European Journal of Special Education Research
ISSN: 2501 - 2428
ISSN-L: 2501 - 2428
Available on-line at: www.oapub.org/edu
10.5281/zenodo.61493
Volume 1│Issue 2│2016
INTERACTIONAL STYLE AND SUBJECTIVE STRESS IN
MOTHERS OF YOUNG CHILDREN WITH FRAGILE X SYNDROME,
DOWN’S SYNDROME OR TYPICAL DEVELOPMENT
Teresa Sansouri
University of Education in Heidelberg, Institute for Special Education,
Keplerstr. 87, 69120 Heidelberg, Baden-Württemberg, Germany
Abstract:
This study examined the interactional style of mothers of boys with Fragile X syndrome
(FXS) and mothers of children with Down s syndrome DS or developmentally typical
children (N=33 mother-child-dyads) in different situations (matching task; playing with
toys; request). The average developmental age of the children was 29 months. Mothers
completed a self-report scale for maternal stress. Directive and restrictive parenting
behaviors were used more frequently by mothers of children with FXS. Mothers of
children with DS tended to use more confirming and praising behaviors. Directive and
restrictive behaviors were negatively associated with children s attention and positively
associated with child behavioral problems and maternal stress. Implications of these
findings are discussed.
Keywords: maternal interactional style, Fragile X syndrome, Down s syndrome, maternal
stress
Introduction
Factors that Influence Child Development
Vygotskij theorized that early reciprocal interactions between mother and child play an
important role in child development. When playing with a competent partner a child is
supported in regulation processes and may thus succeed at tasks that he or she could not
manage alone (Vygotskij, 2010). According to the transactional model, (Sameroff &
Chandler,
development is shaped not only by the extent to which the child s
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INTERACTIONAL STYLE AND SUBJECTIVE STRESS IN MOTHERS OF YOUNG CHILDREN WITH FRAGILE X
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environment satisfies his or her needs, but also by the extent to which the child can
influence his or her environment. Guralnick s
developmental systems approach
identifies three main, interacting influences on child development: child characteristics,
family interaction patterns and parental resources. Guralnick stressed that alongside
parental intellectual capacities and educational background, family wellbeing and
parental locus of control were important factors in child development. Ispa et al. (2002)
revealed a positive correlation between maternal alienation and stress reactivity. Further
studies have shown that maternal proneness to stress is negatively associated with
sensitivity and warmth in maternal interactions with the child (Belsky et al., 1995;
Mangelsdorf et al., 1990).
Research findings suggest that it is not simply the mother s presence that provides
support for the child; how she interacts with the child is also important. Roggman et al.
(2013) identified four aspects of supportive parenting:
-
“ffection: Warmth, physical closeness, and positive expressions towards child
-
Responsiveness: Responding to child’s cues, emotions, words, interests, and behaviors
-
Encouragement: Active support of exploration, effort, skills, initiative, curiosity, creativity,
and play
-
Teaching: Shared conversation and play, cognitive stimulation, explanations, and
questions
(Roggman et al., 2013, p. 2)
Mothers who show these or similar qualities in interactions with their children tend to
have children who achieve language milestones earlier (Tamis-LeMonda et al., 2011) and
display better cognitive (Bernier et al., 2010; Hubbs-Tait et al., 2002) and social (Ispa et al.,
2004; Kelley et al., 2000) skills.
Knowledge of Syndrome-Specific Interactions
Down s syndrome DS and Fragile X syndrome (FXS) are the main genetic causes of
intellectual disability. Both syndromes are associated with a specific behavioral
phenotype. Children with FXS have mild to moderate intellectual disability, an inhibitory
control deficit and show hyperactivity and gaze avoidance (Backes et al., 2000; Hatton et
al., 2002; Kau et al., 2004; Langthorne & McGill, 2012). These specific behaviors may be
caused by deficits in executive functions (Cornish et al., 2004). Children with DS often
have problems with emotion regulation. Children with DS show lower task motivation
and reduced tolerance of frustration compared with children with other forms of an
intellectual disability. There is also evidence that children with DS have relatively good
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social skills (Berger & Cunnigham, 1981; Dykens, 1994; Fidler et al., 2006). As a
consequence, children with DS are more likely to seek help from adults when confronted
with challenging tasks (Jahromy et al., 2008; Kasari & Freeman, 2001; Pitcairn & Wishart,
1994).
