Developmental Medicine & Child Neurology

Volume 66, Issue 6 pp. 702-712

SCOPING REVIEW

Open Access

Lived experience of fetal alcohol spectrum disorder: A qualitative scoping review

Holly Wilson,

Holly Wilson

orcid.org/0000-0001-8701-3872

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

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Jessica C. McCormack,

Jessica C. McCormack

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

Food Science, University of Otago, Dunedin, New Zealand

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Nikki Singh,

Nikki Singh

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

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Joanna Ting Wai Chu,

Corresponding Author

Joanna Ting Wai Chu

[email protected]

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

Correspondence

Joanna Ting Wai Chu, Social and Community Health, School of Population Health, The University of Auckland, Private Bag 92019, Auckland Mail Centre, Auckland 1142, New Zealand.

Email: [email protected]

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Holly Wilson,

Holly Wilson

orcid.org/0000-0001-8701-3872

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

Search for more papers by this author

Jessica C. McCormack,

Jessica C. McCormack

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

Food Science, University of Otago, Dunedin, New Zealand

Search for more papers by this author

Nikki Singh,

Nikki Singh

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

Search for more papers by this author

Joanna Ting Wai Chu,

Corresponding Author

Joanna Ting Wai Chu

[email protected]

Social and Community Health, School of Population Health, The University of Auckland, Auckland, New Zealand

Correspondence

Joanna Ting Wai Chu, Social and Community Health, School of Population Health, The University of Auckland, Private Bag 92019, Auckland Mail Centre, Auckland 1142, New Zealand.

Email: [email protected]

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First published: 04 October 2023

https://doi.org/10.1111/dmcn.15761

Citations: 6

This scoping review is commented on by Andrew on pages 683–684 of this issue.

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Abstract

Aim

To explore the lived experiences of individuals with fetal alcohol spectrum disorder (FASD), their caregivers, and professionals working with individuals with FASD.

Method

We conducted a scoping review using qualitative methods to explore what it is like to live with FASD from the perspective of those living with FASD, their caregivers, and stakeholders experienced in working with individuals with FASD. We searched electronic databases and grey literature for research published between 2005 and 2022.

Results

The 47 studies included in this scoping review show that FASD affects people's lives on a daily basis. Individuals with FASD are aware of their disability and its effects. Caregivers face daily challenges raising children with FASD and are often left unsupported by professionals. Professionals are unprepared and frustrated when supporting someone with FASD.

Interpretation

FASD affects individuals, caregivers, and families on a daily basis; and they require improved support and funding.

What this paper adds

Professionals are overwhelmed and frustrated because of a lack of training and organizational support with regard to fetal alcohol spectrum disorder (FASD).
Extends on previous reviews with the inclusion of grey literature and professionals.
FASD is a highly stigmatized disorder that affects individuals with FASD, and their caregivers and families, on a daily basis.
Research is needed exploring the experiences of adults living with FASD and their siblings.
There is a lack of research involving indigenous populations and their experience with FASD.

What this paper adds

Professionals are overwhelmed and frustrated because of a lack of training and organizational support with regard to fetal alcohol spectrum disorder (FASD).
Extends on previous reviews with the inclusion of grey literature and professionals.
FASD is a highly stigmatized disorder that affects individuals with FASD, and their caregivers and families, on a daily basis.
Research is needed exploring the experiences of adults living with FASD and their siblings.
There is a lack of research involving indigenous populations and their experience with FASD.

This scoping review is commented on by Andrew on pages 683–684 of this issue.

Abbreviations

FASD: fetal alcohol spectrum disorder
MMAT: Mixed Methods Appraisal Tool
PAE: prenatal alcohol exposure

Prenatal alcohol exposure (PAE) can result in a range ofneurodevelopment impairments, which can affect an indi-vidual's physical, behavioural, and cognitive functioning,while also having an impact on everyday life.¹ When theseimpairments have a significant impact on three or more cog-nitive domains, an individual may be diagnosed with fetal alcohol spectrum disorder (FASD).¹ FASD is estimated to affect 7.7 per 1000 children globally.² Diagnosing FASD is complex, often requiring expertise from multiple disciplines and complex assessments; yet, there are few trained profes-sionals. Symptoms of FASD include diminished neurologi-cal and cognitive functioning, as well as behavioural prob-lems.¹ These symptoms result in primary effects that impactindividuals with FASD, such as difficulties with social skills, memory, academic achievement, cognition, and attention.³ These difficulties affect people's ability to function in sev-eral aspects of life, including education, employment, mental health, and engagement with the justice system.^{4, 5}

