle-Income Countries

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A Review of Screening Tools for the Identification of Autism Spectrum Disorders and Developmental Delay in Infants and Young Children: Recommendations for Use in Low- and Middle-Income Countries

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Marlow, M., Servili, C. and Tomlinson, M. (2019), A review of screening tools for the identification of autism spectrum disorders and developmental delay in infants and young children: recommendations for use in low- and middle-income countries. Autism Research, 12: 176-199. https://doi.org/10.1002/aur.2033

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Language practices in the UAE: Perspectives of Arab mothers of children with autism spectrum disorders

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Language practices in the UAE: Perspectives of Arab mothers of children with autism spectrum disorders

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Bakhsh, M. (2020). Language Practices in the UAE: Perspectives of Arab Mothers of Children with Autism Spectrum Disorders. Arab Journal of Applied Linguistics, 5(2), 55–83.

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Autism assessment in low- and middle-income countries: Feasibility and usability of western tools.

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Autism assessment in low- and middle-income countries: Feasibility and usability of western tools.

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Evaluating autism diagnostic and screening tools for cultural and linguistic responsiveness.

# data[].attributes.subject
Evaluating autism diagnostic and screening tools for cultural and linguistic responsiveness.

# data[].attributes.body
Harris, B., Barton, E. E., & Albert, C. (2014). Evaluating autism diagnostic and screening tools for cultural and linguistic responsiveness. Journal of Autism and Developmental Disorders, 44(6), 1275-87. https://doi.org/10.1007/s10803-013-1991-8

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Journal of Autism and Developmental Disorders 44, 1275-1287 (2014) Cite this article

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Abstract

While clear guidelines and best practices exist for the assessment of autism spectrum disorders (ASD), little information is available about assessing for ASD in culturally and linguistically diverse (CLD) populations. CLD populations might be misidentified and under-identified with ASD due to the assessment practices that we employ. Four autism diagnostic tools and six autism screeners were selected and evaluated for their cultural and linguistic responsiveness. Although the evaluation of ASD within CLD populations is highly complex, this study identified the need for improved autism assessment tools and practices. Without the appropriate assessment of these populations, CLD children will likely continue to be misidentified (or not identified at all) and will miss crucial intervention opportunities.

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Author information

Authors and Affiliations

1. University of Colorado Denver, 1380 Lawrence Street #1114, Denver, CO, 80217, USA

Bryn Harris

2. University of Colorado Denver, 1380 Lawrence Street #1115, Denver, CO, 80217, USA

Chantel Albert

3. Vanderbilt University, 230 Appleton Place, Peabody 228, Nashville, TN, 37203, USA

Erin E. Barton

Corresponding author

Correspondence to Bryn Harris.

About this article

Cite this article

Harris, B., Barton, E.E. & Albert, C. Evaluating Autism Diagnostic and Screening Tools for Cultural and Linguistic Responsiveness. J Autism Dev Disord 44, 1275-1287 (2014). https://doi.org/10.1007/s10803-013-1991-8
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Keywords

Autism

Diagnostic assessment

Screening

Diverse populations

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# data[].attributes.attachment_link.title
Evaluating Autism Diagnostic and Screening Tools for Cultural and Linguistic Responsiveness - Journal of Autism and Developmental Disorders

# data[].attributes.attachment_link.description
While clear guidelines and best practices exist for the assessment of autism spectrum disorders (ASD), little information is available about assessing for ASD in culturally and linguistically diverse (CLD) populations. CLD populations might be misidentified and under-identified with ASD due to the assessment practices that we employ.

# data[].attributes.headline
Systematic review: cultural adaption and feasibility of screening for autism in non-English speaking countries

# data[].attributes.subject
Systematic review: cultural adaption and feasibility of screening for autism in non-English speaking countries

# data[].attributes.body
Al Maskari, T.S., Melville, C.A. & Willis, D.S. Systematic review: cultural adaptation and feasibility of screening for autism in non-English speaking countries. Int J Ment Health Syst 12, 22 (2018). https://doi.org/10.1186/s13033-018-0200-8

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International Journal of Mental Health Systems 12, Article number: 22 (2018) Cite this article

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Abstract

Background

Screening children for autism has gained wider acceptance within clinical practice, and early intervention has improved outcomes. Increasingly, adapting an existing screening instrument is a common, fast method to create a usable screening tool, especially for countries with limited resources and/or expertise. However, concerns have been raised regarding adaptation adequacy and the feasibility of screening across cultural groups. This study systematically examined the levels of cultural adaptation and feasibility aspects considered when screening for autism in non-English speaking countries to build upon the sparse knowledge that exists on this topic in the literature.

Methods

Nineteen studies, obtained from five electronic databases, were examined. PRISMA guidance was used for this review. The Ecological Validity Framework model, and Bowen Recommendations for Feasibility were adopted to extract relevant data, which was synthesised narratively.

Results

Cultural adaptation within the included studies mostly involved language translation with little information offered to enable conclusions on how the processes were guided and maintained. Few cultural adjustments involved modifying screening methods; clarifying difficult concepts and changing instrument content were employed to address the core values, competence, beliefs, and norms of the adapted culture. However, less attention was given to adapt the screening goals within the context of cultural values, and customs or to consider interactional match between the clients and assessors. The review also highlighted an acceptable level of practicality to screen for autism but did not encourage integrating autism screening within routine practice or beyond the study context for different cultures.

Conclusion

Concurring with previous literature, we agree that knowledge on cultural adaptation for autism screening instruments is limited and not sufficiently documented to establish adaptation levels (process and/or contents), and prove adequacy. However, this review provides an infrastructure to improve future adaptation processes. Integrating autism screening as routine medical practice is not encouraged and warrants further feasibility studies to minimize wasted resources and improve screening effectiveness in various health care systems.

Background

Most research on screening instruments has been conducted in Western, English-speaking industrialised countries, where it is recognised that cultural issues impact this process [ 15, 16, 17, 18]. Disparities in parental reporting, availability, or lack, of services for ethnic minorities, socioeconomic status and heterogeneity were noted as potential issues hindering the screening process [ 19, 20]. Literature supports using a valid and reliable screening test that considers the cultural context of a country [ 21, 22, 23]. Effective cultural adaptation can strengthen screening programmes, promote instrument validity at a conceptual level across different cultures, increase confidence in outcomes [ 24, 25] and guide future work. Increasingly, adapting an existing screening instrument to the population to be screened is becoming a common, quick and efficient method to provide a valid screening tool. This is especially true for those with limited resources or expertise in the field.

A growing body of research shows attempts to adapt various screening instruments for autism across cultural groups. However, cultural adaptation is recognised as a complicated and challenging process that goes well beyond language translation, and involves careful consideration of cultural values, customs and traditions, using appropriate knowledge and skill. There is no one right way to adapt the cultural validity of an instrument. The literature often suggests adaptation of culture through content and/or process, to increase congruence between the client's ethno-cultural view and the adapted intervention [ 25, 26]. Content adaptation requires a deeper structural change, to address the core values, competence, beliefs and norms that match, both the investigator, and the targeted participants [ 25]. Process adaptation is usually considered a surface change and involves minor modification to interventions, such as literal language translation and changes in ethnicity in intervention materials [ 25, 26]. Consistently, the literature debates whether to undertake both levels of adaptation or to achieve adequate adaptation through the use of surface modification only [ 23, 24, 27, 28]. The most frequently advocated approach was to justify the use of a required level of adaptation through confirming the availability of data and resources (i.e. cost, expertise). This allowed for the proper testing of the cultural validity of the screening instruments [ 23].

In the field of autism screening, the adaptation process was insufficiently documented to justify its validity, and adaptations were limited to linguistic revision and surface modifications [ 29]. The level of adaptation that requires deep structural changes and incorporates cultural values within the adapted instrument were not investigated. Examining the content level of adapting an autism screening tool may extend existing knowledge of cultural adaptation on screening and guide the validation of future cultural adaptation work.

Little information is also available to justify integrating autism screening within routine practice. Although the feasibility of autism screening is not the principle aim of this study, the researchers thought shedding light on this important aspect was an appropriate secondary goal, to build upon the sparse knowledge that exists on this topic in the literature. In addition, information on the feasibility of a newly introduced procedure was thought to be relevant in improving, refining and adapting screening processes [ 30]. Bird, Le Boutillier [ 31] also indicated the importance of recognising "what is and is not feasible" within a practice, to minimise wasted resources, inform and prioritise decisions and improve effectiveness in health care systems.

To sum up, this review aims to determine the extent to which content level of adaptation is considered when screening for autism in non-English speaking countries. It also highlight the feasibility aspects involved in screening for autism in non-English speaking countries.

Methods

The review followed the guidance in the PRISMA statement for reporting systematic reviews and meta-analysis [ 32].

Eligibility criteria

All publications relating to the screening of children under 7 years of age for autism in non-English speaking countries were examined for this review. Only studies that used level I screening instruments and described relevant aspects of cultural adaptation, such as translations and/or cultural modifications, were included. No limitations on publication type or study design were imposed to ensure an adequate number of studies were identified.