Hodapp (1997) distinguished between the direct and indirect effects of a genetic
disposition: direct effects are behaviors which are more probable in people with the
syndrome whereas indirect effects can be described as reactions of the environment to a
person with the syndrome. Although there is strong evidence that parent-childinteractions have a great impact on child development little is known about specific
relationships between behavioral phenotypes and parenting styles.
Until now there has been little data available on the indirect effects of genetic
disorders on mother-child-interactions. Wheeler et al. (2007) logged the frequency of
maintaining behavior, directive behavior, scaffolding and restriction in 24 mother-childpairs observed in a 10-minute play context and a 1-hour unstructured, everyday context.
The mothers primarily used maintaining behaviors. They made less use of directive
behavior in the unstructured, naturalistic context than in the structured play context. In
another study Wheeler et al. (2010) observed 46 children with FXS and their mothers in a
frustrating task situation. The children were given a box containing an attractive toy which
could only be opened using a key which was given to the mother. Mothers were instructed
only to intervene when the child got too frustrated. The mothers mainly showed
encouraging and directive behaviors, making demands and suggestions. The only
investigation to compare the interactional style of mothers with children with FXS with
developmentally typical children (Sterling et al., 2012) used a within-family design.
Sterling et al. compared how mothers interacted with their children with FXS and with
their siblings without disability. Mothers used a responsive style with both children but
used more behavior management strategies and a less conversational style with the child
with FXS. These findings indicate that mothers react to the child s inhibitory control
deficits by regulating task performances more closely.
Considerably more studies have investigated the interaction of mothers and their
children with DS. Roach et al. (1998) compared the interactions of 28 mothers and their
children with DS with those of mothers and their developmentally typical children.
Mothers of children with DS were more directive but also more supportive. In a
longitudinal study Blacher et al. (2013) examined parenting behavior during structured
and unstructured activities. The 183 participants included mothers of children with autism
spectrum disorders, cerebral palsy, DS, undifferentiated developmental delay and typical
cognitive development. The group of mothers with children with DS used more positive,
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encouraging behaviors. These findings suggest that mothers of children with DS combine
a directive interactional style with supportive and motivating behaviors.
What data we have indicate that mothers of children with a specific syndrome
adapt to the behavioral characteristics of their child. Mothers of children with FXS are
more likely to use behavior management strategies, presumably in response to their
children s impulsivity, whereas mothers of children with DS seem to encourage their
children more often to try to increase their motivation and endurance.
The aim of the study was to examine maternal parenting in three different groups
of mother-child dyads: mothers and their children with FXS, mothers and their children
with DS and mothers and their developmentally typical (TYP) children. The conceptual
framework for the study recognizes various influences on maternal interactional style:
situation or context (matching task; free play; request), child characteristics during the
various situations (attention; behavioral adaption) and maternal wellbeing (parenting
stress).
The following research questions guided the study:
-
Do mothers of boys with FXS use a different interactional style from mothers of children
with DS or TYP children? If so, how does their interactional style differ?
-
What influence does situation have on maternal parenting?
-
Is maternal parenting style correlated with child behavior variables?
-
Does maternal wellbeing of mothers with FXS boys differ from maternal wellbeing of
mothers of children with DS or TYP children?
-
Is maternal wellbeing correlated with parenting style?
Material and Methods
Participants
The participants were 11 mothers of children with FXS, 11 mothers of children with DS
and 11 mothers of developmentally typical children. Families were recruited from Fragile
X and Down s syndrome listservs and websites in Germany and Switzerland. The families
of the control group were contacted via postings in day care centers. None of the mothers
of the children with FXS had full mutation status. Demographic information for all groups
is provided in Table 1.
Children s developmental age was determined from a child development
questionnaire (Elternfragebogen zur kindlichen Entwicklung; Brandtstetter, Bode & Ireton,
2003, the German version of the Child Development Inventory, Ireton, 1992) which was
filled out by the mother. As there is evidence that children with DS have significantly
lower expressive language skills than receptive skills (Sarimski, 2014), developmental age
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was determined without considering score on the expressive language scale. The ShapiroWilk test was used to confirm that developmental age was normally distributed in all
groups. ANOVA confirmed that there were no group differences in developmental age.