Individuals with FASD have difficulties that persist throughout their life, from early life through to adulthood.⁴ These difficulties range from everyday challenges, such as keeping appointments and understanding complex social situations, to tertiary difficulties that result from failures to accommodate and support individuals affected by FASD, such as school disruption, substance misuse, homelessness, unemployment, and greater risk of being involved with the justice system.^{4, 6} Compounded by a lack of support and stigma, individuals living with FASD may also experience challenges with their mental and physical health;^{5, 7} average life expectancy is significantly below the general population at 34 years.⁸ Stigma and misconceptions about FASD are pervasive; individuals with FASD may be perceived by others as lazy, troublesome, or bad,⁹ while caregivers are perceived as bad and uncaring parents,¹⁰ and biological parents as careless.⁹ Stigma is felt by those affected by FASD, resulting in isolation and difficulties accessing support.⁹

Given the difficulties individuals with FASD experience, early diagnosis and effective support and interventions, are essential to support individuals with FASD.⁴ Professionals are key in providing support directly to those affected by FASD; however, professionals often feel unprepared, while lacking the training, organizational support, and confidence needed to support individuals with FASD.^11-13 For example, a recent survey of educators found that most were aware of FASD but had significant gaps in their knowledge; very few felt well prepared to educate someone with FASD.¹² Being unprepared could in part contribute to difficulty accessing diagnosis and the lack of support individuals with FASD experience, despite early intervention being the key to success.⁴

Although emerging research is beginning to explore how individuals with FASD experience everyday life, it is complicated by diverse views. Therefore, a synthesis of the existing literature is useful to understand how people experience everyday life while living with FASD. A previous systematic review examined the lived experiences of individuals with FASD, demonstrating that individuals with FASD experience a range of difficulties that impact their everyday functioning, while caregivers experience concerns, anxiety, stigma, and a lack of support.¹⁴ However, the review did not include grey literature or consider the views of professionals who have experience working with individuals with FASD, views that are vital because they can shape access to early intervention and support. Therefore, we conducted a scoping review to provide an up-to-date synthesis of the literature on the lived experiences of individuals with FASD, their caregivers, and those working with individuals with FASD to understand their everyday experiences to better inform services. A scoping review enables a more comprehensive examination of the evidence and synthesis of any existing gaps.¹⁵

METHOD

Protocol and registration

This scoping review was conducted according to the Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) extensions for Scoping Reviews¹⁶ and was registered on PROSPERO (CRD42022321615).

Information sources and search strategy

We conducted an electronic search to identify studies that examined the lived experiences of individuals with FASD, their caregivers, and those working with individuals with FASD. The search was conducted between 26th April 2022 and 30th April 2022. Published literature was identified by searching the following databases: PubMed, Scopus, CINAHL, Embase, APA PsycInfo, and Cochrane. We searched for grey literature using the ProQuest Dissertation and Thesis database, WorldCat, TROVE (National Library of Australia), NZ Research (https://nzresearch.org.nz/), the first 200 results on Google Scholar, government websites (Australia, Canada, New Zealand, and USA), World Health Organization, and FASD support sites (FASD-Can, CanFASD, FASD Hub Australia). The reference list of each included article was screened for additional sources. Searches were restricted to those published in English since 2005, when comprehensive diagnostic criteria for FASD were first established.¹⁷ The search string combined FASD and related terms (e.g. alcohol-related neurodevelopmental disorders, prenatal alcohol exposure) with terms related to daily activities, life and lived experiences, and adult, child, or adolescent populations (see Appendix S1 for an example of the search strategy used in the database search). The search strategy was modified to suit grey literature searches where it was not possible to combine search terms.

Eligibility criteria

Studies were eligible if they met the following criteria: (1) included participants with a diagnosis of FASD or suspected FASD, their caregivers, or any stakeholders with experience of working with individuals with FASD; (2) examined an aspect of lived experiences (e.g. experience with health care or daily challenges); (3) used a qualitative research design; (4) were published after 2005; and (5) were published in English. We excluded studies that used animals, explored the biology of FASD, or used quantitative designs.

Study selection and data charting

Search results were imported into EndNote (Clarivate, Philadelphia, PA, USA) and duplicates were removed. Once duplicates were removed, the title and abstract were independently screened against the inclusion criteria by two reviewers (HW, NS) to determine eligibility. The full text for eligible studies was obtained and screened by one reviewer (HW) with 10% checked by another reviewer (NS). Any disagreement was discussed among all reviewers until a consensus was reached.

Data were extracted to a bespoke Excel spreadsheet by the first author; a random sample of 10% of the articles was extracted by another author (NS) to check reliability. Data that were extracted included: author, date, full citation, number and characteristics of participants, diagnostic information, country, research approach, methods, data analysis, and main findings.

Critical appraisal of individual sources of evidence

The quality of the studies included in this scoping review was assessed using the Mixed Methods Appraisal Tool (MMAT¹⁸). MMAT was chosen because it can be applied to different study designs. It consists of three sections, that is, screening, design, and quality based on research approach, methodology, and results, which are applied to each study based on the criteria of each study established in step 2. Studies are not given an overall score; rather, the response to each item is considered to determine the quality of the study.^{18, 19} The first author applied the quality assessment tool to all articles, with a 10% random sample reviewed by another author (NS); any disagreements were discussed among all reviewers.