Information sources

Five databases, reflecting the topic, were selected: Psych INFO (EBSCOhost), MEDLINE (Ovid), CINAHL (EBSCOhost), EMBASE (Ovid), and ERIC (ProQuest); hand searches were also undertaken. Autism search terms were combined with 'screening' and 'culture'. In March 2016, the first search was conducted. Reference lists of key studies and other reviews were scanned for potentially relevant articles. Google Scholar was also used as a source of 'grey literature'. An updated search was done September 5, 2017. Examples and results from the literature search are provided in Table 1.

Study selection

Search results were imported into Endnote software X7.7, which was used to remove duplicates. Initially, only the title and abstract of each study were scrutinised for relevance independently by two reviewers (TAM, CM). Then the full text of eligible papers was retrieved if it met inclusion criteria or if the abstract did not provide adequate detail to warrant rejection. The full text of the paper was examined by the same two reviewers' independently. The reviewers disagreed on 10 studies that failed to meet the age criteria (children exceeding 7 years of age). Through discussion agreement was reached to include six studies because more than 50% of their participants were under 12 years of age. Another eight papers were questionable due to lack of detail on the process of cultural adaptation; After discussion four papers that consider language translation were included.

Extracted data

Three forms were developed to extract the relevant data: (1) a study characteristic form, (2) a contents level of adaptation form and (3) a feasibility form. The intention of the first form was to provide general information regarding the included studies, such as: author(s), country of publication, participant's age group, assessor(s) and demonstrated screening instrument involved (see Table 2).

The second form was adapted from the Ecological Validity Framework (EVF) [ 33] model, to examine the extent of content level adaptation across the studies investigated within the review. The EVF was adopted because it is one of the first and most widely cited frameworks used to identify the critical elements in which cultural adaptation can play a role, and to address both surface and deep-level adaptations [ 34]. The model highlights effectiveness in a number of studies, when used to adapt culturally sensitive treatments [ 35, 36, 37, 38]. This model has eight dimensions which focus on: language, metaphors, person, contents, concepts, methods, goals and context. To extract the relevant data for each dimension and standardise the focus among the research team, questions to facilitate the process were developed (see Table 3).

The third form highlighted the aspects of feasibility that included studies may have reported and or considered while screening for autism. For that, the researchers adapted recommendations to facilitate investigation and provide a comprehensive understanding of screening feasibility [ 39]. These recommendations also included eight dimensions that focused on accessibility, demands, implementation, practicality, adaptation, integration, expansion, and limited efficacy. Similar to the cultural adaptation form, a questionnaire was developed to assess each dimension individually (see Table 4). Relevant information concerning cultural adaptation and feasibility was extracted by TAM and DW independently. Disagreements were encountered between the two reviewers on a few occasions but were resolved through discussion.

Quality assessments

The selected studies were critically appraised through the use of a straightforward validated assessment tool, known as 'QUALSYST' [ 40]. This tool comprised 14 items and each study was scored, in terms of the degree to which it met the criteria of the item. The results were reported as "yes" = 2, "partial" = 1, "no" = 0. It was also possible to score a particular study design as 'not applicable' ("n/a"), which would exclude it from the calculation of the total score. The maximum total score is 28. The total score of rated items was then divided by the total possible score, to produce a percentage value for each paper. Using this tool the quality of studies included in was assessed by two reviewers independently (TAM and DW). Disagreements were identified between the two reviewers on scoring two papers' methods and outcomes. Both reviewers examined studies critically and discussed quality scores until agreement was reached. No study were excluded due to study quality as all studies were scored above 0.6 which seen as an acceptable level for inclusion in a systematic review [ 40] (see Table 5).

Results

Search outcomes

The database search yielded 585 papers and an additional eight papers were retrieved from the reference list search. Removed were 344 papers, which were duplicates, leaving 249 papers. Paper titles and abstracts were scrutinised for relevance by two reviewers (TAM, CM) and 49 papers were retained. The full text of these 49 papers was examined and checked against the inclusion criteria by the same two reviewers. Thirty three papers were excluded for illegibility. Three more papers that met the inclusion criteria for the second search were included, whereupon a total of 19 papers were selected. The updated search revealed three more studies, culminating in the inclusion of 19 papers as part of this review (see Fig. 1).

Selection process using PRISMA 2009 flow diagram

Study characteristics

Almost 80% of the studies used the Modified Checklist for Autism in Toddlers (M-CHAT n = 15), as well as its revised version, with the follow-up interview (M-CHAT R/F; n = 1) as a screening instrument. However, other screening instruments, such as the First Year Inventory (FYI) in Israel [ 13], Social Responsiveness Scale-Preschool (SRS) in Mexico [ 49], Quantitative Checklist for Autism in Toddlers (Q-CHAT) in Iran [ 45], and Pictorial Autism Assessment Schedule (PAAS) were also recognised in this review.

Parents were the main informants in all studies, especially mothers, although in some cases (n = 6) a trained assessor, such as a medical/health science student, nurse, family physician or psychologist was also involved. The trained assessor involvement was for validation purposes, or to meet cultural preferences [ 44, 47, 48, 53, 54, 55, 57, 58]. Training (seminars, workshops and a special study module) or aids (pamphlet slides, oral presentations, instruction booklets) were used to promote awareness of autism among both professionals and parents. However, the nature of implementation, training programmes, assessors' roles, and detailing such awareness varied from author to author and was not fully documented.

Cultural adaptation

The EVF model [ 33] was used to investigate the extent of cultural adaptation within the study. This model suggested addressing eight dimensions when culturally adapting an intervention. They are explained in following sections.

Language

Metaphors

This dimension addressed incorporating verbal (e.g., folk saying) and visual symbols such as images, pictures, or figures in the screening process to convey a meaning tailored to the cultural values. In this review, two studies used culturally relevant metaphors within the screening process. For example, Canal-Bedia et al. [ 53] developed a Spanish version of the M-CHAT and, after piloting, included an adaptation, using Spanish cultural idiosyncrasies. Items 3, 5 and 23 were modified to include examples of Spanish toys. Perera et al. [ 19] introduced photographs within their screening instruments, to illustrate the text of the screening items and to promote understanding.

Person

This dimension addresses the ethnic or interactional match between study participants and the investigator. "Person" was only considered on two occasions. The first was in Ben-Sasson and Carter [ 13], where only parents who were proficient in Hebrew were involved, which enabled them to complete the adapted version of FYI in the Hebrew language and culture with more ease. Perera et al. [ 19] used local children's photographs to promote parental understanding of the screened items.

Contents

In some studies, the culture, values, customs and traditions of participants were integrated into the content of the adapted instruments and/or screening process. Only one study investigated the value of cultural information from the study groups and incorporated that into the screening instrument. Specifically, Wong et al. [ 44] modified the original instrument, from a checklist format to a graded score system, as a result of a pilot study which found many Chinese parents struggled to answer the original yes/no questions. The modified CHAT-23 involved selecting answers, such as "never", "seldom", "usually," and "often".

Concepts

Ten studies described how the authors' framed the adapted instruments into formats that were more understandable and consistent with the specific culture and context. This involved re-wording, describing and generalising/specifying difficult concepts or supporting them with clarifying examples, in a written or demonstrable format, or by deleting confusing, less well understood items. For example, in Canal-Bedia et al. [ 53] three items from the screening instrument were re-worded after piloting to promote parental understanding (items 5, 8, and 17). Albores-Gallo et al. [ 50] described the meaning of the "peek-a-boo" game because some parents, such as Mexican parents, did not have a name for it. In Kamio et al. [ 47] and Kondolot et al. [ 55], trained interviewers provided parents with specific examples for each failed item, to facilitate a better understanding and enable them to judge their responses. Samadi and McConkey [ 7] provided a general definition for some items, when translated to the Kurdish and Persian languages, to promote parental understanding. For example, item 9, "finger flicking," was presented in the Kurdish instrument as "any unusual finger and hand movements." Item 10, 'fearful behaviours', was explained during the follow-up interview, as reactions to social situations and new experiences. Seung et al. [ 48] also re-worded three items (3, 5 and 11), and included examples for each and deleted the three most confusing and misunderstood items (4, 8 and 22). More explicit words for a number of unspecified items were also included, to promote instrument adequacy and an understanding for Korean parents, consistent with Nygren et al. [ 39], who used interpreters to describe items 11, 22, and 23. Perera et al. [ 19] incorporated relevant photographs within their study, to clarify item concepts and improve recognition.

Goals

From the studies reported here, it was not possible to identify whether the screening goals were constructed within the context and knowledge of values, customs and traditions, or if there was any similarity among the assessors and participants in terms of screening being desirable within the study context. This is with the exception of one study in Spain, in which the authors reported that both professionals and parents expressed an interest in routine autism screening [ 53].

