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Fetal alcohol spectrum disorder resources for health professionals: a scoping review protocol
  1. Josephine Chidinma Okurame1,
  2. Lisa Cannon1,2,
  3. Emily Carter3,
  4. Sue Thomas3,
  5. Elizabeth J Elliott1,4,
  6. Lauren J Rice1
  1. 1Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
  2. 2Telethon Kids Institute, Nedlands, Western Australia, Australia
  3. 3Marulu Unit, Marninwarntikura Women's Resource Centre, Fitzroy Crossing, Kimberley, Australia
  4. 4Kids Research, The Sydney Children's Hospitals Network, Westmead, New South Wales, Australia
  1. Correspondence to Dr Josephine Chidinma Okurame; josephine.agu{at}sydney.edu.au

Abstract

Introduction People with fetal alcohol spectrum disorder (FASD) encounter a range of health and allied health providers and require specialised support to ensure health services are provided safely and effectively. Not all health professionals possess the knowledge or expertise required for the identification, assessment, referral and management of FASD. Accessible resources for understanding and managing FASD can help create awareness in health professionals and ensure patients receive the correct diagnosis and timely access to the necessary supports and services. The aim of this scoping review is to identify and analyse FASD resources for health professionals.

Methods and analysis A comprehensive search of eight databases (MEDLINE, Scopus, PsycINFO, CINAHL, PubMED, EMBASE, Web of Science and Trip Medical Database) and nine grey literature databases (FASD Hub, NOFASD Australia, National Organisation for FASD, FASD United, HealthInfoNet, Proof Alliance, Child Family Community Australia, Foundation for Alcohol Research & Education and the Australian Department of Health websites) will be conducted using three search engines including PubMed, Ovid and Google advanced search (search dates: October 2021 to May 2022). Consultations will also be carried out with international and national experts in the diagnosis/management of FASD to obtain any additional relevant published or unpublished resources. Inclusion criteria were developed to guide the selection of resources that are publicly available, primarily focused on FASD and curated for health professionals for the identification, management or referral of FASD. Critical appraisal process will be executed using the Appraisal of Guidelines for REsearch & Evaluation II (AGREE II) tool to assess the quality of selected resources.

Ethics and dissemination Ethical approval is not required for the scoping review. Scoping review results will be presented at relevant national and international conferences and published in peer-reviewed journals. Search results will be made available to ensure reproducibility and transparency.

  • fetal medicine
  • community child health
  • medical education & training
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Strengths and limitations of this study

  • This scoping review will be the first of its kind to examine fetal alcohol spectrum disorder (FASD) resources for health professionals to aid with appropriate management.

  • The review will follow an established methodological framework for conducting scoping reviews in the JBI Manual for Evidence Synthesis and use the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews checklist to improve the reporting of scoping reviews.

  • Our review will highlight the resources that are deemed high quality from the critical appraisal and recommendations on their use and access will be provided.

  • The primary focus on FASD resources may limit the inclusion of resources for other overlapping neurodevelopmental disorders.

  • The strength and relevance of this scoping review is limited by the quality of the resources identified.

Introduction

Fetal alcohol spectrum disorder (FASD) is one of the leading causes of developmental disabilities that are non-genetic in nature, with a global prevalence of approximately 0.81.1 A closer observation using conservative analytical approaches reveal higher crude prevalence rates in Western countries like the USA (1.1%–5%),2 UK (1.8%)3 and Canada (1.8%).4 These prevalence statistics highlight the importance of allocating more attention and resources to FASD screening and diagnostic services as well as health promotion initiatives to advance the awareness and prevention of maternal alcohol consumption.

FASD is a form of acquired brain injury which occurs in utero and manifests as a neurodevelopmental disorder and as a term, it collectively describes a range of prenatal alcohol exposure-related symptoms including stunted growth, physical and behavioural abnormalities, birth defects, craniofacial anomalies and neurodevelopmental impairments.5–7 FASD is often referred to as an invisible disability as only 10% of people have the facial features,8 with most scoring in the low-average range of intellectual ability. Individuals with FASD experience learning and academic challenges, behavioural issues, attention deficit hyperactivity disorder, problems with physical health, mental health, speech, motor skills, hearing and vision impairments. As a result, people with FASD encounter a range of health and allied health providers and require specialised support to ensure health services are provided safe and effectively.

