Families of children with Down syndrome:What we know and what we need to know
Monica Cuskelly, Penny Hauser-Cram and Marcia Van Riper
This paper provides a brief overview of what is currently known about families of children with Down syndrome. In addition, it highlights a number of issues that require further research if we are to have a thorough understanding of the impact of a child with Down syndrome on families as a system and on the individuals who make up that system. Some of these issues include the need for: 1) a more balanced perspective – one that acknowledges both positive and negative aspects of the experience, 2) greater attention to the experiences of fathers, 3) more cross-cultural research and studies focused on cultural perspectives, 4) increased attention to the change in demands for families as the individual with Down syndrome ages, 5) more longitudinal studies, 6) greater variety in methodological approaches, for example greater use of qualitative approaches and observational methods, and 7) an increase in the use of statistical approaches that model change and test hypotheses about predictors of change in both parents and children.
From a family systems perspective, the relational life of families provides the
central ecological context in which children are nurtured.
The family system is influenced by the attributes each individual brings to family
relationships as well as by family members' perceptions of those attributes. The
birth of a child with Down syndrome is likely to affect the family system in many
ways, from the micro level of dyadic interaction to the macro level of the cultural
views guiding parent perceptions about a developmental disability.
Much research has indicated that a child with Down syndrome has effects on the family. Some effects relate to the likelihood that
a child will display the cognitive and behavioural phenotypes considered to be typical
of children with Down syndrome. These include specific patterns of strengths and
weaknesses in information processing, social interaction, expressive language, receptive
skills, motor skills, and motivation[3,4,5].
Such patterns and/or the expectations of such patterns may influence the behaviour
of caregivers in dyadic interaction with the child. Other effects may be due to
increased difficulties and/or diminished satisfactions in the parental role and
to parents' reduced opportunities in other spheres of life such as career success.
In contrast, positive effects are possible through the parents' identification of
a particular purpose in life, or the development of particular talents that may
not have been called upon in other circumstances[6,7,8].
Parental adaptation to a child with Down syndrome has also been studied extensively
in relation to parental well-being, especially the psychological reactions of parents.
Many studies on parental adaptation to a child with Down syndrome have focused primarily
on stress. The literature is often unclear about the precise meaning of stress.
At least three interpretations have been applied to research on parental adaptation.
Some authors use stress to refer to the emotional responses of parents to the demands
of the parenting role (e.g., feeling isolated, entrapped, overwhelmed with responsibility).
Others focus on the demands brought about by the child's temperament and behaviours
(e.g., demandingness, soothability, activity level). A third approach focuses on
parental mental health and psychological functioning (e.g., depression, anxiety,
self-acceptance, mastery). This latter approach provides more useful data as it
is possible for families to face increased demands but to meet these effectively
(see, for example,
ref 8). Clearly, however, parenting responsibilities, child demands
and psychological outcomes are related.
While there is some evidence that parents of children with Down syndrome experience
similar levels of well-being to those experienced by parents of typically developing
children, most studies suggest that parents
of children with Down syndrome experience lower levels of well-being than parents
of typically developing children of similar age (see, for example, refs 10,11). However,
it should be noted that, in these studies, the majority of the parents fell into
the non clinical range on instruments measuring psychological functioning (e.g.,
Nevertheless, as Singer pointed out, even mild levels of depression can have detrimental
effects on individuals and on their families.
Despite these increased difficulties in comparison to families where all children
are developing typically, as a group, parents of a child with Down syndrome experience
fewer negative effects and more positive effects than parents of children with other
disabilities[14-17]. Many studies compare
parents of children with Down syndrome with those who have a child with autism (e.g.,
18,19), a comparison that may increase the likelihood of this outcome.
There are also studies which use families of children with other conditions as a
comparison group (e.g.,
refs 16,17) and the results generally show that parents of children
with Down syndrome have higher levels of well-being. In a recent study examining
"the Down syndrome advantage," Stoneman reported that the findings generally
replicated the Down syndrome advantage found by other researchers.
However, once the variance attributable to income was removed, the Down syndrome
advantage disappeared. Another possible explanation for the Down syndrome advantage
is that families whose children with Down syndrome are functioning more poorly than
the average or those who have severe behaviour problems (possibly as a consequence
of co-morbid conditions such as autism) may choose not to participate in research
There have now been several important studies that have collected longitudinal data
related to the parenting experience of parents of a child with Down syndrome. Hauser-Cram,
Warfield, Shonkoff and Krauss found that over a seven year period (child age 3 years
to 10 years) the demands associated with raising a child with Down syndrome increased
for both mothers and fathers, and for mothers, this increase was greater than that
experienced by mothers of a child with a motor impairment or mothers of a child
with a developmental delay of unknown origin.
At the initial data point (3 years) the mothers of a child with Down syndrome had
lower child related demands, but these demands increased so that by age 10 they
were higher than those reported by the other two groups. Very similar findings were
reported by Most, Fidler, LaForce-Booth and Kelly
who compared the trajectories of child-related demands in mothers of a child with
Down syndrome with that of mothers of a comparison group of children with intellectual
disability of mixed aetiology using the child related aspects of the Parenting Stress
Index (PSI). The children were between
12 and 15 months on the first occasion, 30 months on the second occasion and were
45 months when the third wave of data was collected. While mothers of a child with
Down syndrome reported lower stress levels on the PSI than the mothers in the comparison
group at 12 months, when their child was 45 months of age there were no differences.
The mothers of a child with Down syndrome showed, at the group level, an increase
in stress that was not apparent in the other group. While the groups were initially
matched on developmental level, by Time 3 the children with Down syndrome were functioning
at a lower cognitive level than the children in the comparison group.
An increase in stress as the child with Down syndrome ages was also reported by
Eisenhower, Baker and Blacher. These
authors initially saw families when the child was aged 36 months and then collected
data again at 48 and 60 months and found an increase in problem behaviour in the
children with Down syndrome which was associated with an increase in maternal stress.