Synthesis of the results

A narrative review of the main findings from each study was conducted. Specifically, the main findings from each study were exported into NVivo (QSR International, Denver, CO, USA) and they were coded for common patterns in the data. All authors were consulted to discuss the common patterns in the data set. A narrative synthesis was chosen as the best approach for synthesizing qualitative data and findings from multiple studies.

RESULTS

Selection of sources of evidence

Our search produced 5561 results. After duplicates were removed and the criteria were applied, 47 studies meet the inclusion criteria and were included in this scoping review (Figure S1).

Characteristics of the sources of evidence

Of the 47 studies included in this scoping review (see Table S1 for the individual studies), most studies (n = 38) reported the experiences of caregivers (biological, foster, or adoptive), 13 studies involved individuals living with FASD, and 11 included the experience of professionals. Most studies (n = 36) explored the general lived experiences of individuals with FASD, while a few (n = 11) focused on specific aspects of the lived experience, such as mental health or sleep. As shown in Table 1, of 122 individuals with FASD most (63.12%) were younger than 30 years; of the 808 caregivers, 85 (10.39%) were biological caregivers. The 11 studies exploring the experiences of professionals captured the views of 162 professionals, of which only six (3.70%) worked in education and 14 (8.64%) in the justice system. Most of the studies included participants with self-reported FASD (n = 36); only 11 studies were restricted to participants with an official FASD diagnosis. Most studies were conducted in Canada (n = 36), with a few studies exploring FASD in New Zealand (n = 4), Australia (n = 4), the USA (n = 3), and the UK (n = 4). Only two studies explored the lived experiences of indigenous people with FASD;^{20, 21} several studies had participants who self-reported as indigenous, but their experiences were not explored separately.

TABLE 1. Characteristics of participants (n = 1092) in the studies that met the inclusion criteria of this scoping review.

Characteristic	n (%)
Individuals with FASD	122 (11.17)
Age, years
0–10	7 (5.74)
11–20	40 (32.79)
21–30	30 (24.59)
>31	12 (9.84)
Adults	33 (27.0)
Professionals with lived experience	162 (14.84)
Sector
Health care	71 (43.82)
Social	71 (43.82)
Justice system	14 (8.64)
Education	6 (3.70)
Caregivers	808 (74.00)
Biological	85 (10.39)
Non-biological (adoptive or foster parent)	505 (62.96)
Not specified	218 (26.65)

Abbreviation: FASD, fetal alcohol spectrum disorder.

Identification of themes and subthemes

Our narrative analysis identified four themes: (1) raising a child with FASD is both challenging and rewarding; (2) individuals with FASD experience a range of difficulties throughout their lives; (3) individuals are aware of their disability (4) and lack of knowledge and support is a major challenge (Tables 2 and 3 for the main themes and subthemes).

TABLE 2. Themes and subthemes of lived experiences of FASD.

Themes	Subthemes
Raising a child with FASD is both challenging and rewarding	Caregivers use an adaptive parenting style to support the individual living with FASD. Caregivers often experience stigma due to FASD. Caregivers experienced increased burden. Caregivers have concerns about the future.
Individuals with FASD experience a range of difficulties throughout their life	Individuals with FASD experience a range of primary effects for PAE. Individuals with FASD difficult and traumatic childhoods. Secondary effects due to FASD. Tension between desire for independence and support. Individuals with FASD have difficulties with employment. Individuals with FASD can be involved with the justice system.
Individuals with FASD are aware of their disability	Individuals with FASD are aware of their FASD and its effects. Individuals with FASD recognize factors that support them.
Lack of knowledge and support is a major challenge	Knowledge is important for caregivers. Caregivers value and depend on informal support. Caregivers' experience with formal support is negative. Caregivers are required to advocate. Professionals feel unprepared and unsupported to help individuals with FASD.

Abbreviations: FASD, fetal alcohol spectrum disorder; PAE, prenatal alcohol exposure.

TABLE 3. Subthemes of the lived experience of FASD.

