Correlates of maternal behaviours in mothers of children with fragile X syndrome

Journal of Intellectual Disability Research doi: 10.1111/j.1365-2788.2006.00896.x

pp

–

©

The Authors. Journal Compilation ©

Blackwell Publishing Ltd

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Blackwell Publishing LtdOxford, UKJIRJournal of Intellectual Disability Research

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©

The Author. Journal compilation ©


6447462

Original Article

Maternal behaviours and FXSA. Wheeler

et al.

Correspondence: Dr Anne Wheeler,

S. Greensboro Street, CB#

, Chapel Hill, NC

-

, USA (e-mail: [email protected]).

Correlates of maternal behaviours in mothers of children with fragile X syndrome

Anne Wheeler,

1

Deborah Hatton,

1

Alison Reichardt

2

& Don Bailey

3

1

FPG Child Development Institute, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA

2

Durham County Public Schools, Durham, NC, USA

3

RTI International, Durham, NC, USA

Abstract

Background

The behaviours of

mothers of chil-dren with fragile X syndrome (FXS) with their affected children were examined during planned observations in their homes. The goal of this study was to describe concurrent maternal interactive behaviour and the factors that influence the type and frequency of these behaviours within this group.

Methods

The frequency of

maintaining

,

directive

and

scaffolding

behaviours and the extent to which the mothers displayed

warm sensitivity

and

restrictions

were examined within a

-min naturalistic observa-tion and a

-min toy play observation. Rating scales and parent questionnaires were used to assess selected maternal mental health factors, including depression, anxiety, stress and social support. The cognitive status of mothers and developmental and behavioural abilities of children were also examined.

Results

The women in this study used primarily maintaining behaviours in interactions with their children. Maintaining behaviours and warm sensitiv-ity were consistent across structured and unstruc-tured settings, while directive, scaffolding and restricting were not correlated across the two settings. Child receptive language skills were related to higher rates of maintaining and scaffolding behaviours. The

women reported clinically significant levels of stress, depression and anxiety at a prevalence rate higher than that of the general public. Child behaviour prob-lems contributed to maternal stress, and mothers with higher stress engaged in fewer interactions with their children.

Conclusions

The relations between maternal stress, child problem behaviour and number of interactive behaviours exhibited by the mothers in this study provide information that can inform interventions and provide direction for future research exploring environmental influences on the development of chil-dren with fragile X syndrome.

Introduction

Fragile X syndrome (FXS) is the leading hereditary cause of developmental disability. Children with FXS are generally characterized as having mild to moder-ate intellectual disabilities (Bailey

et al.

; Maz-zocco

), attention deficit hyperactivity disorder, autistic behaviours and poor eye contact (Merenstein

et al.

; Hatton

et al.

); however, there is con-siderable variability in the phenotypic expression. Although the genetic basis of FXS has received con-siderable attention over the past decade, there have been few studies aimed at exploring environmental influences that might explain the wide variability in developmental and behavioural outcomes. No studies to date have explored maternal–child interactions in

Journal of Intellectual Disability Research

A. Wheeler

et al

. •

Maternal behaviours and FXS448

©



dyads affected by FXS and the within-group variables that may influence this relationship.

However, there is strong evidence that mother–child relationships early in life can have long-term effects on both the mother and child. Early reciprocal parent–child interactions play an important role in the development of children’s affect regulation and subsequent cognitive, social and behavioural out-comes in kindergarten (NICHD Early Child Care Research Network

). In addition, positive or negative everyday interactions, as well as the long-term outcomes of those interactions on the child, contribute to the parents’ perceptions of success and well-being (Dumas

et al.

; Naerde

et al.

).The purpose of this study was to examine maternal

interactive behaviours in dyads affected by FXS. The conceptual model used in this study recognizes the interrelations among maternal interactive behaviours and two categories of variables thought to influence maternal behaviours – maternal well-being (e.g. stress, depression, anxiety, social support) and child characteristics (e.g. age, behaviour, developmental level). Therefore, three primary research questions guided this study: (

) To what extent do mothers of children with FXS use specific behaviours (maintain-ing, directing, scaffolding and warm sensitivity) in interactions with their young children with FXS? (

) What is the association of these behaviours, as exhib-ited during a

-day observation, with child character-istics such as developmental status, challenging behaviours and autistic symptoms? and (

) What influence does maternal mental health have on the type and frequency of interactive behaviours?