Both Most and colleagues, and Eisenhower,
Baker and Blacher, suggested that the
comparative protective effect often attributed to Down syndrome (in contrast to
other types of disability) may apply only in the early years of life. The longitudinal
studies indicate that stress levels of parents of children with Down syndrome increase
over the early to middle childhood period, suggesting that more research should
be directed at delineating moderators of stress beyond the infant and toddler years.
As Most and colleagues point out, however, the majority of studies are cross sectional
and match children on developmental level at the age being studied.
If trajectories of development differ, over time, the children will cease to be
matched, which may account for the changes in parental status vis a vis the comparison
The same association between behaviour problems and parental stress is evident in
these parents as in other parents of a child with a disability[14,16].
Generally, however, children with Down syndrome exhibit fewer behaviour problems
than children with intellectual disability from other causes[25,26],
although more than typically developing children and siblings. Both
the Hauser-Cram and Eisenhower studies reported that the increase in stress levels
were related to the increase in child-related demands experienced by parents[22,25].
It is still the case that the majority of research regarding family functioning
is conducted with mothers. Several factors contribute to this pattern. One is that
mothers continue to take the primary child caring role with respect to children
with Down syndrome. In an interview study
with fathers of a child with Down syndrome that focused on their experiences of
parenting, 46% spontaneously stated that their wives took almost all responsibility
in relation to the child with Down syndrome.
Nevertheless, research on fathers of children with Down syndrome indicates that
they are contributing to family life in a variety of ways. Hedov and colleagues
found that fathers of a child with Down syndrome took a greater share of the child
care (when days off work to care for a sick child were examined) than did fathers
in families where all children were developing typically.
In one of the few studies focusing on fathers' perceptions, Ricci and Hodapp found
that fathers of children with Down syndrome reported their child to have more positive
personality traits and fewer behaviour problems than fathers of children with other
types of intellectual disabilities. The older
children with Down syndrome, however, were found to be less reinforcing and acceptable
to fathers than the younger children.
There are a number of variables that have been found to contribute to parental outcomes,
and it seems likely that mothers and fathers respond differently to the pressures
associated with raising a child with Down syndrome (see, for example,
Krauss reported that although mothers and fathers of infants and toddlers with developmental
disabilities (including Down syndrome) did not differ in overall levels of stress,
their patterns of stress were distinctly different.
Mothers reported higher levels of stress related to the parenting role whereas fathers
reported more stress related to their feelings of attachment to the child. Similarly,
Keller and Honig found that maternal stress was more related to the demands of child
care, while fathers' stress was more related to the child's acceptability. Further support for differences between mothers
and fathers is provided by Saloviita, Itälinna and Leinonen.
They found that, although for both parents a negative view of their situation was
most associated with the experience of stress, mothers were most influenced by behavioural
problems in the child while fathers were more influenced by their child's low social
acceptability. It is clearly important to learn more about fathers' experiences
of parenting a child with Down syndrome and to examine their contributions to family
life in ways that move beyond the limited possibilities of providers of economic
and spousal support.
Parental satisfaction and self-efficacy
Studies on parenting efficacy focus on parents' confidence in their ability to fulfil
the parenting role and successfully nurture children's development. Parenting efficacy
incorporates dimensions of confidence in parenting with satisfaction with the parenting
role. Greater parenting efficacy has been associated with more positive perceptions
of the child. Hassall, Rose and McDonald
found that parents of a child with intellectual disability who felt satisfied with
their parenting role reported lower levels of stress than did those who were less
satisfied. Gilmore and Cuskelly compared the scores of mothers of a child
with Down syndrome with normative data they had collected for the Parenting Sense
of Competence measure and found that,
when children were young (between the ages of 4 and 6), mothers' reports of their
satisfaction with parenting were not different from those of mothers of children
who were developing typically. Confidence in their parenting skills, however, was
lower for the mothers of a child with Down syndrome.
Self-efficacy in the parenting role reflects parents' confidence that they have
the skills to provide the necessary guidance to their child.
Laws and Millward suggested that, for parents of a child with Down syndrome, parental
identity is tied up with being their child's educator.
Parental self-efficacy for these parents may therefore be partially based on their
child's acquisition of skills. In the study discussed above, Gilmore and Cuskelly
found that confidence was not related to competence as measured by an IQ test. Measures of IQ, however, may be inadequate
to capture increases in skills, and adaptive behaviour measures may be more useful
for this purpose.
An additional view of parental adaptation can be gained by a consideration of family
functioning. The importance of family relationships to the well-being of all family
members has been highlighted by the review of scientific evidence of early childhood
by Shonkoff and Phillips. The way in
which the family functions may serve as a buffer or promoter of parenting stress.
For example, trajectories in maternal parenting stress of children with developmental
disabilities have been found to be reduced in cohesive families (i.e., families
in which members feel connected to and supported by each other).
Another aspect of family functioning, perceived quality of family life, has recently
been investigated. Investigations of quality of life provide a broad view of several
domains of family life including health, financial well-being, family relationships,
spiritual and cultural beliefs, social support, leisure enjoyment, and community
involvement. Brown, MacAdam-Crisp, Wang, and Iarocci investigated the quality of
life for families in which a child had Down syndrome or autism in comparison to
families of typically developing children.
Although parents of children with autism reported the lowest overall quality of
life, the families of children with Down syndrome reported a lower quality of life
in relation to health, financial well-being, social support, and career opportunities
(for parents) than those in the comparison group.
One of the central relationships within families is that between mothers and fathers.
A meta-analysis conducted by Risdal and Singer found that the published data on
marital functioning in families with a child with a disability were somewhat skewed
by the negatively biased assumptions of researchers, and that when these biases
were addressed, the negative consequences for this relationship were substantially
less then heretofore thought. Van Riper
and colleagues found no differences in marital functioning (or family functioning)
between families with a child with Down syndrome and comparison families where all
children were developing typically. Kersh,
Hedvat, Hauser-Cram and Warfield also reported that married mothers and fathers
of 10-year-old children with motor impairment, Down syndrome or developmental delay
reported no differences in their marital quality, although all three groups had
lower levels of marital quality than couples in the general population.