Subtheme	Description of the subtheme
Motivation to foster children with FASD	Foster parents choose to foster children with FASD to support the child and to develop their own parenting skills.⁴⁷
Benefits of respite care but concerns	Respite care is useful to give caregivers a break and can help manage difficult behaviours;^{22, 24, 36, 46} however, some caregivers are hesitant to use it because of the risk to the child.²⁴
Professional strategies to support individuals with FASD	Professionals use several strategies to support individuals with FASD: Diagnosis: professionals recognize the importance of diagnosis to ensure that individuals have the right support and fair treatment in the justice system.⁶¹ Health care professionals will sometimes work with a different diagnosis, in part due to lack of knowledge, but sometimes to gain access to funding and support that is unavailable when people are diagnosed with FASD.50, 60, 61 A strength-based approach by professionals has been recognized as helpful and there needs to be advocacy to ensure that the individual with FASD is accessing the support they require;^{60, 61} however, often there is limited support available to individuals with FASD.³⁵ Inexperienced educators report not doing anything different to educate individuals with FASD, which can create frustration.⁴⁶ On the other hand, experienced teachers adapt the curriculum to the needs of the individual with FASD.^{37, 46}
Lived experiences with receiving an official diagnosis (from a caregiver perspective)	Caregivers reported that when the diagnosis is associated with positive interactions and support from health care professionals, it can be an empowering and positive experience.³⁸ However, accessing a diagnosis is often a negative experience, consisting of reluctance to diagnose and stigma, such as health care professionals not wanting to ask about prenatal alcohol consumption.^{14, 35, 41, 57} An official diagnosis is important to be able to access the limited supports available.^{14, 34, 35, 43} For caregivers, diagnosis comes with a range of emotions, including guilt for biological mothers,³² a sense of validation, a feeling that their concerns were real, an understanding of the child's behaviour,³⁸ and access to some support.⁴⁶
Primary disabilities experienced by individuals with FASD	Difficulties with emotional regulation, having tantrums.^{25, 32, 35, 71} Cognitive: lacking insight of the consequences of actions,^{14, 25, 32, 41, 71} difficulty maintaining attention,⁷¹ difficulty with memory,^{25, 34, 71} difficulty comprehending or understanding information,^{40, 71} confabulation,^{35, 71} difficulty learning from mistakes,^{25, 32} indecisiveness.³⁴ Behaviour: difficulty with behaviour regulation,^{30, 32, 40, 41, 46, 58, 71} disruptive behaviours,⁵⁸ destructive behaviours,^{14, 30} running away,^{35, 46} stealing,^{35, 71} setting fires.⁴⁶ Executive functioning: difficulties academically. Social skills: difficulty in interpreting verbal cues,^{35, 58, 71} friendly to strangers.⁷¹ Motor skills: clumsy, difficulty with fine motor skills.⁷¹ Physical health: difficulties with sleeping,^{30, 42, 71} eating, and speaking.⁴⁰ Mental health: depression and anxiety are common,⁵³ suicide attempts or ideations,^{52, 53, 62} difficulty dealing with negative thoughts,⁵³ self-harm;^{34, 45} can be managed with treatment and support.^{30, 52}

Abbreviation: FASD, fetal alcohol spectrum disorder.

Raising a child with FASD is both challenging and rewarding

Twenty-eight studies explored the experiences of caregivers raising children with FASD. Most studies included the views of both biological and adoptive caregivers, whereas some studies explored the views solely of biological or adoptive caregivers. From these studies, four subthemes were identified that concerned the experiences of caregivers raising children with FASD.

Caregivers use an adaptive parenting style to support the individual living with FASD

In several studies,^21-37 adoptive and biological caregivers described using adaptive parenting styles to support individuals with FASD. Specifically, biological and adoptive caregivers reported that maintaining structure and routine are key. To achieve this, caregivers monitor and modify the environment to support individuals with FASD using strategies consisting of, but not limited to, setting consistent rules and using a range of communication strategies. Caregivers adapted their parenting based on previous success or failure. Specifically, caregivers felt key characteristics that support success include having patience, optimism, and setting expectations in line with ability. Although caregivers felt that parenting a child with FASD was a challenge, they could identify positive aspects, such as positive experiences and parenting skills.

Caregivers often experience stigma due to FASD

Adoptive and biological caregivers experienced stigma because their child had FASD, leaving them feeling blamed, misunderstood, and isolated; this stigma contributed to caregiver burden.^{9, 10, 20, 21, 25, 32, 35, 36, 38-44} Adoptive caregivers can be blamed for their child's negative behaviours and considered as bad parents, especially if the child does not show the facial features of FASD. While biological caregivers are stigmatized as uncaring and bad parents, when in reality most are unaware of the potential effects of PAE during pregnancy. This stigma leaves biological caregivers feeling responsible, and feeling guilt and shame. For all caregivers this stigma can be a barrier to seeking or receiving help. Caregivers reported experiencing stigma from health care^40-42 and education³² professionals, and the general population.^{9, 25, 38}

Caregivers experience increased burden

In 11 studies, biological and adoptive caregivers reported the burden experienced from having a child with FASD.^{14, 27, 30, 36, 37, 39, 40, 45-48} These burdens included increased parenting burden and social isolation due to the demands of parenting a child with FASD, stigma, and behavioural challenges. Caregivers had concerns that these burdens extended to the entire family, potentially resulting in marital strain or breakdown, strained sibling relationships, or the caregiver spending less time with siblings, often leaving caregivers frustrated and exhausted.

