MENTAL RETARDATION AND DEVELOPMENTAL DISABILITIES
RESEARCH REVIEWS 13: 293 – 301 (2007)
RECOGNIZING THE ROLE OF PARENTS IN
DEVELOPMENTAL OUTCOMES: A SYSTEMS
APPROACH TO EVALUATING THE CHILD WITH
DEVELOPMENTAL DISABILITIES
Lara S. Head* and Leonard Abbeduto
Waisman Center, University of Wisconsin-Madison, Madison, Wisconsin
When developmental concerns arise in children, a clinical assessment focuses on the child’s developmental profile to achieve three goals
(1) determine diagnosis, (2) develop interventions, and (3) evaluate progress. Parents often have needs during this time that are not addressed
by professionals because of the exclusive focus on the child during the
evaluation. In this article, we suggest that clinicians take a ‘‘systems’’
approach to the assessment process by recognizing how the well being
of family members can impact a child with a developmental disability.
We review systems theory and its conceptualization of individual functioning and discuss how parental well-being differs according to child diagnosis. Finally we suggest a systems-based model to use during the
' 2007 Wiley-Liss, Inc.
assessment process.
MRDD Research Reviews 2007;13:293–301.
Key Words: assessment; systems; families
L
anguage delays, problems in gross motor development,
difficulties in participating in reciprocal social interaction, significant behavioral challenges, the possibility of
an autism diagnosis—these and other concerns about their
children’s development are among the reasons that parents seek
professional help [Simeonsson and Scarborough, 2001]. When
such concerns arise, parents often turn to their pediatricians to
provide initial guidance in establishing an explanation or
obtaining confirmation of their child’s delays. However, a
more comprehensive clinical assessment, often conducted by a
multidisciplinary team (e.g., clinical psychologist, speech-language pathologist, occupational therapist), is typically necessary
to address questions about the origins of existing delays, their
nature and extent, treatment options, responses to treatment,
and anticipated outcomes for the child [Simeonsson and Scarborough, 2001]. In this article, we make recommendations for
further broadening of this assessment process.
For many parents, reactions to initial suspicions and then
confirmation of an existing developmental disability are similar
to those reactions experienced in response to a trauma or crisis
[Sanders and Morgan, 1997; Seligman and Darling, 1997]. In
some cases, elevated levels of psychological stress lead to
marked and sustained impairments in functioning among
parents and other members of the family. In contrast, some
' 2007 Wiley -Liss, Inc.
families deal effectively and even thrive in the face of the challenges associated with a child’s developmental disability. The
factors that lead to these differences in family response are not
fully known [Hassall and Rose, 2005]. Family responses, however, can play an important role in their ability to implement
recommended treatments for the child, to seek out additional
resources for the child, and generally to support the child’s
development.
It can be difficult for professionals to fully appreciate
and address the needs of parents and other family members
because the assessment process is traditionally child focused.
Current assessment practices often limit a clinician’s or evaluation team’s ability to expand their clinical evaluation services
beyond establishing a diagnosis for the child and providing a
set of recommendations for treatment and care, again, focused
on the child [Greenspan and Meisels, 1996]. Large case loads
and lengthy waiting lists have become the norm as increasing
numbers of children are identified as needing evaluations to
investigate developmental concerns. Add to this the financial
and staffing restrictions in many healthcare environments, and
it can be difficult for clinicians and multidisciplinary evaluation
teams for envision and expansion of the assessment process
[Greenspan and Meisels, 1996].
We believe, however, that, despite these challenges,
clinicians and evaluation teams should take a systems approach
to the assessment process and thereby acknowledge that the
well-being and experiences of other family members can
impact a child with a developmental disability. In the systems
approach, the ‘‘patient’’ is no longer simply the child with suspected developmental delays, but his or her family system and
its individual members, including parents and siblings, each of
Grant sponsor: National Institute of Child Health and Human Development; Grant
numbers: T32 HD007489, R03 HD048884, P30 HD003352.
*Correspondence to: Lara S. Head, 533 Waisman Center, University of WisconsinMadison, 1500 Highland Ave., Madison, WI 53705.
E-mail: [email protected]
Received 20 August 2007; Accepted 21 August 2007
Published online in Wiley InterScience (www.interscience.wiley.com).
DOI: 10.1002/mrdd.20169
whom presents with unique needs and
challenges to be assessed, interpreted,
and ultimately treated.
In this article, we describe systems
theory, how parental well-being differs
according to child diagnosis, and a
model for clinicians to use in applying a
systems approach to the clinical assessment process. Because of space constraints, we limit our discussion largely
to parents, but it is important to recognize that siblings and other extended
family members (e.g., grandparents) also
are affected by, and can affect the child
with a disability, and thus they too
should be considered in a systems-based
assessment.
SYSTEMS THEORY
Until relatively recently, relationships among family members have been
viewed as comprised of associations
between two individuals, such as
mother and child or husband and wife,
independent of any other relationships
within the family [Seligman and Darling, 1997]. However, family systems
theory asserts that relationships among
family members are interdependent and
that the family is more than the sum of
its individual members. Individuals
assert influence on other family members while being influenced by other
family members and the contexts in
which interactions among all family
members are occurring [Minuchin,
1974]. Relationships are thus described
as a ‘‘system’’ [Minuchin, 1974], operating so that all members are affected in
some way by the experiences of any
one individual in the system, in much
the way that a machine’s function
‘‘emerges’’ from the mutually dependent
actions of its various parts.
Martin Seligman, Ann Turnbull,
and others have argued that families
with a child who has a disability can be
best understood through family systems
theory application [Turnbull et al.,
1986; Turnbull and Turnbull, 1990;
Seligman and Darling, 1997]. However,
little empirical research has been conducted on families having a child with a
developmental disability within a systems framework. Further, there have
been few studies focused on the functioning of family systems according to
the nature of the child’s diagnosis
[Turnbull and Turnbull, 1990; Patterson
and Garwick, 1994].
Translating a systems approach
into clinical practice requires incorporating ‘‘family-centered’’ policies and
practices into healthcare services
throughout the diagnostic and treatment
294
process [Johnson, 2000]. A systemsbased approach to assessment considers
the family within a context of normality, while still acknowledging the structural and functional characteristics that
create differences in experiences and
well-being among families [Seligman
and Darling, 1997]. Seligman and Turnbull’s work has emphasized the need to
develop family-centered support and
delivery services that recognize the parent and other family members as both
partners in treatment provision for the
child with a disability and as benefactors
of services.
THE IMPACT OF PARENTAL
WELL-BEING ON CHILD
DEVELOPMENT
When parents establish a supportive, caring, and positive family environment, developmental outcomes for children with disabilities are optimized
Translating a systems
approach into practice
requires incorporating
‘‘family-centered’’ policies
and practices into
healthcare services
throughout the diagnostic
and treatment process.