Higher marital quality was related to lower levels of stress and fewer depressive
symptoms for both mothers and fathers as well as to greater efficacy for mothers.
In a study of the perceived burden of care of mothers of children with a developmental
disability (one third of whom had Down syndrome), Erickson and Upshur found that
the mother's perception of the caretaking burden was lighter when the father participated
in tasks and provided emotional support.
Building partially on that study, Simmerman, Blacher, and Baker studied marital
satisfaction of parents with a child with severe intellectual disabilities in middle
to late childhood. They found that mothers'
satisfaction with fathers' help, not the actual amount of help provided, predicted
both mothers' and fathers' marital adjustment. Fathers' help was most frequently
reported to be in the areas of playing, nurturing, discipline and decision-making
about services and less frequently in areas of hygiene, dressing, feeding, teaching,
therapy and taking a child to appointments. These studies suggest that, although
the dyadic relationship between mothers and fathers has multiple dimensions, maternal
satisfaction with fathers' participation may guide the relationship.
The sibling experience
The relationship between siblings is considered to be one of the most enduring relationships
within families. There are a number of studies that have examined the impact on
siblings of having a brother or sister with Down syndrome, although these generally
focus on children who are in middle childhood or older. These studies typically
focus on behaviour or other adjustment problems in the sibling and/or on the relationship
between the typically developing child and the child with Down syndrome. While there
were some initial reports of adjustment difficulties (e.g. ref 44) more recent
research has found that the siblings have favourable self-concepts
and that many believe they have developed additional strengths because of their
sibling with Down syndrome[45,46]. Also,
findings suggest that there are no important differences in the adjustment of the
siblings of a child with Down syndrome and children in families where all are developing
and that relationships are as good as or better than in these families[48,49].
Good sibling relationships are often perceived by mothers as evidence of good parenting. We have very little understanding of how
parents accomplish this task, although it is clear that the majority do so.
Some of the challenges for researchers interested in examining the sibling experience
include the lack of longitudinal studies with large, diverse samples. Most of the
existing sibling studies are cross-sectional studies conducted with small, convenience
samples of white, middle to upper-class siblings. Many families of children with
Down syndrome have more than one typically developing sibling, so another challenge
for researchers in this area is deciding which sibling to include in the study.
A final challenge for sibling researchers is the lack of good measures to evaluate
the sibling experience, especially measures designed to assess positive outcomes.
For a more complete review of the challenges facing researchers interested in the
sibling experience, see reviews by Hodapp, Glidden and Kaiser, Stoneman, and Van
Positive outcomes for families
When given the opportunity, parents identify a range of positive contributions that
the child with Down syndrome makes to family life (see ref 53). Unfortunately,
that opportunity is often not provided as researchers have generally focused on
the negative consequences experienced by family members. Most relationships comprise
both positive and negative aspects, and the dominant quality may change over time
as circumstances vary. Family life in families of children with Down syndrome is
likely to contain some mix of hassles and uplifts, disappointments and great satisfactions,
and it important that the uplifts and satisfactions be considered if an accurate
picture of family life is to be gained. These positive aspects of family life are
also likely to contribute to the ability to cope with the difficulties families
may face. Parents report increased feelings
of empowerment, personal growth, and a rearrangement of priorities, as examples
of the positive changes they attribute to their experience of parenting a child
with a disability.
Poehlmann and colleagues found that mothers saw their child with Down syndrome as
having a number of very positive personal characteristics that acted to maintain
and develop connections between family members and with others.
Hodapp, Ly, Fidler and Ricci found no difference between parents of a child with
Down syndrome and parents of typically developing children with respect to 'rewardingness'. Cuskelly and colleagues found that mothers
of a child with Down syndrome reported significantly more reinforcing aspects of
their relationship with their child than did mothers of typically developing children.
More work on investigating the satisfactions associated with parenting a child with
Down syndrome needs to be done. This is important in order that a balanced perspective
can be provided to families who are beginning their lives as a family with a child
with Down syndrome. It is also important that the broader society is appraised of
these experiences as they may contribute to attitude change and to therefore increasing
the inclusiveness of our society.
Connections to community
Disability is typically viewed as a burden, and the general community holds fairly
negative views about parenting a child with Down syndrome[57,58,59].
Naturally, many individuals who become the parent of a child with Down syndrome
shared these values prior to the birth of their child. While many parents alter
their view after the birth of their child with Down syndrome[60,61],
they remain embedded within a community for whom having a child with a disability
can be viewed only as a tragedy. This is clearly somewhat of an overstatement –
there are many individuals who do hold this opinion and families of a child with
Down syndrome will have contact with individuals with a range of views. The impact
of this dissonance between family and community values has rarely been examined
explicitly; however, the enmeshment experienced by some families of a child with
a disability and the realignment of friendships that many parents report are likely,
in part, to be due to this mismatch.
At the macro-level, parents are embedded in a set of cultural and often spiritual
beliefs. The broader culture in which families live and with which values are shared
is likely to play some part in the ways in which parents cope and their level of
coping with the increased demands associated with raising a child with a disability.
Belief systems about the perceptions of developmental disability are integral to
parents' views of the effect of their child with Down syndrome on their lives. The role of spirituality or religiosity in
the family is also often central to such perceptions.
Some cultures are more secular than others and different religious traditions view
disability and its meaning quite differently (e.g., ref 64).
Religion is not the only way in which cultures differ with respect to their responses
to and understandings of disability. An example of possible cultural effects on
coping processes comes from work by Blacher and McIntrye who found that Latina mothers
reported higher depression and lower morale.
However, Latina mothers also reported more positive perceptions of parenting and
a more positive impact of the child on their family. The authors speculated that
this association may be a product of differing attributions about the cause of the
child's behaviour from those of the comparison Anglo-American mothers. Lam and Mackenzie
discussed the highly competitive nature of Hong Kong society and the impact this
may have on parents of a child with Down syndrome in that milieu.
