ResearchPDF Available

The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder

Authors:
  • NHS Healthcare Improvement Scotland
Research

The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder

Abstract and Figures

This qualitative synthesis explores caregivers’ experiences of supporting individuals with FASD. It aims to describe the impact of the condition for caregiving from the experiences across a range of caregiver and family contexts and how this helps to inform the provision of quality of care and services. Available from: https://www.sign.ac.uk/sign-156-children-and-young-people-exposed-prenatally-to-alcohol.html
Content may be subject to copyright.
The experiences
of caregivers looking
after individuals
with Fetal Alcohol
Spectrum Disorder
A rapid synthesis of qualitative studies
January 2019
© Healthcare Improvement Scotland 2019
Published January 2019
This document is licensed under the Creative Commons Attribution Noncommercial-NoDerivatives 4.0
International Licence. This allows for the copy and redistribution of this document as long as Healthcare
Improvement Scotland is fully acknowledged and given credit. The material must not be remixed,
transformed or built upon in any way. To view a copy of this licence, visit https://creativecommons.org/
licenses/by-nc-nd/4.0/
www.healthcareimprovementscotland.org
Contents
Acknowledgements
1 Introduction 01
2 Methods 03
2.1 Literature search 03
2.2 Selection of the literature 04
2.2.1 Inclusion criteria 04
2.2.2 Exclusion criteria 04
2.3 Selection of studies 04
2.4 Quality assessment 04
2.5 Data extraction 05
2.6 Data synthesis 05
2.7 Review and consultation 06
3 Results 07
3.1 Description of studies 07
3.1.1 Design 07
3.1.2 Sample sizes 07
3.1.3 Setting and dates of studies 07
3.1.4 Participants 07
3.1.5 Study quality 07
3.2 Results of the framework synthesis 08
4 Synthesised themes 09
4.1 Living and coping with FASD 09
4.1.1 Impact on carer’s wellbeing as a result of caring for a child with FASD 09
4.1.2 Positive caregiving experiences 09
4.1.3 Concerns about and hope for the future 10
4.1.4 Parenting strategies 10
4.2 Stigma and blame 11
4.3 Diagnosis 12
4.3.1 Difficulties in obtaining a diagnosis 12
4.3.2 Formal diagnosis and awareness can help 12
4.4 Support 12
4.4.1 Lack of professionals’ knowledge and appropriate support 12
4.4.2 Formal and informal support can help 13
5 Discussion 14
6 Conclusion 16
6.1 Implications for practice 16
6.2 Implications for policy 16
6.3 Implications for further research 17
Abbreviations 18
Appendices 19
Development team 32
References 33
Acknowledgements
The development team would like to thank kinship carers from
Grandparents Parenting Again and Kinship Carers (Midlothian) for
their valuable contribution to the findings of the synthesis in sharing
their experiences of looking after individuals with FASD. In particular,
the team acknowledges the help of Mrs Anne Wilson, Development
Officer at Children’s Health Scotland, for liaison work with the kinship
carers and the co-ordination of feedback. The team is also grateful
to Mrs Karen Graham, Public Involvement Officer at Healthcare
Improvement Scotland for running focus groups to identify patient
and carer issues as part of the development of the national guidance
on identification, screening and diagnosis of children and young
people exposed prenatally to alcohol. These sessions helped to
provide a contextual foundation for this work.
1 | Introduction
01
1 Introduction
Diagnostic criteria for fetal alcohol syndrome (FAS), the most severe end of a spectrum characterised
by physical, neurodevelopmental and behavioural impairments, were first defined in 1973 to account
for biological anomalies associated with consumption of alcohol during pregnancy.1 Over time, other
definitions have been introduced to attempt to provide better descriptions of a range of outcomes
manifesting in clinically diverse presentations. Such terms have included fetal alcohol effects,2
alcohol-related biological defects, alcohol-related neurodevelopment disorder and partial fetal alcohol
syndrome.3 This wider constellation of effects, along with FAS, constitutes the continuum of structural
anomalies and neurocognitive and behavioural disabilities associated with prenatal exposure to alcohol
which is termed fetal alcohol spectrum disorder (FASD).
There are significant challenges for caregivers of individuals with FASD due to the wide range of
ways the condition can affect individuals. Research has noted that parents and carers may struggle to
cope with the significant demands of supporting children, particularly in the absence of an accurate
diagnosis to explain the child’s behaviour.4-6 The consistent requirement for supervision and support
can have a deleterious effect on carers’ well-being and family functioning.7
Studies have suggested that type and volume of stress associated with raising a child with FASD
may be different from that of other developmental disabilities.8,9 Parents of children with FASD, for
example, report more difficult behaviours and expressed more concerns regarding their children’s future
abilities to become self sufficient compared with parents of children with autism spectrum disorder.9
It has been suggested that both family and child characteristics contribute to the experience of stress
for families of children with FASD.10,11 Beyond economic resources, researchers have found that having
fewer personal resources to advocate for and support a child with FASD predicted higher stress in
mothers from low socioeconomic status families.11
Studies have suggested that a number of factors play an important part in determining the success of
raising children with FASD. These include involving the extended family, access to relief for caregivers,
collaborating with the school to establish support and consistency for their child, receiving respect
and help from professionals, being generally informed about FASD and what the condition entails,
and developing a personal network of support with other parents of children with FASD.12,13
There is uncertainty about whether the underlying cause of developmental impairments is the
prenatal alcohol exposure or the impact of a detrimental social environment. In many cases, these
increase the likelihood that such children will be identified at risk by social services and placed into
care. There is evidence that up to 75% of children in the foster care system have a family history of
mental illness or substance abuse14 with estimates of the prevalence of FASD among children in care
in the region of 78–80%.3,15,16
To mitigate such risks, the careful and comprehensive assessment and description of a child’s
impairments at time of entry into the foster system will increase the likelihood of appropriate
placements and allow foster or adoptive carers the opportunity to prepare for the specific challenges
associated with each child’s care. Within this context, the value of early assessment and diagnosis is
particularly important.17
Despite the range of challenges faced by caregivers of children with FASD, foster parents have
reported the impact of positive experiences on their motivation for maintaining and improving care,
for example observing the child overcome problems and sharing their personal achievements, and
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
02
the desire to improve the child’s life and future prospects.13,18 Other studies have emphasised the
potential fulfilment gained by carers when raising children affected by FASD and have highlighted
the role played by children in teaching parents important life lessons and enjoying the unique gifts
of each individual child.10,19
This qualitative synthesis explores caregivers’ experiences of supporting individuals with FASD. It
was conducted using a rapid review methodology in order to allow publication alongside national
guidance on screening, identification and diagnosis of children and young people exposed prenatally
to alcohol, which has also been developed by Healthcare Improvement Scotland (HIS).
The synthesis aims to describe the impact of the condition for caregiving from the experiences across
a range of caregiver and family contexts and how this helps to inform the provision of quality of
care and services.
03
2 | Methods
2 Methods
This report synthesises primary qualitative studies which have used methods including interviews,
focus groups and participatory action research (PAR), to ask carers (biological or adoptive parents,
foster parents, kinship carers or others) about their experiences of supporting an individual with FASD.
2.1 Literature search
A number of techniques can be used to identify qualitative literature, these include the use of qualitative
filters within conventional databases, ‘snowballing’ or citation chaining, browsing of core journals,
and the knowledge of colleagues or field experts as sources of information.20,21
A qualitative literature search was carried out by an information specialist in December 2017, using
a pragmatic approach to identify as much relevant English-language literature as possible within a
short time span. Three techniques were used: formal literature searching, snowballing, and experts
from the HIS guidance group were invited to provide examples of further relevant literature in early
2017. Papers identified in the experts’ list were used to generate citation chaining in Google Scholar
to augment formal database searching and also checked against the literature search results.
The search methodology combined both the use of a published qualitative search filter with general
key search terms for FASD (FASD, fetal alcohol spectrum disorder, prenatal alcohol exposure) to identify
studies relating to the impact of FASD on carers. No date limits were applied. The following Databases
from the Scottish Knowledge Network were used:
yASSIA
yCINAHL
yEmbase
yEducation Resources Information Center (ERIC)
yMEDLINE
yMidwives Information and Resource Service (MIDIRS)
yProquest public health
yProquest social abstracts
yPsycINFO
yPsychology and Behavioural Sciences
yWeb of Science
The search filter used to identify potential qualitative studies was applied to each database.22
Alongside the terms for FASD to help identify studies, the snowballing method included the following
terms: ‘parent, carer, family, adopt, foster, kinship, relation, impact, qualitative and experience’. Results
were scanned by the information specialist and all potentially-relevant items were added to Google
Scholar results.
The reference lists of included studies were not reviewed due to the rapid nature of the review.
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
04
2.2 Selection of the literature
2.2.1 Inclusion criteria
Primary studies were included in the synthesis if they:
y included carers (biological or adoptive parents, foster parents, kinship carers or others) of individuals
with FASD
yexplored the impact on carers of supporting an individual with FASD
y used a qualitative study design (for example, ethnographic research, phenomenology, studies
utilising focus group, and/or interview methods). Studies using mixed methods were only included
if the qualitative element was reported separately to the quantitative findings
ywere published in a peer-reviewed journal.