Health professionals play a significant role in the prevention of alcohol exposure in pregnancy and development of FASD by providing interventions and education to pregnant women. In Australia, the majority of women want health professionals to ask pregnant women about alcohol use during pregnancy and advise them about the possible effects.9 However, health professionals have voiced their hesitance to have discussions around alcohol consumption with pregnant women mainly due to a lack of confidence and insignificant resources available to provide follow-up services or address alcohol-related issues.10–12 We believe there are limited resources13 available to educate health professionals on how to work with people with FASD and most health professionals possess limited expertise or knowledge base for the identification, assessment, referral and management of FASD in the absence of consistent and standardised screening tools.14 To ensure the appropriate management and referral of such patients, health professionals need to be equipped with efficient tools.

We will conduct a scoping review to identify and evaluate resources currently available to health professionals about the recognition, diagnosis and management of FASD. The working definition of the term ‘resources’ in this review refers to the successive itemisation of instructions in the form of frameworks, guides, tools, instruments, applications or models that are developed for health professionals in the management and prevention of FASD.

Methods

Protocol design

This scoping review utilises a version of Arksey and O’Malley’s15 methodological framework for conducting scoping reviews,15 which has been further developed by Levac and colleagues in 201016 and the Joanna Briggs Institute (JBI) in 201417 and 2020,18 emphasising on the importance of conducting trustworthy, rigorous and transparent scoping reviews.

This scoping review will be reported according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews (PRISMA-ScR) and the latest JBI guidance for authors of scoping reviews will be used to organise the review process15 into nine stages (table 1).

Table 1

Stages of the scoping review process

Stage 1: define the research questions and objectives

The primary research question of this scoping review is as pertains to diagnosis, assessment, referral and/or management of FASD, what resources or guidelines are available for health professionals? Secondary research questions include:

  1. What is the primary purpose of each resource/guideline for example, screening, diagnosis, behavioural management or referral and can they be implemented?

  2. What is the quality of these resources as assessed using a validated critical appraisal tool?

Stage 2: develop the inclusion criteria

The eligibility criteria for this scoping review allow capture of a wide range of existing literature, including but not limited to meta-analyses or systematic reviews, primary research studies, books, policies, guidelines, programmes, professional development and information-based resources for example, factsheets, podcasts, apps, videos and websites. Hereafter all sources of information will be referred to as resources. There are no limits on the time frame of publications, but resources must be in English and targeting health professionals. All resources must address FASD and should be usable by all health and allied health professionals. Inclusion criteria are outlined in table 2.

Table 2

Inclusion criteria for resources

Stage 3: planned approach to evidence searching (search strategy)

The evidence search will be conducted by sourcing resources from both peer-reviewed and grey literature (published or unpublished) using search engines including OVID (for Medline) and Google advanced search including but not limited to, primary research studies, systematic reviews or meta-analyses, guidelines and policies, books, programmes and professional development and information-based resources, for example, factsheets, videos, websites and through consultation with experts in FASD, as outlined in table 3.

Table 3

Information sources for search or consultation

An iterative process will inform the comprehensive search approach.5 First, keywords will be identified by conducting an initial search of one bibliographic database and some grey literature to ascertain search terms and keywords that are utilised in the article title, abstract or study description. Next, we will identify synonyms of these keywords and subject headings for each database to be searched. Finally, advanced search syntax will be used to develop a search strategy for each database using the key terms and database-specific subject headings for example, Medical Subject Headings. Due to various website structures, each grey literature search will require bespoke consideration. Where possible, search terms will be combined in the same way as when conducting databases searches. Where multiple terms cannot be searched, the availability of primary and secondary navigation menus and/or basic search bars will be used to examine for relevant links and resources.