[Mink et al., 1983; Mink and Nihira,
1986; Hauser-Cram et al., 2001]. In a
series of studies examining the role of
the home environment on the development of children with disabilities, Mink
et al. [1983] focused on 115 families of
children with mental retardation, 48%
of whom were diagnosed with Down’s
syndrome. Results indicated that children raised in families that were cohesive and harmonious or child-oriented
and expressive in nature demonstrated
higher levels of adaptive behavior, fewer
behavior problems, and experienced less
peer isolation compared to families
lower in these qualities.
Mink and Nihira [1986] extended
these findings to the prediction of a
child’s later social and emotional development and functioning. Using a sample of 218 children identified as having
mild to moderate intellectual disabilities
and their families, these investigators
found that higher levels of family cohe-
MRDD Research Reviews DOI 10.1002/mrdd
sion or harmony along with high levels
of parent involvement resulted in children subsequently demonstrating higher
levels of independent community functioning and increased understanding of
their role and responsibility in social situations [Mink and Nihira, 1986].
In a study examining the relationship between home environment and
child functioning utilizing data samples
from the Early Intervention Collaborative Study database [Hauser-Cram et al.,
2001], increased levels of family cohesion reported at age 3 predicted fewer
behavioral problems in children with
disability when measured at age 5 [Warfield, 1995]. Similarly, preschool children raised in environments with higher
levels of family cohesion were also
found to demonstrate improved social
interactions with peers [Hauser-Cram
et al., 1999].
These studies demonstrate that
positive, cohesive, engaging family relationships can lead to better family functioning and positive development in
children with developmental disabilities.
When parental functioning is compromised, the negative effects can be seen
within the family system as changes in
parenting approach and the emergence
of interactions that are increasingly negative or disengaged [Mink and Nihira,
1986; Hauser-Cram et al., 2001]. Already compromised due to medical,
congenital, and developmental difficulties, the child with a developmental disability is particularly vulnerable to the
effects of a less than optimal family
environment. Therefore, identifying the
needs of parents and families and determining how those needs can be met
should be an important goal for professionals [Seligman and Darling, 1997].
FACTORS AFFECTING
PARENTAL WELL-BEING
Parents’ ability to process diagnostic information, seek out resources, participate in their child’s course of treatment, and support the child’s development can be compromised if they
experience stress or mental health challenges of their own as a result of the
child’s disability. In fact, many parents
with a child with disabilities report
higher levels of stress and perform
poorly on measures of individual emotional well-being and family functioning
compared to families raising a typically
developing child [Olsson and Hwang,
2002; Baker et al., 2005; Singer, 2006],
although some report adjusting well to
caregiving responsibilities and having
positive outcomes or benefits [Taunt
Systems Approach to Evaluation
Head and Abbeduto
and Hastings, 2002]. The factors
explaining why some individuals and
families fare better than others, include
parental gender, coping style, and parental genotype in explaining response
differentiation [Hassall and Rose, 2005].
The differences between mothers
and fathers and their experiences in
raising a child with a developmental
disability have been reasonably welldocumented across different child disabilities, with those differences being
grounded in gender roles, particularly
in relation to caregiving responsibilities
[Seltzer et al., 2001; Hassall and Rose,
2005; Herring et al., 2006]. In comparison to fathers, mothers of children
with disabilities typically report higher
levels of stress [Herring et al., 2006],
higher levels of depression [Olsson and
Hwang, 2001], and increased caregiving
responsibilities [Simmerman et al.,
2001]. Some studies, however, have
demonstrated that parental well-being is
similar for both mothers and fathers
[Heller et al., 1997; Hastings, 2003;
Keller and Honig, 2004].
Parental coping style is also a determinant of differences in parental psychological well-being [Glidden et al.,
2006]. Parents who use a problemfocused coping approach, defined by
the use of cognitive and behavioral
problem-solving strategies and the seeking of social support to address stressful
issues, display relatively high levels of
emotional well-being [Essex et al.,
1999; Abbeduto et al., 2004]. Parents
who employ a problem-focused coping
style demonstrate lower levels of depression, less pessimism and increased positive affect in comparison to reliance on
an emotion-focused coping style, which
is characterized by the use of strategies
to manage some form of psychological
distress through avoidance, denial, or
escape [Abbeduto et al., 2004].
Some parents and other family
members may be at risk for lower levels
of well-being by virtue of their genotypes. In light of the genetic bases of
autism, for example, other family members can display the ‘‘broader autism
phenotype,’’ which includes language
impairment, limited social reciprocity,
difficulties establishing relationships, behavioral rigidity, and the presence of
obsessive and compulsive behaviors
[Piven et al., 1997; Piven, 1999]. Similarly, biological mothers of children
diagnosed with fragile X syndrome,
who are carriers of either the full mutation or premutation of the FMR1 gene,
are at elevated risk for executive functioning difficulties, higher rates of
depression and anxiety, and social interaction difficulties, as well for physical
health problems, such as premature
ovarian failure [Sobesky et al., 1994;
Franke et al., 1996]. In each of these
conditions, genetic variations may contribute to differences in parental wellbeing, which may in turn prevent
parents from implementing preferred
adaptation strategies compared to
parents without such predisposing
genetic variations [Piven et al., 1997;
Abbeduto et al., 2004].
Findings regarding the lower levels of well-being, on average, for
parents of children with disabilities and
the factors responsible for individual
variation in well-being illustrate the
need for clinicians to recognize the
ongoing experience of adaptation and
how a systems approach to evaluation
considers the changing needs of the
family in this process [Seligman and
Darling, 1997].
and Morgan, 1997; Herring et al.,
2006], increased levels of pessimism
regarding their child’s future [Abbeduto
et al., 2004], and increased levels of
depression in comparison to parents
raising children with other types of
neurodevelopmental disorders [Olson
and Hwang, 2001; Hastings et al.,
2005]. Mothers report higher levels of
distress than fathers [Hastings, 2003;
Hastings et al., 2005]. Although fathers
consistently report lower levels of stress
in comparison to mothers of children
with autism [Herring et al., 2006], corresponding father reports of stress
remain significantly higher in comparison to other disability groups [Hastings
et al., 2005]. Hastings [2003] further
indicated that the greater caregiving
responsibilities assumed by mothers,
coupled with traditional household
activities and the somewhat poorer
mental health outcomes of fathers, is responsible for the higher levels of stress
and symptoms of anxiety in mothers.
The child’s behavioral difficulties are
also a critical factor in predicting parent
mental health in this group [Baker
et al., 2002; Hastings et al., 2005].
Children with autism typically display
higher levels of behavioral difficulties in
comparison to children with other disability conditions [Herring et al., 2006],
with mothers being particularly vulnerable to child behavioral difficulties
[Hastings, 2003].