2.2.2 Exclusion criteria
Primary studies were excluded from the synthesis if they:
y used quantitative methods to gather qualitative data, for example qualitative data derived from a
largely quantitative questionnaire. Such data lack the conceptual depth to contribute to a qualitative
synthesis23
y were published in a language other than English
y referred to the design or evaluation of an intervention or a service for individuals with FASD
y were not published in a peer-reviewed journal.
2.3 Selection of studies
One author reviewed all identified records by title and abstract to evaluate their eligibility. This initial
sift was undertaken to exclude papers that were clearly unrelated to the aim of the synthesis. The full
texts of all potentially relevant papers were obtained and assessed for eligibility by one author. A full
quality appraisal of the identified papers was conducted independently by two authors. Disagreements
between authors were resolved by discussion.
See Appendix 2 for a summary of eligible studies (included and excluded).
2.4 Quality assessment
The inclusion criterion that studies need to use appropriate qualitative data collection and analysis
methods was used as an initial quality assessment attribute for the included studies. In addition,
further quality appraisal of each eligible study was carried out. The selection of methods for quality
appraisal was informed by the guidance laid out by the Cochrane Qualitative Research Methods Group.24
Appraisal was performed using the Critical Appraisal Skills Programme (CASP)25 quality assessment
tool for qualitative studies. Two authors independently applied the CASP tool to each eligible study.
A quality level of low, moderate or high (based on the Swedish Council on Health Technology
Assessment’s handbook for qualitative evaluation and synthesis)26 was jointly applied to each
study guided by the independent CASP ratings. Disagreements were resolved by discussion.
See Appendix 3 for details on the quality assessment of the studies.
05
There is currently no consensus among qualitative authors on how quality criteria should be applied
and how they should be used in qualitative syntheses.27 It has been noted that studies considered
‘low quality’ due to methodological flaws may still provide valuable insights arising from the data
and, conversely, methodologically strong studies may suffer from poor or limited interpretation of
their data, leading to limited insights into the phenomena under investigation.24,28 It has further been
acknowledged that poor reporting of methods does not equate with poorly-conducted research.24,29
Despite lacking clear description of the analysis, three studies30-32 were included because they still
made a valuable contribution to the synthesis (see Appendix 3 for further details). Two low-quality
studies33,34 were excluded from this synthesis because they did not have a clear statement of findings
or were not adding any substantial findings beyond the research findings of the other included studies
(see Appendix 3).
2.5 Data extraction
A data extraction table was populated. Information extracted included author and year of publication;
study sample characteristics; number and age of participants; method of data collection; analysis
approach and study setting (see Appendix 2).
The authors’ interpretations of the included studies represent the data analysed in this synthesis.27
All text labelled as discussion or results was thematically analysed and synthesised in themes (see
section 4). The approach of analysing both the discussion and result section of the studies is thought
to increase the transparency and ‘auditability’ of the synthesis.35
QSR NVivo® 10 software for qualitative analysis was used to manage and analyse the data thematically.
The results of all included studies were entered verbatim into NVivo® for analysis.
2.6 Data synthesis
Framework analysis36 was used to allow the identification of common and variable patterns of themes
within and across different studies. Framework analysis is a matrix-based method which involves the
construction of thematic categories into which data from primary qualitative studies can be coded.36
Framework synthesis has been widely utilised in the synthesis of qualitative health data and is one of
the approaches to qualitative synthesis recommended by the Cochrane Qualitative Review Methods
Group.24 This approach involves several overlapping stages: familiarisation, identifying a thematic
framework, indexing, charting and interpretation. However, not all stages were carried out in the
current synthesis due to a limited timescale for conducting the synthesis and reduced research capacity.
y Familiarisation – Two authors read all of the included studies, with reference to the review questions
y Identifying a thematic framework – In this stage, one author examined all the findings from these
papers to identify the initial themes for the thematic framework. The four overarching themes
which were identified arose consistently across the findings of multiple studies or were identified
by one or more study as a key theme (see section 3.2)
y Indexing – One author systematically applied the thematic framework to the results and discussion
section of all studies. The included papers were imported into Nvivo® and the results and discussion
sections were coded. The stages were iterative and additional themes which emerged at this stage
further informed the thematic framework
y Charting – This stages includes the creation of a matrix of findings summaries for each theme by
study. Given the rapid approach of the synthesis this stage was not conducted.
y Interpretation – Refinement of the subthemes and search for patterns and connection between
themes was performed by two authors.
2 | Methods
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
06
2.7 Review and consultation
The discussion section of the synthesis report was presented at a Kinship Carers group meeting in
Midlothian, Scotland to gather carers’ feedback based on their own experiences of looking after an
individual with FASD in the Scottish context. The carers’ responses are summarised in Appendix 4. The
final synthesis report was reviewed by the Lead Health Services Researcher and underwent editorial
review by senior management from Healthcare Improvement Scotland.
07
3 | Results
3 Results
6,145 studies were identified through electronic database searching. Of these studies, 30 were
selected for full text screening and 12 were subsequently included in the synthesis. See Appendix 1
for a figure illustrating the screening process.
3.1 Description of studies
3.1.1 Design
All studies used qualitative methods of data collection and analysis. Four of the included studies
conducted semi-structured interviews as a method of data collection,37-40 six conducted unstructured
interviews,19,30,32,41-43 one used participatory action research31 and one conducted focus groups.44 The
methods of data analysis used by the studies were: interpretative phenomenological analysis,32,37-41
phenomenological-hermeneutic approach,19 thematic analysis,19,44 narrative analysis,43 and constant
comparison.42 Two studies did not provide information about their approach to data analysis.30,31
3.1.2 Sample sizes
Study sample sizes ranged from 432 to 84 participants.37
3.1.3 Setting and dates of studies
The studies were published between 2001 and 2016 and were conducted in four countries:
Canada,19,30,37,39,40 USA,41,43 UK32,44 and New Zealand.42
3.1.4 Participants
Four studies19,30,32,44 had a mixed sample of biological, adoptive and/or foster parents. In three
studies37,39,40 participants included biological parents, foster parents, adoptive parents, step-parents,
and custodial grandparents. Three studies38,41,43 included only foster and/or adoptive parents and two
studies31,42 included only biological parents.
The age range of participants and their children varied across studies.
3.1.5 Study quality
Five of the included studies were rated as high quality,37,38,40,41,43 four as moderate quality,19,39,42,44 and
three studies were difficult to appraise due to reporting issues.30-32 See Appendix 3 for more details
about the quality assessment of the included studies.
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
08
3.2 Results of the framework synthesis
Analysis of the twelve included studies identified four themes, three of which included subthemes
(Table 1).
Table 1. Themes and subthemes
Theme Subtheme
Theme 1: Living and coping with FASD 1.1 Impact on carers’ well-being as a result of caring for
a child with FASD
1.2 Positive caregiving experiences
1.3 Concerns about and hope for the future
1.4 Parenting strategies
Theme 2: Stigma and blame
Theme 3: Diagnosis 3.1 Difficulties in obtaining diagnosis
3.2 Formal diagnosis and awareness can help
Theme 4: Support 4.1 Lack of professionals’ knowledge and appropriate support
4.2 Formal and informal support can help
09
4 | Synthesised themes
4 Synthesised themes
Studies identified various different caregiving experiences associated with looking after an individual
with FASD. Although individual studies reported that parenting a child with FASD is a unique experience
due to the various developmental difficulties that are manifested in the child’s life,19,40,44 caregivers
shared similar experiences across studies which were synthesised in the four themes described in
sections 4.1 to 4.4 (see also Table 1).
4.1 Living and coping with FASD
4.1.1 Impact on carers’ well-being as a result of caring for a child with FASD
It’s probably like Post Traumatic Stress Syndrome, to be honest because you dread the police coming
to the door, you dread the phone ringing, you absolutely dread it, well, nobody rings us up anymore
because they know that we didn’t used to answer the phone and it’s like it’s left us with that and I
remember the police coming to my door one day and I’m thinking, and I looked at them and I said is he
still alive? What’s happened?… but it was like that all the time, there was always this dread and fear
inside of what is going to happen next (biological mother).” [Whitehurst 2011, p.191]
Studies revealed that caring for an individual with FASD can have an impact on carers’ well-being.
Stress was identified as a common experience of caregivers19,30-32,39,42,44 particularly when parents
had to cope with children’s externalising behaviour problems, such as hyperactivity, aggression or
violence.19,42,43 Studies highlighted that caregivers’ stress could be caused by constantly fighting with
the child about accepting support because of the child’s inability to accept their own limitations.31,32
The need for constant supervision for their children was seen by some adoptive and foster parents
as emotionally demanding.43 Several parents explained that a further cause for concern for them is
having to cope with multiple disabilities in their children in addition to FASD.19,41 Factors explored under
other themes, such as poor or incorrect support from school and health professionals,19,42,44 or missing
knowledge and lack of training about FASD diagnosis,31 can further contribute to caregivers’ stress. In
one study, biological, adoptive and foster parents referred to their experience as a ‘continual battle’.32
Several studies identified that caring for an individual with FASD can have a negative effect on the
overall family functioning.19,37,44 In one study biological families indicated that they were living in
constant crisis, struggling to move from adjustment to adaptation.42 For some families, having a child
with FASD can have a significant impact on family members’ mental health and relationships.19,37,44
The challenging behaviour that children with FASD demonstrate can also create a chaotic home
environment where “it’s very hard to make a family seem like a family” [Sanders 2010, p. 314]. Some
parents spoke about their difficulties balancing their parenting responsibilities with other family
demands, such as carrying for an ageing family members or a sick spouse.37 The challenges in looking
after an individual with FASD, which can be increased by the lack of support, in some cases led to
marital breakdown.43,44
4.1.2 Positive caregiving experiences
I think I’ve been blessed that I have these two girls in my life; they are very special in their own way.