Caregivers have concerns about the future

In eight studies, biological and adoptive caregivers had concerns about the future of their child with FASD.^{25, 31, 32, 35, 38, 39, 43, 49} Concerns caregivers held included: the provision of ongoing support when caregivers were unable to provide; the ability to live independently; continuing in education; being involved in the wrong social group; falling into criminal or gang involvement; and concerns about the lack of available support. Despite these concerns, caregivers still hoped that their child would thrive and live a fulfilling life in the future.

Individuals with FASD experience a range of difficulties throughout their life

Across studies exploring the lived experiences of individuals with FASD and their biological or adoptive caregivers, difficulties throughout life was a common theme. This theme incorporates specific challenges encountered by individuals living with FASD; within this theme, six subthemes were identified.

Individuals living with FASD experience a range of primary effects from PAE

Across several studies, individuals living with FASD and their adoptive and biological caregivers identified a range of primary effects from PAE. Caregivers commonly reported difficulties with regulating emotions, cognition, harmful or challenging behaviours, executive functioning at school, social skills, motor skills, and health. Individuals living with FASD reported difficulties with mental and physical health and social skills, as well as academic challenges at school (see Table 2 for a full list of primary disabilities).

Difficult and traumatic childhoods

In four studies,^{30, 50-52} individuals living with FASD reported difficult or traumatic childhoods. Those who grew up in their biological homes often reported that they regularly experienced neglect, abuse, poverty, and abandonment, and grew up in homes with parents who suffered alcohol and drug addiction. Often children were adopted and fostered, which can expose them to abuse and separation from their families. For some individuals, these childhood experiences shaped negative perceptions of alcohol and drugs, and caused alcohol and drugs to be used as a coping mechanism from a young age, creating ongoing mental health challenges.

Secondary effects due to FASD

The primary effects of FASD leave individuals with FASD vulnerable to ongoing daily challenges across school, independent living, employment, and involvement with the justice system. While at school, adolescents and adults with FASD reported that they struggle academically^{30, 50-53} and are often placed in special classrooms,^{30, 50} contributing to isolation from their peers, although some individuals viewed this as necessary for academic success.⁵⁰ These adolescents and adults with FASD recognized that they have difficulty with peer relationships, while a desire to fit in leaves them vulnerable to risky behaviour.^{30, 50, 53} Birth, adoptive, and foster caregivers reported similar concerns, feeling that there was a lack of support in schools and the set-up did not support success for individuals with FASD,^{21, 28, 32, 35, 36} along with concerns that individuals with FASD were left socially isolated and excluded from their peers.

Tension between desire for independence and support

In three studies involving adolescent and adults with FASD^{23, 30, 52} and six involving biological, adoptive, and foster caregivers,^{26, 31, 32, 34, 35, 54} a tension between a desire to be independent and the need for ongoing support was reported. These caregivers felt that their child with FASD would require lifelong support, but had concerns because there is limited support available; managing the tension between providing support and maintaining their relationship with the child with FASD could be difficult.

Difficulties with employment

Adolescents and adults living with FASD^{30, 53} and biological and adoptive caregivers explained that individuals with FASD have difficulties with employment.^{14, 21, 34, 49} Individuals with FASD struggle with gaining and maintaining employment because of their difficulties with cognition, behaviour, and transport issues. Some individuals with FASD had experienced discrimination from employers and felt this was a barrier to successful employment. Likewise, biological and adoptive caregivers had employment concerns due to risky behaviour, but felt that supportive workplaces and understanding co-workers can support employment.

Involvement with the justice system

Five studies^{21, 23, 41, 52, 53} exploring the lived experiences of adults with FASD and their biological parents reported on experiences with the justice system. Those with FASD reported that they struggle with authority, dislike being told what to do, and can become involved in risky behaviour, such as stealing, placing them at increased risk of being involved in the justice system. FASD can go unrecognized in the justice system, with people often unsupported and undiagnosed or receiving a diagnosis later in life.

Individuals with FASD are aware of their disability

Thirteen studies explored the lived experiences of people with FASD using interviews. Analysis of these studies revealed two subthemes unique to individuals with FASD.

Individuals living with FASD are aware of their FASD and its effects

In six studies, people with FASD reported self-awareness of their diagnosis and the effects they experienced.^{14, 30, 51-53, 55} Some individuals with FASD were aware of their diagnosis, and when learning about their diagnosis they experienced frustration and a greater understanding of the challenges they face. Others were aware of the challenges they faced because of FASD, such as remembering and ongoing mental health challenges, and were aware of some of the impacts FASD had on their family. These challenges leave individuals with FASD feeling different to others and isolated from their peers.