PARENTAL WELL-BEING AND
SYNDROME-RELATED
VARIATION
Considerable research has demonstrated that disabilities of differing etiology often vary in their profiles of
behavioral/psychological strengths and
weaknesses, as well as in the probability
of comorbid psychiatric conditions
[Hodapp and Dykens, 2001]. These
child behavioral phenotypes may tax
parents and families to different degrees
and in different ways, resulting in condition-related differences in mental
health and well-being outcomes in
parents and other family members
[Seligman and Darling, 1997; Hassall
and Rose, 2005]. To illustrate, we present four different conditions each posing unique challenges to the adaptation
process for parents and to assessment for
professionals.
Down Syndrome
Down syndrome is the most frequently occurring chromosomal disorder, occurring in 1/800 to 1,000
births, with 5,100 children born each
year in the U.S. with Down syndrome
[Canfield et al., 2006]. Most cases
(95%) arise from nondisjunction during
meiosis resulting in three copies of all
or part of chromosome [Gardiner and
Costa, 2006]. Not surprisingly, therefore, the syndrome is associated with
abnormalities in multiple organ systems
and a characteristic phenotype, which
includes physical as well as behavioral
features [Roubertoux and Kerdelhue,
2006]. The behavioral phenotype
includes impairments in cognitive ability and especially serious delays in language development [Abbeduto et al., in
press]. In general, however, individuals
with Down syndrome are highly sociable and display low rates of challenging
behaviors, such as aggression and hyperactivity, compared to other neurodevelopmental disorders [Antonarakis and
Epstein, 2006].
Autism
Autism is a neurodevelopmental
disorder characterized by qualitative
impairments in communication and
social reciprocity and the presence of
restricted and perseverative behavioral
features [Piven, 1999]. A comprehensive
longitudinal study conducted by the
U.S. Centers for Disease Control and
Prevention suggests 1 in 150 children in
the United States have an autism spectrum disorder, including autistic disorder, pervasive developmental disorder
not otherwise specified, and Asperger
syndrome [Kuehn, 2007].
Parents of children with autism
report higher levels of stress [Sanders
MRDD Research Reviews DOI 10.1002/mrdd
Systems Approach to Evaluation
Head and Abbeduto
295
Parents of children with Down
syndrome have typically been characterized by higher levels of adaptive parental and familial functioning in comparison to other disability groups, particularly autism [Stoneman, 1997]. This
‘‘Down syndrome advantage’’ has been
described as including lower levels of
stress and higher levels of positive
impact or rewarding uniquely attributed
to the experience of raising a child with
Down syndrome [Hodapp et al., 2001,
2003; Sellinger and Hodapp, 2005].
The sociability and low rates of challenging behaviors in children with
Down syndrome as compared to children with other disabilities have been
cited as causal features of the advantage
[Hodapp et al., 2003].
More recent evidence, however,
suggests that the Down syndrome
advantage may attenuate somewhat over
time for parents [Hodapp et al., 2001,
2003]. In a study examining the relationship between maternal well-being
and syndrome-specific behavior among
preschool children with Down syndrome, autism, and cerebral palsy, findings suggested that mothers of children
with Down syndrome, on average,
reported lower levels of stress, potentially attributed to fewer behavioral
problems at the preschool age [Eisenhower et al., 2005]. However, mothers
of children with Down syndrome
reported increased maternal stress and
behavioral problems in their children
over time [Eisenhower et al., 2005].
Similar findings were demonstrated in a
study examining maternal stress trajectories over three different time points at
12–15 months, 30 months, and 45
months of age [Most et al., 2006].
Fragile X Syndrome
Fragile X syndrome is an Xlinked disorder caused by an expansion
of repetitive sequence of nucleotides in
the FMR1 (Fragile X Mental Retardation 1) gene. The syndrome is the most
common inherited cause of mental retardation [Sherman, 1996], with an estimated prevalence of 1 in 4,000 males
and 1 in 8,000 females have the syndrome [Reiss and Dant, 2003]. Virtually
all males with syndrome meet diagnostic
criteria for intellectual disability [Loesch
et al., 2002]. Females are less intellectually impaired than males, with about
half of females having an intellectual
disability and the remainder having normal range IQs, but often with learning
disabilities, especially in math [Loesch
et al., 2002]. The phenotype also
includes hypersensitivity to a variety of
296
sensory stimuli, hyperactivity, attentional
problems, impairments in executive
function, and social anxiety [Freund
et al., 1992; Hagerman, 1999; Reiss
and Dant, 2003]. Social problems are
particularly severe and include gaze
avoidance and excessive delays in the
pragmatic dimension of language
[Abbeduto et al., 2007]. There is also a
high-rate of comorbidity between fragile X syndrome and autism, with
10–40% (with a consensus being
about 25%) of individuals with the syndrome warranting an autism diagnosis
[Feinstein and Reiss, 2001; Rogers
et al., 2001; Demark et al., 2003].
The mothers of individuals with
fragile X syndrome appear to be at risk
for poor psychological outcomes. These
The systems approach
would . . . enable
clinicians to consider
intervention options
against the broader
context of the family’s
needs and expectations . . .
understanding the
variability among parents
and families with regard
to their psychosocial
circumstances is an
important step in this
process.
mothers report higher levels of parenting stress and lower levels of psychological well-being than normative samples
[Franke et al., 1996, 1998; Sarimski,
1997; van Lieshout et al., 1998; von
Gontard et al., 2002; Johnston et al.,
2003]. Mood disorders, especially depression and anxiety disorders, are frequent among these mothers [Freund
et al., 1992; Sobesky et al., 1994;
Franke et al., 1998; Abbeduto et al.,
2004]. Mothers of individuals with fragile X syndrome also fare more poorly
than mothers of adolescents and adults
with several other disabilities [Sarimski,
1997; van Lieshout et al., 1998; von
Gontard et al., 2002; Abbeduto et al.,
2004]. For example, mothers of adolescents and young adults with fragile X
MRDD Research Reviews DOI 10.1002/mrdd
syndrome report more symptoms of
depression, greater pessimism about the
child’s future, and feel less reciprocated
closeness from their children than do
mothers of matched individuals with
DS [Abbeduto et al., 2004; Lewis et al.,
2006].
There is considerable variation in
well-being, however, among mothers of
individuals with fragile X syndrome.
Much of this variability is related to differences in the behavioral challenges
displayed by the child with fragile X
syndrome, with more extensive autism
symptoms being associated with greater
risk to maternal well-being [Abbeduto
et al., 2004; Lewis et al., 2006]. Contextual factors, including the number of
affected children in the family, also
influence the well-being of mothers of
individuals with fragile X syndrome.
Maternal genotype may also contribute,
however, as there is evidence that some
women who carry the FMR1 premutation (i.e., an expansion of the FMR1
gene beyond normal but smaller than
needed to cause the syndrome) may at
risk for poorer mental health outcomes
independent of having an affected child
[Franke et al., 1996; Johnston et al.,
2001; Hessl et al., 2005].