I don’t know what I would do without them if they weren’t in my life. It was a long journey! But it
was worth it… I could be helping them the rest of their lives… They probably remember all the things
I helped them on and what I did for them. I think they will appreciate all things I did. I give myself a
pat on the back!” [Sanders & Buck, 2010, p. 321]
10
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Multiple studies described that caregivers’ experiences can change over time when they start
appreciating their children’s differences and uniqueness.19,31,37,40,41,44 Three studies mentioned biological
parents’ positive outlook of raising a child with FASD,31,37,44 with one mother sharing the experience of
staying sober in order to be able to look after her child.37 Two studies indicated that adoptive parents
increasingly appreciate their children’s accomplishments, gifts and skills over time.19,41 Another two
studies highlighted biological and non-biological caregivers’ experiences of personal growth and a
‘changed worldview’ as a result of looking after a child with FASD.19,37 One study highlighted that both
biological and non-biological parents try to focus on the positive contributions that their children
bring in their lives and often described their lives as an ‘adventure’.42
4.1.3 Concerns about and hope for the future
“I’m just kind of afraid of the future… I try not to think about the future.” [Watson 2013, p.81]
A number of studies described caregivers’ concerns about their children’s future.19,31,39,41 One study
identified that for some carers letting go of their hopes for the child’s future was described as an
experience of grief.39 Several studies discussed caregivers’ concerns in relation to their children’s
ability to live independently as an adult.12,20,22 The risk of teenage pregnancy,39 as well as the lack
of support services when children with FASD transition into adulthood12,31,39,41 were identified by
both biological and non-biological caregivers as possible barriers for independent living. Anticipated
difficulties varied depending on whether the child is transitioning into primary or secondary school,
or into adulthood.44 One study showed that concerns for the child’s future were also influenced by
the lack of information and diagnosis that caregivers receive from professionals.41
Caregivers were also able to describe hope for the future of their children.19,31,39 One study described
the hope of an adoptive mother who stated: “I think she’ll be pretty independent. I think she’ll be… I
think, like I said, once she’s got it she’s got it. And I think she’ll, she’s going to be okay. I think she’ll
be okay” [Watson 2013, p. 85]. Several studies identified that parents had hopes for their children
achieving independence.19,31,39 The experience of being hopeful for the future for some caregivers was
related to external factors such as the child finding the right partner or receiving adequate support.31,39
4.1.4 Parenting strategies
It’s taken us a while to adapt to having these kids…so now that we’re on a routine and understand
that kids need that routine, it’s easier for us now as well… If you have everything as a routine, it’s very
simple.” [Coons et al. 2016, p. 156]
Studies described caregivers using a range of parenting strategies for their children with FASD. Both
biological and adoptive carers found parenting a child with FASD to be a very different experience
from parenting other children.31,43 Two studies suggested that for some biological and non-biological
carers it is important to set realistic expectations for their children in light of their deficits.19,39 One
study found that many carers are adapting to specific challenges on a day-to-day basis and that this
adaptation is influenced by various different factors (for example, age of the child, family type, etc).37
The study also indicated that both biological and non-biological carers preferred to ‘pick their battles’
with their children which, in some cases, led to improved relationships. For example, arguing about
daily issues was not considered as important as dealing with serious behaviour problems, such as
aggression or depression.37,38 Parents, furthermore, considered the importance of maintaining a routine
and consistency to aid the challenges of everyday life.37 Maintaining a routine in the home, as well
as using visual prompts and signs was seen by parents as a way to co-ordinate their children’s day
and to prevent children’s challenging behaviour. Two studies indicated that some parents find altering
the home environment to be a more effective strategy in preventing feelings of overstimulation
than prescribing consequences for behaviour or trying to change behaviour.31,37 Foster and adoptive
11
parents, as well as one biological mother, elaborated on their need to protect and even ‘control’ their
children’s environments in order to keep them away from harm.19,43 Biological mothers in another study,
however, identified that providing supervision which is balanced with an unconditional acceptance
is a necessary condition for children to stay home.31
Biological and non-biological caregivers highlighted the importance of obtaining information and
knowledge about FASD in order to better understand their child’s behaviour and to adopt appropriate
parenting strategies.19,37,38,43 Behavioural strategies such as following up bad behaviour with
consequences were seen as ineffective because of challenges in learning and memory associated with
FASD.19,32 Two studies identified that parenting strategies and coping improved as parents learned
about the impact of FASD on the behaviour and development of children.19,38 Acquiring new skills or
support was recognised by parents as important in coping and adapting to their children’s behaviour.37
The ability to effectively look after children with FASD was considered by adoptive and foster parents
as something that develops over time and with experience.43
In addition to having to educate themselves about FASD, parents also identified having to take on a
role of advocate for their children.19,37,40 Parents described this advocating role as a ‘fight’, especially
when having to argue in favour of support services or adequate support at school.19,37
4.2 Stigma and blame
It’s very hard to forgive yourself when you’ve actually damaged someone’s life, irrevocably, you know,
you can’t turn the clock back… I’ve said sorry to him so many times and said I really, really wouldn’t
have wanted this life for you and I really am so sorry… but all I can do is be there for you to help you
get through it, you know.” [Whitehurst 2011, p.191]
Guilt for drinking during pregnancy was identified as common experience for biological mothers.19,31,32,40,42
Mothers also felt guilty for assuming that their children were purposefully misbehaving and felt that
they were ‘mishandling’ situations by not using appropriate parenting strategies.19 In one study the
experience of guilt was related to the inability to meet the complex needs of children with FASD.31
Some biological mothers also shared the experience of having to ask their children to leave home
when their behaviour became unmanageable.31 The mothers expressed a sense of failure for not being
able to continue caring for their children.31 One study reported a feeling of guilt from an adoptive
mother who shared: “So when they say that biological moms who are raising these kids feel guilty, the
adopted moms feel just as guilty because you want to do so much [Morrissette 2001, p.11].