Methods

Methods takes into consideration the incorporation of cultural knowledge into screening methodology. Five studies incorporated cultural knowledge and modified screening methods to ensure screening falls within that country's cultural context. For example, a study by Kara et al. [ 41], found that when Turkish parents filled in the M-CHAT [ 59], 49% of participants' screened were positive for autism. As a result, in the second study trained nurses and psychologists interviewed parents to completed the Turkish version of the M-CHAT questionnaire, where they were able to probe and clarify issues. This method proved more effective and followed a recent study [ 55] where the M-CHAT [ 59] was completed using information gathered in face-to-face interviews. This, again, was found to be useful in the Turkish culture and resulted in fewer false-positive screening results. Another example of methodological modification to meet cultural preferences and improve instrument reliability, was in a study by Wong et al. [ 44], where Chinese parents did not complete the entire questionnaire checklist. An observational section, completed by a trained assessor, was found to reduce false-positive results. For the same purpose, other studies incorporated the screening instrument, M-CHAT, with different instruments [ 52, 54], or with a follow-up interview, to enhance reliability and meet cultural needs.

Context

Context is the last dimension of the framework and takes into account the contextual issues that may affect the screening process within each culture. This review found authors of the described studies attempted to address issues which might have challenged autism screening and they suggest potential efforts to overcome these challenges. For example Kara et al. [ 41], Kondolot et al. [ 55], identified a context issue among the Turkish population: the general population was not used to completing checklists and, hence, preferred verbal interview formats. Low and middle-income families in Turkey may also have difficulty in understanding the written questionnaire. The number of years spent in education is lower (not specified) in Turkey than in Western nations. Seif Eldin et al. [ 42] produced an Arabic version of the M-CHAT to screen children for autism in nine Arabic countries. Participating countries were classified into four sub-groups (the Gulf area, East Mediterranean, Egypt and Tunisia) based on cultural, ethnic, political and social structure similarities, in order to reduce the impact of cultural diversity and help generate concrete conclusions. However, the authors did not report how they accounted for other cultural influences.

Feasibility

In Bowen et al. [ 39] taxonomy of feasibility constructs were used to evaluate feasibility aspects for each study. Some information relevant to these aspects was identified and detailed in the following sections for each dimension.

Acceptability

With the exception of one study, the perception of suitability or satisfaction towards autism screening was not documented. In their two-phase study, Canal-Bedia et al. [ 53] adapted and validated the M-CHAT for the Spanish population, highlighted the "great interest" that both parents and professionals showed in routine screening for communicative and social development in Spain.

Demand

Only one study documented interest in using autism screening within their current practice. Nygren et al. [ 54] trained doctors and nurses in child health care settings to screen children for autism, within the two and a half years of age check-up window. The study highlighted that the trained assessors continued to use their newly acquired skills to refer suspected cases of autism (in children both younger and older than two and a half years) for evaluation, even after completion of the study.

Implementation

Practicality

Besides M-CHAT, this review identified other instruments that lent themselves to being completed by parents in a short time frame. For example, the First-Year Inventory (FYI) includes 60 items, takes about 20 min to rate the 60 items as: never, seldom, sometimes and often, or includes multiple choice questions to identify children at risk of autism or a related developmental disability. Similarly, SRS, a 65-item rating scale, ranging from 1 (not true) to 4 (almost always true), requires 15-20 min to complete. In contrast, Quantitative Checklist for Autism in Toddlers (Q-CHAT), scored on a 5-point scale (0-never to 4-always) contains 25 items, and takes 5-10 min to complete. Finally, the 21 PAAS items with "yes" or "no" choices, can be completed in 15-20 min.

Adaptation

Adaptations were made in all studies, with variations to accommodate cultural values and traditions, depending on the study aims and perspective.

Integration

Expansion and limited efficacy

Discussion

Nineteen papers (incorporating 20 studies), from different geographical regions were included in this review, aiming to determine the level of content adaptation that was considered when adapting instruments to screen for autism in non-English speaking countries. The review also highlighted feasibility aspects of screening for autism in the countries included, if any were reported. Despite variation in description and documentation of the investigated points, there were some commonalities across findings which helped the reviewers to draw relevant conclusions. They are explained in following sections.

Surface versus deeper level of adaptation

In this review, it was clear that most of the studies used surface modifications, the main focus being translation, with only a few studies also implementing deeper level adaptations. Various steps and measures were undertaken to ensure verbal and written language used in the screening process was clear, understandable, culturally appropriate and syntonic to the individual culture. However, the authors concurred with Soto et al. [ 29], in that little information was offered to enable conclusions to be drawn on how such adaptations were maintained or guided. For example, the majority of studies mainly reported back and forth translations. Other aspects of surface modifications (e.g., metaphors) were less recognisable, and/or reported in the reviewed studies.

Translation is the first step involved in the adaptation of an instrument. It requires careful planning and equal treatment of linguistic, cultural, contextual and scientific information [ 60, 61]. Yet, despite the significance of this step, some authors failed to report basic details, such as how many translators were involved and what their qualifications were. Recent evidence indicates the need for a minimum of two bilingual translators, with a cultural background and proficiency in both languages, to minimise the risk of linguistic, psychological, cultural and understanding (i.e., theoretical and practical) biases [ 60, 62]. Some studies failed to include an expert review or a pilot study. Both steps are essential in synthesising the suitability of an instrument for the targeted cultural context or in approving its readiness for use.

Reviewed studies lacked the justification for favouring a surface type of cultural adaptation. This might be due to the absence of available information on autism screening in each contexts. However, the body of research on autism screening is growing, rapidly and globally. Future studies might be able to identify the level of cultural adaptation and the resources required beforehand. The lack of feasibility studies in this area might be another reason researchers were prevented from conducting deep-level cultural adaptation investigation. This may be due to an inability to estimate expected expenses and required resources for this level of adaptation. In addition to the lack of data on the practicalities of implementing autism screening and the acceptability of screening in the targeted population, cost effectiveness analysis and random controlled trial studies, comparing the satisfaction levels of autism screening groups with that of control groups, might be valuable in advancing this area. Lack of investigator knowledge, interest and expertise on cultural adaptations might be another reason for inadequate documentation and justification for adapting a screening instrument.

Additionally, studies in this review lacked details on the particular cultural adaptation framework that was followed, as well as the efforts taken to avoid bias. This issue was also revealed by Baumann et al. [ 34], who advocated for an unambiguous description of what had been adapted, why it had been adapted, and how it was adapted. Adequate reporting is necessary for future studies, to promote an effective outcome, maintain high fidelity and avoid decrements in screening impact [ 34]. The literature also provides a number of guidelines to ensure adequate adaptation is achieved at the process [ 24, 60, 62, 63] and/or content levels [ 33, 64]. Noting such guidelines and integrating them within the screening process, may reduce discrepancy among results and enable researchers to replicate studies, and investigate differences between instruments within an increasingly diverse population [ 62].

Aspects of feasibility on screening for autism

With advances in knowledge of autism screening, identifying the feasibility of this programme has become essential in minimising resource waste, in prioritising decisions and in improving the strength of health care organisations [ 31]. The studies investigated as part of this review, are generally concerned with the practicalities of screening instruments, in terms of their adequacy, time, cost, and training required to deliver effective screening.

In non-English speaking countries, M-CHAT was a popular screening instrument [ 7, 41, 42, 43, 44, 48, 51, 53, 55, 56], albeit with a number of language and cultural adaptations, as discussed earlier. Adaptation suggests an effectiveness in improving instrument properties (sensitivity, specificity and PPV values), reducing false identification and unnecessary burden. However, this raises concerns regarding costs of training staff and allocating follow-up interviews for parents, especially in those countries with limited staff and resources.

Responding to global use of M-CHAT, a new version of this instrument with 20 items, referred to as a Modified Checklist for Autism in Toddlers, Revised with Follow-Up (M-CHAT-R/F) [ 65], was released. Despite the existence of this version, all studies in this review, with the exception of a recent one [ 56] adopted the original version of the M-CHAT, with 23 items. Adopting the new version might reduce the challenges of dealing with some difficult items (i.e., 1 and 4) [ 43, 50, 53] and providing supporting examples to reduce any future misunderstanding and to improve instrument properties. Carakovac et al. [ 56] reported less positivity and improved results, when compared with previous M-CHAT studies. Using the revised version of the M-CHAT in future studies might improve its practicality.

Previous M-CHAT studies attributed cultural impacts for any inconsistencies between item responses. However, recent evidence revealed additional reasons, such as demographic characteristics. For example, the level of education might reduce a parents' ability to understand items in the questionnaire [ 41, 50, 52]. A lack of parental comprehension might also be the result of parents sharing some autistic characteristics with the child, making it difficult to recognise the abnormal signs of autism [ 50]. This, therefore, could reduce their credibility, as the sole informants for autism screening and might explain the improved result achieved when a trained assessor or follow-up interview was incorporated into the parental self-report in M-CHAT studies. Another potential reason for discrepancy was the problem of reversed coding for certain items in the M-CHAT, such as items 11, 18 and 22 [ 48]. Seung et al. [ 48] recommended adapting and using these items with caution. To improve the practicality of adapting the M-CHAT, issues like these should be considered and investigated, to avoid wasting time, effort, and resources.