Individuals living with FASD recognise factors that support them

In three studies,^{23, 52, 55} individuals with FASD reported the factors that supported them. For individuals living with FASD, it was important to have an understanding support network and a strength-based approach. Individuals with FASD were aware of their own strengths, such as helpfulness and compassion. Individuals living with FASD felt they were successful when they had positive experiences and made contributions to their communities and societies, helping them to cope with the difficulties of living with FASD.

Lack of knowledge and support is a major challenge

Several studies explored the experiences of professionals supporting individuals with FASD and the experiences of adoptive and biological caregivers with this support; one main theme was identified. This theme encompasses the lack of knowledge of caregivers and professionals have about FASD and barriers to support. This theme contains five subthemes.

Knowledge is important for caregivers

Biological and adoptive caregivers believe that information about FASD is essential to help them understand, develop successful parenting techniques, and make an informed decision before fostering a child with FASD.^{22, 23, 26-28, 32, 33, 36, 38, 39, 41, 43, 48} However, often caregivers lack knowledge about FASD, particularly prior to diagnosis; therefore, most caregivers had sought knowledge about FASD, which can be impaired by a lack of available information.

Caregivers value and depend on informal support

In several^{22, 24, 26-28, 33, 35, 36, 43} of the studies included in this scoping review, biological, adoptive, and foster caregivers reported that informal support, usually found in friends and family and support groups, is vital for well-being. Informal support provides caregivers with an accepting, non-judgmental place to discuss shared experiences and concerns. Also informal supports often provide practical support, such as looking after the child with FASD. Yet, informal support was not sufficient on its own and caregivers also needed formal support.

Caregiver experience with formal support is negative

Several studies^{14, 22, 24-29, 32, 33, 36, 40, 41, 44-46, 56-59} explored the experience of adoptive and biological caregivers with formal support. Formal support was vital to provide advice, information, and support to the whole family, as well as preventing caregiver burnout; caregivers often experienced a lack of support. Interactions were often negative, due to professionals' lack of knowledge, being misinformed, or holding stigmatizing beliefs. These interactions leave caregivers feeling unheard, ignored, or unappreciated, that is, hampered by misinformation or the stigmatizing beliefs held by professionals. Caregivers reported being bounced around services and only having access to support at times of crisis due to policy-based issues and lack of available services. Of the few positive interactions, caregivers valued the support, effort, and being heard by professionals and felt that a strength-based approach is key.

Caregivers are required to advocate

Across 11 studies with biological and adoptive caregivers,^{14, 23, 26-29, 36, 38, 39, 50, 59} advocacy was key to their child's success. Caregivers felt that advocacy is important so that their child is not misunderstood or unsupported because of lack of knowledge among professionals, and to share the insights they have learned through their lived experience. This advocacy is particularly important in education, to support teachers and ensure that education is matched to the child's ability, and in health care where lack of knowledge among health care professionals can impede support. Caregivers reported that their advocacy was not always well received by health care professionals, describing it as a ‘fight’ to access support and as adding to caregiver burden.

Professionals feel unprepared and unsupported to help individuals with FASD

Seven studies explored the experiences of professionals working in the health care, social, education, and justice sectors with experience supporting individuals with FASD.^{35, 37, 42, 46, 58, 60, 61} Professionals reported difficulty supporting individuals with FASD because of a lack of professional training, institutional support, and unclear professional boundaries, leaving professionals feeling unprepared and overwhelmed. For health care professionals, they felt that the focus is on the prevention of PAE, leaving some health care professionals feeling reluctant to diagnose FASD due to a lack of confidence, holding stigmatizing beliefs, and the benefits of diagnosis being unclear due to a lack of funding. Professionals were aware of a lack of support and funding for individuals with FASD, therefore, caregivers become the advocate and support for individuals with FASD. Education professionals had to figure out themselves how to best support individuals with FASD, with new teachers finding this overwhelming, whereas experienced teachers had developed strategies to support individuals with FASD.

Critical appraisal

The MMAT tool was applied to 45 of the 47 studies to assess quality. Quality assessment could not be applied to two studies because MMAT cannot be applied to case studies.^{48, 62} The results of the quality assessment are shown in Appendix S2. Of the studies assessed for quality, most met all the relevant criteria of MMAT (n = 34, 75.56%). A few studies (n = 11, 25%) did not provide sufficient detail of the methods used to determine their suitability in addressing the research question, therefore making it difficult to determine study quality. Two studies^{37, 52} met only two of the five criteria because of insufficient information needed to derive the methodology of the study to assess its quality.

DISCUSSION

Our scoping review is largely consistent with a previous systematic review;¹⁴ inclusion of the current literature expands on the experiences from the perspective of individuals with FASD demonstrating self-awareness, feelings of isolation, and mental health challenges. The experiences of caregivers are largely consistent with the previous review;¹⁴ the inclusion of up-to-date research shows that the challenges and experiences of caregivers remain largely unchanged or have not improved.