Several studies indicated that carers of individuals with FASD feel isolated and judged by the public
and professionals.32,37,42,44 Biological and non-biological carers often felt criticised by community
members and professionals for their ‘poor parenting skills’ because of the assumption that child’s
behaviour is caused by poor parenting.37,44 Some non-biological carers expressed feelings of anger
or judgement toward biological parents for their drinking during pregnancy.19,38
In one study adoptive parents reported feelings of uneasiness and embarrassment to have to explain
themselves when people make assumptions and judge them for drinking during pregnancy or when
taking their child with FASD to public places.32 Some families expressed a concern that the difficulties
which are experienced with their children are belittled or distorted.44 In one study parents shared that
community members and professionals perceived the child to be deliberately acting poorly37 although
noted that these judgements are a result of that the lack of knowledge about FASD, as well as the
fact that FASD is not always physically recognisable.37 Parents expressed the view that educating the
public and raising awareness about FASD and its accompanying behaviours is needed for parents to
feel a greater amount of community support.37
4 | Synthesised themes
12
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
4.3 Diagnosis
4.3.1 Difficulties in obtaining a diagnosis
“It’s [the diagnostic process] like hitting a brick wall.” [Watson et al. 2013, p.108]
More than half of the studies highlighted that families experienced challenges in regards to obtaining
a diagnosis of FASD for their children.19,30,32,37,38,40-42,44 Carers expressed that healthcare professionals
(HCPs) may fail to make a timely diagnosis because of lack of awareness, understanding or training
about functional diagnosis of FASD.19,30,32,38,40,42 One study indicated that sleeping problems in people
with FASD are also underdiagnosed as a result of ‘inadequate explanatory models’.30 Studies also
mentioned that children with FASD could receive an incorrect diagnosis, such as attention deficit
hyperactivity disorder (ADHD) or conduct disorder.30,37,42 Furthermore, both biological and non-
biological carers felt that HCPs do not provide consistent messages regarding alcohol consumption
during pregnancy which can further impact the diagnosis of FASD.38,40 One study, for example, reported
several mothers who were encouraged by HCPs to consume alcohol during pregnancy.37
Alongside professionals’ lack of knowledge and awareness (see section 4.4.1), studies identified further
factors such as the lack of confirmed maternal drinking and the variability in children’s characteristics
which can make the diagnosis of FASD more challenging.19,40 Because FASD is a spectrum disorder, the
lack of physical impairments can sometimes lead to unrealistic assumptions and expectations about
children’s development.19 Studies highlighted that the lack of, or incorrect, diagnosis led to caregivers
being unaware of the impact of alcohol consumption during pregnancy on the child’s development.19,37
This can have further implications on family adaptation. Several studies, for example, identified that
adoptive parents were not given a formal FASD diagnosis or enough information about the condition
prior to adoption, which left them feeling that they were not able to make an informed decision or that
they had a false sense that the child will develop normally.32,38,41,44 In two studies, however, parents
started investigating the possibility of child’s prenatal alcohol exposure and initiated the process of
diagnosis as they became aware that ‘something was not right’.19,32
4.3.2 Formal diagnosis and awareness can help
Without the name there’s no place, there’s no place to begin and there’s no way to try and get other
people to understand.” [Watson et al. 2013, p.109]
Several studies highlighted that formal diagnosis of FASD can help caregivers to understand children’s
unique needs and challenges, and be able to access appropriate support services for their children.19,37,40
Two studies identified that a formal diagnosis can provide relief for both biological and non-biological
caregivers and alleviate family tension as they gain more understanding about the cause of their
children’s behaviour and adapt their parenting strategies.19,42 One study, however, highlighted that
parents were struggling to obtain support services for their children even after they were diagnosed
with fetal alcohol effects45 which is considered as a less severe diagnosis than fetal alcohol syndrome
within the FASD spectrum.40
4.4 Support
4.4.1 Lack of professionals’ knowledge and appropriate support
I would want someone to say you’re not going to have to fight social services, you’re not going
to have to fight education to get a placement, you’re not going to have to argue with health over
resources or whatever, and then I would have felt more confident about carrying on.” [Mukherjee
& Wray 2013, p.50]
Lack of knowledge and awareness of FASD among HCPs was one of the most common themes in the
13
studies.19,30,32,37-40,43,44 Some caregivers talked about lack of trust in professionals because of their lack
of understanding of the behavioural challenges of children with FASD.19,32,37 Caregivers often referred
to their experience of obtaining HCP support as a ‘struggle’ and a ‘fight’.19,32,44 Professionals’ lack of
knowledge and understanding could lead to lack of support to meet the needs of caregivers.32,38,44 In
one study both biological and non-biological carers expressed their concerns that the lack of support
can further lead to their children experiencing difficulties in establishing independence.39
Carers sometimes felt that they were ‘not listened to’ and that children’s needs were neglected or
forgotten by professionals.39,42 One study pointed out the lack of recognition of the challenge of sleep
problems of children with FASD and the failure to integrate information about these problems into
the clinical assessment of FASD which can have a negative effect on the whole family.30
Caregivers also spoke about school professionals’ lack of awareness and knowledge about FASD
and the associated child’s behaviour19,32,37,38,40,42,43 which made some parents feel that they need
to educate professionals or advocate for their children’s schooling.37,38 Parents also stated that
the lack of knowledge can lead to inappropriate schooling support and failure to meet children’s
needs.32,38,40,42 Parents described experiences of frustration, resentment or lack of trust towards school
professionals.19,38,42,43
4.4.2 Formal and informal support can help
“It’s nice to know you are not alone.” [Coons et al. 2016, p.158]
In more than half of the studies caregivers shared experiences of the benefits of receiving support from
professionals in managing their child’s well-being.19,31,32,37,38,40,42,43 Professional support was perceived
by caregivers as necessary in promoting successful family adaptation and functioning.19,38 Professional
support, both for children and their caregivers, was seen as crucial particularly in transition periods,
such as from childhood into teenage years, or from teenage into adulthood.43 Studies acknowledged
that by providing support to the children and understanding not only their challenges but strengths,
professionals were able to provide better assistance to parents.19,37 Three studies highlighted that
caregivers value the support that is provided by paediatricians and other clinicians as a point of
reference in accessing services or for prescribing medications.32,37,38 In one study biological and
adoptive parents reported that the support received from counsellors helped them to alleviate stress.37
Furthermore, three studies identified formal support groups as valuable in providing caregivers with
an opportunity to build relationships, support each other and exchange information about parenting
strategies.31,37,42 One study, however, highlighted that support can be beneficial only if it is relevant
and meaningful to carers’ needs, with one mother reporting that she had to stop the support she was
receiving in the mornings as this created ‘more of a hassle’.40
Studies also identified the importance of informal support in the upbringing of individuals with FASD.
For example, caregivers placed importance on the mutual support between spouses in the upbringing
of their children,43 as well as the support they receive from extended family or friends.37 A biological
great-grandmother shared: “It takes a community to raise a child [with FASD]. And I believe it, I really
believe it now. There’s nobody. I don’t care where you come from or where they go, there’s nobody that
can do it on their own” [Coons et al. 2016, p.157].
4 | Synthesised themes
14
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
5 Discussion
This report synthesised qualitative studies looking at the experiences of caregivers of individuals with
FASD. The aim of the synthesis was to integrate the findings from twelve primary qualitative studies
by staying close to the descriptive study data. The synthesis identified four main themes: living and
coping with FASD, stigma and blame, diagnosis and support.
The most prevalent theme in the synthesis related to caregivers’ experience of obtaining a diagnosis
of FASD. The theme of diagnosis identified two subthemes: ‘difficulties in obtaining a diagnosis’ and
‘formal diagnosis and awareness can help’. The findings from the synthesis indicated that obtaining
a formal diagnosis of FASD can be beneficial for both the individuals with the condition and their
caregivers in bringing understanding into individuals’ unique needs and challenges, helping caregivers
adopt appropriate parenting strategies and access support services. The potential advantages of having
a diagnosis in providing access to different resources and appropriate support services was also
identified by a review of studies investigating the ethical aspects and consequences of diagnostics,
interventions, and family support in relation to FASD.46 The synthesis, however, identified caregivers’
experiences of healthcare professionals who undermined or avoided the diagnosis of FASD because it
was perceived by them as a ‘label’. Some disadvantages related to FASD diagnosis were also identified
by the review which found that diagnosis of FASD can have negative consequences for families
related to the experience of stigma and blame, as well as feelings of guilt.46 In contrast, the synthesis
reported that some caregivers considered the experience of judgement by community members and
professionals to be a result of the lack of knowledge and awareness about FASD.
The second most common theme in the synthesis captured the attitudes of parents towards receiving
appropriate support to assist them in caring for an individual with FASD. Two subthemes were identified
under this theme: ‘lack of professionals’ knowledge and appropriate support’, and ‘formal and informal
support can help’. The synthesis found that appropriate support promotes family adaptation and
functioning, helps children transition into adulthood and maintain their independence. This reflects
the findings from a recent systematic review of FASD intervention studies which found that involving
caregivers as ‘change agents in outcome of children with FASD’, by supporting them in improving
their parenting skills, understanding their child’s behaviour and connecting them with appropriate
resources, can impact the developmental and behavioural outcome of their children.47 Furthermore,
studies in the systematic review showed that increasing parents’ knowledge about the condition and
the associated children’s behaviour alters caregivers’ attitudes and behaviours towards the child.
The synthesis identified that caring for an individual with FASD can have a deleterious impact on
caregivers’ individual well-being, as well as on the overall family functioning. This finding is consistent
with previous research which showed that children’s behaviour problems are associated with parental
wellbeing.48 Studies in the synthesis showed that contributing factors to caregivers’ experiences of
stress could be caused by children’s externalising behaviour, fighting with the child about accepting
support, having to cope with multiple disabilities in addition to FASD or poor or incorrect support
from school and health professionals or missing knowledge and lack of training about FASD diagnosis.
The synthesis identified a range of strategies that caregivers used to look after their with children
FASD, such as setting realistic expectations for their children, maintaining a routine and
consistency in the home, picking up their battles, adapting on a day-to-day basis. The synthesis
suggested that caregivers can experience positive change and improve their parenting strategies
when they receive more information about the condition and the associated child’s behaviour
and start appreciating their children’s differences and uniqueness.
15
Limitations
More than half of the studies included in the synthesis were conducted in Canada. Findings
from qualitative research are often dependent on the context in which they were produced49
and, therefore, transferability to other sociocultural environments needs to be considered
carefully. The nature of the spectrum disorder and the complexity of the behaviours and symptoms
associated with it adds a further challenge for findings to be representative of all potential caregivers’
experiences of looking after individuals with FASD.
5 | Discussion
16
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
6 Conclusion
The findings from this synthesis suggest that there are numerous impacts being experienced by
caregivers and their families as they navigate the difficulties in recognising and understanding
the symptoms of FASD and obtaining a diagnosis; and the consequences and management of FASD
throughout the life course. A lack of knowledge and understanding of FASD among healthcare
professionals is experienced as a key barrier to formal diagnosis and receiving meaningful support
which suggests the need for professional interventions such as improved training. While there can
also be negative consequences of FASD diagnosis including stigma, the synthesis suggests that an
accurate diagnosis is important for receiving timely support that can help caregivers make positive
changes towards managing the symptoms of FASD and improving the outcomes for their families.
Furthermore, support that is meaningful and that meets the need of individuals with FASD and their
caregivers also depends on the availability of appropriate support interventions.