Reducing the number of items in other screening instruments found in this review (SRS and FYI) might help speed up the screening process, facilitate its integration into a busy clinical setting, promote the cooperation of parents and make it easier for both parents and professionals, with limited experience, to comprehend the questions and complete them with ease. There appears to be a movement towards the development of screening instruments with less items (e.g., 10 items). The studies have identified the most definitive items that would accurately pinpoint a symptom of autism [ 7, 11, 46] and proposals to increase their use in screening instruments in the future. Incorporating visual aids (i.e., photographs or pictures) and conceptualising the instrument items using the original language, as was the case in Perera et al. [ 19], may potentially facilitate a parent's comprehension and reduce cultural as well as adaptation barriers.

It was evident recent screening scales are moving towards quantitative measurements, with items reorganised as Likert scale types. This was established on the assumption autistic traits normally are distributed in the general population, not only in parents, but also in individuals with no previous diagnosis of autism in their families [ 50]. Despite advancing knowledge in this area and promising results, these abbreviated quantified instruments warrant further validation globally, considering a participant's characteristics, such as social factors, cognitive level, and medical history [ 66]. This will enrich our understanding of the factors that might influence the accuracy of the instruments from a global perspective.

Due to limitations in the screening instruments, scholars have not encouraged integrating autism screening within routine practice and warranted further investigation for individual cultures. For the same reasons, expanding the screening programme beyond the study context and for different populations was not favoured by most researchers, as it indicated limited efficacy.

Cost effectiveness on autism screening is an important practicality to consider when introducing a new programme [ 39]. However, research in the field of autism screening cost is limited. Assessing the costs of screening might provide a comprehensive insight into the eventual financial burden of both direct (e.g., medical expenditures) and indirect, (e.g., special education/training services, lost productivity by family caregivers [ 66], parental stress and the hassle of following positive screen participants) factors. Future research is recommended, to adequately compare various screening strategies and potentially identify, the most cost-effective methods for each individual study context. Countries vary significantly in their medical facilities and services. Regions with limited capacity for mental health assessment and services should ensure adequate resources, the sufficient coordination of services in the primary sector and early intervention prior to introducing any autism screening [ 41, 50, 53]. Future research should investigate the resources and cost effectiveness of introducing autism screening processes into clinical settings, as this will inform and direct future policy decisions. It is also noteworthy that coordination between healthcare and specialised services, in terms of follow-up, along with adequate preparation for early intervention, are crucial in enhancing the benefits of early identification of ASD.

Despite concerns regarding cost, increasing professional awareness and training of professionals to screen for autism were found to be useful in the studies reviewed here, in terms of facilitating the screening process and improving the rigorous nature of the instruments. Kondolot et al. [ 55] also highlighted the benefits of training staff to screen for autism and the fact it might reduce the high positivity that results from screening instruments used by inexperienced staff or parents. However, studies varied in their documentation of the training received and therefore comparisons could not be made regarding the level of training required or the expenses incurred to facilitate effective screening. Training professionals to recognise early signs of autism is recommended in the clinical guidelines [ 67], as without standardised training, vital signs and differences in screening results may occur.

Ignored areas in cultural adaptation and feasibilities model

Ultimately, investigators failed to capture some common areas in both models: cultural adaptation (i.e., 'person and goals') and feasibility (i.e., 'acceptability, and demands'). In most studies, participants' interests, views, perceptions, understanding and agreement, in relation to autism screening goals and implementations, were not discussed or documented much. These areas are essential to bring acceptance to the adapted programme, reducing ethnic and racial discrepancies between investigators and participants, promoting cooperation, increasing demands for autism screening, as well as producing a flexible screening programme, framed within the values, customs and traditions of the targeted population [ 33]. Future studies should consider both domains when adapting screening programmes to accommodate cultural discrepancies, raising investigator credibility and improving participant and investigator relationships towards an effective outcome.

Strengths and limitations

This review is the first to consider exploring content level of cultural adaptation and the feasibility of screening for autism in non-English-speaking countries, and therein lies its strength. It may serve as a baseline for future practitioners considering adapting an autism screening process for these populations. In terms of rigour, all stages of the process (data selection, extractions and quality validation) were cross-checked by two individuals.

Nonetheless, this review had a number of limitations. No studies written from the non-English speaking literature were included, due to limited resources for translation. The small number of identified studies represents only 13 cultures and thus has limited efficacy globally. However, important and relevant investigated aspects do emerge from the reviewed studies that may guide future work.

References

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Authors' contributions

TAM undertook this review, as part of a Ph.D. project. DW helped to conceptualise the paper, guided the quality appraisal of the study, advised on the review analysis and interpretation, and provided feedback on drafting the manuscript. CM guided the search process and provided feedback on the results, analysis and interpretation of the review. All authors read and approved the final manuscript.

Acknowledgements

None.

Competing interests

The authors declare that they have no competing interests.

Availability of data and materials

The data of this study can be obtained by contacting the corresponding author.

Consent for publication

Not applicable.

Ethics approval and consent to participate

This work did not involve human subjects.

Funding

Funding was provided to first author, TAM, by the Oman Ministry of Higher Education, as part of a Ph.D. project.

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Authors and Affiliations

1. Nursing & Healthcare School, College of Medical Veterinary and Life Sciences, University of Glasgow, 59 Oakfield Avenue, Glasgow, G12 8LL, UK

Turkiya S. Al Maskari

2. Institute of Health and Wellbeing, College of Medical Veterinary and Life Sciences, University of Glasgow, 1st Floor, Admin Building, Gartnavel Royal Hospital 1055 Great Western Road, Glasgow, G12 0XH, UK

Craig A. Melville

3. Faculty of Health, Life and Social Sciences, Edinburgh Napier University, Sighthill Campus, Edinburgh, EH11 4BN, UK

Diane S. Willis

Corresponding author

Correspondence to Diane S. Willis.

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Al Maskari, T.S., Melville, C.A. & Willis, D.S. Systematic review: cultural adaptation and feasibility of screening for autism in non-English speaking countries. Int J Ment Health Syst 12, 22 (2018). https://doi.org/10.1186/s13033-018-0200-8
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Keywords

Autism Screening

Feasibility Aspects

Screening Goals

Screening Children

Interactive Matching

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Systematic review: cultural adaptation and feasibility of screening for autism in non-English speaking countries - International Journal of Mental Health Systems

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Background Screening children for autism has gained wider acceptance within clinical practice, and early intervention has improved outcomes. Increasingly, adapting an existing screening instrument is a common, fast method to create a usable screening tool, especially for countries with limited resources and/or expertise.

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Research on Autism Acceptance in Other Countries

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Research on Autism Acceptance in Other Countries

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The estimated prevalence of autism in school-aged children living in rural Nepal using a population-based screening tool

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The estimated prevalence of autism in school-aged children living in rural Nepal using a population-based screening tool

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Heys, M., Gibbons, F., Haworth, E., Medeiros, E., Tumbahangphe, K. M., Wickenden, M., Shrestha, M., Costello, A., Manandhar, D., & Pellicano, E. (2018). The estimated prevalence of autism in school-aged children living in rural nepal using a population-based screening tool. Journal of Autism and Developmental Disorders, 48(10), 3483-3498. https://doi.org/10.1007/s10803-018-3610-1

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Introduction

Autism is a global phenomenon. An estimated 1-2% of children worldwide lie on the autism spectrum, with approximately 52 million autistic1 individuals across the globe (Baxter et al. 2015). These estimates are largely driven, however, by prevalence estimates from high-income countries (HIC). Virtually no data exist on the population prevalence of autism in low-income countries (LIC) and only one in a LIC rural setting (Uganda: Kakooza-Mwesige et al. 2014). In fact, there is a paucity of research examining autism generally in these regions at all (Abubakar et al. 2016). Studies that have been conducted in lower-middle and upper-middle income countries have produced varied results reporting prevalence estimates ranging from 0.32 (China: Tao 1987) to 250 per 10,000 (China: Ren et al. 2003) and more recently 90 per 10,000 (India: Raina et al. 2017). These discrepancies are possibly due to a variety of reasons, including the fact that autism is a spectrum condition (American Psychiatric Association 2013), the variety of traits; changing definitions of autism; varying levels of awareness in different countries; cultural variation in expectations and understandings of children's behavior; different methodological approaches used to assess prevalence; and the lack of availability of culturally-sensitive diagnostic tools and year of assessment (Elsabbagh et al. 2012).