Multiple Disabilities
Families raising a child with multiple disabilities involving physical limitations potentially in addition to cognitive and behavioral impairments, such as
cerebral palsy, spina bifida, or other
neuromuscular disorders, may also experience higher stress levels than families
raising children with typical development. Among families with a child with
spina bifida, for example, increased psychosocial difficulties and family distress
have been reported [Holmbeck et al.,
2003; Greenley et al., 2006]. Further,
increased stress levels in individual
members of families that include a child
with spina bifida are associated with less
cohesive, less engaging patterns of interaction among family members, including between parents and their child
with spina bifida [Holmbeck et al.,
2002; Greenley et al., 2006]. In contrast
to the case for autism and fragile X syndrome, there is not a genetic basis for
spina bifida [Greenley et al., 2006].
Summary
Each of these four neurodevelopmental is associated with a different
child phenotype, a different level of
parent vulnerability to stress and mental
health challenges and thus, a very different adaptation experience for families.
Systems Approach to Evaluation
Head and Abbeduto
Although there is considerable variability even within a disorder, clinicians
working within a systems approach to
assessment can nevertheless use knowledge of the child’s diagnosis to guide
the assessment process. They can probe
those dimensions of child functioning
that are part of the disorder’s phenotype
and that have special relevance to parental adaptation. At the same time,
they can be sure to assess those dimensions of parental well-being and perceptions that are most likely to be problematic for the disorder and consider
the ways in which those dimensions
might affect reactions to the diagnosis
or to planned treatments and follow up
for the child. Moreover, because the
phenotype of each disorder emerges and
changes with age, a systems approach
would require that clinicians help families cope with such changes.
A MODEL FOR CLINICAL
PRACTICE
The diagnostic evaluation process
is an opportunity to evaluate a child’s
developmental progress and determine
what interventions and resources might
be useful in facilitating and supporting
his or her continued growth. A systems
approach would address this goal by
broadening the concepts of assessment,
interpretation of findings, and development of treatment recommendations
[Seligman and Darling, 1997; Rosenthal
et al., 2001; Hassall and Rose, 2005].
The systems approach would expand
what has traditionally been regarded as
a child-focused practice and enable
clinicians to consider intervention
options against the broader context of
the family’s needs and expectations
[Seligman and Darling, 1997; Rosenthal
et al., 2001]. Further, understanding the
variability among parents and families
with regard to their psychosocial circumstances is an important step in this
process. Clinicians would maximize a
family’s ability to implement interventions in support of the child’s development if they approach the diagnostic
evaluation experience with greater sensitivity to each family’s circumstances
and their ability to ‘‘digest’’ and implement evaluation results and recommendations, which would minimally require
an appreciation for the current psychological circumstances and well-being of
the parents and other family members
[Patterson and Garwick, 1994; Rosenthal et al., 2001].
In the following sections, we
briefly sketch a systems model of clinical practice. By integrating information
gathered through clinical interview,
existing records, self-report questionnaires, observational tasks, and diagnostic testing, clinicians can develop an
assessment protocol that ebbs and flows
with the needs of the family and their
needs as professionals in implementing a
systems approach. Clinicians must be
flexible and willing to use existing
materials while still recognizing the
need to probe further and ask additional
questions and consider other issues that
will assist in establishing rapport, but
most importantly, facilitate information
gathering and create a comprehensive
picture of the family’s experience.
tioning [Greenspan and Meisels, 1996;
Sattler, 2002].
Individual psychological well-being
The assessment of families should
be designed to allow the clinician to
evaluate parents and other family members along the dimensions of stress, coping, and other variables reflective of
psychological well-being, such as depression and anxiety. The format of the
clinical interview allows parents a voice
in describing how they feel in response
to the challenges of raising a child with
a developmental disability and is an opportunity to share how those challenges
are experienced within the context
of their everyday lives [Seligman and
Darling, 1997].
Interview questions should also
address parental perceptions about the
role children assume within the family
and feelings of acceptance towards the
child with developmental concerns.
Assessing parental perceptions of competency to execute parental tasks, particularly as they pertain to caregiving of
the child with developmental concerns
and to balancing the needs of other
family members, also provides insights
into the family climate. Examining how
parents view the child in relation to
expectations for their growth, development, parental caregiving, and the
child’s participation within the family
can also provide a useful context for
understanding parent and family adaptation and parental locus of control.
Self-report measures are also useful as a supplement to the clinical interview and provide quantitative measures
of many important dimensions of parental well-being to better understand
the parent’s current psychological state,
response to the child with the disabilities, and the degree to which necessary
supports are in place. Although not a
complete list of those used by clinicians,
Table 1 includes a sampling of selfreport measures that are available to
assess individual psychological wellbeing, parenting perceptions, and functioning within the family context.
Assessment
Evaluating the family context
The clinical interview is an excellent tool for clinicians to examine the
attitudes, expectations, personal traits,
levels of stress and emotional wellbeing, and coping mechanisms of
parents and siblings, individually and as
they pertain to the greater family unit
[Patterson and Garwick, 1994; Rosenthal et al., 2001]. Although the assessment process traditionally begins only
after the child has been born and demonstrated developmental delays, prenatal
diagnosis is now possible and allows
clinicians to identify and assess the
needs of families during pregnancy
rather than waiting until the child has
been born to begin providing support
services to assist families in the adaptation process. In any event, establishing a
relationship with parents that is sensitive
to their experiences and to the challenges they have encountered prior to
the evaluation appointment is important. Doing so ensures that parents feel
comfortable within the interview and
are willing to respond openly to questions that are individual and familyfocused [Berman and Shaw, 1996;
Rosenthal et al., 2001].