Consultation will then proceed with national/international experts in FASD by email or telephone. Consultations in the form of qualitative one-on-one phone interviews will be conducted to obtain information about any relevant resources for healthcare professionals, including unpublished resources, as well as any suggestions and contact details of other individuals or colleagues that may know of other resources. To track these strategies and searches, a concept table will be developed in excel for each database and website searched. The concept table will capture the name of the database, database coverage, date exported, reference management software (EndNote), total number of results and search strategy. Resources will then be imported into Covidence where any duplicates will be removed.

Search terms

The search terms and synonyms identified in table 4 will be modified and used for each database depending on the advanced search rules and search functions for respective databases. Truncations, wildcards, proximity, and Boolean operators will be used to expand on and combine these search terms to capture a broad range of possible resources and ensure a comprehensive search process.

Table 4

Search category, terms and synonyms

Stage 4 and 5: evidence screening and selection

Resources will be imported into two reference management software systems (Endnote and Covidence). Evidence screening will commence with one member of the research team reviewing the titles and abstracts of studies following the inclusion criteria specified in table 2. A second reviewer will screen 20% of the titles and abstracts. The next step will involve obtaining full text for review of relevant evidence and screening for eligibility.18 This step will be conducted by two reviewers and additional reviewers will be available to resolve any disagreements or disparities in the screening process. The final step will involve one team member searching the reference lists of all identified reports and articles to identify any additional studies of potential relevance to be screened for eligibility using the inclusion criteria as done in the aforementioned steps.5

The study selection process, including the number of studies selected for inclusion in the final scoping review, will be described in a narrative description of the search process and a diagrammatic decision flowchart.18 A flow diagram as indicated in the PRISMA-ScR statement and checklist (figure 1) will be used to detail the screening and selection pf resources; including search results, removal of duplicate citations, study selection, retrieval of full-text articles and additional articles found in the reference list search.19 Detailed reasons will be provided for exclusion of articles.

Figure 1

Preferred Reporting Items for Systematic Reviews and Meta-Analyses flow diagram for the scoping review process.

Stage 6: evidence extraction (charting the data)

The data extraction stage in a scoping review is commonly referred to as charting the data, as results will be summarised in a logical and descriptive way in alignment with the review question(s) and objectives. Information and characteristics from the eligible resources such as references, authors, findings and results will be inserted into a charting table developed specifically for this scoping review and piloted for any refinement or modification of the tables that may be required. The pilot will involve two team members charting four randomly selected resources (two each) in an attempt to test the efficiency of the charting table for extracting all the relevant data of interest.5 The data charting tool used in this scoping review will be modified from the tool utilised by Lees et al5 in their scoping review of FASD resources for educators5 to suit our target sample of health professionals. Additional categories will be added from the JBI’s recommended standardised extraction fields.20 Data extraction is also an iterative process, often requiring multiple refinements in order to meet and address the research question(s) or study objectives. Therefore the charting table may need to be refined when conducting the full review.18 The draft data charting is outlined in table 5.

Table 5

Draft data extraction/charting table for health professionals’ FASD resources

Quality appraisal

Assessment of resource quality will be undertaken using the AGREE II tool.21 The AGREE II comprises 23 items organised into six quality domains including scope and purpose, stakeholder involvement, rigour of development, clarity of presentation, applicability and editorial independence (table 6). Each of the 23 items addresses a different aspect of resource quality. At the end of the AGREE II appraisal tool, there are two questions that require each appraiser to give an overall judgement of the guideline or resource, considering how they have rated the 23 items. Each item will be allocated a score by the appraisers using a Likert scale ranging from 1 (Strongly Disagree) to 7 (Strongly Agree).

