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SYSTEMATIC REVIEW PROTOCOLS

Parents’ and guardians’ experiences of barriers and facilitators in accessing autism spectrum disorder diagnostic services for their children: a systematic review protocol of qualitative evidence

Smith-Young, Joanne1,3; Murray, Cynthia1,3; Swab, Michelle2,3

Author Information
JBI Database of Systematic Reviews and Implementation Reports: May 2018 - Volume 16 - Issue 5 - p 1141-1146
doi: 10.11124/JBISRIR-2017-003437
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Abstract

Introduction

Autism spectrum disorder (ASD) is a range of neurodevelopmental disorders identified by the presence of persistent deficits in social interactions and communication, as well as restricted repetitive patterns of behaviors.1 Autism spectrum disorder is an umbrella term that covers conditions such as childhood autism, atypical autism and Asperger syndrome.1 The etiology of ASD is multifactorial.2 There is a genetic basis for ASD and it is associated with immune dysregulation and inflammation, oxidative stress, environmental toxicant exposures and mitochondrial dysfunction.3 Older paternal age, complications during pregnancy, premature birth, low birth weight and jaundice in neonates are associated with ASD.2 Individuals with ASD are at higher risk for other disorders, including fragile X syndrome, allergies, asthma, epilepsy, gastrointestinal disorders, persistent viral infections, feeding disorders, anxiety disorder, bipolar disorder, attention deficit hyperactivity disorder (ADHD), Tourette syndrome, obsessive-compulsive disorder, sensory integration dysfunction, sleeping disorders, immune disorders, autoimmune disorders, neuroinflammation, and pediatric autoimmune neuropsychiatric disorders associated with streptococcal infection.4

The economic and social burden of ASD is high. For example, in the United States it was estimated that the total annual direct medical, direct non-medical and productivity costs for ASD in 2015 were US$268 billion, and it is projected that those costs will balloon to US$461 billion by 2025.5 The most recent published data from the United States indicate that ASD affects one in 66 children in that country.6 Other developed countries have reported a rising prevalence in autism cases: 65 cases per 10,000 children in Canada, 72 cases per 10,000 children in Sweden, 94 cases per 10,000 children in the United Kingdom, and an alarming 161 cases per 10,000 children in Japan.6 In developing countries, ASD rates are often underestimated because of under-reporting and under-diagnosis; for example, a recent pilot study indicated that the prevalence of pervasive developmental disorder, which includes ASD, is 27 cases per 10,000 children in Brazil.7 Globally, in 2010, there were an estimated 52 million cases of ASD or one in 132 persons diagnosed with this disorder.8

Diagnostic services are those services organized and provided by physicians for the purpose of providing a diagnosis. Since there is no specific medical test to diagnose ASD, specially trained physicians and psychologists administer ASD-specific behavioral evaluations that may include evaluations from a pediatrician, psychologist, speech and language pathologist, occupational therapist, and geneticist.9

ASD is usually detected in early childhood and it can be reliably diagnosed by 18 months of age.10 However, later diagnosis occurs, especially in children and adolescents who have lower socioeconomic status, are non-White, and exhibit less severe symptoms.10 Four to five times as many boys are diagnosed with ASD in comparison to girls.4 This disorder is a lifelong developmental disability that can negatively influence a person's educational and social attainments, as well as employment opportunities. While some people with ASD are able to live independent and productive lives, others have severe dysfunctions and require lifelong care and supportive services. The level of intellectual functioning is extremely variable, extending from profound impairment to superior cognitive skills. Experts agree that early intensive behavioral interventions can positively affect overall health outcomes.11,12 Earlier diagnosis means more intensive therapy can begin sooner, resulting in improvements in cognitive, language and adaptive skills.13,14 Despite this, most countries report the median age of ASD diagnosis to be more than 24 months. Canadian data indicate a median age in ASD diagnosis between 39 and 55 months.15 A study in the United Kingdom reported a median age of diagnosis of 55 months.16