This scoping review, which is consistent with the previous literature,^{14, 63} mainly focuses on the challenges and difficulties faced by individuals with FASD and their caregivers. Although this scoping review reveals the strengths and positive qualities individuals with FASD possess and the benefits for caregivers raising a child with FASD, it mainly focuses on the difficulties and challenges faced by those with FASD. A recent review found that individuals with FASD are aware of their strengths, such as resilience, and a desire to be supported and connected with others.⁶³ The focus in the literature on the challenges and difficulties of FASD creates a negative discourse and contributes to stigmatizing beliefs. Our scoping review shows that FASD is still a highly stigmatized disability and stigma is often experienced by both caregivers and individuals with FASD. Additionally, this stigma creates barriers to support and accessing a diagnosis, despite early intervention being the key to success.⁴ Therefore, there is a need for more strength-based research to demonstrate the qualities of individuals with FASD and create a positive and supportive environment for individuals with FASD and their families.

Of the studies included in this scoping review, only 13 explored the lived experiences from the perspective of individuals with FASD. Although, it is likely difficult to conduct research with individuals living with FASD because of their difficulties with communication, memory, and the confabulation³ associated with PAE. Our scoping review demonstrates that several studies have been conducted with individuals with FASD and provide insight into their experiences that would not be available without this approach, such as insight and feelings of isolation and exclusion. Therefore, it is vital that research understands and responds to the lived experiences of individuals with FASD. Methods beyond interviews could be used to capture the lived experiences of individuals with FASD, such as a photovoice methodology.⁶⁴ A recent study used photovoice methodology to explore the lived experiences of adolescents with FASD.⁶⁵ This study identified barriers to engagement in daily activities, such as anxiety, and the need for positive relationships;⁶⁵ it emphasized the importance of participatory research to capture the experiences of individuals with FASD.

Likewise, no studies included in this scoping review directly explored the lived experiences of siblings of individuals with FASD. FASD burdens the entire family and caregivers have concerns about the impact of FASD on siblings and sibling relationships;^{30, 47} yet, no studies specifically explored the impact of FASD on siblings. In fact, no studies explored the impact or effects of being a sibling of an individual with FASD, despite siblings of individuals with other neurodevelopmental disorders having poorer outcomes.^{66, 67} Therefore, future research needs to be drawn from the perspective of the individual with FASD while exploring the experiences of their siblings.

Many of the studies included in this review drew from the experiences of children and adolescents with FASD; only four^{23, 30, 52, 53} studies included participants older than 31 years. One of these studies used case reports to explore the lived experiences of two participants in their 60s, revealing ongoing challenges with mental health, employment, housing, and involvement with the criminal justice system throughout adulthood.⁵² This lack of research into adult experiences of FASD is concerning given that individuals with FASD experience significantly lower life expectancies⁸ and difficulties that persist into later life.^{4, 6} Therefore, it is vital that future research explores the experiences of adults with FASD. As transition, employment, a desire for independence, and healthy relationships were identified as common challenges for individuals with FASD that are more pervasive as people get older, it is important to explore further the experiences of adults with FASD to improve the support and services available to them and to improve the quality of life of individuals with FASD and their caregivers.

Our review provides an insight into how professionals feel and the difficulties they face when supporting someone with FASD. Professionals who can provide key support often feel unprepared and unsupported to support individuals with FASD.^{11-13, 68} This scoping review shows a lack of knowledge compounded by a lack of organizational support, policies, and support available for individuals with FASD, leaving professionals frustrated and overwhelmed. This increased burden and the negative emotions professionals experience could enforce negative stereotypes and misinformation by creating negative perceptions of individuals with FASD and their caregivers.⁹ These negative perceptions could be compounded by caregivers advocating for their child, often perceived as not well received by professionals, and the lack of government recognition and support for individuals with FASD. Therefore, there is a need for improved training, organizational support, and funding to support individuals with FASD and professionals.

Despite indigenous populations likely experiencing a greater burden of PAE and therefore high rates of FASD, there is limited research on the lived experiences of indigenous people with FASD. Although, indigenous participants were included in several studies, only two studies^{20, 21} explored the experiences of indigenous caregivers independently, making it difficult to understand the lived experiences of FASD within indigenous populations. It is likely that indigenous people with FASD may face greater barriers to accessing support given the difficulties in accessing care, and stigma, with some people holding the perception that FASD is more common in indigenous populations.⁹ Therefore, it is important to explore the experiences of indigenous populations with regard to support and look for culturally appropriate interventions, which are few and far between, to support individuals with FASD.⁶⁹

Study limitations

FASD covers a range of different diagnoses and effects that stem from PAE;^{1, 70, 71} therefore, the lived experiences of individuals with FASD and their caregivers are likely to be different due to the different effects. A scoping review enables findings across studies to be compared, looking for common themes across studies, and producing common lived experiences; however, some aspects of the lived experience could be missed because they are less common and have not been captured in the studies included in this scoping review.