Developmental and background
history forms are often completed by
parents prior to a child’s evaluation
appointment and used to inform clinicians of a child’s development, medical
and educational history, psychosocial
experiences, and description of behavioral problems. Self-report questionnaires
assessing important facets of parent functioning may also be administered prior to
the appointment. If reviewed during a
clinical interview, this material can be an
avenue for rapport building between
parents and clinicians, creating the opportunity for increasingly focused questions about individual and family func-
MRDD Research Reviews DOI 10.1002/mrdd
Systems Approach to Evaluation
Family environment
The assessment should also address
the family’s environment and the nature
of family interactions. Obtaining information from family members during
the clinical interview that describes
how warm and supportive they feel
towards each other will help clinicians
understand the types of support that are
available for individuals within the family unit and the extent to which interventions designed to change patterns of
Head and Abbeduto
297
Table 1. Sample Self-Report Assessment Measures of Individual
and Family Contexts
Domain
Measure
Individual functioning
Beck Depression Inventory II [Beck et al., 1996]
Beck Anxiety Inventory [Beck and Steer, 1993]
Parenting Stress Index [Abdin, 1995]
Questionnaire on Resources and
Stress—Short Form [Friedrich et al., 1983]
Multidimensional Coping Inventory [Carver et al., 1989]
Parenting Locus of Control Scale [Campis et al., 1986]
Family Environment Scale [Moos and Moos, 1994]
Family Impact Questionnaire
[Donenberg and Baker, 1993]
Family Adapatibility & Cohesion Evaluation Scale II
[Olson et al., 1982]
HOME Inventory [Bradley, 1994]
Dyadic Adjustment Scale [Spanier, 1989]
Positive Affect Index [Bengtson and Black, 1973]
Stress
Coping
Locus of control
Family context
Marital relationship
Positive engagement
interaction might be necessary [Baer,
2002]. Understanding family members’
perceptions of support availability can
also be informative as clinicians begin
to consider possible interventions following the evaluation. Questions or
measures that highlight quantitative and
qualitative aspects of communication
among family members are useful to
determine how often parents and children interact and if such interactions are
positive, negative, or neutral and are reflective of a warm and receptive family
environment. Clinicians should attempt
to discern patterns of interaction, such
as differences in frequency and quality
of verbal interactions between mother
and child, father and child, and among
all family members [Summers et al., 2005].
Medical records, developmental
history questionnaires, and school
records are additional resources to consider beyond the clinical interview.
Parent–child and family–child observational interaction tasks, such as observing parents and child in play or engaged
in a structured conversation, or other
planned activity, these can also be
incorporated into the assessment process. These types of tasks allow for qualitative assessment of specific interactional features and patterns through the
coding of observed behavior in the
context of the task. Finally, parental
self-report measures can systematically
document perceptions on a variety of
different domains using a standardized
format for the purposes of evaluating
parent and family functioning against a
normative reference group.
Interpretation of Findings
Having gathered information
about parent and family functioning,
298
the clinician using a systems approach
can now consider how the child’s diagnosis and even more importantly, his
or her phenotypic behavioral profile, is
eliciting particular responses from
parents and other family members.
This approach examines how the
child’s diagnosis and phenotypic behavioral profile is affecting individual
and family well-being and functioning
and how the various processes are
shaped by other contextual variables,
such as the family’s socioeconomic status and resource availability and access.
By integrating the information collected, the clinician can begin to
anticipate how the family might
respond to being told the diagnosis
and the supports that need to be established, including interventions targeting parents and other family members,
to ensure that the family can follow
recommendations for follow up or
treatment.
Adapting to a child with a disability is often a slow and, perhaps, even
life-long process, and clinicians have an
opportunity through assessment to evaluate where the family is in that process,
what their needs are, and how to
address those needs. For example, clinicians can identify families or family
members who may be at risk for
increasingly significant problems in coping and/or the development of psychiatric issues (e.g., clinical depression).
Families may also require assistance in
seeking external support networks, particularly over time. Contextual changes
in raising a child with a developmental
disability, such as changes in parental
perceptions of child functioning, access
to social supports, and employment
changes can impact parents’ ability to
MRDD Research Reviews DOI 10.1002/mrdd
sustain effective coping mechanisms. In
a longitudinal study examining coping
strategies used by parents of children
with autism, it was found that parental
use of problem-focused coping mechanisms declined over time, with a corresponding increase in emotion-focused
coping responses [Gray, 2006]. Possible
explanations for the decline in problemfocused coping included changes in
social support systems as the child ages,
with fewer parents and children having
routine interventions available to them
beyond the school-age years, and
changes in parental expectations for
improvement in their child’s overall
functioning and behavior [Gray, 2006].
Findings such as those of Gray [2006]
illustrate the need for clinicians to recognize the ongoing experience of adaptation and how a systems approach to
evaluation considers the changing needs
of the family in this process [Seligman
and Darling, 1997].
Treatment
Determining and implementing a
course of treatment and establishing a
procedure for evaluating treatment progress are the remaining pieces of an
assessment and the systems approach can
help here as well [Seligman and Darling,
1997]. A multidisciplinary evaluation
team is very beneficial in this process as
each discipline is able to attest to the
needs of the child and family and provide
materials, resources, referrals, and therapy recommendations that are concrete,
specific, and individualized. Further,
team members are able to consider the
parents and family’s ability to implement
services for the child and other family
members. Medical care, educational programming, behavioral management,
pharmacological support—such interventions can require significant effort on
the part of parents to implement and
even to monitor. Clinicians who have
assessed how parents and family members
are coping with current caregiving and
adaptation issues are better able to tailor
recommendations and treatment options
in recognition of the family’s status, their
ability to seek out treatment recommendations, and their ability to participate in
treatment implementation. In fact, the
clinician might make different recommendations for treatment for the child
with disabilities based on expectations
about the family’s current state of wellbeing. If possible, recommendations for
treatment of family members in addition
to the child with a disability can also be
suggested.
Systems Approach to Evaluation
Head and Abbeduto
In many cases, treatment recommendations will not be implemented by
the clinician(s) conducting the assessment, but instead by outside agencies
and professionals. Team members can
be incorporated as part of follow-up
care to monitor the family’s journey
through the treatment process and to
provide case management support following the evaluation. Assessment clinics that include a significant social work
element or family resource program are
especially prepared to do this by maintaining a relationship with the family
and assisting them in answering questions and providing contacts as they
navigate treatment during this critical
period.
IMPLICATIONS FOR SERVICE
DELIVERY IMPLICATIONS
Although progressive in its perspective, implementing a systems
approach can be challenging and numerous obstacles exist that can impede its
acceptance into practice. Clinicians often encounter logistical constraints,
including lack of resources, time, and
money that make a systems-based assessment process difficult. Changing longtime standards of assessment procedure
as well as soliciting professionals from
other disciplines to endorse the holistic
nature of a systems approach and
actually participate in its implementation
can also present significant barriers.
Thus, although a family systems
approach to assessment is comprehensive
in its attempts to recognize the needs of
the family as a means of supporting the
child’s development, implementing
social, healthcare, and educational policies and procedures that reflect this
approach have only begun to be initiated with the advent of early intervention and special education services
[Seligman and Darling, 1997]. These
promising new developments encourage
professionals to support the use of a
family systems approach to assessment.
The Individuals with Disabilities
Education Improvement Act of 2004
P.L. 108-446, Part C, includes the Individual Family Service Plan (IFSP),
which is a model early intervention
process for children birth to age three
that is designed to foster child development by recognizing and supporting the
family and their needs, concerns, and
priorities. In developing and implementing the IFSP, education professionals and families work together in partnership from the onset of the child’s
initial evaluation and entry into services.