A number of barriers in accessing ASD diagnosis have been identified in several primary studies (e.g. stigma;17,18 living in a rural community;17,19 transportation issues;20,21 ineffective screening tools;18 dismissive, hesitant or unskilled health professionals;17,22 inadequate insurance coverage;17,18 cultural/immigrant status;10,19,23 and difficulty navigating the system20,21). Some of the facilitators in accessing ASD diagnostic services that were identified in primary studies include higher levels of parental education10,24 and higher socioeconomic status.10,18,21,24 A good understanding of the barriers and facilitators in accessing ASD diagnostic services gained from a systematic review of the literature could assist healthcare professionals and policy makers in breaking down the barriers to timely diagnosis and early intervention. We completed a preliminary search of databases (PubMed, CINAHL, the JBI Database of Systematic Reviews and Implementation Reports, the Cochrane Library, the Campbell Library and PROSPERO) and found that to date, no reviews have been conducted on our phenomenon of interest to our knowledge. Instead, other reviews focused on: barriers and facilitators of parenting programmes for childhood behavior problems;25 the experience of advocating for a child with autism;26 lived experience of parents of children with ASD;27 the experience of parenting or caring for a child with autism;28-30 parenting stress in parents of children with and without ASD;31 and early interventions for children with ASD.32 Furthermore, our phenomenon of interest was not addressed as a subtopic within these previously mentioned reviews. Thus, there are primary studies17-24 in the literature that have investigated our phenomenon of interest that have yet to be synthesized, which justifies this proposed systematic review.

Inclusion criteria

Participants

The review will consider studies conducted worldwide that include parents and guardians of children up to 18 years of age and who have accessed or who are attempting to access ASD diagnostic services for their children and adolescents on an inpatient or outpatient basis. In some countries, pediatric services go up to the age of 18. The diagnosis of ASD is considered to be a significant life event for families;33 therefore, we anticipate that their experiences can be recalled. The studies may also include other participants as well as parents or guardians if the data from the parents or guardians can be separated from the larger sample.

Exclusion criteria

We will exclude all studies that are not written in English or do not have an English translation; studies that include parents/guardians of children over 18 years of age; studies where the parents’/guardians’ voices cannot be distinguished from other study participants; and studies where the voices of parents/guardians of children cannot be distinguished from the voices of parents/guardians of children with other diagnoses. Purely quantitative studies will also be excluded.

Phenomena of interest

The phenomenon of interest is parents’ and guardians’ experiences of barriers and facilitators in accessing ASD diagnostic services for their children and adolescents.

Context

The context for this systematic review is inpatient and outpatient settings in any country worldwide. Regions/countries may have different health system contexts; however, the barriers and facilitators in accessing ASD diagnostic services for parents/guardians of children with ASD could be similar. We will report any differences in parents’/guardians’ experiences of barriers and facilitators that can be attributed to dissimilarities in health system contexts (e.g. developed and underdeveloped countries).

Types of studies

This review will consider studies that focus on qualitative data about the experiences of barriers and facilitators for parents and guardians in accessing ASD diagnostic services for their children and adolescents. These studies include, but are not limited to, designs such as phenomenology, grounded theory, ethnography, action research and feminist research.

Methods

Search strategy

The search strategy will aim to find both published and unpublished studies. An initial limited search of MEDLINE and CINAHL has been undertaken followed by analysis of the text words contained in the title and abstract, and of the index terms used to describe the article. This informed the development of a search strategy which will be tailored for each information source. A full search strategy for CINAHL is detailed in Appendix I. The reference list of all studies selected for critical appraisal will be screened for additional studies. Studies published in the English language will be searched and the search will not be limited by publication dates.

Information sources

The databases to be searched include: CINAHL, PubMed, Embase, PsycINFO, Social Services Abstracts and ERIC.