Magaña, Seltzer and Krauss found that family problems were more strongly associated
with depression for Puerto Rican mothers than for comparison mothers drawn from
non- Latina Anglo groups.
There is little cross cultural work or work within cultural psychology to guide
our understanding of what aspects of culture are supportive or undermining of family
functioning when there is a child with a disability in the family. The majority
of studies on families of children with Down syndrome are focused on only a subset
of families, largely those who are white and living in Western industrialised nations.
This gap limits our knowledge about the many ways in which parents adapt to a child
with Down syndrome and deserves a priority in setting an agenda for future research.
While early intervention is usually viewed as an intervention aimed at improving
child developmental outcomes, there is general recognition that it is part of a
developmental system and as such often
has important effects on other family members, particularly mothers. In a review
of studies of early intervention (parent training models) with a child with autism
McConachie and Diggle identified positive impact upon mothers as one of the outcomes. Pelchat, Bisson, Ricard, Perreault and
Bouchard found that parents of children with Down syndrome or cerebral palsy who
were involved in early intervention had more positive perceptions of their child
and of their parenting situation, had lower levels of distress and felt more supported. The benefits accruing from involvement with
early intervention are likely to reflect a number of processes including the mother's
perceptions of progress for her child, the development of self-confidence in her
skills for working with her child, and an increased sense of support as a result
of sharing her concerns with professionals (see ref 71) and other parents.
In an explicit acknowledgement of the role of early intervention in supporting families,
Bailey and colleagues identified five outcomes to aid in determining the effectiveness
of early intervention, all of which were aimed at the family functioning rather
than at child skill development:
"(a) families understand their child's strengths, abilities and special needs;
(b) families know their rights and advocate effectively for their child; (c) families
help their child develop and learn; (d) families have support systems; and (e) families
are able to gain access to desired services and activities in their community"[72:p.227].
Bailey, Scarborough, Hebbeler, Spiker and Mallik conducted a national study of early
intervention services in the U.S. and reported that parents had a very positive
view of the help provided to their family by early intervention services. Most families indicated that early intervention
had a significant positive impact on their family; 59% of those sampled stated that
their family was "much better" because of early intervention services.
Although this study was not based on a randomised design (due to ethical reasons),
it was large in both its scope and the diversity of families and early intervention
There is evidence that support by friends, relatives, and health care professionals
plays an important role in assisting mothers in their role as parents of a child
with a disability[74,75,76]. Van Riper found
that maternal well-being and mothers' perceptions of family functioning were associated
with their perceptions of the quality of the support they received from the professionals
working with them and their child with Down syndrome.
We have discussed the important role of perceived spousal support earlier, especially
for mothers, and will not repeat this here. Other family members, particularly grandparents,
play a central supporting role for many families.
We know little, however, about their needs with respect to support. In addition,
more work on understanding how best to provide support for fathers is clearly needed.
Economic resources contribute to individual and family well-being,
and employment is the major source of these resources for most families. Employment
contributes more than merely money to individuals, however. It also provides an
important avenue of connection to the community, as well as an arena to demonstrate
competence outside the parenting role.
There are a number of studies that have established that mothers of children with
a disability are less involved in employment outside the home than are other mothers
ref 79), although few studies have investigated this area specifically
with respect to mothers of a child with Down syndrome. In one of the studies to
do so, Hedov and colleagues found that this group of mothers were less engaged in
outside work than were mothers of a child without a disability.
Warfield reported that about two thirds of mothers of 5-year-old children with Down
syndrome, motor impairment or developmental delay were employed, and that employment
status did not vary with the child's type of disability, although more mothers of
children with Down syndrome were employed full time.
She further found that greater parenting demands related to greater absenteeism
at work but not to lower levels of work quality. In contrast, greater interest in
work and less work intensity were associated with lower parenting stress.
Thus, the relations among maternal employment, parenting demands, and workplace
expectations are complex, and the needs of working parents of children with Down
syndrome and other disabilities demand more investigation. The impact of differences
in employment is also not clearly established. Shearn and Todd have identified a
number of consequences for mothers including a curtailing of their ambitions. The role that employment outside the home
(or lack of it) in mothers' lives requires more focused attention.
There appears to be little research on direct interventions aimed at reducing stress
or increasing well-being in parents of a child with Down syndrome. In one report
Greaves compared an eight week group programme
teaching from Rational Emotive Therapy with
teaching related to Applied Behaviour Analysis and a no treatment comparison group.
All mothers reported a reduction of stress arising from their responsibilities for
the care and management of their child with Down syndrome over the course of the
intervention. While there were significant improvements for the Rational Emotive
Therapy group on some measures of stress immediately post training in contrast to
both comparison groups, no follow-up data were able to be collected.
There is a well established association between the use escape-avoidance coping
strategies and poorer outcomes for parents (for example ref 74) It seems
that a more sophisticated view of problem solving strategies may be helpful if research
is to provide assistance to families with a child with a disability. A closer examination
of the helpfulness of which strategies under which circumstances is likely to be
productive. There are some circumstances that cannot be changed and it may be less
debilitating for parents to avoid thinking about that particular problem and to
focus their problem solving on problems that are amenable to change. Both longitudinal
studies of the benefits of certain problem solving approaches and systematic intervention
studies are required to provide information to guide the practice of those providing
What do we still need to know?
The review above has identified a number of issues that require further research
if we are to have a thorough understanding of the impact of a child with Down syndrome
on families as a system and on the individuals who make up that system. These will
be briefly summarised here.
The nature of the association between phenotypic characteristics as displayed by
the child and the impact on parents needs to be investigated from the perspective
of parental adjustment, not solely from the perspective of the impact on those parenting
behaviours likely to influence child development. Consideration needs to be given
to parental expectations based on the stereotypic view of children with Down syndrome
as well as to the 'true' phenotypic characteristics.