FXS
DS
Typ
n = 11
n = 11
n = 11
Maternal age in years M
34
41
33
German ethnicity
10
10
9
1
1
2
10
7
11
Number of mother-child-pairs
Other ethnicity
In a stable relationship
Single parent
1
4
0
2.2
2.2
1.9
Maternal educational attainment: junior high school
2
0
1
Maternal educational attainment:
5
3
0
4
8
10
Full-time employment
1
2
0
Part-time employment
7
3
8
Homemaker
3
5
3
In further education or training
0
1
0
Number of boys
11
6
3
Number of girls
0
5
8
57
51
32
(13.71)
(9.70)
(5.03)
28 (5.21)
27(4.62)
31(5.03)
Ø Number of children in family
Middle / High school
Maternal educational attainment:
Matriculation standard
Child’s age in months,
M (SD)
Child’s developmental age excluding expressive speech in
months, M (SD)
Table 1: Demographic profile of mother-child dyads (n=33)
Procedures
Mother-child dyads were visited in their homes. Behavior was videotaped in three
situations: a structured 10-minute session in which the mother was instructed to explain a
matching task to her child, a 10-minute play session for which toys (vehicles, a house,
Lego and figurines) were available and, finally, a request situation, in which mothers were
instructed to ask the child to tidy up the toys. After the observation period mothers were
asked to fill out questionnaires about the child s development and their own wellbeing.
The average length of visit was 2 hours.
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Instrumentation
Maternal measures. Maternal behavior was observed and coded using an adaptation of
the coding system developed by Wheeler and colleagues (2007). There were four main
categories, each containing four or five items: maintaining behaviors, regulation of emotions,
directive behaviors and restriction. A rest category served to code maternal behaviors which
did not involve interaction with the child e.g. cleaning one s nose . Maintaining behaviors
tell the child that his or her interests, thoughts and actions are important (Wheeler et al.,
. This category was used when the mothers made comments on the child s activities
or made suggestions about how the child could extend his or her activity. Regulation of
emotions refers to maternal attempts to increase the child s positive affect or reduce or
prevent negative affect e.g. encouraging, praising or comforting the child or supporting
the child to handle toys. Directive behaviors serve to structure the situation and to focus
the child s attention on what is relevant. Directive behaviors can be described as
responsive to the child s behavior or unresponsive if the mother is not taking into account
her child s activity Landry et al.,
. Maternal demands or requests for information
were coded as directive behaviors. Restrictions are another way of directing or restricting
the child s actions but this term is used for behaviors which cannot be described as helpful
to the child s development. This category was used when the mother limited the child s
actions, e.g. by holding the child, or when the mother disciplined the child. All videos
were coded using a time-sampling method with 5-seconds segments.
We also used the PICCOLO rating scale (Roggman et al., 2013) to rate four aspects
of supportive parenting: affection, responsiveness, encouragement and teaching. Each domain
consists of 7-8 behavioral items. Items are rated as not observable (0), partly observable (1)
or full observable (2). PICCOLO was developed for play situations with pretend play toys
or manipulative toys and picture book situations. Although PICCOLO was only
appropriate to our play situation according to the PICCOLO Handbook (Roggman et al.,
2013), maternal behavior in all three situations was rated using it. PICCOLO is a reliable
instrument. Based on data from over 2000 families the inter-rater reliability for pairs of
observers averaged 75% across all four domains; the internal consistency (average
Cronbach s α = .
is also very good.
Maternal stress was measured by using the Elterliches Belastungsinventar (EBI;
Tröster, 2011), a German version of the Parenting Stress Index (PSI; Abidin, 1995). The EBI
consists of 48 items organized into two domains: stress linked with the child s
characteristics (child domain) and parental distress (parent domain). In this study the EBI
had satisfactory internal consistency average of Cronbach s α for both domains = .
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Child measures. Children s behavior was coded using two categories: attention and
behavioral adaption. Attention was coded as on- and off-task behaviors in relation to
engagement with the materials available in the situation (matching task or toys).
Children s behavior was coded as on-task when the child was dealing with the material in
a non-specific way. Behavioral adaption was coded as rule-consistent or not rule-consistent.