Table 6

The AGREE II quality appraisal tool (summarised from the original AGREE II document)21

An overall quality grade will be calculated from the individual scores. The AGREE II manual document21 will be used to guide allocation and calculation of scores. Scoring will help identify limitations in the resource and select high-quality resources for recommendation, implementation and use in practice. Quality appraisal will be conducted by two members of the research team to ensure consistency and reliability of the process. Scoring differences will be assessed and considered by the two appraisers and when no consensus is reached, the larger research team will be involved to make a final decision. The AGREE PLUS is a platform that allows multiple users to complete, contribute to, coordinate and track appraisals online. This website operates using the email addresses of registered and invited researchers only, making it easier for multiple members to contribute to the appraisal process on one interface.

Stage 7: evidence (data) analysis

Analysis in a scoping review does not require data synthesis as used, for example, in reviews of quantitative data. Descriptive analysis of data extracted is advised and will be used for the current review.18 Descriptive analysis will collate frequency counts of populations, concepts, location of studies, type of resources, and so on which can then be mapped out for visual presentation using bar or pie charts, tables or word clouds as the research team deems fit. The way data are analysed is highly dependent on the purpose of the scoping review. For scoping reviews with a primary purpose of identifying concepts or clarifying definitions, qualitative descriptive analysis involving basic coding to set categories may be utilised with a thematic approach.22 23 However, given that the purpose of our review is to identify specific resources, basic descriptive analysis as described previously will be implemented.7

Stage 8: evidence (data) reporting and presentation

This scoping review will be reported in line with the (PRISMA-ScR) checklist24 and a PRISMA flowchart (figure 1) will be used to show search results of the screening and selection process. Diagrammatic (bar/pie charts), descriptive or tabular formats will be used to report and present findings from the quality appraisal and charting stages, depending on the contents of included evidence. Data reported and presented will align with the objectives and scope of the review.

Stage 9: evidence summary

The main findings, including any concepts and the type of evidence available, will be summarised in relation to the review objectives. Relevance to key health professional groups will be considered and conclusions and recommendations made for future research and resource development based on identified gaps. In accordance with the aim of the review, outcomes will help identify resources that can be used by health professionals. To facilitate access to resources by Australian health professionals, any published, high-quality resources identified in the scoping review will be posted and freely available on the FASD Hub (www.fasdhub.org.au). The scoping review should be completed in 2022 and results will be published in a peer-reviewed publication and on the FASD Hub.

Patient and public involvement

The development of this research protocol did not require any involvement with patients or the public. However, during the scoping review process, consultation with experts in the diagnosis and management of FASD will occur to identify any additional, relevant resources not already included.

Ethics and dissemination

This is the first scoping review on this topic and will provide important insights for health professionals working with people with FASD. The methodological process is detailed and is guided by an established and referenced framework ensuring its reproducibility and transparency. Dissemination of data and results will be through conferences, peer-reviewed journals and relevant seminars to health professionals. Results from the review will also help inform the development of new resources to support health professionals. Scoping review is anticipated to be completed by the end of 2022 and results disseminated early in the year 2023. Ethical approval is not required for the scoping review.

Ethics statements

Patient consent for publication

References

Footnotes

  • EJE and LJR are joint senior authors.

  • Contributors This review is part of a larger project designed by EC, LJR, ST and EJE. JCO, LJR designed the scoping review and EJE and LC provided critical feedback on the protocol. JCO wrote the first draft of the review protocol which was revised and approved by LJR, EJE, EC, ST and LC. All authors have had training and experience in conducting systematic reviews, including use of checklists and as a result, were able to provide significant contributions.

  • Funding This work was supported by a National Disability Insurance Scheme Mainstream Capacity Building Grant (#4-DWLFU8L). LJR is supported by the Ian Potter Foundation (#31110414) and a 2021 Westpac Research Fellowship. EJE is supported by an Australian Medical Research Futures Fund Next Generation Fellowship (#1135959).

  • Competing interests EJE chairs the FASD Hub advisory board, and some authors have been involved in the development of some FASD resources. To address this, the identification and quality appraisal of resources will be conducted by two reviewers who are independent of the development of any such existing FASD resources.

  • Patient and public involvement Patients and/or the public were not involved in the design, or conduct, or reporting or dissemination plans of this research.

  • Provenance and peer review Not commissioned; externally peer reviewed.