Maternal interactive behaviours

Although child characteristics and the reciprocity of the interaction are clearly related to effective caregiv-ing, there is some evidence that maternal behaviours have a significant impact on child development (Yoder

et al.

; Hutchings

et al.

). As mothers tend to have the most contact with very young chil-dren, they are often the most important and effective teachers of their children. For example, when moth-ers actively maintain their child’s attention by asking questions, commenting, or physically guiding their child towards an object of interest, they provide chil-dren with knowledge that their interests and thoughts are important. Children whose mothers display more

maintaining and responsive behaviour during the first few years achieve language milestones earlier and with better proficiency (McCathren

et al.

; Yoder

et al.

; Tamis-LeMonda

et al.

), have higher scores on cognitive tests (Bornstein & Tamis-LeM-onda

,

; Landry

et al.

,

a,

), and have better developed social skills and are more resilient to emotional problems (Goldberg

et al.

; Landry

et al.

b; Calkins

et al.

; Kochanska

et al.

).Other maternal behaviours that are readily observ-

able and that have been shown to influence child development are directive behaviours, scaffolding and warm sensitivity. Directive behaviours involve structured requests by a mother to her child, and can either be responsive (redirecting) or non-responsive (do not consider the object or activity to which the child is currently attending). Responsive directive behaviour allows mothers to increase their children’s arousal and help them focus on relevant stimuli. This may be an especially useful technique for mothers of younger children, or children whose development is delayed (Marfo

). Mothers may have specific difficulties responding to children with disabilities who are likely to behave less responsively and maturely (Barnard

). In contrast, unresponsive directive behaviours have been shown to negatively influence independence, social skills and initiating abilities in typically developing children over

.

years of age (Landry

et al.

). In general, a combination of responsive maintaining and directing may be the optimal strategy for children who are chronologically or developmentally younger than

years old (Rosen-berg & Robinson

).Scaffolding helps children to focus and organize

their verbal and physical responses to their environ-ment (Smith

et al.

) and helps guide children’s behaviour and problem solving by providing them with natural links among objects, actions and catego-ries (Bridges

). Examples of scaffolding behav-iour include using questions, directives or statements that associate objects and specific locations; relating an object, activity or topic in which the child is engaged to a previous experience; associating feelings and emotions with a reason for the emotion; and linking objects with specific categories. Maternal scaffolding has been linked to higher vocabulary and mental development in toddlers (Stevens

et al.

), school readiness (Price

et al.

), increased


A. Wheeler

et al

. •

Maternal behaviours and FXS449

©



problem-solving skills (Freund

) and higher lev-els of independent social and cognitive functioning (Smith

et al.

).Positive affect, when combined with maternal

warmth (physical and verbal affection) and flexibility (sensitivity and promptness to child’s cues, patience), also has been shown to have a positive influence on child development (Bornstein & Tamis-LeMonda

; DeWitt

et al.

; Landry

et al.

a; Hammond

et al.

; Miller-Loncar

et al.

).

Factors that influence maternal behaviours

Child characteristics

Parents often employ a combination of behaviours at any given time in order to assist in teaching and managing their child’s behaviour. Many factors, both internal and external, may impact how and when parents utilize these strategies. For example, the lim-itations inherent in a developmental disability, like FXS, may make it more challenging for mothers to respond to their children (Marfo

; Marfo

et al.

; Brophy & Dunn

). Mothers’ ability to interpret and respond to children’s emotional and communicative cues depends on children’s ability to send undistorted signals. Therefore, any cognitive, sensory, emotional or behavioural factors that influ-ence the child’s actions could in turn challenge the parent’s ability to respond (Dunst & Trivette

). In dyads in which the child has a disability, mothers tend to be more directive, provide more stimulation, and take a more dominant role in interactions than do mothers of typically developing children (Marfo

; Doussard-Roosevelt

et al. ).

Maternal mental health

Maternal depression has been repeatedly shown to influence the ways mothers interact with their chil-dren (Jouriles et al. ; Goldsmith & Rogoff ; Murray et al. ; Gondoli & Silverberg ; Jame-son et al. ; Stanley et al. ). Mothers who have depressive disorders typically use fewer verbal interactions with their children (Jouriles et al. ), look at and talk to their infants less, show fewer positive facial expressions, and provide less overall stimulation than non-depressed mothers (Field , ). Depression may influence the mother’s expec-tations and ability to respond to her child’s cues. As

a result, maternal depression may negatively affect child social and language development (NICHD ).