6.1 Implications for practice
Caregivers believed that healthcare professionals often had poor knowledge and awareness of FASD
which may limit opportunities for diagnosis, prevention and early intervention.
y It is therefore important that efforts are made to provide training and guidance, and increase
awareness of prenatal alcohol exposure and FASD among healthcare professionals, as well as other
relevant professional stakeholders such as social work, criminal justice and education.
y Training on the prevention, diagnosis and management of FASD should be integrated into
undergraduate and postgraduate curricula and continued professional development.
Caregivers reported that families experienced challenges in obtaining a diagnosis of FASD for their
children, which presented a barrier to fully understanding the children’s unique needs and challenges,
and the ability to access appropriate support services. The British Medical Association Board of Science
has published a report50 calling for the development of national guidance on diagnosis and referral
of FASD, based on the Canadian diagnostic guidelines as a model of best-practice.
y Guidance on the diagnosis of the full range of fetal alcohol spectrum disorders should be developed
and made available to all healthcare professionals. Guidance on diagnosis should emphasise the
need for testing to exclude other disorders.
6.2 Implications for policy
There are no specific services funded to oversee the diagnosis or management of individuals exposed
prenatally to alcohol in the UK. The National Clinic for Fetal Alcohol Spectrum Disorders (the only
specialist FASD behavioural clinic of its type in the UK) relies on individual funding requests, whereby
clinicians in England have to apply to their local clinical commissioning group for funding in order
to refer individual patients. The lack of proper commissioning acts as a barrier to the development
of services required to adequately address the needs of individuals affected by prenatal alcohol
exposure. Scottish Government has funded a pilot to set up a novel multidisciplinary clinic in NHS
Ayrshire and Arran for the assessment and support of children with suspected FASD. The aim of the
team is to work with multidisciplinary teams in Health Boards across NHS Scotland to improve access
to diagnostic services and improve clinician confidence.
17
y The Fetal Alcohol Advisory and Support Team in NHS Ayrshire and Arran should publish the service
evaluation of the pilot project, showing how the experience gained can be used to support clinicians
to develop appropriate services for families and individuals affected by FASD.
6.3 Implications for further research
No qualitative studies were identified describing the experiences of individuals in Scotland affected by
prenatal exposure to alcohol and only a single study was identified in the UK setting. Further research
involving Scottish caregivers is required to inform the context of care provision within Scotland.
6 | Conclusion
18
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Abbreviations
ADHD attention deficit hyperactivity disorder
CASP Critical Appraisal Skills Programme
FAE fetal alcohol effects
FAS fetal alcohol syndrome
FASD fetal alcohol spectrum disorder
HCP healthcare professional
HIS Healthcare Improvement Scotland
PAR participatory action research
SIGN Scottish Intercollegiate Guidelines Network
UK United Kingdom
USA United States of America
19
Appendices
Appendix 1: Prisma flow chart of articles
Appendices
Articles identified
through
database searching
(n=6,145)
Articles retained
after keyword sifting
for relevance
(n=188)
Full text articles
assessed for eligibility
(n=30)
Included in
qualitative synthesis
(n=12)
Articles excluded by
title and abstracts
(n=158)
Full text articles
excluded for:
• eligibility (n=16)
• quality (n=2)
20
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Appendix 2: Summary of eligible studies (included and excluded)
Foster parents (n=4),
adoptive parents (n=1),
legal guardians (n=5)
Married (n=5), single
(n=2), widowed (n=1),
divorced (n=1), domestic
partnership (n=1)
n/aChamberlain et al.
(2017)33
Adoptive parents (n=5) n/a n/aGranitsas (2004)41
Adoptive, foster or
biological mothers (n=5)
n/a n/aMichaud & Temple (2013)31
Foster and adoptive
parents (n=6); 1 male/5
female
Mean number of years of
experience as a foster/
adoptive parent: 10
n/aMorrissette (2001)43
Biological parents (n=1),
adoptive parents (n=2);
foster parents (n=3)
n/a n/aIpsiroglu et al. (2013)30
Foster mothers (n=8) Age range: 30 – 60 Age range – 2 – 16
(n=17)
Gardner (2000)34
Adoptive mothers (n=37),
adoptive fathers (n=14)
Mothers: average age –
51.73; married – 86.1%
Fathers: average age –
55.45%; married 100%
Average age: 15.85 (n=53)
Gender (% female): 58.49
Coons et al. (2016)38
Mothers (n=58): biological
(n=12), step-parent (n=2),
adoptive (n=39),
foster (n=5).
Fathers (n=26): biological
(n=4), step-parent (n=3),
adoptive (n=16),
foster (n=3)
Mothers’ average age –
50.74, married – 67.2%
Father’s average age –
54.35, married – 80.8%
Age range: 1 – 36Coons et al. (2016)37
n/a – not available
Study sampleAuthor and year Participant characteristics Children: number and age
21
Appendices
Semi-structured
interviews conducted
at two months post
diagnosis
Excluded. Lacking
depth in analysis
and findings.
Thematic analysis Specialist diagnostic
service, Australia
Low to moderate
Unstructured
interviews
IncludedInterpretative
phenomenological
analysis
Developmental
clinic,
Commonwealth of
Massachusetts, USA
High
Participatory Action
Research
Included because
of valuable insights
provided by PAR
methodology
No detail given Sioux Lookout,
Ontario, Canada
Difficult to appraise
due to reporting
Unstructured
interviews
Included Narrative analysis Missoula, Montana,
USA
High
Unstructured
interviews
Included because of
its specific focus on
sleep
No detail given Pediatric Sleep
Assessment Clinic
of British Columbia
Children’s Hospital,
Vancouver, Canada
Difficult to appraise
due to reporting
Unstructured
interviews
Excluded. Doesn’t
add any substantial
findings beyond
body of existing
literature
Content analysis n/a Low
Semi-structured
interviews
IncludedInterpretative
phenomenological
analysis
Northern Ontario,
Canada
High
Semi-structured
interviews
IncludedInterpretative
phenomenological
analysis
Ontario, Canada High
Appendix 2 continues on page 22
Method of data
collection
Included/excludedMethod of data
analysis
Recruitment/ Setting Quality rating
22
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Appendix 2: Summary of eligible studies (included and excluded)
continued
Biological mothers (n=8) n/a Age range: 8.5 – 30 (n=9).
Sample included two
males and two females
diagnosed with FAS, three
males and one female
diagnosed with fetal
alcohol effects (FAE)45,
and one female who had
partial FAE and ADHD.
Salmon (2008)42
Biological (n=3), adoptive
(n=7), and foster (n=1)
parents. 2 male/9 female
Married (n=6), separated
(n=1), single (n=1),
divorced (n=3). Age range:
32 - 59
Mean age: 13.5 (n=14)Sanders (2010)19
Biological, foster, adoptive,
step-, and custodial
parents (n=31)
n/a Age range: 1 – 3, mean
age: 16.51
Watson et al. (2013)40
Adoptive mothers (n=3),
biological mother (n=1)
n/a Mean age: 10.28 (n=7)
Gender: 4 female/3 male
Whitehurst (2011)32
Mothers (n=42): biological
(n=5), step-mother (n=2),
adoptive (n=32),
foster (n=3)
Fathers (n=15): biological
(n=2), adoptive (n=11),
foster (n=2)
Mothers: married – 80.95%
Fathers: married – 93.33%
Mean age: 15.25Watson et al. (2013)39
Foster and adoptive
parents (n=28), biological
mothers (n=2)
Age range: 30 – 62 n/aMukherjee et al. (2013)44
n/a – not available
Study sampleAuthor and year Participant characteristics Children: number and age
23
Unstructured
interviews
Included Constant comparison Hamilton,
New Zealand
Moderate
Unstructured
interviews
Included Phenomenological-
hermeneutic
approach and
thematic analysis
Alberta, Canada Moderate
Semi-structured
interviews
Included Interpretative
phenomenological
analysis
Disability support
organisations,
Northern Ontario,
Canada
High
Unstructured
interviews
Included. Valuable
study given its UK
setting
Interpretative
phenomenological
analysis
UK Difficult to appraise
due to reporting
Semi-structured
interviews
Included Interpretative
phenomenological
analysis (not
reported in methods
section, only in
results)
Disability support
organisations,
Northern Ontario,
Canada
Moderate
Appendices
Focus groups Included Thematic analysis England, London and
Oxfordshire, UK
Moderate
Method of data
collection
Included/excludedMethod of data
analysis
Recruitment/ Setting Quality rating
24
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Appendix 3: Quality assessment of included and excluded studies
Author and year Clear
statement
of aims
Qualitative
methodology
appropriate
Research
design
appropriate
Recruitment
strategy
appropriate
Data
collected
appropriately
Relationship
between
author and
participant
considered
Yes Yes Yes Yes Yes YesChamberlain et al.
(2017)33
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes Yes Yes Yes Yes Yes
Granitsas (2004)41
Yes
Yes
Yes
Yes
Yes
Yes
Canʼt tell
Canʼt tell
Yes
Yes
No
YesMichaud & Temple
(2013)31
Ipsiroglu et al.
(2013)30
Yes Yes Yes Yes Yes NoGardner (2000)34
Coons et al. (2016)38
Coons et al. (2016)37
25
Appendices
Summary ratingEthical issues
taken into
consideration
Data
analysis
sufficiently
rigorous
Statement
of findings
clear
ExclusionValue of research
Low to moderate quality.