The child s behavior was coded as rule-consistent when it was in accordance with the
object of the task (matching task situation) or when it was non-destructive (play situation).
In the request situation behavior was coded as rule-consistent when the child obeyed the
mother s instruction to tidy up. When the child was coded as off-task his or her behavior
was by definition not rule-consistent. An event-sampling method was used to code
children s behavior.
Data Analysis
Two researchers observed all three situations and inter-rater agreement were calculated
for 12% of the video data. The two researchers were trained in use of the coding system
and the PICCOLO rating scale. A minimum inter-rater agreement coefficient of .70 was
maintained for all interaction behaviors across all the data.
As a 5s coding segment could contain behaviors from several categories the total
number of codes assigned was not the same for all situations or dyads and so inter-groupcomparisons could not be presented as percentages. We therefore used the average
number of instances of a given code per minute of maternal behavior in all analyses. This
was a way of transforming observations of specific behaviors into meaningful, comparable
data. Since the data were not normally distributed we used a non-parametric test, the
Kruskal-Wallis test. This analysis shows via post hoc pairwise comparisons which groups
differ from each other.
Spearman s rank correlations were run to identify relationships among the
variables (e.g. maternal stress and interactional style . Effects were rated as follows . ≤ r s
≤ . , no correlation . < rs ≤ . , little correlation . < rs ≤ . , clear correlation and . < rs ≤ . ,
high correlation.
Results
Maternal Behavior
Analysis of the distribution of maternal interactive behaviors indicated that in all groups
the most common behaviors were maintaining behaviors (range: 35-49% across all groups)
and directive behaviors (range: 37–
% across all groups . Regulation of the child s
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emotions (range: 10–12% across all groups) was less frequent and restrictive behaviors
(range: 1–7% across all groups) were rarely observed. Over all situations mothers of
children with FXS or DS made greater use of more directive behaviors than mothers of
developmentally typical children.
Mothers of children with FXS made less use of maintaining behaviors than mothers
in the other two groups but made more frequent use of directive and restrictive behaviors.
The interactional behavior of mothers of children with DS and mothers of
developmentally typical children was similar with respect to the main categories; however
mothers of children with DS tended to use encouraging behaviors, such as praising and
confirming their children, more often. Table 2 gives the average number of instances of
each code per minute for mothers for all the situations combined (also displayed in Figure
1).
Category
Maintaining behavior
Regulation of emotions
Directive behavior
Restrictive behavior
Non-interactive behavior
FXS
DS
Typ
M (SD)
M (SD)
M (SD)
6.38
8.44
8.66
(1.83)
(1.37)
(0.58)
1.85
2.10
2.07
(0.65)
(0.90)
(0.58)
8.64
7.20
6.49
(1.67)
(1.31)
(1.42)
1.20
0.28
0.11
(1.21)
(0.39)
(0.09)
0.17
0.20
0.14
(0.14)
(0.14)
(0.11)
Pairs
Significance
Test statistics
FXS – DS
.006
-11.273**
FXS – Typ
.003
-12.455**
DS – Typ
.774
-1.182
n.s.
.565
1.143
FXS – DS
.103
6.727
FXS – Typ
.004
11.818**
DS – Typ
.217
5.091
FXS – DS
.018
9.773*
FXS – Typ
.001
13.818**
DS – Typ
.326
4.045
n.s.
.651
.858
Table 2: Group comparisons (n = 11 per group) for main categories; average instances of each code
per minute across all situations; *p < .05; **p < .01; n.s. = not significant; FXS = Fragile X;
DS = Down s syndrome Typ = developmentally typical
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Figure 1: Group comparisons of maternal behavior (n = 11 per group) for main categories;
average instances of each code per minute across all situations; FXS = Fragile X;
DS = Down s syndrome Typ = developmentally typical
In all three groups the distribution of behavior varied according to situation. Directive
behavior was used more frequently in the more challenging situations (matching task and
request situation) (range: 51–57%) than in the play situation (range: 19–30%). For reasons
of space detailed situation-specific data are not presented.