The search for unpublished studies will include: ProQuest Dissertations and Theses, Google Scholar, Google, OpenGrey, and other online resources, including government and organizational websites, such as the International Society for Autism Research and the Centers for Disease Control and Prevention, ASD Research.

Study selection

Following the search, all identified citations will be collated and uploaded into Endnote and duplicates removed. Titles and abstracts will then be screened by two independent reviewers for assessment against the inclusion criteria for the review. Studies that meet or could potentially meet the inclusion criteria will be retrieved in full and their details imported into Joanna Briggs Institute System for the Unified Management, Assessment and Review of Information (JBI SUMARI). The full text of selected studies will be retrieved and assessed in detail against the inclusion criteria. Full text studies that do not meet the inclusion criteria will be excluded and reasons for exclusion will be provided in an appendix in the final systematic review report. Studies that meet the inclusion criteria will undergo a process of critical appraisal. The results of the search will be reported in full in the final report and presented in a PRISMA flow diagram. Any disagreements that arise between the reviewers will be resolved through discussion, or with a third reviewer.

Assessment of methodological quality

Studies that meet the inclusion criteria will be critically appraised by two independent reviewers at the study level for methodological quality in the review using the JBI Critical Appraisal Checklist for Qualitative Research.34 Any disagreements that arise between the reviewers will be resolved through discussion, or with a third reviewer. The results of critical appraisal will be reported in narrative form and in a table.

If there is an insufficient number of high quality studies, then all studies regardless of the results of their methodological quality, will undergo data extraction and synthesis (where possible).

Data extraction

Qualitative data will be extracted by two independent reviewers from papers included in the review using the standardized data extraction tool from JBI SUMARI.34 The data extracted will include specific details about the populations, context, culture, geographical location, study methods and the phenomena of interest relevant to the review question and specific objectives. Findings, and their illustrations, will be extracted and assigned a level of credibility.

Data synthesis

Qualitative research findings will, where possible, be pooled using JBI SUMARI with the meta-aggregation approach.34 This will involve the aggregation or synthesis of findings to generate a set of statements that represent that aggregation, through assembling the findings and categorizing these findings on the basis of similarity in meaning. These categories will then be subjected to a synthesis in order to produce a single comprehensive set of synthesized findings that can be used as a basis for evidence-based practice. Where textual pooling is not possible the findings will be presented in narrative form.

Assessing certainty in the findings

The final synthesized findings will be graded according to the ConQual approach for establishing confidence in the output of qualitative research synthesis and presented in a Summary of Findings.35 The Summary of Findings includes the major elements of the review and details how the ConQual score is developed. Included in the table is the title, population, phenomena of interest and context for the specific review. Each synthesized finding from the review is then presented along with the type of research informing it, a score for dependability, credibility, and the overall ConQual score.

Acknowledgements

We would like to express our sincere thanks to the School of Nursing, Memorial University of Newfoundland for supporting our Joanna Briggs Institute training in systematic reviews.

Appendix I: Search strategy

CINAHL

(MH “Autistic Disorder” OR MH “Asperger Syndrome” OR TI autism OR TI autistic OR TI asperger OR AB autism OR AB autistic OR AB asperger)

AND

(MH “Parents” OR MH “Adoptive Parents” OR MH “Foster Parents” OR MH “Fathers” OR MH “Mothers” OR TI parent OR TI father OR TI mother OR TI guardian OR AB parent OR AB father OR AB mother OR AB guardian)

AND

(MH “Diagnosis+” OR MW “di” OR TI diagnos OR AB diagnos)

AND

(MH “Qualitative Studies+” OR MH “Phenomenology” OR MH “Audiorecording” OR MH “Focus Groups” OR MH “Interviews+” OR MH “Narratives” OR MH “Observational Methods+” OR MH “Life Experiences” OR MH “Thematic Analysis” OR MH “Parental Attitudes+” OR TI qualitative OR AB qualitative OR TI interview OR AB interview OR TI “mixed method” OR AB “mixed method” OR TI “mixed methods” OR AB “mixed methods”)