Variation in methodological approaches to assessing autism prevalence includes differences in case-finding techniques, from population-based sampling (Baird et al. 2006) to sampling from clinics and healthcare registers (Croen et al. 2002). Variation is also found in the method of diagnosing or screening cases for autism, including a mix of relying on healthcare or educational reports (Idring et al. 2012) and/or researchers assessing for autism first-hand using comprehensive diagnostic tools including the Autism Diagnostic Observation Schedule (ADOS) and the Autism Diagnostic Interview (ADI) (Kim et al. 2011), to less sensitive screening tools or questionnaires, based on different standards and diagnostic criteria (Stewart and Lee 2017). Furthermore, while many studies have investigated the prevalence of autism in Western societies, the general consensus is that there is an urgent need for more studies investigating the prevalence of autism in LICs (Abubakar et al. 2016; Elsabbagh et al. 2012; Mpaka et al. 2016; Ruparelia et al. 2016), in particular LIC rural settings using low-cost, community-based screening tools that can be administered by lower cadres of health workers. Without these tools and studies, it is impossible to draw an accurate prediction of the global prevalence of autism and develop appropriate services that can cater for the needs of autistic children and adults and their families.

Sixty-one percent of the world's population of children and young people live in LICs or lower- to middle-income countries (LMIC). Nepal is one of the poorest LICs (World Bank 2016) and one of only four LICs outside of sub-Saharan Africa. The majority (estimated 82%) of its population lives rurally (World Bank 2014). Nepal has a growing population of about 26.7 million people due to high birth rate and declining death rate. The Autism Care Nepal Society website states that there is "no reliable estimate for the prevalence of autism in Nepal as autism is not known to many people" (Autism Care Society Nepal). Indeed, recent qualitative evidence from this population shows limited understanding of many aspects of atypical child development, in particular autism-but a strong desire to advocate for and increase support for these children and their families (Heys et al. 2016).

The overarching aim of the current study was to establish the prevalence of autism in school-aged children living in Nepal. In a preliminary screening-adaptation and acceptability study, we first sought to identify a low cost, short, population-based screening tool with good sensitivity and specificity that could potentially be delivered by all cadres of health care workers to detect possible autism in children and young people in a rural Nepali setting. We then sought to develop a Nepali-language adaptation of the identified screening tool, the AQ-10 (Allison et al. 2012), which would be acceptable for Nepali parents.

In the prevalence study, we used the resulting tool to estimate the prevalence of autism in 9-13 year-old Nepali children living in Makwanpur District, a rural hill area in the central region in which most households are dependent on subsistence agriculture (population > 500,000 in 2014). We also assessed the validity of the adapted AQ-10 within the same population by comparing results of a screening tool for childhood disability, which included questions on social and communication skills, and was delivered 6 months previously to the same cohort of children.

Our research aims were twofold: (1) to examine whether the AQ-10 is acceptable for use with Nepali parents and (2) to determine the estimated prevalence of autism in a rural population of Nepali children using the adapted AQ-10.

Method

Study Overview

Here, we describe two interrelated studies. The first study was designed to assess the adaptation and acceptability of a population-based screening tool. The second was a population-based prevalence study using the adapted screening tool. Data collection for the prevalence study occurred in two waves, wave 1 from Jan 2014 to July 2014 (during which a wide range of demographic and clinical data were obtained, including disability prevalence) and wave 2 from Sept 2014 to Jan 2015 (during which the autism screening was conducted). Description of sample size, inclusion and exclusion criteria and variables of interest are outlined in the two study sections.

Preliminary Screening Tool Adaptation and Acceptability Study

There were three stages to this adaptation study: (1) the identification of the best fit screening tool, (2) translation and back translation of the chosen tool, and (3) a qualitative study on its acceptability.

Identification of Screening Tool

To begin, we conducted a review of available literature on population-based screening tools for autism using the search terms, 'autis*', 'ASD' and 'screening' in PubMed and the reference lists of existing articles. The key characteristics of these tools, including length of administration, cost, population target, evidence base, specificity and sensitivity and reporter characteristics (e.g., parent versus clinician report), were identified. Our criteria for tool selection for the current study included: (1) being population based, (2) for use across childhood and adolescence; (3) being freely available; (4) being short in length of administration; (5) requiring parent report only; and (6) having demonstrated sufficiently high sensitivity and specificity. Supplementary Table 1 shows the 12 screening tools that were identified, of which only one (the AQ-10; Allison et al. 2012) fitted best our criteria.

Adaptation and Translation of the Screening Tool

The Autism Quotient-10 (AQ-10; Allison et al. 2012) was identified in the review of screening tools as the most feasible for use in this population-based study, where the tool would be administered by trained Nepali field research assistants. It is a short version of the AQ-50, which was originally developed as a tool for screening for autistic traits in intellectually able adults (Baron-Cohen et al. 2001). A cut-off score of 6 or more out of 10 is used to identify children and young people who have a likely diagnosis of autism, with excellent sensitivity (93%) and specificity (97%) in UK populations (Allison et al. 2012). It has a child, adolescent and adult version, requires only limited specific knowledge of ASD, is administered in a short amount of time and is freely available.

Next, we translated (and back-translated) the AQ-10 in discussion with UK and Nepali pediatricians and clinical psychologists in line with the gold standard WHO guidance on adaptation of English language tools (World Health Organization 2013). All of the team involved in the translation were bilingual Nepali/English except for one (UK pediatrician). In Nepal, we discussed the translation with a small group of European and Nepali researchers, pediatricians and clinical psychologists. Team members' experience of autism varied widely from those for which a major part of that person's role related to diagnosis and management of autistic children and young people (e.g., Nepali clinical psychologist, pediatrician) to those who had little to no experience of working with autism (Nepali researchers). We reviewed the cultural equivalence of each item and underlying concepts in light of the Nepalese constructions of childhood, maturity and the ethnopsychological concepts of the mind (Kohrt and Harper 2008; Medeiros 2014). This allowed us to elicit the most appropriate experiential equivalent in the Nepali language.

Acceptability Study

The final, qualitative arm of the adaptation study was conducted in both rural (Makwanpur) and urban (Kathmandu) Nepal in the spring of 2014 as part of a larger qualitative study exploring Nepali parents and professionals' understanding of typical and atypical child development (Heys et al. 2016). The translated version was tested for acceptability in three focus groups of parents (n = 28; 10 Kathmandu-based mothers, fathers and/or carers with a child with a known diagnosis of autism and 18 rural mothers, fathers and carers from lower caste and ethnic groups [ janajatis] with a child(ren) without a known diagnosis of autism).

The main outcome of interest for this qualitative arm was the acceptability of the AQ-10 for use as a screening tool. The Kathmandu-based participants were purposefully recruited by our collaborators Autism Care Nepal Society (ACSN) with the inclusion criteria being: parent and/or main carer of a child (aged up to 18 years) with a known diagnosis of autism (confirmed by the clinical team at ACSN from their records). There were no exclusion criteria. These parents were mostly from a high caste, middle class and educated background living in the capital, Kathmandu. The Nepali caste system is a method of social stratification practiced by most but not all ethnic groups in Nepal.

The Makwanpur-based parents were recruited in collaboration with our partners Mother and Infant Research Activities (MIRA). The inclusion criteria were parent/carer and/or grandparent of a typical child of any age. The exclusion criterion was any known diagnosis of atypical child development (including autism). Participant characteristics for these focus groups are shown in Table 1.

The same research team adapted the content and the approach used in the focus groups to ensure cultural validity. We also ensured that the content discussed was accessible for the participants who had lower literacy levels, by simplifying the language, and limiting reference to abstract concepts. The approach used in the delivery of the focus group was also tailored to address the power imbalance between some of the participants (rural uneducated women) and the representations of the researchers (high caste, some male, educated middle class from the capital, Kathmandu)-by ensuring the lead facilitators were female and of a lower caste. A fourth additional workshop was held with parents of autistic children to help clarify any uncertainties around Nepali language terms for atypical child behaviors as autistic symptomatology is common among children with atypical child development.

In the initial focus groups, each of the ten questions in the AQ-10 were considered in turn, participants were asked to discuss each statement/question and consider whether each statement/question described usual or unusual child behaviors. We used the translated version of the tool with both groups of parents for consistency as this was the version we were intending to use in the prevalence study. This procedure was essential for understanding whether the final translated version (not previous untranslated/original versions) was acceptable. All participants were asked how they felt about discussing the questions and whether other parents (of children with or without a known diagnosis of autism) would be willing and able to answer the questions about their child. This discussion was facilitated using prompts such as "Do you think these questions are easy or difficult to answer?" and "Do you think parents will mind answering these questions about their children?". Each item was discussed until there was consensus from focus group participants that the items would be acceptable for completion by Nepali parents. All participants consented for the focus group discussions to be recorded.

Analysis of Qualitative Data

Interview recordings were transcribed verbatim and subjected to thematic analysis following Braun and Clarke ( 2006) through NVivo10 (QSR International Pty Ltd 2012) as well as manually by three of the authors. The analytic process was iterative and inductive in nature. The authors independently familiarized themselves with the data and met regularly to discuss preliminary themes and codes, resolve discrepancies and decide on the final themes and subthemes.