Scores from the PICCOLO rating scale indicated that mothers of children with DS
or FXS were less likely to support their children to do things on their own than the
mothers of developmentally typical children. Mothers of children with FXS had lower
scores for the item “djusts pace or activity according to child’s interests or needs, which is part
of the responsiveness category. This group also had lower score for the items Speaks in a
warm tone of voice and Shows emotional warmth. During the matching task situation mothers
of children with FXS spent less time following what their child was trying to do. Table 3
presents data for a selection of the PICCOLO items.
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Item (Domain)
Situation
Speaks in a warm
S1
tone of voice
FXS
DS
Typ
M (SD)
M (SD)
M (SD)
1.45
1.91
2.00
(0.52)
(0.30)
(0.00)
(Affection)
S2
S3
Shows emotional
S1
warmth
1.64
2.00
2.00
(0.51)
(0.54)
(0.00)
1.36
1.91
2.00
(0.67)
(0.30)
(0.00)
1.45
2.00
2.00
(0.52)
(0.00)
(0.00)
(Affection)
S2
S3
Adjusts pace or
S1
activity according
1.55
1.91
2.00
(0.52)
(0.30)
(0.00)
1.45
2.00
2.00
(0.69)
(0.00)
(0.00)
1.27
1.91
2.00
(0.65)
(0.30)
(0.00)
to child s interests
Pairs
Significance
Test
statistics
FXS – DS
.010
-7.500*
FXS – Typ
.002
-9.000**
DS – Typ
.608
-1.500
FXS – DS
.010
-6.000*
FXS – Typ
.010
-6.000*
DS – Typ
1.000
.000
FXS – DS
.010
-7.591*
FXS – Typ
.002
-9.045**
DS – Typ
.620
-1.455
FXS – DS
.001
-9.000**
FXS – Typ
.001
-9.000**
DS – Typ
1.000
.000
FXS – DS
.029
-6.000*
FXS – Typ
.006
-7.500**
DS – Typ
.586
-1.500
FXS – DS
.003
-7.500**
FXS – Typ
.003
-7.500**
DS – Typ
1.000
.000
FXS – DS
.003
-9.091**
FXS – Typ
.001
-10.545**
DS – Typ
.636
-1.455
FXS – DS
.029
-7.636*
FXS – Typ
.024
-7.909*
DS – Typ
.938
.273
FXS – DS
.586
-2.091
FXS – Typ
.002
-11.955**
DS – Typ
.010
-9.864*
FXS – DS
.157
-4.500
FXS – Typ
.005
-9.000**
DS – Typ
.157
-4.500
FXS – DS
.744
-1.227
FXS – Typ
.011
-9.545*
DS – Typ
.027
-8.318*
or needs
(Responsiveness)
Follows what the
S1
child is trying to do
0.45
1.00
1.00
(0.52)
(0.45)
(0.63)
(Responsiveness)
Supports the child
S1
to do things on his
0.55
0.73
1.55
(0.52)
(0.79)
(0.52)
or her own
(Encouragement)
S2
S3
1.45
1.73
2.00
(0.52)
(0.47)
(0.00)
0.36
0.45
1.18
(0.67)
(0.69)
(0.75)
Table 3: Scores per group (n = 11 per group); *p < .05; **p < .01; n.s. = not significant;
FXS = Fragile X DS = Down s syndrome Typ = developmentally typical
S1 = matching task situation, S2 = play situation, S3 = request situation
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Children’s Behavior
On average off-task behaviors were most frequent in children with FXS. Children with
FXS spent more time in off-task behaviors during the matching task situation and the play
situation than developmentally typical children. Both groups of children with disability
spent a lower proportion of their time on task than the group of developmentally typical
children. For all groups the percentage of on-task behavior was highest in the play
situation. Table 4 presents data for on-task behavior.
Situation
Matching task
Play situation
Request situation
FXS
DS
Typ
M (SD)
M (SD)
M (SD)
88%
91%
97%
(15)
(7)
(10)
95%
97%
100%
(6)
(7)
(1)
83%
95%
93%
(19)
(11)
(16)
Pairs
Significance
Test statistics
FXS – DS
.929
-.364
FXS – Typ
.007
-10.955**
DS – Typ
.009
-10.591**
FXS – DS
.033
-7.909*
FXS – Typ
.014
-9.136*
DS – Typ
.740
-1.227
n.s.