References

1. American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 5th edn.Washington, DC: American Psychiatric Publishing; 2013.
2. Ng M, de Montigny JG, Ofner M, Do MT. Environmental factors associated with autism spectrum disorder: a scoping review for the years 2003–2013. Health Promot Chronic Dis Prev Can 2017; 37 1:1–23.
3. Rossignol DA, Frye RE. A review of research trends in physiological abnormalities in autism spectrum disorders: immune dysregulation, inflammation, oxidative stress, mitochondrial dysfunction and environmental toxicant exposures. Mol Psychiatr 2012; 17 4:389–401.
4. National Autism Association. Autism fact sheet. 2017; [internet]. Available from: http://nationalautismassociation.org/resources/autism-fact-sheet/. [Accessed 14 March 2017]
5. Leigh JP, Du J. Brief report: forecasting the economic burden of autism in 2015 and 2025 in the United States. J Autism Dev Disord 2015; 45 12:4135–4139.
6. World Atlas. World facts. Countries with the highest rates of autism. 2017 [internet]. Available from: http://www.worldatlas.com/articles/countries-with-the-highest-rates-of-autism.html. [Accessed 14 March 2017]
7. Uwaezuoke SN. Autism Spectrum Disorder in children: the disparities between the developed and developing countries. Autism Open Access 2015; 5 3:152.
8. Baxter AJ, Brugha TS, Erskine HE, Scheurer RW, Vos T, Scott JG. The epidemiology and global burden of autism spectrum disorders. Psychol Med 2015; 45 3:601–613.
9. Autism Speaks. How is autism diagnosed? 2017; [internet]. Available from: https://www.autismspeaks.org/what-autism/diagnosis. [Accessed 6 October 2017]
10. Fountain C, King MD, Bearman PS. Age of diagnosis for autism: individual and community factors across 10 birth cohorts. J Epidemiol Commun H 2011; 65 6:503–510.
11. Magiati I, Tay XW, Howlin P. Early comprehensive behaviorally based interventions for children with autism spectrum disorders: a summary of findings from recent reviews and meta-analyses. Neuropsychiat 2012; 2 6:543–570.
12. Nadel S, Poss JE. Early detection of autism spectrum disorders: screening between 12 and 24 months of age. J Amer Acad Nurs Pract 2007; 19 8:408–417.
13. Dawson G, Rogers S, Munson J, Smith M, Winter J, Greenson J, et al. Randomized, controlled trial of an intervention for toddlers with autism: the Early Start Denver Model. Pediatrics 2010; 125 1:e17–e23.
14. Orinstein AJ, Helt M, Troyb E, Tyson KE, Barton ML, Eigstiê M, et al. Intervention for optimal outcome in children and adolescents with a history of autism. J Dev Behav Pediatr 2014; 35 4:247–256.
15. Ip A. Screening practices and factors influencing autism spectrum disorder (ASD) screening by community pediatricians. Paper presented at the 2016 International Meeting for Autism Research; 2015, May; Baltimore, MD, USA. [internet]. Available from: https://imfar.confex.com/imfar/2016/webprogram/Paper22871.html. [Accessed 14 March 2017]
16. Brett D, Warnell F, McConachie H, Parr JR. Factors affecting age at ASD diagnosis in UK: no evidence that diagnosis age has decreased between 2004 and 2014. J Autism Dev Disord 2016; 46 6:1974–1984.
17. Harrison Elder J, Brasher S, Alexander B. Identifying the barriers to early diagnosis and treatment in underserved individuals with Autism Spectrum Disorder (ASD) and their families: A qualitative study. Issues Ment Health Nurs 2016; 37 6:412–420.
18. Hildago NJ, McIntyre LL, McWhirter EH. Sociodemographic differences in parental satisfaction with an autism disorder diagnosis. J Intellect Dev Disabil 2015; 40 2:147–155.