Prevalence Study

Prevalence Study Design and Timeframe

The prevalence study was a cross-sectional study of 4098 children from Makwanpur district, rural Nepal conducted between Jan 2014 and Jan 2015 in two waves of interviews and data collection. Each wave of data collection item was preceded by a pilot of data collection procedures. These 4098 children were recruited within the broader context of a long-term follow-up study (Heys et al. 2016) of a cluster randomized controlled trial (RCTcRCT) of participatory women's groups in rural Nepal to improve neonatal survival (2001-2003) (Manandhar et al. 2004). The follow-up study was designed to examine the long-term impact on disability-free survival in mothers and their offspring enrolled in the original trial. Participants were recruited from Makwanpur district only. The inclusion criteria were all children whose mothers had been pregnant during the original trial period (2001-2003) and who were alive and willing to participate in the follow-up study.

Full details of the original trial can be found elsewhere (Manandhar et al. 2004). In brief, the initial trial intervention consisted of community-based perinatal participatory women's groups facilitated by local women (non-health professionals). The groups investigated health issues around pregnancy, childbirth and newborn health through monthly meetings over 3 years. The primary outcome was neonatal mortality rate. Secondary outcomes included maternal mortality at 4 weeks. The study demonstrated a 30% reduction in neonatal mortality and a 78% reduction in maternal mortality in intervention clusters, compared with controls. (Manandhar et al. 2004). Pregnancies were followed until 4 weeks after delivery. (Manandhar et al. 2004; Osrin et al. 2003). The study was registered as an International Standard RCT (ISRCTN31137309).

Prevalence Study Procedures

In 2014-2015, two rounds of household interviews were conducted 6 months apart on all surviving and willing participants of the original trial (Haworth et al. 2017; Heys et al. 2016). Children from all clusters were included.

In the first wave of data collection, the questionnaire administered assessed survival, age and working memory for both mothers (10-word recall) and children (digit recall forward and backward). Mothers were also asked about reproductive history, literacy, smoking, diagnosis of hypertension and diabetes and psychological wellbeing. Additional child-specific topics included questions about childhood development and disability, school attendance and performance. Topics relating to the family overall included socio-economic status (as measured by household asset, land, house and animal ownership) and household occupation. Blood pressure and anthropometric measures were taken from both mothers and children.

In the second wave of data collection 6 months later, child-specific topics included the Nepali AQ-10 (written version, described herein; see Appendix Table 5) and child pubertal status (Petersen et al. 1988); maternal-specific questions included an assessment of maternal empowerment (Cunningham et al. 2015) and perceived social support (Kvaal et al. 2014); and family-specific questions focused on an assessment of family socio-economic status using the Oxford Multi-poverty Index (Alkire 2010). Critically, data pertaining to this autism prevalence study were collected in round 2 of the follow-up study only.

Prevalence Study Sample Size

In total, we conducted face-to-face interviews with 4222 (wave 1) and 4098 (wave 2) maternal-infant dyads. Figure 1 shows the flow of participants from the original trial population through to the AQ-10 follow-up in wave 2.

Participant flow chart from baseline to follow-up in rounds 1 and 2

Prevalence Study Pilot Studies

Pilot studies were carried out for both waves of data collection. Research tools for the first round were piloted with 531 mother-child pairs, chosen randomly from original records in six clusters (three control and three intervention). The six clusters were randomly selected stratified by original trial allocation status. The first 100 mother-child pairs were then selected by birth date from the six clusters for the pilot study, with an aim to assess approximately 500 mother-child pairs. Of the 531 mother-child pairs who participated in wave 1 data collection, 152 of these were selected at random to participate in wave 2, 6 months later. Analysis of pilot data and field interviewer feedback were used after both pilots to revise the final questionnaires and databases and to provide additional field worker training (e.g., around measurement of body size and blood pressure). Following the pilot studies, minor edits were made only to other parts of the questionnaire and none to the disability questions (round 1) and Nepali AQ-10 (round 2). Therefore, results from the pilot and main study are presented together for the purposes of these analyses.

Prevalence Study Outcome Measures

Two screening tools were used for the primary and secondary outcomes, namely the adapted Nepali version of the AQ-10 and the Module on Child Functioning and Disability (MCFD), respectively. The primary outcome of interest was likelihood of autism measured by the Nepali AQ-10 and defined as a score of 6 or more out of 10 (cf. Allison et al. 2012).

The secondary outcome of interest, collected during wave 1 of data collection, was report of social and communication difficulties, as well as physical, learning and behavioral disability using the Module on Child Functioning and Disability (MCFD) produced by UNICEF and the Washington Group on disability statistics (UNICEF 2014) for use in children and young people aged 2-17 years. The MCFD is a new screening tool which builds on the established short set of questions for adult disability screening (Washington Group on Disability Statistics 2006; Washington Group on Disability Statistics and; UNICEF 2014), and is being developed as a gold standard epidemiological measure of disability prevalence. The screening tool is in the final phases of validation testing and we received permission to use it in the current study. It is carried out by interview with the child's main caregiver to assess functioning across seven core functional domains, with a total of 19 questions, namely: speech and language, hearing, vision, cognition, mobility, self-care and emotions and behavior (see Appendix Table 6). With the exception of three questions, participants rate each item on a scale ranging from no difficulty to cannot do at all. The three exceptions are questions around the use of hearing aids/glasses (yes/no response) and the question around propensity to feelings of sadness (three possible responses: the same or less, more or a lot more than age matched comparisons). Owing to the current absence of validity data on the questions around emotions and behaviors the UNICEF and the Washington Group on disability statistics have advised the definition of disability to be the report of at least some difficulty in at least one of speech and language, hearing, vision, learning, mobility and motor skills-the core functional domains, termed here as MCFD-core (Washington Group on Disability Statistics). Additional to questions on these seven domains, questions on behavior, attention, relationships, playing and coping with change were used as a comparative with the AQ-10 interview-termed here as MCFD-extended. A positive screen for these questions was defined as reporting at least some difficulty in each area (Washington Group on Disability Statistics). Sensitivity analysis was conducted using more stringent definition of a positive screen defined as a lot of difficulty or cannot do at all in at least one of these domains (as is typically used in the adult Washington disability screening tool).

Supplementary Questions

In wave 1 of screening, following the administration of the MCDF, parents were asked a series of questions around what support they received for their child, if they had screened positive for disability. In particular, they were asked if they had applied for, had received and/or had used a government disability card. These cards have four color coded categories: red (completely affected); blue (severely affected); yellow (moderately affected); white (persons with mild or ordinary disabilities). Persons who have these cards have additional financial benefits from the government and also priority over some government transport and health care. Participants were also asked about school attendance (historical and current) and whether they had received any support (in the form of counselling, medical equipment, transport or financial support) from non-government organizations (NGOs). Weight and height measured in wave 1 were used to estimate stunting of growth.

Statistical Analysis

Quantitative statistical analyses were performed using Stata version 14.0 (StataCorp., 2015). Fischer's exact test and Chi-squared tests were used as appropriate for univariate analysis of categorical variables and t-tests for univariate analysis of continuous variables. A p-value of less than or equal to 0.05 was interpreted as being statistically significant.

Ethical approval for the follow-up study was obtained both from Nepal Health Research Council and the University Research Ethics Committee. All participants gave informed verbal consent prior to participation, which is more culturally appropriate in this setting than written consent and suited to the low educational achievement/level and literacy rates of the participants.

Results

Preliminary Screening-Tool Acceptability Study

Parents of children with and without a known diagnosis of autism considered the AQ-10 items to be acceptable and straightforward to answer (see Table 2)-but not without caution. We identified two themes in parents' responses reflecting the potential challenges related to probing for atypical behaviors and/or autism. They suggested that parents of children with no known diagnosis of autism (1) would not know enough about child development to be able to recognize atypical behaviors and (2) would find a potential diagnosis of autism challenging to accept or endorse. One parent of an autistic child summed up this latter point: "They will not take it [i.e., a diagnosis of autism] easy. Nowadays, all the parents expect that my son will be a big businessman, engineer and doctor. But they don't expect anything from the autistic children but it does not mean that autistic children have no capacity. On average, they think autistic children can't do anything and they feel them as a useless thing of society. If we try to make them aware, they will not hear us. They think spending money on autistic children is like putting water on sand."

Prevalence Study

Of the 4098 parents of surviving children interviewed in round 2 for this autism prevalence study, 14 scored 6 or greater out of 10, indicative of autistic symptomatology. Mean AQ-10 scores for those who screened positive on the AQ-10 was 7.9 (SD 1.5), with median of 8 (interquartile range 7-9); for those screening negative, 0.1 (SD 0.5), median 0, 95.4% of the cohort scored 0 out of 10. In these 4098 children, the prevalence of disability using the most inclusive definition of disability as at least some difficulty in one or more of the core domains was 7.4%. Prevalence of disability using more stringent cut offs provided the following prevalence: MCFD-core reporting a lot of difficulty in at least one domain: 1.0% and MCFD-core reporting cannot do at all in at least one domain: 0.3%.