.223
3.003
Table 4: Children s on-task behavior by group (n = 11); *p < .05; **p < .01; n.s. = not significant;
FXS = Fragile X DS = Down s syndrome Typ = developmentally typical
There were also situational differences in behavioral adaption: in all groups rule-consistent
behavior was more common in the play situation than the other situations. Children with
FXS were less rule-consistent than the other groups. There were group differences in
behavioral adaptation in the play situation. Table 5 presents data on rule-consistent
behavior.
Situation
Matching task
Play situation
Request situation
FXS
DS
Typ
M (SD)
M (SD)
M (SD)
78%
90%
94%
(31)
(8)
(6)
88%
96%
100%
(19)
(10)
(1)
55%
66%
63%
(34)
(32)
(27)
Pairs
Significance
Test statistics
n.s.
.449
1,600
FXS – DS
.005
-10,864**
FXS – Typ
.001
-13,136**
DS – Typ
.557
-2,273
n.s.
.656
.842
Table 5: Children s rule-consistent behavior by group (n = 11); *p < .05; **p < .01; n.s. = not
significant FXS = Fragile X DS = Down s syndrome Typ = developmentally typical
Relationship between Child s and Mother s Behavior
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In order to identify relations between maternal and child variables Spearman s rank
correlations were used to evaluate relationships between maternal and child variables.
There was a small positive correlation between children s on-task behavior and maternal
maintaining behaviors (r = .370, p < .05) and a clear correlation between on-task behavior
and maternal encouragement (r = .581, p < .01). On-task behavior was clearly negatively
correlated with maternal restriction (r = -.618, p < .01). Rule-consistent behavior was
positively correlated with maternal maintaining behavior (r = .473, p < .01), regulation of
emotion (r = .475, p < .01) and encouragement (r = 676, p <.
. Children s rule-consistent
behavior was negatively correlated with maternal directive (r = -.522, p < .01) and
restrictive (r = -.656, p < .01) behaviors.
Maternal stress
The EBI the German version of the PSI differentiates between stress caused by the child s
behavior (child domain) and parental distress (parent domain). Total parental stress levels
were similar in mothers of children with FXS and DS. Mothers with developmentally
typical children reported the lowest levels of stress. Mothers of children with FXS reported
high stress arising from the hyperactivity or distractibility of their child. Mothers of children
with DS reported more stress related to how demanding their child was than mothers in
the other groups. Table 6 shows the differences between the three groups. Both groups of
mothers of children with disability reported high levels of stress in the role restriction
subscale of the parent domain. Mothers of children with FXS also reported high levels of
stress in the competence and depression subscales. Mothers of children with FXS reported
more competence-related stress than mothers of children with DS. These data are presented
in TablesTable 6 and Table 7.
EBI Subscales
FXS
DS
Typ
Child domain
M (SD)
M (SD)
M (SD)
8.36
6.64
4.73
(1.12)
(1.96)
(1.35)
Hyperactivity or distractibility
Mood
Acceptability
Demandingness
6.73
4.55
4.27
(2.15)
(1.57)
(1.90)
8.09
7.45
5.00
(0.30)
(1.04)
(0.89)
8.55
7.91
6.45
(0.93)
(2.02)
(1.21)
Pairs
Significance
Test statistics
Typ – DS
.038
8.318*
Typ – FXS
.000
16.227***
DS – FXS
.049
7.909*
Typ – DS
.798
1.045
Typ – FXS
.009
10.682**
DS – FXS
.018
9.636*
Typ – DS
.001
13.227**
Typ – FXS
.000
18.237***
DS – FXS
.197
5.045
Typ – DS
.005
10.864**
Typ – FXS
.001
13.409**
DS – FXS
.515
2.545
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Adaptability
7.18
5.91
5.27
Typ – DS
.263
4.545
(1.47)
(2.34)
(1.55)
Typ – FXS
.012
10.182*
DS – FXS
.165
5.636
Table 6: Group comparisons of maternal stress (EBI subscales, child domain); Stanines are
presented; *p < .05; **p < .01, ***p<.001; n.s. = not significant; FXS = Fragile X;
DS = Down s syndrome Typ = developmentally typical
Subscales EBI
FXS
DS
Typ
Parent domain
M (SD)
M (SD)
M (SD)
7.00
6.45
5.00
(1.34)
(2.12)
(1.55)
Parental attachment
Isolation
Competence
Depression
Health
Role restriction
Spouse
6.09
6.82
4.18
(2.47)
(1.66)
(1.08)
8.00
5.73
4.82
(0.89)
(2.20)
(1.83)
8.00
6.64
6.73
(1.34)
(2.58)
(1.10)
6.73
6.55
5.36
(1.01)
(2.25)
(2.06)
7.27
7.09
4.82
(1.74)
(1.38)
(1.25)
6.00
6.44
5.73
(1.55)
(1.88)
(1.35)
Pairs
Significance
Test statistics
Typ – DS
.069
7.364
Typ – FXS
.009
10.636**
DS – FXS
.419
3.273
Typ – DS
.003
12.045**
Typ – FXS
.031
8.818*
DS – FXS
.429
-3.227
Typ – DS
.332
3.955
Typ – FXS
.000
14.455***
DS – FXS
.010
10.500*
n.s.