By identifying the family’s needs and
resources, the evaluation process better
reflects a complete and accurate picture
that include the child’s strengths, needs,
and available supports. Recognizing the
evolving needs of children as they grow
and develop requires that professionals
across healthcare, education, and
employment settings work together
with individuals, parents, and families to
establish goals and interventions that
enhance the child’s capacity for development by strengthening the family’s
ability to participate in that process.
The family-centered approach evidenced in IFSP meetings and implemented through daily programming and
intervention efforts must be uniformly
extended to the Individual Education
Program and vocational rehabilitation
processes, to further recognize the continued role of families in an individual’s
development over the lifespan.
investigations into the use of these
measures within the assessment process
is needed. Development of increasingly
sophisticated measures that allow the
relational aspects between different family members to be assessed with
increased objectivity would provide a
tremendous wealth of information that
extends beyond the individual perspectives available through self-report questionnaires.
In conjunction with research that
examines the impact of diagnosis delivery on parents in terms of specific medical conditions [Garwick et al., 1995;
Ferguson et al., 2006], additional
research is needed to understand the
impact of diagnosis delivery within the
clinical setting following an evaluation,
with an emphasis placed on identifying
the role of a systems approach to assessment within the context of conveying
the evaluation’s results and recommendations to family members. Studies are
needed to investigate the role professionals play in shaping parent perceptions
of their child’s condition and their
child’s developmental outlook or prognosis in this process. By looking at variations among the interactions between
professionals and families, we can better
understand what characteristics have the
greatest impact in short-term adjustment and long-term adaptation. These
issues become particularly important
when working with culturally diverse
families whose familial structure often
encompasses individuals beyond just
parents and siblings to include extended
family members. Further, the inclusive
nature of a family systems approach
suggests that research is needed to
examine how culturally diverse families
respond to this approach given social
norm variations and differences in coping and if adaptations to the approach
are needed based on these unique characteristics.
Finally, it is important to consider
how a family systems approach to assessment can be integrated into the larger
service delivery system so that the needs
of families as well as the child can be
addressed over time. Adaptation to caring for an individual with a developmental disability is an evolving process
with unique considerations at various
timepoints in the life of the child and
the family’s experience. A family systems approach recognizes that needs
exist and change over time. Future
research should consider the obstacles
to this integration process and examine
how a family systems approach to assessment must change with as well to
DIRECTIONS FOR FUTURE
RESEARCH
A family systems approach to
assessment affords researchers the opportunity to examine a number of key
questions with the goal of understanding how to approach families in this situation in a way that is most effective
and comprehensive, yet sensitive to
their unique experience.
We have learned much in the past
three decades about how families
respond to raising a child with a disability. Studies have demonstrated that child
maladaptive behavior, syndrome-specific
behavior, and cognitive and adaptive
functioning levels are associated with
individual and family dysfunction
[Hauser-Cram et al., 2001; Herring
et al., 2006]. However, few studies have
examined these relationships at the time
of initial diagnosis, when specific interventions could be initiated [HauserCram et al., 2001]. Studies are needed
to further define variables that contribute to the prediction of parental wellbeing and family functioning, consider
the impact of ongoing developmental
concerns on families, and examine contextual factors that differentiate families
prior to diagnosis.
In order to do this with increasing specificity, research is needed to
determine how to develop new tools to
operationalize and assess family systems
variables. Although a number of different self-report questionnaires exist and
various methods, including use of
audiotape, videotaping, and observational coding systems, have been suggested
as ways to quantify observed interactions among family members, future
MRDD Research Reviews DOI 10.1002/mrdd
Systems Approach to Evaluation
Head and Abbeduto
299
accommodate the needs of families successfully. Such research will continue to
foster an understanding of the variations
in the trajectory of the adaptation process and potentially contribute to the
development of interventions at the earliest stage of adaptation and as it occurs
over the lifespan.
CONCLUSIONS
When concerns arise regarding a
child’s development, all family members
are potentially affected [Seligman and
Darling, 1997]. Typically, clinical assessments have been child-focused in an
effort to determine a diagnosis and develop specific interventions that target
the child’s needs. However, parents also
have needs that are often not addressed
within the assessment process. In this article, we suggested a systems approach as
a way of expanding the assessment process to consider family member functioning and its relationship to child development. A systems approach likewise,
emphasizes understanding the variation
across different disability conditions with
which parents and family members
respond to the challenges of raising a
child with developmental issues. Condition-related differences in mental health
and well-being outcomes among parents
suggest that clinicians should use the
assessment process to recognize and
acknowledge the needs of parents and
family members and to consider condition-related differences in order to tailor
interventions that can effectively meet
the child and the family’s needs. n
REFERENCES
Abbeduto L, Brady N, Kover ST. 2007. Language
development and fragile X syndrome: profiles, syndrome-specificity, and within-syndrome differences. Ment Retard Dev Disabil
Res Rev 13:36–46.
Abbeduto L, Seltzer MM, Shattuck P, et al. 2004.
Psychological well-being and coping in
mothers of youths with autism, Down syndrome, or fragile X syndrome. Am J Ment
Retard 109:237–254.
Abbeduto L, Warren SF, Conners FA. 2007. Language development in Down syndrome: from
the prelinguistic period to the acquisition of
literacy. Ment Retard Dev Disabil Res Rev
13:247–261.
Abdin RR. 1995. Parenting Stress Index, 3rd ed.
Odessa, FL: Psychological Assessment Resources.
Antonarakis SE, Epstein C. 2006. The challenge
of Down syndrome. Trends Mol Med 12:
473–479.
Baer J. 2002. Is family cohesion a risk or protective factor during adolescent development?
J Marriage Fam 64:668–675.
Baker BL, Blacher J, Crnic KA, et al. 2002.
Behavior problems and parenting stress in
families of three year old children with and
without developmental delays. Am J Ment
Retard 107:433–444.
300
Baker BL, Blacher J, Olsson MB. 2005. Preschool
children with and without developmental
delay: behaviour problems, parents’ optimism and well-being. J Intellect Disabil Res
49:575–590.
Beck AT, Steer RA. 1993. Beck Anxiety Inventory. New York: Psychological Corporation.
Beck AT, Steer RA, Brown GK. 1996. Beck
Depression Inventory, 2nd ed. New York:
Psychological Corporation.
Bengston VL, Black KD. 1973. Solidarity between
parents and children: four perspectives on
theory development. In: Paper Presented to
the Theory Development Workshop, National
Council on Family Relations, Toronto.
Berman C, Shaw E. 1996. Family-directed evaluation and assessment under the Individuals
with Disabilities Education Act (IDEA). In:
Meisels SJ, Fenichel E, editors. New visions
for the developmental assessment of infants
and young children. Washington, DC:
National Center for Infants, Toddlers, and
Families. p 361–390.
Bradley RH. 1994. The HOME Inventory:
review and reflections. In: Reese HW, editor. Advances in child development and
behavior, Vol. 25. San Diego, CA: Academic
Press. p 241–288.