Lacking depth in analysis
and findings.
Yes No No ExcludedThe focus on diagnosis
could have been more
valuable if a greater
depth of findings had
been achieved.
High quality
High quality
Yes
Yes
Yes
Yes
Yes
Yes
Included
Included
Detailed description
of analysis given.
Conclusions well
supported by quotes
and triangulated by
other research where
identified.
Good quotes and
stayed close to lived
experience. Some
themes emphasised
are different to other
included studies.
High qualityYes Yes Yes IncludedFocus on adaptation and
well conducted.
Reporting makes difficult
to appraise probably
because a part of mixed
method study but a
valuable study for this
analysis.
Reporting makes difficult
to appraise due to PAR
methodology but a
valuable study for this
analysis.
Canʼt tell
Canʼt tell
Canʼt tell
Canʼt tell
Yes
Yes
Included
Included
Valuable given specific
focus on sleep, which
is not covered in detail
in any other studies
included.
Findings have a different
emphasis due to use of
PAR methodology. Voice
close to participants.
May have limited
transferability due to
small, rural, Canadian
sample but does cover
common ground with
other studies.
Low qualityCanʼt tell Canʼt tell No ExcludedDoesn’t add any
substantial findings
beyond body of
existing literature.
Appendix 3 continues on page 26
26
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Mukherjee et al.
(2013)44
Appendix 3: Quality assessment of included and excluded studies
continued
Author and year Clear
statement
of aims
Qualitative
methodology
appropriate
Research
design
appropriate
Recruitment
strategy
appropriate
Data
collected
appropriately
Relationship
between
author and
participant
considered
Morrissette (2001)43 Yes Yes Ye s Yes Ye s Yes
Yes Yes Yes Canʼt tell No No
Yes Yes Yes Yes Yes NoSalmon (2008)42
Yes Yes Yes Canʼt tell Yes NoSanders (2010)19
Yes Yes Yes Yes Yes YesWatson et al.
(2013)40
27
Summary ratingEthical issues
taken into
consideration
Data
analysis
sufficiently
rigorous
Statement
of findings
clear
ExclusionValue of research
High quality with a
valuable focus on
how best to support
families. Sample may
be unrepresentative of
broader group of families
supporting children with
FASD.
Can't tell Yes Yes IncludedHas some unique
contributions, focus
on detail of how might
support families better
and discussion of
family dynamics such
as 'manipulation' and
splitting of parents by
children with FASD.
Moderate quality.
Valuable study in UK
setting.
Canʼt tell Canʼt tell Yes IncludedYes does contextualise
in evidence base and is
a UK setting so valuable
for this analysis.
Moderate quality,
with possible problem
concerning bias of solo
author.
Yes Yes Yes IncludedValuable exploration
of biological mothers
experiences (under-
represented in most other
studies). Contextualised
with extensive literature
review.
Moderate qualityCanʼt tell No Yes IncludedStudy does make an
effort to separate
biological and non-
biological parents
experience and describes
these experiences in
detail, which is useful
given lack of sampling
of biological mothers
in most other included
studies.
High qualityYes Yes Yes IncludedYes. Good comparison of
ASD/FASD.
Appendix 3 continues on page 28
Appendices
28
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Appendix 3: Quality assessment of included and excluded studies
continued
Author and year Clear
statement
of aims
Qualitative
methodology
appropriate
Research
design
appropriate
Recruitment
strategy
appropriate
Data
collected
appropriately
Relationship
between
author and
participant
considered
Yes Yes Yes Yes Yes Canʼt tellWatson et al.
(2013)39
Whitehurst (2011)32 Yes Yes Ye s Canʼt tell Canʼt tell Canʼt tell
29
Summary ratingEthical issues
taken into
consideration
Data
analysis
sufficiently
rigorous
Statement
of findings
clear
ExclusionValue of research
Moderate qualityCanʼt tell Canʼt tell Yes IncludedOne of two studies in
sample that compares
ASD and FASD.
Interesting application
of hope theory. Clear
description of lower
hope for future
outcomes for children
with FASD and degree
to which this group is
ignored and unsupported
relative to ASD.
Reporting makes
difficult to appraise but
a valuable study for this
analysis.
Canʼt tell Ye s Yes IncludedYes. Valuable given UK
setting and in-depth and
explicit findings with
good supporting quotes.
Appendices
30
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Appendix 4: Feedback from carers
A focus group with members of the Grandparents Parenting Again and Kinship Carers (Midlothian),
consisting of family members of individuals affected by prenatal alcohol exposure, was conducted
in November 2018 to find out whether they identify with caregivers’ experiences reported in the
discussion section of the report. Carers provided feedback which was summarised according to each
individual theme. This consultation aims to demonstrate what relevance each theme identified in the
discussion might have for carers in Scotland.
Living with and coping with FASD
Carers identified with the experience of stress of living life through their emotions all the time and
the impact that stress has on their mental and physical health as a result of caring for an individual
with FASD. They further spoke about how stress about their child causes them to feel a failure and
indirectly affects the relationship with their partners or other family members. Carers shared feelings
of exhaustion and also shame due to unawareness about the reasons for the child’s behaviour. One
carer did not identify with the positive caregiving outlook described in the synthesis but shared that
they would have not taken the child if they had known he/she was affected by FASD. The caregiver,
however, appreciated that the child is safe and looked after.
Some carers shared concerns about their own mortality and the impact it could have on their children
not being able to cope on their own. One carer expressed worry about their child’s developmental
delay which could have an impact on their future but noted that one-to-one support through school
has helped their child.
Carers agreed with the concept described in the parenting strategies subtheme that parenting a
child with FASD was a very different experience from conventional parenting. One carer made the
point that what was needed was not just ‘good enough parenting’ but ‘exceptional parenting’. Carers
felt they needed to attend parenting courses such as Incredible Years and Growing with Confidence.
Carers shared using different parenting strategies to cope with their child’s behaviour. Carers avoided
accusation but still sanctioned the behaviour of the child while reaffirming their love for them.
Stigma and Blame
Consistent with the theme synthesised from the published evidence, carers shared experiences of
being criticised by community members and professionals in relation to the child’s behaviours being
due to poor parenting. They also shared their own criticism of their own children for their lifestyle
choices, where one carer showed clear disapproval towards their daughter’s behaviour.
Diagnosis
One carer shared the experience of being told by child and adolescent mental health services that
due to the lack of the facial features, the child does not have FASD. Professionals suggested that the
terrifying breakdown in the child’s regulation and prolonged tantrums lasting hours were due to early
puberty which caused the carers to feel frustrated. Sometimes carers were told that child’s behaviour
could be partly due to attachment issues and partly due to autism spectrum disorder (ASD). Some
carers shared the experience of their child refusing to be assessed or having to wait long periods of
time for the results of assessment and subsequently being informed of no impairment. Overall, carers
shared the experience of being frustrated both because of the lack of formal diagnosis but also left
uncertain whether a formal diagnosis could help.
31
Support
One carer shared the experience of passing from one professional to another without being able to find
out what is wrong with their children. Lack of knowledge and awareness of FASD among healthcare
professionals was one of the most common themes identified in the studies within the literature
included in the synthesis. Similarly, Scottish carers felt that both social workers and healthcare
professionals should be informed about FASD. Furthermore, carers pointed out the lack of awareness
and knowledge of FASD among school professionals and wanted every school and member of staff
to have the pack from the National Organisation for Foetal Alcohol Syndrome-UK (NOFAS-UK) which
contains a Teacher’s Tookit with checklist of things which will help the child manage on a day-to-day
basis in the classroom.
Caregivers identified the significant and unique role that the Kinship Care group has in their lives
because it provides them with an opportunity to support each other and exchange invaluable
information around parenting, benefits, arrangements with the local authority, entitlements and
many more. In regards to the importance of mutual support between spouses, one carer talked about
the lack of emotional support provided by her spouse and shared the experience of feeling as being
‘only one person’.
Appendices
32
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
Development team
Ms Nikolina Angelova Health Services Researcher, Healthcare Improvement Scotland
(Lead author)
Ms Juliet Brown Evidence and Information Scientist, Healthcare Improvement Scotland
Ms Naomi Fearns Health Services Researcher, Healthcare Improvement Scotland
Ms Sarah Harley Health Services Researcher, Healthcare Improvement Scotland
Dr Moray Nairn Programme Manager, SIGN
33
References
1 Jones KL, Smith DW. Recognition of the fetal alcohol syndrome
in early infancy. Lancet 1973;302(7836):999-1001.
2 Clarren SK, Smith DW. The fetal alcohol syndrome. N Engl J Med
1978;298(19):1063-7.
3 Institute of Medicine. Fetal Alcohol Syndrome: Diagnosis,
Epidemiology, Prevention, and Treatment. Washington, DC:
National Academies Press; 1996.
4 Dorris M. The Broken Cord. New York, NY: Harper & Row
Publishing; 1989.
5 Buxton B. Damaged Angels. Toronto, ON: Alfred A. Knopf of
Canada; 2004.
6 Malbin D. Fetal alcohol spectrum disorder: A collection of
information for parents and professionals. 2nd. Portland, OR:
FASCETS, Inc.; 2007.