.078
5.102
n.s.
.215
3.075
Typ – DS
.004
11.727**
Typ – FXS
.001
12.955**
DS – FXS
.763
1.227
n.s.
.790
.472
Table 7: Group comparisons of maternal stress (EBI subscales, parent domain); Stanines are
presented; *p < .05; **p < .01, ***p<.001; n.s. = not significant; FXS = Fragile X;
DS = Down s syndrome Typ = developmentally typical
Maternal Stress and Parenting Style
There was a negative correlation (r = -.463, p < .01) between total EBI score and maternal
maintaining behaviors. Total EBI score was positively correlated with maternal directive
behavior (r = .502, p < .01) and restrictive behavior (r = .502, p < .01). EBI score was also
related to the PICCOLO ratings. EBI score was clearly negatively correlated with maternal
responsiveness (r = .482, p < .01) and encouragement (r = .582, p < .01).
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Discussion
Mother-Child Interaction
The goal of the study was to explore patterns of maternal behavior in mother of children
with two different genetic syndromes. We found that mothers of children with FXS or DS
adapt their behavior to the syndrome-specific characteristics of their child.
Wheeler et al. (2007) showed that mothers of children with FXS tend to use an
interactive style that combines high proportions of maintaining (67%) and directive (27%)
behaviors; we corroborated this finding in a play context; however we found that in a
matching task situation and a request situation these mothers made much more use of
directive behavior compared to the play situation. Wheeler et al. (2010) confronted
children with FXS with an unsolvable task; in this situation 26% of the mothers used
negative behavioral control and 20% used only directive and negative control behaviors.
Taken together these findings suggest that in challenging situations mothers of children
with FXS make more extensive use of directive and controlling behaviors than mothers of
children with DS or mothers of developmentally typical children. We found that
compared with mothers of children with DS and mothers of developmentally typical
children the mothers of children with FXS used a generally more controlling style during
interactions with their children. Based on observations of mothers with children with FXS
and their developmentally typical sibling Sterling et al. (2012) reached a similar
conclusion; their mothers were responsive to both children but were less flexible when
interacting with the child with FXS. Sterling et al. characterized the behavior of mothers
towards the child with FXS as less conversational and more directive. Our results, based
on the coding system and the PICCOLO rating scale confirm the earlier findings. One
possible explanation for the pattern of maternal interactions with children with FXS is that
these children tend to be impulsive and mothers feel a greater need to structure and direct
their child s activity. The results of this study indicate that mothers of children with DS
made more use of directive behaviors than mothers of developmentally typical children;
however on the whole there were few differences between the interactions of mothers of
children with DS and mothers of developmentally typical children. Directive and
responsive behavior are not mutually exclusive and both types of behavior were used
extensively by mothers of children with DS. Tannock (1989) and Roach et al. (1998)
reported similar findings. We also observed that mothers of children with DS confirmed
and praised their children more often than mothers in the other groups. This may be
because children with DS often have motivational problems and can be described as
externally oriented. Confirming and praising the child continuously may be the mother s
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way of trying to motivate her child to persist. It is likely that children with DS will remain
dependent on this form of external regulation.