Campis LK, Lyman RD, Dunn SP. 1986. The parental locus of control scale: Development
and validation. J Clin Child Psychol 15:
260–267.
Canfield MA, Honein MA, Yuskiv N, et al. 2006.
National estimates and race/ethnic-specific
variation of selected birth defects in the
United States 1999–2001. Birth Defects Res
A Clin Mol Teratol 76:747–756.
Carver CS, Scheier MF, Weintraub JK. 1989.
Assessing coping strategies: a theoretically
based approach. J Pers Soc Psychol 56:
267–283.
Denmark JL, Feldman MA, Holden JJ. 2003. Behavioral relationship between autism and
fragile X syndrome. Am J Ment Retard 108:
314–326.
Donenberg G, Baker BL. 1993. The impact of
young children with externalizing behaviors
on their families. J Abnorm Child Psychol
31:179–198.
Eisenhower AS, Baker BL, Blacher J. 2005. Preschool children with intellectual disability:
syndrome specificity, behaviour problems,
and maternal well-being. J Intellect Disabil
Res 49:657–671.
Essex EL, Seltzer MM, Krauss MW. 1999. Differences in coping effectiveness and well-being
among aging mothers and fathers of adults
with mental retardation. Am J Ment Retard
104:545–563.
Feinstein C, Reiss AL. 2001. Autism: the point of
view from fragile X studies. J Autism Dev
Disord 28:393–405.
Ferguson JE, Kleinert HL, Lunney CA, et al.
2006. Resident physicians’ competencies and
attitudes in delivering a postnatal diagnosis
of Down syndrome. Obstet Gynecol 108:
898–905.
Franke P, Leboyer M, Gansicke M, et al. 1998.
Genotype-phenotype relationship in female
carriers of the premutation and full mutation of FMR-1. Psychiatry Res 80:113–
127.
Franke P, Maier W, Hautzinger M, et al. 1996.
Fragile-X carrier females: evidence for a distinct psychopathological phenotype? Am J
Med Genet 64:334–339.
Freund LS, Reiss AL, Hagerman R, et al. 1992.
Chromosome fragility and psychopathology
MRDD Research Reviews DOI 10.1002/mrdd
in obligate female carriers of the fragile X
chromosome. Arch Gen Psychiatry 49:
54–60.
Friedrich WN, Greenberg MT, Crnic K. 1983. A
short-form of the questionnaire on resources
and stress. Am J Ment Defic 88:41–48.
Gardiner K, Costa A. 2006. The proteins of
human chromosome 21. Am J Med Genet
C Semin Med Genet 142:196–205.
Garwick AW, Patterson J, Bennett FC, et al.
1995. Breaking the news: how families first
learn about their child’s chronic condition.
Arch Pediatr Adolesc Med 149:991–997.
Glidden LM, Billings FJ, Jobe BM. Personality,
coping style and well-being of parents rearing children with developmental disabilities.
J Intellect Disabil Res 50:949–962.
Gray DE. 2006. Coping over time: the parents of
children with autism. J Intellect Disabil Res
50:970–976.
Greenley RN, Holmbeck GN, Rose BM. 2006.
Predictors of parenting behavior trajectories
among families of young adolescents with
and without spina bifida. J Pediatr Psychol
31:1057–1071.
Greenspan SI. Meisels SJ. 1996. Toward a new
vision for the developmental assessment of
infants and young children. In: Meisels SJ,
Fenichel E, editors. New visions for the developmental assessment of infants and young
children. Washington, DC: National Center
for Infants, Toddlers, and Families. p 9–52.
Hagerman RJ. 1999. Neurodevelopmental disorders. Oxford: Oxford University Press.
Hassall R, Rose J. 2005. Parental cognitions and
adaptation to the demands of caring for a
child with an intellectual disability: a review
of the literature and implications for clinical
interventions. Behav Cogn Psychother 33:
71–88.
Hastings RP. 2003. Child behaviour problems and
partner mental health as correlates of stress
in mothers and fathers of children with
autism. J Intellect Disabil Res 47:231–
237.
Hastings RP, Kovshoff H, Brown T, et al. 2005.
Coping strategies in mothers and fathers of
preschool and school-age children with autism. Autism 9:377–391.
Hauser-Cram P, Warfield ME, Shonkoff JP, et al.
1999. Family influences on adaptive development in young children with Down syndrome. Child Dev 70:979–989.
Hauser-Cram P, Warfield ME, Shonkoff JP, et al.
2001. Children with disabilities: a longtitudinal study of child development and parent
well-being. Monogr Soc Res Child Dev
66:1–131.
Heller T, Hsieh K, Rowitz L. 1997. Maternal and
paternal caregiving of persons with mental
retardation across the lifespan. Fam Relat
46:407–415.
Herring S, Gray K, Taffe J, et al. 2006. Behaviour
and emotional problems in toddlers with
pervasive developmental disorders and developmental delay: associations with parental
mental health and family functioning. J
Intellect Disabil Res 50:874–882.
Hessl D, Tassone F, Loesch DZ, et al. 2005.
Abnormal elevation of FMR1 mRNA is
associated with psychological symptoms in
individuals with the fragile X permutation.
Am J Med Genet B Neuropsychiatr Genet
139:115–121.
Hodapp RM, Dykens EM. 2001. Strengthening
behavioral research on genetic mental retardation syndromes. Am J Ment Retard 106:
4–15.
Systems Approach to Evaluation
Head and Abbeduto
Hodapp RM, Ly TM, Fidler DJ, et al. 2001. Less
stress, more rewarding: parenting children
with Down syndrome. Parent Sci Pract 1:
317–337.
Hodapp RM, Ricci LA, Ly TM, et al. 2003. The
effects of the child with Down syndrome on
maternal stress. Br J Dev Psychol 21:137–151.
Holmbeck GN, Coakley RM, Hommeyer JS,
et al. 2002. Observed and perceived dyadic
and systemic functioning in families of preadolescents with spina bifida. J Pediatr Psychol 27:177–189.
Holmbeck GN, Westhoven VC, Phillips WS,
et al. 2003. A Multimethod, multi-informant, and multidimensional perspective on
psychosocial adjustment in preadolescents
with spina bifida. J Consult Clin Psychol
71:782–796.
Johnson BH. 2000. Family-centered care: four
decades of progress. Fam Syst Health 18:
137–156.
Johnston C, Eliez S, Dyer-Friedman J, et al. 2001.
Neurobehavioral phenotype in carriers of
the fragile X premutation. Am J Med Genet
103:314–319.
Johnston C, Hessl D, Blasey C, et al. 2003. Factors associated with parenting stress in mothers of children with fragile X syndrome. J
Dev Behav Pediatr 24:267–275.
Keller D, Honig AS. 2004. Maternal and paternal
stress in families with school-aged children
with disabilities. Am J Orthopsychiatry 74:
337–348.