Figure 2 shows the proportion of children by AQ-10 score from 0 to 10 and the proportion of children by AQ-10 score from 1 to 10 inclusive, thus more clearly visualising the distribution in scores of 1 and above. These scores are shown for four groups, namely: (1) all children, (2) children screening positive for the MCFD-core disability questions, (3) children screening positive for the MCFD-extended questions and (4) children screening positive for the 3 questions most intuitively associated with autistic symptomology-denoted in Fig. 2 as MCFD-autism. The AQ-10 scores for children who scored postitive for the extended MCFD questions and for the MCFD-autism questions were spread more evenly across the potential AQ-10 scores from 0 to 10, with a greater number of children showing higher AQ-10 scores. Mean AQ-10 scores for the four groups of children were as follows: (1) all children: M = 0.12 (SD = 0.66); (2) MCFD-core disability: M = 0.55 (SD = 1.82) and (3) MCFD-extended: M = 1.36 (SD = 2.88) and (4) MCFD-autism: M = 1.94 (SD = 3.31).

Proportion of children by AQ10 score. Proportions are shown for four groups of children, namely: (1) all children, (2) children who screen positive for MCFD-core; (3) children who screen positive for MCFD-extended and (4) children who screen positive for MCFD-extended questions most intuitively associated with autism symptomatology-MCFD-autism. AQ-10 Autism quotient-10, MCFD module of child functioning and disability

Table 3 describes key characteristics of the 14 children who scored 6 or more out of 10 in the AQ-10. Almost two-thirds (65%) were male, which compares to 51% males in the cohort as a whole, and their mean age, 11.4 years (range 10.5-12.3), was similar to the entire cohort. Thirteen out of 14 screened positive for disability on the MCFD and 12 screened positive for difficulties in communication, forming relationships with others, coping with change, playing with other children and attention (see Table 4). None of the children had a known diagnosis of autism. One child screened positive for AQ-10 (score 6 out of 10) but negative for all of the disability screen (Table 3: Child 1). Out of the 14 children screening positive, this child was one of only 4 children who attended school and the only child of the 14 who was able to complete the digit recall forward and backward scoring below average on both scores (digit recall forward 4: M for cohort = 4.8, SD = 1.1; digit recall backward 3: M = 3.1, SD = 1.6). The majority of the 14 children screening positive (71%) did not attend school, compared with 4% of the cohort, and 83% had stunted growth compared with 39.6% of the cohort. A child is said to have stunted growth when their height for age is < 2 standard deviations of the WHO child growth standards median. Five families had applied for the government disability card (see Table 3) and, of the four who had received one, two had used it. The same two families had received financial support from an NGO. Of note, children who screened positive for the AQ-10 were equally as likely to be in the intervention or control arm of the original trial with 7 children in each trial arm (p-value for chi-squared, 0.945).

Using questions from the MCFD extended screen, we created a composite score by summing the three questions most intuitively associated with autism symptomatology, namely coping with changes, social relationships and playing with others-denoted in Fig. 2 as MCFD-autism. Figure 2 shows a histogram of this composite score from the subset disability screening, excluding those children with a score of zero. Of the 4222 children, this composite score identified 66 (1.6%; 39 males; M age = 11.4 years, SD = 0.6) who screened positive for reporting a problem in one or more of these areas. Of these 66 children, 62 (94%) received the AQ-10 in round 2, 6 months later, with mean AQ-10 scores of 1.94 (SD = 3.31, range 0-10) - which were significantly higher (p < 0.001) than the scores from the remaining cohort (n = 3,822; M = 0.1, SD = 0.47). Of these 62 children, 20 (32%) scored 1 or more out of 10 on the AQ-10.

Discussion

This is one of only two published studies to date to have estimated the prevalence of autism in a rural LIC setting. The study showed that the adapted AQ-10 was acceptable to groups of parents of children both with and without a known diagnosis of autism. Of the 4098 children sampled, 14 scored positive for autistic symptomatology. The demographic, anthropometric, clinical and educational characteristics and gender ratio of these 14 children indicate that the AQ-10 administered with a threshold score of 6 or above in this population is likely to identify children with complex needs and more likely more severe autism. 66 out of 422 children screened positive for the three questions most intuitively associated with autism symptomatology from the MCFD disability screening tool. A comparison of scores from the AQ-10 to those from the MCFD administered 6 months earlier provided some evidence of the clinical validity of the AQ-10. Likewise, the comparison of scores from these two tools provided evidence of the potential of MCFD, at least the section pertaining to child behavior, to identify successfully children within the population with atypical child development and behaviors.

There is no population-based study with which to compare these AQ-10 scores. The original paper describing the AQ-10 as a short screening tool was tested on UK cases (autistic children and adults) and controls (children and adults without a known diagnosis of autism) (Allison et al. 2012). The autistic adolescents scored a mean of 8.40 (SD 1.69) and adolescent controls a mean of 1.78 (SD 1.80). The AQ-10 scores for UK cases were therefore not dissimilar to those from this cohort who screened positive with AQ-10 scores (M = 7.9; SD 1.5). However, the Nepali population mean scores were much lower than those of the control group tested in the UK study.

If the AQ-10 screening tool is as sensitive and specific in the Nepali population as it is in the UK, the current results would give an estimated true prevalence of 3 in 1000 (95% confidence interval: 2-5 in 1000) (Brown et al. 2001). The current population of children under 18 years in Nepal is a little over 11.6 million. If confirmed, these prevalence estimates would equate to 34,803 children and young people currently living in Nepal (range 23,203-58,007) with a potential diagnosis of autism. The number of children with a current diagnosis of autism (n = 107); Kathmandu Valley, 2012 estimates (Autism Care Society Nepal) is substantially lower than this figure. This estimated prevalence of 3 per 1000 is lower than that in HICs, which is reported as 10-20 per 1000 (Elsabbagh et al. 2012), and lower than the only other LIC study showing Ugandan population estimates in 1-10-year-olds to be 12-13 per 1000 (Kakooza-Mwesige et al. 2014). Nevertheless, this estimate is higher than global median estimates (1.7/1000) and estimates from a recent study in rural India 0.9 per 1000 for all children in the full cohort of 11,000 children aged between 1 and 10 years and 1.1 per 1000 in the rural cohort (Raina et al. 2017). In the latter study, urban rural abode and higher socio-economic class were associated with reduced prevalence. The closest areas geographically, other than India, in which there is some (but still not a significant amount of) research on autism prevalence available are Sri Lanka and Indonesia. One, relatively old Indonesian-based study estimated 1.7 cases of autism per 1000 of the population (Wignyosumarto et al. 1992). In comparison, a study based in Sri Lanka placed the estimate as high as 10 per 1000 (Perera et al. 2009).

Of those children who screened positive for autism symptomatology, almost all also screened positive for physical, learning and behavioral disabilities. Given these two screening tools were delivered approximately 6 months apart, this finding provides preliminary evidence that our modified AQ-10 is a valid measure of atypical child behavior. It also suggests that those children who screened positive for autism symptomatology on the AQ-10 have evidence of multiple impairments and so most likely represent the severe end of the spectrum of potential clinical presentations of autism. This is also reflected in the gender ratio (1.4:1, M:F), showing a much higher number of females than is typically reported in the full spectrum of autism, around 4:1 (Fombonne 2009); though see (Loomes et al. 2017). In autistic children who are cognitively less able, the gender ratio falls from 10:1 to be closer to 1:1 (Volkmar et al. 1993). It is therefore likely that our estimated prevalence of 3 per 1000 is an underestimation of the true prevalence of autism within this population. This is also reflected in the number of children (66 of 4222) screening positive for difficulty in at least one of the areas of coping with changes, social relationships and playing with others. These questions have not yet been validated or indeed tested in any way as a screen for autism. Yet if it were assumed that a positive screen was indicative of likelihood of autism, this figure would equate to an estimated prevalence of 16 per 1000, much more similar to HIC estimates and those from Uganda. Indeed, the Ugandan study used an adaptation of the Ten questions questionnaire that was the basis for the MCFD used in this study (Kakooza-Mwesige et al. 2014).

In addition to being more likely to screen positive for physical, learning and behavioral disabilities, children who screened positive for autism symptomatology were more likely to be stunted and to have cognitive difficulties (as measured by marked difficulties completing the forward and backward digit recall, a measure of working memory). The majority of these children were not attending education and were unlikely to be receiving any financial support despite having significant difficulties. An 83% stunting rate is substantial. Nutritional deficits in children with disabilities and learning difficulties are common and can not only be a cause of cognitive deficits, but also contribute to the failure to reach full developmental potential in the presence of a developmental condition. These limited, but important descriptive data support the general opinion that children with all kinds of atypical child development and disability are a highly vulnerable, disadvantaged group, especially in resource-limited settings such as Nepal (UNICEF 2013).