19. Coo H, Ouellette-Kuntz H, Lam M, Yu CT, Dewey D, Bernier FP, et al. Correlates of age at diagnosis of autism spectrum disorders in six Canadian regions. Chronic Dis Inj Can 2012; 32 2:90–100.
20. Pickard KE, Ingersoll BR. Quality versus quantity: The role of socioeconomic status on parent-reported service knowledge, service use, unmet service needs, and barriers to service use. Autism 2016; 20 1:106–115.
21. Khanlou N, Haque N, Mustafa N, Vazquez LM, Mantini A, Weiss J. Access barriers to services by immigrant mothers of children with autism in Canada. Int J Ment Health Addict 2017; 15 2:239–259.
22. Zuckerman KE, Sinche B, Mejia A, Corbian M, Becker T, Nicolaidis C. Latino parents’ perspectives of barriers to autism diagnosis. Acad Pediatr 2014; 14 3:301–308.
23. Valicenti-McDermott M, Hottinger K, Seijo R, Chulman L. Age at diagnosis of autism spectrum disorders. J Pediatr 2012; 161 3:554–556.
24. Goin-Kochel RP, Mackintosh VH, Myers BJ. How many doctors does it take to make an autism spectrum diagnosis? Autism 1006; 10 5:439–451.
25. Koerting J, Smith E, Knowles MM, Latter S, Elsey H, McCann DC, et al. Barriers to, and facilitators of, parenting programmes for childhood behavior problems: a qualitative synthesis of studies of parents’ and professionals’ perceptions. Eur Child Adoles Psy 2013; 22 11:653–670.
26. Boshoff K, Gibbs D, Phillips RL, Wiles L, Porter L. Parents’ voices: ‘why and how we advocate’. A metasynthesis of parents’ experiences of advocating for their child with autism spectrum disorder. Child Care Hlth Dev 2016; 42 6:784–797.
27. Corcoran J, Berry A, Hill S. The lived experience of US parents of children with autism spectrum disorders: a systematic review and meta-synthesis. JOID 2015; 19 4:356–366.
28. DePape AM, Lindsay S. Parents’ experiences of caring for a child with autism spectrum disorder. Qual Hlth Res 2015; 25 4:569–583.
29. Ooi KL, Ong YS, Jacob SA, Khan TM. A meta-synthesis on parenting a child with autism. Neuropsychiat Dis Treat 2016; 12 1:745–762.
30. Keok CA. Parental experience of having a child diagnosed with autistic spectrum disorder: an integrative literature review. Singapore Nurs J 2012; 39 1:8–18.
31. Hayes SA, Watson SL. The impact of parenting stress: a meta-analysis of studies comparing the experience of parenting stress in parents of children with and without autism spectrum disorder. J Autism Dev Disord 2013; 43 3:629–642.
32. Tachibana Y, Miyazaki C, Ota E, Mori R, Hwang Y, Kobayashi E, et al. A systematic review and meta-analysis of comprehensive interventions for pre-school children with autism spectrum disorder (ASD). PLOS ONE. 2017. Available from https://doi.org/10.1371/journal.pone.0186502. [Accessed 6 December 2017]
33. Autism Help. Autism spectrum disorders. Fact sheet 2008; [internet]. Available from: http://www.autism-help.org/child-diagnosed-autism.htm. [Accessed 6 October 2017]
34. The Joanna Briggs Institute. Joanna Briggs Institute Reviewers’ Manual: 2014 edition. Australia: The Joanna Briggs Institute; 2014.
35. Munn Z, Porritt K, Lockwood C, Aromataris E, Pearson A. Establishing confidence in the output of qualitative research synthesis: the ConQual approach. BMC Med Res Methodol. 2014; 14: 108. [internet] Available from: http://bmcmedresmethodol.biomedcentral.com/articles/10.1186/1471-1188-14-108. [Accessed 14 March 2017]
Keywords:

Autism spectrum disorder; barriers and facilitators; children; diagnostic services; parents

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