There is an increase in the number of studies that include fathers; however, there
is much we do not know about the experiences of fathers in their parenting role.
These require further exploration if we are to understand what services and supports
would assist them to adjust to the circumstances of having a child with Down syndrome.
In addition, a deeper knowledge of their contributions to family functioning would
provide a more complete picture of the influences on other family members. It is
important that this information is collected from fathers themselves, rather than
using maternal views as a substitute. This will require some additional work on
the part of researchers as they look for ways to involve fathers in research.
Investigations of family life must take a balanced perspective and allow all aspects
to be acknowledged. This includes some concentration on the satisfactions of parenting
a child with Down syndrome (or of being a sibling or grandparent). Grandparents
are often an important resource for parents and as they are an integral part of
the family, they also should be included in research. We know very little about
how grandparents respond to having a grandchild with Down syndrome and how this
changes over time. There are a number of questions about the most useful ways in
which they can be involved in the family that is satisfactory for themselves and
helpful for the family with the child with Down syndrome, as well as questions about
how they might be better supported in their roles.
The demands associated with parenting a child with Down syndrome appear to increase
with age and it may be that the focus of research should be on these older age groups.
The contributors to this increase have not been established, although child behaviour
problems, child acquisition of skills and the parental relationship are potential
candidates. Families with a child with Down syndrome who also has other conditions
that increase the demands on families, such as autism or significant sensory disabilities,
need to be included in research if we are to have a complete understanding of the
implications for families. If we fail to do this, these families will be even more
marginalised, as they will not be represented in the usual picture of families of
a child Down syndrome presented through our work.
Cross cultural research on family adaptation to a child with Down syndrome, as well
as investigations that reveal cultural ideology in relation to family adaptation,
should be a priority. In these investigations the processes or mechanisms that underpin
differences should be the guiding question. Finally, the investigation of direct
therapeutic approaches to assisting parents, and other family members, deal effectively
with the additional demands associated with having a child with Down syndrome in
the family, should be undertaken.
Several issues emerge in consideration of the methodology employed in studies on
parental adaptation to a child with Down syndrome. As exemplified in this review,
most studies on parental adaptation to raising a child with developmental disabilities
are cross-sectional and comparative. The comparisons are made in relation to the
parents of typically developing children (e.g., ref 27) or to the parents of children
with other disabilities (e.g., ref 14). Such studies have yielded important
information about the comparative well-being of parents. Although many of the early
studies employed maternal reports as representative of the parent voice, current
trends involve the reports of both mothers and fathers (e.g., refs 22,
Comparative studies usually employ conventional analytic approaches, but the dependent
nature of the mother-father data requires more complex analyses. Mother-father pairs
are often influenced by each other's responses and behaviours. Models that incorporate
both individual level and dyadic level predictors (or correlates) of outcomes are
needed. Moreover, the outcomes themselves are not independent of each other and
are more accurately modelled as bi-directional. In a study on the contributions
of family resources, parenting challenges, work rewards, and work demands on the
parenting stress of both mothers and fathers in two-earner families in which a child
had a disability, Warfield addressed the critical issue of data dependency. She employed hierarchical linear modelling to account for the effects of mother
and father reports nested within couple-level data. The analytic approach used in
that study is likely to provide a more accurate approach to examining the mother-father
dyad and may serve as a model for future work.
Another methodological issue relates to the type of measures selected to investigate
parent adaptation. Parent report on scaled instruments has been the method of choice,
and Likert scales are the most common response format. Ease in data collection and
analysis is one of the advantages of such scales. Nevertheless, Likert scales have
been criticised for limiting a respondent's ability to accurately express opinions. If respondents have difficulty choosing between
two options, they are likely to leave an item blank, producing possible bias in
the set of recorded responses. The problems of missing data and dilemmas about ways
to address 'missingness' are considered by Schafer, and although the approaches
he suggests (e.g., imputation) are preferable to prior approaches (e.g., mean replacement),
researchers need to aim for as little missing data as possible.
Other than studies of mother-child interaction, other methodologies, such as observational
studies, are seldom used in investigations of parental adaptation. Observational
studies have been employed with other special populations, however. For example,
Jacobs and Fiese observed family interaction during mealtime using a well-developed
observational instrument to determine if families of children with asthma who were
overweight displayed distinct interactional patterns.
Observational studies may provide more nuanced information about parental adaptation
to a child with Down syndrome.
The field of psychology has been slow to respond to research designs that deviate
from conventional quantitative comparative approaches. Qualitative research studies,
though relatively few in number, have added a new dimension to the work on family
adaptation to a child with a disability. Drawing from an ecocultural framework which
emphasises the use of the 'activity setting' as the unit of analysis, Gallimore
and colleagues studied how parents adapt daily routines to accommodate their child
with a developmental disability[90,91].
Others, such as Harry, have used ethnographic approaches to elucidate ethnic and
cultural differences in views of normative development and disability.
Investigations using qualitative approaches may offer new and distinct ways of delineating
the ethno-theories that mothers and fathers themselves bring to the experience of
parenting a child with Down syndrome. Such ethno-theories are critical to a more
complete understanding of parent adaptation and may serve to assist in the development
of improved interventions to enhance parental functioning.
In addition, population studies deserve priority if we are to ensure that a complete
representation of family life is captured. Without this, the experiences of families
with a child who is functioning less well than his/her peers with Down syndrome
may be overlooked. Finally, longitudinal studies, although costly and difficult
to maintain, have an important place in the study of parent adaptation. Carr's study
serves as foundational work for descriptive information on children and parents
over time. More recent studies (e.g.,
have taken advantage of statistical advances that model change and test hypotheses
about predictors of change in both parents and children. The field is now well poised
to investigate more thoroughly the mediation and moderation of such trajectories.
Such information is critical to a more fully developed science on parent adaptation
to a child with Down syndrome.
- Minuchin PP. Families and individual development: provocations
from the field of family therapy. Child Development. 1985;56:289-302.