The strengths of our study include (1) its novelty-to our knowledge there is only one other published study of prevalence estimates in a LIC; (2) the interview of families within households, thus including children who were not present in school; (3) our restricted age range, thus rendering age range less of a confounding variable; and (4) our cultural adaptation of the screening tool using qualitative methods in collaboration with local stakeholders (Stewart and Lee 2017). Also, to our knowledge, no other study has incorporated cultural constructions of mind/emotions and local notions of childhood/children in the translation and piloting of the AQ-10 or similar tools. We also situated our prevalence estimate within the context of other important data, including disability screen, school attendance and growth with which to explore characteristics of those screening positive for AQ-10.

There are, however, limitations to our study. First, the cohort is derived from mother-infant dyads who were enrolled in a perinatal trial. Nevertheless, children who screened positive for the AQ-10 were equally as likely to have been born into the villages enrolled in the intervention as those villages enrolled in the control. Second, the MCFD is under development and indeed since the commencement of this study minor edits have been made to the questions. In addition, early testing in India and Cameroon, coupled with the prevalence found here, suggest cultural interpretation of degree of difficulty may greatly influence reporting (Mactaggart et al. 2016). For instance, prevalence of reporting at least some difficulty in at least one domain was 35% and 64% in India and Cameroon, respectively, whereas prevalence of reporting a lot of difficulty or cannot do in at least one domain was 4 and 9% again in India and Cameroon, respectively (Mactaggart et al. 2016). Thus, our comparison of MCFD-extended questions with AQ-10 here should be considered exploratory. Notably, however, our prevalence estimate of 7.4% is in keeping with a systematic review of the global estimates of childhood disability in LMICs which suggested that despite a wide range in estimates (0.5-18%), the majority clustered around 5-10% (Maulik and Darmstadt 2007).

Future research is required to validate the AQ-10 and the MCFD-core and -extended modules through in-depth comprehensive assessments of high-scoring children and a representative sample of low-scoring children. Given the wide range of perinatal data available for this cohort, we also have the unique opportunity to conduct an exploratory study around the association of perinatal factors with likelihood of autism symptomatology in a population of children with poor nutrition (40% stunting at mean age of 11.5 years). Such prevalence research, however, also raises serious ethical concerns, including the possibility of disclosure of likelihood of and/or even a diagnosis of autism in a population for whom there is no term for autism (see Heys et al. 2016) and there is very little health or education provision to support children and families with a diagnosis of autism. These issues surrounding the ethics and logistics of diagnostic disclosure were precisely the ones that our focus group participants perceived to be of concern. There are also no validated diagnostic tools available for use in the Nepali population, rendering a validation study even more challenging. Thorough examination of these issues with Nepali parents and practitioners is essential prior to pursuing further the validation study of the AQ-10, including examining the potential impact of disclosure of a diagnosis of, or likelihood of a diagnosis of autism about which very little is known in most resource-poor settings like Nepal. Finally, future research in this area should explore the potential impact of caste/ethnicity and rural/urban divide on understanding of autism and its implications.

In our qualitative study of Nepali parents' and professionals' understanding of typical and atypical child development (with an emphasis on autistic symptomatology), we found that parents of children without a diagnosis of autism and professionals in general had little explicit awareness of autism (Heys et al. 2016). Only parents of autistic children, pediatricians and the disability sector worker identified behaviors typically associated with autism as 'autistic'. Other participants, including parents of children without an autism diagnosis, primary and early child development teachers, community health workers and faith healers, used distinctive terms, such as "stubborn" and "insisting" to distinguish vignettes of autistic children from vignettes of children with other developmental conditions. Most participants felt that environmental factors, including in-utero stressors and birth complications, parenting style, and home or school environment, were key causes of atypical child development and further called for greater efforts to raise awareness and build community capacity to address autism. Thus, the preliminary prevalence estimate reported herein, combined with complementary research showing the lack of awareness of autism by Nepali professionals and parents, demonstrates a substantial unmet need and stresses the importance of developing services to support families and children with atypical development in LIC settings.

Notes

Identity-first language (i.e. autistic person), opposed to person-first language (i.e., person with autism), is preferred by many autistic people and their allies (see Kenny et al. 2016) and (Sinclair 1999). Therefore, in this article, the authors use predominantly identity-first language.

Acknowledgments

We are grateful to all of the participants who so generously gave up their time to take part in the study, to Autism Care Society Nepal for hosting training days for the researchers and to the Mother and Infant Research Activities, Nepal, senior research team: Mangalmaya Manandhar, Rita Shrestha, Dhurba Adhikari, Jyoti Bamjan, Bharat Budhathoki, Dej Krishna Shrestha and Sagar Khadka. We also thank Dr. Melissa Neuman for her data cleaning of the original trial dataset and Claudia Cappa, Statistics and Monitoring Specialist Statistics and Monitoring Section, Division of Policy and Strategy, UNICEF, USA and Mitchell Loeb from the Office of Analysis and Epidemiology, National Center for Health Statistics, USA, and the UN/Washington Module on Child Health Functioning group for sharing the Module on Child Functioning and Disability (MCFD) with us and Prof. J. Katz for sharing their group's Nepali translation of the Ten questions questionnaire and Amy Alexander for her help with analysis of the qualitative data. The facilitator training and focus group work for this project was funded by a grant awarded to M.H., E.P., and A.C. from the Institute of Education (IOE)/UCL Strategic Partnership Incubator Fund. M.H. was funded by the National Institute for Health Research, UK, during this project. E.P. was funded by a Philip Leverhulme Prize from the Leverhulme Trust, UK. Research at the Centre for Research in Autism and Education (CRAE) is supported by The Clothworkers' Foundation and Pears Foundation. The study was funded by a Wellcome Trust strategic award. Ref: 085417/Z/08/Z, Project Title: "Population Science of Maternal and Child Survival."

Funding

The facilitator training and focus group work for this project was funded by a University Strategic Partnership Incubator Fund awarded to MH, AC and EP. The first author was funded by a National Institute of Health (NIHR) clinical academic lectureship. The final author (EP) was funded by a Philip Leverhulme Prize from the Leverhulme Trust, UK. The field work for the study was funded by a Wellcome Trust strategic award. Ref: 085417/Z/08/Z, Project Title: "Population Science of Maternal and Child Survival."

Author information

Authors and Affiliations

1. UCL Institute for Global Health, University College London, London, UK

Michelle Heys, Emilie Medeiros, Mary Wickenden & Anthony Costello

2. Great Ormond Street Institute of Child Global Health, University College London, London, UK

Michelle Heys & Ed Haworth

3. Chelsea and Westminster NHS Foundation Trust, London, UK

Felicity Gibbons

4. Mother and Infant Research Activities, Kathmandu, Nepal

Kirti Man Tumbahangphe & Dharma Manandhar

5. Autism Care Society, Kathmandu, Nepal

Merina Shrestha

6. Tribhuvan University Teaching Hospital, Department of Child Health, Kathmandu, Nepal

Merina Shrestha

7. Department of Maternal, Newborn, Child and Adolescent Health (MCA), World Health Organization, Geneva, Switzerland

Anthony Costello

8. Centre for Research in Autism and Education (CRAE), UCL Institute of Education, University College London, London, UK

Elizabeth Pellicano

9. Department of Educational Studies, Macquarie University, Sydney, Australia

Elizabeth Pellicano

Contributions

MH, DM, EP and AC conceived of the study and drafted the manuscript. MH, DM, EP, AC, MW, KMT, MS, EM and FG participated in its design and coordination. EH, FG and EM participated in the design and interpretation of the data and helped to draft the manuscript. MH performed the statistical analysis. MH, EM and EP performed the qualitative analysis. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Michelle Heys.

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Conflict of interest

All authors declare that they have no conflict of interest.

Ethical Approval

Ethical approval was obtained from the University College London ethics committee (Project ID: 5143/002) and the Nepal Health Research Council (Reg.no.L99/2OL3). All procedures performed in studies involving human participants were in accordance with the ethical standards of the university and the Nepali national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed Consent

All participants gave informed verbal consent prior to participation, which is more culturally appropriate in this setting than written consent and suited to the low educational achievement/level and literacy rates of the participants.

Electronic supplementary material

Below is the link to the electronic supplementary material.

Appendix

Appendix

See Tables 5 and 6.

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Heys, M., Gibbons, F., Haworth, E. et al. The Estimated Prevalence of Autism in School-Aged Children Living in Rural Nepal Using a Population-Based Screening Tool. J Autism Dev Disord 48, 3483-3498 (2018). https://doi.org/10.1007/s10803-018-3610-1
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Autism

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# data[].attributes.attachment_link.title
The Estimated Prevalence of Autism in School-Aged Children Living in Rural Nepal Using a Population-Based Screening Tool - Journal of Autism and Developmental Disorders

# data[].attributes.attachment_link.description
Few data exist on the prevalence of autism in low-income countries. We translated, adapted and tested the acceptability of a Nepali-language version of a screening tool for autism (Autism Quotient-10). Using this tool, we estimated autism prevalence in 4098 rural Nepali children aged 9-13 years.

# data[].attributes.body
Further investigation into cultural adaptions to screening tools for ASD