Kuehn BM. 2007. CDC: autism spectrum disorders common. JAMA 297:940.
Lewis P, Abbeduto L, Murphy M, et al. 2006.
Cognitive, language and social-cognitive
skills of individuals with fragile X syndrome
with and without autism. J Intellect Disabil
Res 50:532–545.
Loesch DZ, Huggins RM, Bui QM, et al. 2002.
Effect of the deficits of fragile X mental retardation protein on cognitive status of fragile X males and females assessed by robust
pedigree analysis. J Dev Behav Pediatr 23:
416–423.
Mink IT, Nihira K. 1986. Family life-styles and
child behaviors: a study of direction of
effects. Dev Psychol 22:610–616.
Mink IT, Nihira K, Meyers CE. 1983. Taxonomy
of family life styles. I. Homes with TMR
children. Am J Ment Defic 87:484–497.
Minuchin S. 1974. Families and family therapy.
Cambridge, MA: Harvard University Press.
Moos RH, Moos BS. 1994. Family Environment
Scale, 3rd ed. Palo Alto, CA: Consulting
Psychologists Press.
Most DE, Fidler DJ, Booth CL, et al. 2006. Stress
trajectories in mothers of young children
with Down syndrome. J Intellect Disabil
Res 50:501–514.
Olson DH, Portner J, Lavee Y. 1982. FACES II.
St. Paul, MN: University of Minnesota.
drome: diagnosis, treatment, and research.
Baltimore: Johns Hopkins University Press.
p 165–192.
Simeonsson RJ, Scarborough A. 2001. Issues in
clinical assessment. In: Simeonsson RJ,
Rosenthal SL, editors. Psychological and developmental assessment. New York: Guilford
Press. p 17–31.
Simmerman S, Blacher J, Baker BL. 2001.
Fathers’ and mothers’ perceptions of father
involvement in families with young children
with a disability. J Intellect Dev Disabil
26:325–338.
Singer GHS. 2006. Meta-analysis of comparative
studies of depression in mothers of children
with and without developmental disabilities.
Am J Ment Retard 111:155–169.
Sobesky WE, Pennington BF, Porter D, et al.
1994. Emotional and neurocognitive deficits
in fragile X. Am J Med Genet 51:378–
385.
Spanier GB. 1989. Dyadic adjustment scale. Tonawanda, NY: Multi-Health Systems.
Stoneman Z. 1997. Mental retardation and family
adaptation. In: MacLean WE, editor. Ellis’
handbook of mental deficiency, psychology
theory and research. Mahwah, NJ: Lawrence
Erlbaum. p 405–437.
Summers JA, Poston DJ, Turnbull AP, et al. 2005.
Conceptualizing and measuring family quality of life. J Intellect Disabil Res 49:
777–783.
Taunt HM, Hastings RP. 2002. Positive impact of
children with developmental disabilities on
their families: a preliminary study. Educ
Train Ment Retard Dev Disabil 37:410–
420.
Turnbull AP, Summers, JA, Brotherson MJ. 1986.
Family life cycle: theoretical and empirical
implications and future directions for families
with mentally retarded members. In: Gallagher JJ, Vietze PM, editors. Families of
handicapped persons. Baltimore: Brookes.
p 45–65.
Turnbull AP, Turnbull HR. 1990. Families, professionals, and exceptionality, 2nd ed. Columbus, OH: Merrill.
van Lieshout CFM, De Meyer RE, Curfs LM,
et al. 1998. Family contexts, parental behavior, and personality profiles of children and
adolescents with Prader-Willi, fragile X, or
Williams syndrome. J Child Psychol Psychiatry 29:699–710.
von Gontard A, Backes M, Laufersweiler-Plass C,
et al. 2002. Psychopathology and familial
stress—comparison of boys with fragile X
syndrome and spinal muscular atrophy. J
Child Psychol Psychiatry 43:949–957.
Warfield ME. 1995. Early predictors of child
behavior problems: examining the family
environment. In: Paper presented at the
Biennial Meeting of the Society for Research
in Child Development, Indianapolis, IN.
Olsson MB, Hwang CP. 2001. Depression in
mothers and fathers of children with intellectual disability. J Intellect Disabil Res
45:535–543.
Olsson MB, Hwang CP. 2002. Sense of coherence
in parents of children with different developmental disabilities. J Intellect Disabil Res
46:548–559.
Patterson JM, Garwick AW. 1994. Levels of
meaning in family stress theory. Fam Process
33:287–304.
Piven J. 1999. Genetic liability for autism: the
behavioural expression in relatives. Intern
Rev Psychiatry 11:299–308.
Piven J, Palmer P, Joacbi D, et al. 1997. Broader
autism phenotype: evidence from a family
history study of multiple-incidence autism
families. Am J Psychiatry 154:185–190.
Reiss AL, Dant CC. 2003. The behavioral neurogenetics of fragile X syndrome: analyzing
gene-brain-behavior relationships in child
developmental psychopathologies. Dev Psychopathol 5:927–968.
Rogers SJ, Wehner EA, Hagerman RJ. 2001. The
behavioral phenotype in fragile X: symptoms
of autism in very young children with fragile
X syndrome, idiopathic autism, and other
developmental disorders. J Dev Behav
Pediatr 22:409–417.
Rosenthal SL, Cohen SS, Simeonsson RJ. 2001.
Assessment of family context. In: Simeonsson RJ, Rosenthal SL, editors. Psychological
and developmental assessment. New York:
Guilford Press. p 141–152.
Roubertoux PL, Kerdelhu’ B. 2006. Trisomy 21:
from chromosomes to mental retardation.
Behav Genet 36:346–354.
Sanders JL, Morgan SB. 1997. Family stress and
adjustment as perceived by parents of children with autism or Down syndrome: implications for intervention. Child Fam Behav
Therapy 19:15–32.
Sarimski K. 1997. Behavioural phenotypes and
family stress in three mental retardation syndromes. Eur Child Adolesc Psychiatry 6:
26–31.
Sattler JM. 2002. Assessment of children behavioral and clinical applications, 4th ed. San
Diego, CA: Jerome M. Sattler.
Seligman M, Darling RB. 1997. Ordinary families, special children: a systems approach to
child disability, 2nd ed. New York: Guilford
press.
Sellinger MH, Hodapp RM. 2005. Indirect effects
of genetic syndromes: parental reactions to
behavioral phenotypes. Enfance 57:218–
226.
Seltzer MM, Greenberg JS, Floyd FJ, et al. 2001.
Life course impacts of parenting a child with
a disability. Am J Ment Retard 106:
265–285.
Sherman S. 1996. Epidemiology. In: Hagerman
RJ, Cronister A, editors. Fragile X syn-
MRDD Research Reviews DOI 10.1002/mrdd
Systems Approach to Evaluation
Head and Abbeduto
301