- Hodapp RM. Families of persons with Down syndrome: New
perspectives, findings, and research and service needs. Mental Retardation and Developmental
- Fidler DJ. The emergence of a syndrome-specific personality
profile in young children with Down syndrome. Down Syndrome Research and Practice.
2006;10(2):53-60. [ Open Access Full Text]
- Fidler DJ, Hepburn S, Rogers S. Early learning and adaptive
behaviour in toddlers with Down syndrome: Evidence of an emerging behavioural phenotype.
Down Syndrome Research and Practice. 2006;9(3):37-44. [Open
Access Full Text]
- Silverman W. Down syndrome: cognitive phenotype.
Mental Retardation and Developmental Disabilities Research Reviews. 2007;13:228-236.
- Scorgie K, Sobsey D. Transformational outcomes associated
with parenting children who have disabilities. Mental Retardation. 2000;38(3):195-206.
- Van Riper M. A change of plans: the birth of a child
with Down syndrome doesn't have to be a negative experience. American Journal of
- Van Riper M. Families of children with Down syndrome:
Responding to "a change of plans" with resilience. Journal of Pediatric
- Van Riper M, Ryff C, Pridham K. Parental and family
well-being in families of children with Down's syndrome: A comparative study. Research
in Nursing and Health. 1992;15:227-235.
- Hedov G, Annerén G, Wikblad K. Swedish parents of children
with Down syndrome: parental stress and sense of coherence in relation to employment
rate and time spent in child care. Scandinavian Journal of Caring Sciences.
- Scott BS, Atkinson L, Minton HL, Bowman T. Psychological
distress of parents of infants with Down syndrome. American Journal on Mental Retardation.
- Cuskelly M, Jobling A, Chant D, Bower A, Hayes A. Multiple
perspectives of family life. In: Cuskelly M, Jobling A, Buckley S, editors. Down
syndrome across the life span. London: Whurr; 2002: p. 159-173.
- Singer GHS. Meta-analysis of comparative studies of depression
in mothers of children with and without developmental disabilities. American Journal
on Mental Retardation. 2006;111:155-169.
- Abbeduto L, Seltzer MM, Shattuck P, Krauss MW, Orsmond
G, Murphy MM. Psychological well-being and coping in mothers of youths with autism,
Down syndrome, or fragile X syndrome. American Journal on Mental Retardation.
- Hodapp RM, Ricci LA, Ly TM, Fidler DJ. The effects of the
child with Down syndrome on maternal stress. British Journal of Developmental Psychology.
- Ricci LA, Hodapp RM. Fathers of children with Down's syndrome
versus other types of intellectual disability: Perceptions, stress and involvement.
Journal of Intellectual Disability Research. 2003;47:273-284.
- Shonkoff JP, Hauser-Cram P, Krauss M, Upshur CC. Development
of infants with disabilities and their families. Monographs of the Society for Research
in Child Development. 1992;57 (6, Serial No. 230).
- Pisula E. A comparative study of stress profiles in mothers
of children with autism and those of children with Down's syndrome. Journal of Applied
Research in Intellectual Disabilities. 2007;20(3);274-278.
- Sanders JL, Morgan SB. Family stress and adjustment as
perceived by parents of children with autism or Down syndrome: Implications for
interventions. Child and Family Behavior Therapy. 1997;19:15-32.
- Stoneman Z. Examining the Down syndrome advantage: Mothers
and fathers of young children with disabilities. Journal of Intellectual Disability
- Cuskelly M, Hayes A, Grace R. Disability: Characteristics,
contexts and consequences. In: J. Bowes J, Grace R, editors. Children, Families
and Communities. 3rd ed. Oxford University Press. In press 2007.
- Hauser-Cram P, Warfield ME, Shonkoff JP, Krauss MW.
Children with disabilities: A longitudinal study of child development and parent
well-being. Monographs of the Society for Research in Child Development.
2001;66 (3, Serial No. 266).
- Most DE, Fidler DJ, LaForce-Booth C, Kelly J. Stress trajectories
in mothers of young children with Down syndrome. Journal of Intellectual Disability
- Abidin R. Parenting stress index manual. Odessa,
Fl: Psychological Assessment Resources; 1995.
- Eisenhower AS, Baker BL, Blacher J. Preschool children
with intellectual disability: Syndrome specificity, behavior problems, and maternal
well-being. Journal of Intellectual Disability Research. 2005;49:657-671.
- Stores R, Stores G, Fellows B, Buckley S. Daytime behaviour
problems and maternal stress in children with Down's syndrome, their siblings, and
non-intellectually disabled and other intellectually disabled peers. Journal of
Intellectual Disability Research. 1998;42:228-237.
- Hedov G, Wikblad K, Annerén G. Sickness absence in Swedish
parents of children with Down syndrome: Relation to self-perceived health, stress
and sense of coherence. Journal of Intellectual Disability Research. 2006;50:546-552.
- Bower A, Cuskelly M, Jobling A. Fathers' experiences of
parenting a child with Down syndrome. Paper presented at the 1st Asia-Pacific Regional
Congress of International Association for the Scientific Study of Intellectual Disabilities.
Taipei, Taiwan, June 2005.
- Stoneman Z, Gavidia-Payne S. Marital adjustment in families
of young children with disabilities: Associations with daily hassles and problem-focussed
coping. American Journal on Mental Retardation. 2006;111:1-14.
- Krauss MW. Child-related and parenting stress: Similarities
and differences between mothers and fathers of children with disabilities. American
Journal on Mental Retardation. 1993;97:393-404.
- Keller D, Honig AS. Maternal and paternal stress in families
with school-aged children with disabilities. American Journal of Orthopsychiatry.
- Saloviita T, Itälinna M, Leinonen E. Explaining the
parental stress of fathers and mothers caring for a child with intellectual disability:
A Double ABCX model. Journal of Intellectual Disability Research. 2003;47:300-312.