7 Graefe S. Living with FASD: A guide for parents. Vancouver, BC:
Groundwork Press; 2004.
8 Olson HC, Jirikowic T, Kartin D, Astley S. Responding to the
Challenge of Early Intervention for Fetal Alcohol Spectrum
Disorders. Infants & Young Children 2007;20(2):172-89.
9 Watson SL, Coons KD, Hayes SA. Autism spectrum disorder and
fetal alcohol spectrum disorder. Part I: A comparison of parenting
stress. Journal of Intellectual & Developmental Disability
2013;38(2):95-104.
10 Olson HC, Oti R, Gelo J, Beck S. 'Family matters:' Fetal alcohol
spectrum disorders and the family. Developmental Disabilities
Research Reviews 2009;15(3):235-49.
11 Paley B, O'Connor MJ, Kogan N, Findlay R. Prenatal alcohol
exposure, child externalizing behavior, and maternal stress.
Parenting: Science and Practice 2005;5(1):29-56.
12 Brown JD, Bednar LM. Challenges of parenting children with
a fetal alcohol spectrum disorder: A concept map. Journal of
Family Social Work 2004;8(3):1-18.
13 Brown JD, Sigvaldason N, Bednar LM. Foster parent perceptions
of placement needs for children with a fetal alcohol spectrum
disorder. Children and Youth Services Review 2005;27(3):309-
27.
14 Chernoff R, Combs-Orme T, Risley-Curtiss C, Heisler A. Assessing
the health status of children entering foster care. Pediatrics
1994;93(4):594-601.
15 May PA, Hymbaugh KJ, Aase JM, Samet JM. Epidemiology
of fetal alcohol syndrome among American Indians of the
Southwest. Soc Biol 1983;30(4):374-87.
16 Streissguth AP, Clarren SK, Jones KL. Natural history of the fetal
alcohol syndrome: a 10-year follow-up of eleven patients. Lancet
1985;2(8446):85-91.
17 Astley SJ, Stachowiak J, Clarren SK, Clausen C. Application of
the fetal alcohol syndrome facial photographic screening tool
in a foster care population. J Pediatr 2002;141(5):712-7.
18 Brown JD, Rodger S, George N, St Arnault D, Sintzel J. Rewards
of parenting a child with a fetal alcohol spectrum disorder. The
Open Family Studies Journal 2008;1:23-30.
19 Sanders JL, Buck G. A long journey: Biological and non-
biological parents’ experiences raising children with FASD.
Journal of population therapeutics and clinical pharmacology
2010;17(2):e308-e22.
20 Gentles SJ, Charles C, Nichola DB, Ploeg J, McKibbon KA.
Reviewing the research methods literature: principles and
strategies illustrated by a systematic overview of sampling in
qualitative research. Systematic Reviews 2016;5(172).
21 Noyes J, Popay J, Pearson A, Hannes K, Booth A. Qualitative
research and Cochrane reviews. 2008.
22 De Jean D, Giacomini M, Simeonov D, Smith A. Finding qualitative
research evidence for health technology assessment. Qualitative
health research 2016;26(10):1307-17.
23 Ring N, Ritchie K, Mandava L, Jepson R. A guide to synthesising
qualitative research for researchers undertaking health
technology assessments and systematic reviews. 2010;
24 Noyes J LS. Supplemental Guidance on Selecting a Method of
Qualitative Evidence Synthesis and Integrating Qualitative
Evidence with Cochrane Intervention Reviews. 2011. [cited
2014 Jan 13]. Available from url: http://cqim.cochrane.org/
supplemental-handbook-guidance
25 Programme CAS. CASP Qualitative Research Checklist. 2018.
26 Assessment SCoHT. Evaluation and synthesis of studies using
qualitative methods of analysis. 2013.
27 Glenton C, Colvin CJ, Carlsen B, Swartz A, Lewin S, Noyes J, et
al. Barriers and facilitators to the implementation of lay health
worker programmes to improve access to maternal and child
health: qualitative evidence synthesis (Review). The Cochrane
Library; 2013.
28 Dixon-Woods M, Booth A, Sutton AJ. Synthesizing qualitative
research: a review of published reports. Qualitative Research
2016;7(3):375-422.
29 Atkins S, Lewin S, Smith H, Engel M, Fretheim A, Volmink J.
Conducting a meta-ethnography of qualitative literature: lessons
learnt. BMC Med Res Methodol 2008;8:21.
30 Ipsiroglu OS, McKellin WH, Carey N, Loock C. "They silently live
in terror ... " why sleep problems and night-time related quality-
of-life are missed in children with a fetal alcohol spectrum
disorder. Social Science & Medicine 2013;79:76-83.
31 Michaud D, Temple V. The Complexities of Caring for Individuals
with Fetal Alcohol Spectrum Disorder: The Perspective of
Mothers. Journal on Developmental Disabilities 2013;19(3).
32 Whitehurst T. Raising a child with foetal alcohol syndrome:
hearing the parent voice. British Journal of Learning Disabilities
2012;40(3):187-93.
33 Chamberlain K, Reid N, Warner J, Shelton D, Dawe S. A
qualitative evaluation of caregivers’ experiences, understanding
and outcomes following diagnosis of FASD. Research in
Developmental Disabilities 2017;63:99-106.
34 Gardner J. Living with a child with fetal alcohol syndrome.
MCN: The American Journal of Maternal/Child Nursing
2000;25(5):252-7.
35 Thomas J, A H. Methods for the thematic synthesis of qualitative
research in systematic reviews. BMC Medical Research
Methodology 2008;8(45).
References
The experiences of caregivers looking after individuals with Fetal Alcohol Spectrum Disorder
34
36 Ritchie J SL. Qualitative data analysis for applied policy research.
In: Bryman A BR, editor. Analyzing qualitative data. Abingdon:
Routledge; 1994.
37 Coons KD, Watson SL, Schinke RJ, Yantzi NM.
Adaptation in families raising children with fetal alcohol
spectrum disorder. Part I: What has helped. Journal of
Intellectual & Developmental Disability 2016;41(2):150.
38 Coons KD, Watson SL, Yantzi NM, Schinke RJ. Adaptation in
families raising children with fetal alcohol spectrum disorder.
Part II: What would help. Journal of Intellectual & Developmental
Disability 2016:1-15.
39 Watson S, Hayes S, Radford-Paz E, Coons K. 'I'm hoping, I'm
hoping...' Thoughts about the future from families of children
with autism or fetal alcohol spectrum disorder in Ontario.
Journal on Developmental Disabilities 2013;19(3):76-93.
40 Watson SL, Hayes SA, Coons KD, Radford-Paz E. Autism
Spectrum Disorder and Fetal Alcohol Spectrum Disorder Part
II: A Qualitative Comparison of Parenting Stress. 2013.
41 Granitsas J. Experiences of adoptive parents of children
with fetal alcohol syndrome. Clinical Excellence for Nurse
Practitioners 2004;8(1):22-8.
42 Salmon J. Fetal alcohol spectrum disorder: New Zealand birth
mothers’ experiences. Can J Clin Pharmacol 2008;15(2):e191-
213.
43 Morrissette PJ. Fetal alcohol syndrome: Parental experiences
and the role of family counselors. The Qualitative Report
2001;6(2):1-20.
44 Mukherjee R, Wray E, Commers M, Hollins S, Curfs L. The
impact of raising a child with FASD upon carers: findings from
a mixed methodology study in the UK. Adoption & Fostering
2013;37(1):43-56.
45 Tsang TW, Lucas BR, Olson HC, Pinto RZ, Elliott EJ. Prenatal
alcohol exposure, FASD, and child behavior: A meta-analysis.
Pediatrics 2016;137(3):1-20.
46 Helgesson G, Bertilsson G, Domeij H, Fahlstrom G, Heintz E,
Hjern A, et al. Ethical aspects of diagnosis and interventions
for children with fetal alcohol Spectrum disorder (FASD) and
their families. BMC Med Ethics 2018;19(1):1.
47 Olson HC, Oti R, Gelo J, Beck S. "Family matters:" fetal alcohol
spectrum disorders and the family. Dev Disabil Res Rev
2009;15(3):235-49.
48 Hasting RP, Taunt HM. Positive perceptions in families of children
with developmental disabilities. American Journal on Mental
Retardation 2002;107:116-27.
49 SBU. Evaluation and synthesis of studies using qualitative
methods of analysis. Swedish Agency for Health Technology
Assessment and Assessment of Social Services 2016.
50 British Medical Association. Alcohol and pregnancy. Preventing
and managing fetal alcohol spectrum disorders. London: BMA,
2016.
Healthcare Improvement Scotland
Edinburgh Ofce
Gyle Square
1 South Gyle Crescent
Edinburgh
EH12 9EB
0131 623 4300
Glasgow Ofce
Delta House
50 West Nile Street
Glasgow
G1 2NP
0141 225 6999
www.healthcareimprovementscotland.org
ResearchGate has not been able to resolve any citations for this publication.