Analysis of the relationship between child and maternal behavior indicated that
maternal restrictive behaviors were associated with off-task behavior in the child, whereas
on-task behavior was related to maternal behaviors that encouraged the child to handle
toys or to extend his or her activity. Children with FXS tend to have problems switching
attention within a task (Cornish et al., 2004), so it is possible that if a mother directs the
child s attention too forcibly by offering new toys or making demands the child s
attentional system may be overloaded and consequently he or she may turn away.
Attentional problems may also arise if the mother does not respond to the child s interests.
In this study the mothers of children with FXS followed their child s goals and activity to a
lesser extent than mothers in the other groups. This may be because they felt they needed
to be more controlling in order to reduce their child s impulsiveness.
Maternal Wellbeing and Maternal Behaviors
Both groups of mothers with children with disability reported stress related to parenting
and child care. Mothers of both groups reported experiencing stress caused by role
restriction (parent domain). Mothers of children with FXS also reported high stress related
to child behaviors such as hyperactivity. This finding has been reported previously
(Sarimski, 2010; Wheeler et al., 2008). A study based on qualitative interviews (Wheeler et
al., 2008) concluded that mothers of children with FXS experienced stress when dealing
with their child s challenging behaviors and when they did not know how to help or
control their child. Moreover, one third of mothers reported that parenting and family life
in general were stressful. Amongst the reasons given were having too little time and the
feeling of being pulled in too many directions (Wheeler et al., 2008).
In this study both groups of mothers of children with disabilities had similar EBI
scores. The findings that mothers of children with FXS made more use of restrictive
behaviors and that restrictive behavior was positively correlated with maternal stress
suggest that in mothers of children with FXS child characteristics are the main source of
stress.
Limitations
Our results only provide a glimpse into how different genetic syndromes influence
interactional style in mother-child dyads, owing to the use of a small, selective sample and
a single observation session per dyad. It is also important to bear in mind that mothers
participated voluntarily; it seems likely that those who participated were probably
relatively secure in their parenting competence. Another critical issue is that data on
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children s developmental age were based on a questionnaire rather than direct testing,
which would have had greater validity. The matching task may not have been similarly
demanding for all the children as their chronological age varied. Given that behavior may
be modified under open observation it would have been useful to get more detailed
information about the children s behavior, e.g. via the Child Behavior Check List
(Achenbach & Rescorla, 2000). Finally, sequence analysis is needed to determine whether
specific maternal behaviors tend to be followed by specific child behaviors (and vice versa);
such analysis would have provided more insight into interaction patterns.
Conclusion
These limitations notwithstanding, this study provides important information about how
maternal interaction behavior is related to factors such as situation, intellectual disability,
behavioral phenotype and maternal stress.
Firstly, in all three groups maternal behavior was influenced by the context or
situation. Secondly, both groups of mothers of children with disability made more use of
directive behaviors than mothers of developmentally typical children. This finding makes
it clear that some differences in maternal behavior are related to having a child with an
intellectual disability. Our third finding was that maternal behaviors can be related to the
behavioral phenotype associated with the child s genetic syndrome. Mothers of children
with DS made heavy use of confirmation and praise and this could be linked to the
motivational problems and external orientation which are known to characterize DS. The
extensive use of directive and restrictive behaviors by mothers of children with FXS may
be due to the inhibitory control deficit and hyperactivity these children display. Finally,
the study demonstrated that maternal stress affects mother-child interactions.
These findings have implications for practice and research. Families who need help
with interacting with a child with a disability should be offered syndrome-specific
guidance. There also seems to be a need to find ways of reducing parental stress and the
first step towards this is to identify the main sources of stress. Further research might
include an investigation with a larger sample and extending the comparisons to include
other syndromes, e.g. Prader-Willi syndrome. Finally, a longitudinal investigation could
be used to determine how parental factors influence child development.
Acknowledgements
The study presented in the following article was published as a doctoral dissertation at the
University of Education in Heidelberg:
Sansour, Teresa (2016): Analysen des Interaktionsverhaltens von Müttern im Kontext des
Fragilen-X-Syndroms und des Down-Syndroms. Eine empirische Vergleichsstudie.
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SYNDROME, DOWN’S SYNDROME OR TYPICAL DEVELOPMENT
Dissertation an der Pädagogischen Hochschule Heidelberg. Online in: http://opus.phheidelberg.de/frontdoor/index/index/docId/146
The following article was not yet published.
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