Electronic search methods and the selection of databases may have limited the studies that were identified and this scoping review only includes studies published in English. Therefore, some studies may not have been identified; regardless, the scoping methodology allows the inclusion of unpublished work, which can provide an insight into the lived experiences of individuals with FASD and their caregivers.

Most studies included in this review were conducted in Canada. This is likely due to the early recognition of FASD in Canada, as evidenced by early diagnostic guidelines,¹⁷ and the Canada FASD research network facilitating FASD research in Canada. As most of the studies in this scoping review are from Canada, the findings may not reflect the experiences of those living elsewhere. However, synthesis of the literature showed similar experiences and challenges experienced across studies from different countries. Nevertheless, as recognition, awareness, and research in FASD grows, this review may need to be updated to ensure that the lived experiences reflects those from different countries.

Studies in this review included participants who self-reported as having FASD. It is possible that in studies with self-reported FASD, individuals may be waiting for an official diagnosis or may have been misdiagnosed, as identified by one of the subthemes of this scoping review. This difficulty with diagnosis is unsurprising given the barriers to recognition and difficulty in accessing a diagnosis, as well as a lack of trained health care professionals to diagnose FASD. Therefore, given the difficulties with accessing diagnosis and identifying misdiagnosis, it is important to include studies of individuals with self-reported FASD.

Conclusions

FASD is a lifelong disability that affects individuals, their caregivers, and families on a daily basis. Individuals with FASD are aware of their disability and can be left feeling different and isolated, while caregivers have to negotiate the daily challenge of parenting a child with FASD. Despite these challenges, individuals with FASD have talents and qualities; raising a child with FASD can bring joy to both caregivers and families. Professionals feel unprepared and overwhelmed when supporting someone with FASD because of a lack of knowledge, funding, and support for individuals living with FASD. There is a need for greater recognition, acceptance, and funding to improve the daily lives and outcomes of individuals with FASD and their families.

ACKNOWLEDGEMENTS

This study was supported by an Ember Korowai Takitini Research Grant. Open access publishing facilitated by The University of Auckland, as part of the Wiley - The University of Auckland agreement via the Council of Australian University Librarians.

CONFLICT OF INTEREST STATEMENT

The authors declare no conflicts of interest.

Open Research

DATA AVAILABILITY STATEMENT

The data that support the findings of this study are available from the corresponding author upon reasonable request.

Supporting Information

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Citing Literature

Volume66, Issue6

June 2024

Pages 702-712

Filename	Description
dmcn15761-sup-0001-AppendixS1.docxWord 2007 document , 13.5 KB	Appendix S1: Scopus search strategy
dmcn15761-sup-0002-AppendixS2.docxWord 2007 document , 21.7 KB	Appendix S2: Quality assessment of each study
dmcn15761-sup-0003-FigureS1.docxWord 2007 document , 25.7 KB	Figure S1: PRISMA flow chart of the studies included in this scoping review
dmcn15761-sup-0004-TableS1.docxWord 2007 document , 19.3 KB	Table S1: Characteristics of the studies that met the inclusion criteria

Lived experience of fetal alcohol spectrum disorder: A qualitative scoping review

Abstract

Aim

Method

Results

Interpretation

What this paper adds

What this paper adds

Abbreviations

METHOD

Protocol and registration

Information sources and search strategy

Eligibility criteria

Study selection and data charting

Critical appraisal of individual sources of evidence

Synthesis of the results

RESULTS

Selection of sources of evidence

Characteristics of the sources of evidence

Identification of themes and subthemes

Raising a child with FASD is both challenging and rewarding

Caregivers use an adaptive parenting style to support the individual living with FASD

Caregivers often experience stigma due to FASD

Caregivers experience increased burden

Caregivers have concerns about the future

Individuals with FASD experience a range of difficulties throughout their life

Individuals living with FASD experience a range of primary effects from PAE

Difficult and traumatic childhoods

Secondary effects due to FASD

Tension between desire for independence and support

Difficulties with employment

Involvement with the justice system

Individuals with FASD are aware of their disability

Individuals living with FASD are aware of their FASD and its effects

Individuals living with FASD recognise factors that support them

Lack of knowledge and support is a major challenge

Knowledge is important for caregivers

Caregivers value and depend on informal support

Caregiver experience with formal support is negative

Caregivers are required to advocate

Professionals feel unprepared and unsupported to help individuals with FASD

Critical appraisal

DISCUSSION

Study limitations

Conclusions

ACKNOWLEDGEMENTS

CONFLICT OF INTEREST STATEMENT

Open Research

DATA AVAILABILITY STATEMENT

Supporting Information

REFERENCES

Citing Literature

References

Related

Information