Article
Full-text available
This paper discusses the experiences of foster/adoptive parents who raise children diagnosed with Fetal Alcohol Syndrome and the instrumental role family counselors can play regarding child management issues, intimate relationships, larger system collaboration, and research. For the actual study, six parents participated in individual, in-depth tape-recorded interviews. Each interview was transcribed, thematically analyzed, and revealed seven predominant themes including (a) constant vigil, (b) effects on marriages, (c) child management, (d) parental issues, (e) interactions with professional community, (f) emancipation concerns, and (g) medical implications. The ways in which family counselors can intervene are reviewed and include the role of clinician, facilitator, and researcher/educator.
Article
Full-text available
Background Fetal alcohol spectrum disorders (FASD) is an umbrella term covering several conditions for which alcohol consumption during pregnancy is taken to play a causal role. The benefit of individuals being identified with a condition within FASD remains controversial. The objective of the present study was to identify ethical aspects and consequences of diagnostics, interventions, and family support in relation to FASD. Methods Ethical aspects relating to diagnostics, interventions, and family support regarding FASD were compiled and discussed, drawing on a series of discussions with experts in the field, published literature, and medical ethicists. ResultsSeveral advantages and disadvantages in regards of obtaining a diagnosis or description of the condition were identified. For instance, it provides an explanation and potential preparedness for not yet encountered difficulties, which may play an essential role in acquiring much needed help and support from health care, school, and the social services. There are no interventions specifically evaluated for FASD conditions, but training programs and family support for conditions with symptoms overlapping with FASD, e.g. ADHD, autism, and intellectual disability, are likely to be relevant. Stigmatization, blame, and guilt are potential downsides. There might also be unfortunate prioritization if individuals with equal needs are treated differently depending on whether or not they meet the criteria for a specific condition. Conclusions The value for the concerned individuals of obtaining a FASD-related description of their condition – for instance, in terms of wellbeing – is not established. Nor is it established that allocating resources based on whether individuals fulfil FASD-related criteria is justified, compared to allocations directed to the most prominent specific needs.
Article
Full-text available
Background Overviews of methods are potentially useful means to increase clarity and enhance collective understanding of specific methods topics that may be characterized by ambiguity, inconsistency, or a lack of comprehensiveness. This type of review represents a distinct literature synthesis method, although to date, its methodology remains relatively undeveloped despite several aspects that demand unique review procedures. The purpose of this paper is to initiate discussion about what a rigorous systematic approach to reviews of methods, referred to here as systematic methods overviews, might look like by providing tentative suggestions for approaching specific challenges likely to be encountered. The guidance offered here was derived from experience conducting a systematic methods overview on the topic of sampling in qualitative research. ResultsThe guidance is organized into several principles that highlight specific objectives for this type of review given the common challenges that must be overcome to achieve them. Optional strategies for achieving each principle are also proposed, along with discussion of how they were successfully implemented in the overview on sampling. We describe seven paired principles and strategies that address the following aspects: delimiting the initial set of publications to consider, searching beyond standard bibliographic databases, searching without the availability of relevant metadata, selecting publications on purposeful conceptual grounds, defining concepts and other information to abstract iteratively, accounting for inconsistent terminology used to describe specific methods topics, and generating rigorous verifiable analytic interpretations. Since a broad aim in systematic methods overviews is to describe and interpret the relevant literature in qualitative terms, we suggest that iterative decision making at various stages of the review process, and a rigorous qualitative approach to analysis are necessary features of this review type. Conclusions We believe that the principles and strategies provided here will be useful to anyone choosing to undertake a systematic methods overview. This paper represents an initial effort to promote high quality critical evaluations of the literature regarding problematic methods topics, which have the potential to promote clearer, shared understandings, and accelerate advances in research methods. Further work is warranted to develop more definitive guidance.
Article
Full-text available
Introduction: The effects of prenatal exposure to alcohol are wide-ranging and pervasive in nature. In response to growing concerns about the lifelong disabilities related to prenatal alcohol exposure, a fetal alcohol spectrum disorder (FASD) diagnostic clinic was established in 2012. This was the first multi-disciplinary service operating permanently within an Australian health service. The current study aimed to explore the lived experience of the diagnostic process for caregivers of children with FASD. Methods: Twelve caregivers were approached and ten participated in audiotaped interviews about caring for a child with FASD. Qualitative analysis was undertaken on transcribed interviews using NVivo 10 for thematic analysis. Results: The major themes that emerged were: a desire for future support for their child although uncertainty about how this support could be accessed; an understanding of FASD prior to assessment but a concerted concern that this condition was not recognised as a disability across educational and related settings; that a formal assessment process provided validation for caregiver concerns and that caregivers felt respected and understood by the clinical team during this process. Discussion: These findings highlight the concerns of caregivers, emphasising both the importance of diagnostic services and the need for provision of further support beyond diagnosis for a child with FASD. WHAT DOES THIS PAPER ADD?: This study provides information on the lived experiences of caregivers who attended the first multi-disciplinary FASD assessment and diagnostic service in Australia. This study provides valuable insight into the ongoing difficulties that families are experiencing following diagnosis in the current Australian context.
Article
Full-text available
Context: Fetal alcohol spectrum disorders (FASD) and prenatal alcohol exposure (PAE) are associated with behavioral difficulties, although there are no published systematic reviews that summarize and critique the literature. Objective: To describe the behavioral characteristics of children with PAE and/or FASD, assessed using the Achenbach System of Empirically Based Assessments (ASEBA) for school-aged children with parent, teacher, and youth (self-report) forms. Data sources: Electronic literature databases, reference lists, hand-searches. Study selection: peer-reviewed observational studies. Data extraction: Study appraisal and data extraction were undertaken by 2 independent assessors. Meta-analyses were performed for parent-rated Internalizing, Externalizing, and Total problems scales. All other ASEBA scales were summarized qualitatively. Results: Included were 23 articles; 16 were used in meta-analyses. Pooled results showed higher Total (mean difference 12.1, 95% confidence interval [95% CI] 7.7-16.5), Internalizing (6.3, 95% CI 3.1-9.5), and Externalizing problems scores (12.5, 95% CI 7.9-17.0) in FASD than No FASD; and greater odds of scoring in the "Clinical" range in FASD. Pooled results demonstrated higher problem scores in children with PAE (P > .05). Qualitative summaries of other scales from parents, teachers, and self-report show poorer behavior ratings in children with FASD and PAE on composite Problem and Competence scores and many Syndrome subscales. Limitations: Findings were restricted to behaviors assessed using the ASEBA. The published literature was limited, often with only 1 study reporting on a particular scale. Conclusions: Meta-analysis reveals that FASD and PAE are associated with problematic behavior in many, but not all domains. This clearly affects families, and should be considered in clinical practice by providers.
Article
Background Our aim in this paper is to build upon the experience of family adaptation and to describe one unique stressor experienced by adoptive parents of children with fetal alcohol spectrum disorder (FASD) that may hinder successful family adaptation. Method Fifty-one adoptive parents of children with FASD from 36 families in Ontario, Canada, participated in a mixed methods study. Parents completed 2 questionnaires, the Family Crisis Oriented Personal Scales and the Questionnaire on Resources and Stress – Friedrich’s Short Form, and participated in in-depth, semistructured interviews, which were analysed using interpretative phenomenological analysis. Results Parents expressed that improved knowledge and awareness of FASD from healthcare professionals, teachers, and society as a whole would help in their adaptation to raising their children with FASD. Specifically, parents discussed the lack of recognition of the symptoms of FASD, the lack of understanding about what a diagnosis of FASD means, and the need to promote better messages of prevention. Conclusions Our findings display a clear need for more supports for families, particularly in the area of increasing public and professional knowledge about the realities of raising a child with FASD.
Article
Health technology assessment (HTA) agencies increasingly use reviews of qualitative research as evidence for evaluating social, experiential, and ethical aspects of health technologies. We systematically searched three bibliographic databases (MEDLINE, CINAHL, and Social Science Citation Index [SSCI]) using published search filters or "hedges" and our hybrid filter to identify qualitative research studies pertaining to chronic obstructive pulmonary disease and early breast cancer. The search filters were compared in terms of sensitivity, specificity, and precision. Our screening by title and abstract revealed that qualitative research constituted only slightly more than 1% of all published research on each health topic. The performance of the published search filters varied greatly across topics and databases. Compared with existing search filters, our hybrid filter demonstrated a consistently high sensitivity across databases and topics, and minimized the resource-intensive process of sifting through false positives. We identify opportunities for qualitative health researchers to improve the uptake of qualitative research into evidence-informed policy making.
Article
Background There is limited research investigating the lived experiences of parents raising children with fetal alcohol spectrum disorder (FASD). The aim of this paper is to use qualitative analysis to identify how parents have adapted to the experience of raising their child with FASD. Method Eighty-four parents and caregivers of children with FASD in Ontario, Canada, participated in in-depth, semistructured interviews employing a basic interpretive approach. Results Parents of children with FASD report a number of strategies, supports, and transformational outcomes. Using interpretative phenomenological analysis, 5 themes were identified: understanding FASD and advocating on their child's behalf, day-to-day adaptation, transformational outcomes, as well as the importance of informal and formal supports. Conclusions Understanding what families do in order to facilitate adaptation is important when assisting families who may not be adapting as successfully. Continued research looking at the family experience of raising a child with a developmental disability, such as FASD, is necessary.