- Johnston C, Mash EJ. A measure of parenting satisfaction
and efficacy. Journal of Clinical Child Psychology. 1989;18:167-175.
- Hassall R, Rose J, McDonald J. Parenting stress in mothers
of children with an intellectual disability: The effects of parental cognitions
in relation to child characteristics and family support. Journal of Intellectual
Disability Research. 2005;49:405-418.
- Gilmore L, Cuskelly M. Parenting satisfaction and self-efficacy
in mothers of children with Down syndrome: Early childhood to adolescence. Paper
presented in the symposium Parenting an Adolescent with Developmental Disabilities:
Do Developmental Principles Apply? Society for Research in Child Development Conference,
Boston (March, 2007).
- Bandura A. Regulation of cognitive processes through perceived
self-efficacy. Developmental Psychology. 1989;25:729-735.
- Laws G, Millward L. Predicting parents' satisfaction with
the education of their child with Down's syndrome. Educational Research.
- Shonkoff JP, Phillips D. From neurons to neighborhoods;
The science of early childhood development. Washington, DC: National Research
Council and Institute of Medicine; 2000.
- Brown RI, MacAdam-Crisp J, Wang M, Iarocci G. Family quality
of life when there is a child with a developmental disability. Journal of Policy
and Practice in Intellectual Disabilities. 2006;3(4):238-245.
- Risdal D, Singer GHS. Marital adjustment in parents of
children with disabilities: A historical review and meta-analysis. Research and
Practice for Persons with Severe Disabilities. 2004;29:95-103.
- Kersh J, Hedvat TT, Hauser-Cram P, Warfield ME. The contribution
of marital quality to the well-being of parents of children with developmental disabilities.
Journal of Intellectual Disability Research. 2006;50:883-893.
- Erickson M, Upshur CC. Caretaking burden and social support:
Comparisons of mothers of infants with and without disabilities. American Journal
on Mental Retardation. 1989;94(3):250-258.
- Simmerman S, Blacher J, Baker BL. Fathers' and mothers'
perceptions of father involvement in families with young children with a disability.
Journal of Intellectual and Developmental Disability. 2001;26:325-338.
- Cuskelly M, Dadds M. Behaviour problems in children with
Down's syndrome and their siblings. Journal of Child Psychology and Psychiatry.
- Van Riper M. Family variables associated with well-being
in siblings of children with Down syndrome. Journal of Family Nursing.
- Skotko B, Levine SP. What the other children are thinking:
brothers and sisters of persons with Down syndrome. American Journal of Medical
- Cuskelly M, Gunn P. Adjustment of children who have a
sibling with Down syndrome: Perspectives of mothers, fathers and children. Journal
of Intellectual Disability Research. 2006;50:917-925.
- Cuskelly M, Gunn P. Sibling relationships of children
with Down syndrome: Perspectives of mothers, fathers and siblings. American Journal
on Mental Retardation. 2003;108:234-244.
- Hannah ME, Midlarsky E. Helping by siblings of children
with mental retardation. American Journal on Mental Retardation. 2005;110:87-99.
- Hodapp RM, Glidden JM, Kaiser AP. Siblings of persons with
disabilities: Toward a research agenda. Mental Retardation. 2005;43:334-338.
- Stoneman Z. Siblings of children with disabilities: Research
themes. Mental Retardation. 2005;43:339-350.
- Van Riper M. The sibling experience of living with
childhood chronic illness and disability. Annual Review of Nursing Research.
- Poehlmann J, Clements M, Abbeduto L, Farsad V. Family
experiences associated with a child's diagnosis of fragile X or Down syndrome: Evidence
for disruption and resilience. Mental Retardation. 2005;43:255-267.
- Hastings RP, Taunt HM. Positive perceptions in families
of children with developmental disabilities. American Journal on Mental Retardation.2002;107:116-127.
- King GA, Zwaigenbaum L, King S, Baxter D, Rosenbaum P, Bates
S. A qualitative investigation of changes in the belief systems of families of children
with autism or Down syndrome. Child: Care, Health and Development. 2006;32:353-369.
- Hodapp RM, Ly TM, Fidler DJ, Ricci LA. Less stress, more
rewarding: Parenting children with Down syndrome. Parenting. 2001;1:317-337.
- Brasington CK. What I wish I knew then… reflections
from personal experiences in counseling about Down syndrome. Journal of Genetic
- Gilmore L, Campbell J, Cuskelly M. Developmental expectations,
personality stereotypes and attitudes towards inclusive education: Community and
teacher views of Down syndrome. International Journal of Disability, Development
and Education. 2003;50:67-78.
- Lawson KL. Expectation of the parenting experience and
willingness to consider selective termination for Down syndrome. Journal of Reproductive
and Infant Psychology. 2006;24:43-59.
- King LA, Scollon CK, Ramsey C, Williams T. Stories of life
transition: Subjective well-being and ego development in parents of children with
Down syndrome. Journal of Research in Personality. 2000;34:509-536.
- Van Riper M, Selder F. Parental responses to the birth
of a child with Down syndrome. Loss, Grief and Care: A Journal of Professional Practice.
- Garcia Coll C, Magnuson K. Cultural differences as sources
of developmental vulnerabilities and resources. In: Shonkoff JP, Meisels SJ, editors.
Handbook of early childhood intervention. 2nd ed. New York: Cambridge University
- Raghavan C, Weisner TS, Patel D. The adaptive project
of parenting: South Asian families with children with developmental delays. Education
and Training in Mental Retardation and Developmental Disabilities. 1999;34(3):281-292.
- Fadiman A. The spirit catches you and you fall down:
A Hmong child, her American doctors, and the collision of two cultures. NY:
Farrar, Straus and Giroux; 1997.
- Blacher J, McIntrye, LL. Syndrome specificity and behavioural
disorders in young adults with intellectual disability: Cultural differences in
family impact. Journal of Intellectual Disability Research. 2006;50:184-198.
- Lam LW, Mackenzie AE. Coping with a child with Down syndrome:
The experiences of mothers in Hong Kong. Qualitative Health Research. 2002;12:223-237.
- Magaña S, Seltzer MM, Krauss MW. Cultural context of caregiving:
Differences in depression between PuertoRican and Non-Latina White mothers of adults
with mental retardation. Mental Retardation. 2004;42:1-11.
- Guralnick MJ. The developmental systems approach
to early intervention. Baltimore, MD: Paul H. Brookes; 2005.
- McConachie H, Diggle T. Parent implemented early intervention
for young children with autism spectrum disorder: A systematic review. Journal of
Evaluation in Clinical Practice. 2007;13: 120-129.
- Pelchat D, Bosson J, Ricard N, Perreault M, Bouchard JM.
Longitudinal effects of an early family intervention programme on the adaptation
of parents of children with a disability. International Journal of Nursing Studies.
- Van Riper M. Maternal perceptions of family-provider
relationships and well-being in families of children with Down syndrome. Research
in Nursing and Health. 1999;22:357-368.
- Bailey DB, Bruder MB, Hebbler K, Carta J, Defosset M, Greenwood
C, Kahn L, Malli S, Markowitz J, Spiker D, Walker D, Barton L. Recommended outcomes
for families of young children with disabilities. Journal of Early Intervention.
- Bailey D, Scarborough A, Hebbeler K, Spiker D, Mallik S.
National Early Intervention Longitudinal Study: Family outcomes at the end of early
intervention. Menlo Park, CA: Stanford Research Institute; 2004.
- Feldman M, McDonald L, Serbin L, Stack D, Secco ML, Yu
CT. Predictors of depressive symptoms in primary caregivers of young children with
or at risk for developmental delay. Journal of Intellectual Disability Research.2007;51:609-619.
- Skotko B. Mothers of children with Down syndrome reflect
on their postnatal support. Pediatrics. 2005;115:64-77.
- Skotko B, Bedia CB. Postnatal support for mothers of children
with Down syndrome. Mental Retardation. 2005;43:196-212.
- Drew LM, Richard MH, Smith PK. Grandparenting and its relationship
to parenting. Clinical Child Psychology and Psychiatry. 1998;3:465-480.
- Eisenhower A, Blacher J. Mothers of young adults with
intellectual disability: multiple roles, ethnicity and well-being. Journal of Intellectual
Disability Research. 2006;50:905-916.
- Gordon M, Rosenman L, Cuskelly M. Maternal employment:
A comparative study of paid work levels and the desire to work among mothers with
and without dependent children with disabilities. Journal of Applied Research on
Intellectual Disabilities. 2007;20:236-246.
- Warfield ME. Employment, parenting, and well-being among
mothers of children with disabilities. Mental Retardation. 2001;39:297-309.
- Shearn J, Todd S. Maternal employment and family responsibilities:
The perspectives of mothers of children with intellectual disabilities. Journal
of Applied Research in Intellectual Disabilities. 2000;13:109-131.
- Greaves D. The effect of Rational-Emotive Parent Education
on the stress of mothers of young children with Down syndrome. Journal of Rational-Emotive
and Cognitive-Behavior Therapy. 1997;15:249-267.
- Ellis A. Reason and emotion in psychotherapy: Revised
and updated. New York: Carol Publishing Group;1994.
- Roach MA, Orsmond GI, Barratt M. Mothers and fathers of
children with Down syndrome: Parental stress and involvement in child care. American
Journal on Mental Retardation. 1999;104:422-436.
- Warfield ME. Family and work predictors of parenting
role stress among two-earner families of children with disabilities. Infant and
Child Development. 2005;14:155-176.
- Raudenbush SW, Bryk A. Hierarchical linear models:
Applications and data analysis methods. 2nd ed. Thousand Oaks, CA: Sage; 2002.
- Flynn D, van Schaik P, van Wersch A. A comparison of multi-item
Likert and visual analogue scales for the assessment of transactionally defined
coping function. European Journal of Psychological Assessment. 2004;20:49-58.
- Schafer JL. Analysis of incomplete multivariate data.
NY: Chapman and Hall; 1997.
- Jacobs MP, Fiese BH. Family mealtime interactions
and overweight children with asthma: Potential for compounded risks? Journal of
Pediatric Psychology. 2007;32:64-68.
- Gallimore R, Coots J, Weisner T, Garnier H, Guthrie
D. Family responses to children with early developmental delays II: Accommodation,
intensity, activity in early and middle childhood. American Journal on Mental Retardation.
- Gallimore R, Weisner TS, Bernheimer LP, Guthrie, D,
Nihira K. Family responses to young children with developmental delays: Accommodation
activity in ecological and cultural context. American Journal of Mental Retardation.
- Harry B. Trends and issues in serving culturally diverse
families of children with disabilities. The Journal of Special Education.
- Carr J. Down's syndrome: Children growing up. Cambridge,
UK: Cambridge University Press; 1995.
Monica Cuskelly is at the School of Education, University of Queensland, Australia;
Penny Hauser-Cram is at Boston College, Lynch School of Education, USA; Marcia Van
Riper is at the University of North Carolina at Chapel Hill School of Nursing, Carolina
Center for Genome Sciences, USA.
Correspondence to Marcia Van Riper • e-mail:
Paper prepared from presentations and discussions at the Down Syndrome Research
Directions Symposium 2007, Portsmouth, UK. The symposium was hosted by Down Syndrome
Education International in association with the Anna and John J Sie Foundation,
Denver. Major sponsors also included the Down Syndrome Foundation of Orange County,
California and the National Down Syndrome Society of the USA. Information about
the symposium can be found at
Received: 15 February 2008; Accepted: 22 February 2008; Published online: 4 July
See and Learn Speech is designed to help parents and educators support children with Down syndrome to develop clearer speech.
See and Learn Speech offers a structured approach to support speech development, working in small steps towards clearer speech production.
Now available as teaching kits and apps.
Find out more...