Stress and anxiety may also influence maternal behaviour, indirectly by increasing a mother’s risk to develop a depressive disorder and directly by limiting her emotional and physical energy to engage with her child (Rodgers ; Bright & Hayward ). Social support networks may help buffer the influences of stressful situations and help reinforce positive inter-actions between mother and child (Rodgers ).

Mothers of children with disabilities have been shown to have higher levels of depression, stress and anxiety than the general population (Dumas et al. ; Blacher et al. ; Hoare et al. ; Veisson ; Olsson & Hwang ; Baker et al. ; Emerson ; Glidden & Schoolcraft ; Hast-ings ; Saloviita et al. ), and carriers of FXS may have a greater biological predisposition to social anxiety and depression (Frank et al. ; Mazzocco ; Lesniak-Karpiak et al. ). Because of the difficult behaviours exhibited by children with FXS, the potential stress of receiving a diagnosis that impacts the entire family, and the increased chance of experiencing higher levels of anxiety and depres-sion, mothers of children with FXS are at-risk for experiencing higher levels of stress and lower levels of well-being. In fact, Abbeduto et al. () found that mothers of children with FXS experienced increased levels of pessimism about their children’s future and decreased levels of perceived closeness with their children than mothers of children with Down syndrome. However, while levels of depression in mothers of children with FXS were found to be higher than those of the average population, they did not differ from those of mothers of children with Down syndrome or autism.

Therefore, mothers of children with FXS may experience both a biological predisposition for affec-tive disorders through their premutation status as well as increased stress related to raising a child with sig-nificant behavioural difficulties and developmental limitations. The ‘double-risk’ these women face sug-gests that this population may experience a greater vulnerability than mothers of children with other dis-abilities. As a result, there is a great need to better understand the mental health of these mothers and the nature of their interactions with their children in order to inform effective interventions that can lead


A. Wheeler et al. • Maternal behaviours and FXS450

© The Authors. Journal Compilation © Blackwell Publishing Ltd

to improved outcomes for the child and family. We examined maternal behaviours and factors associated with those behaviours in FXS in order to gain insight into the environmental and familial factors that influ-ence the cognitive, behavioural and emotional devel-opment of children with FXS.

Methods

Participants

Families were recruited from an ongoing, longitudi-nal study of children with FXS, as well as from national fragile X listservs and web sites. The partic-ipants were mothers and their biological children with FXS. Confirmation of the FXS diagnosis was made through review of genetic reports. The chil-dren’s ages ranged from to months. Children who were not yet walking were not included in the study. Although all of the mothers in this study were carriers of FXS, premutation or full mutation status was unknown for most of them. With the exception of eight women who had participated in an earlier

study of genetic status, the knowledge of carrier sta-tus was based solely on the mother’s report. Of the eight mothers whose genetic status was known, all were premutation carriers. All mothers scored in the average to superior range of intellectual ability as measured by the Wechsler Abbreviated Scales of Intelligence (WASI; Psychological Corporation ), and the majority of women had some college education. Demographic information for participants is provided in Table .

Instrumentation

Observation of maternal behaviour

Maternal behaviour was observed and coded using adaptations of protocols designed by Landry and col-leagues (Smith et al. ; Hammond et al. ; Landry et al. ; Smith et al. ). Mothers were asked to perform daily activities during an hour-long observation session in their homes in which they were requested to stay in the same room with their child and to engage in typical daily routines with their child

Child age (months) Mean = 45.32SD = 16.74Range = 18–72

Child gender 20 male, 4 femaleChild ethnicity* 88% (21) Euro-American

4% (1) African American4% (1) Hispanic American4% (1) Asian American

Maternal age (years) Mean = 34SD = 10.95Range = 25–48

Marital status 88% (21) married to father of the child at time of assessment

12% (3) were divorcedEmployment status 76% (19) were stay-at-home mothers/worked from home

14% (5) worked out of the home at least part-timeMaternal education 8% high school degree or less

44% some college47% college grad or more

Maternal IQ (WASI)FSIQ (two subtests) Mean = 110.56 SD = 10.95 Range = 90–131VIQ Mean = 106.96 SD = 13.61 Range = 77–137PIQ Mean = 113.84 SD = 11.20 Range = 88–134

*In one family, the mother was Euro-American, father was not, therefore child was of a different ethnicity than the mother.FSIQ, Full Scale IQ; PIQ, Performance IQ; SD, standard deviation; VIQ, Verbal IQ; WASI, Wechsler Abbreviated Scales of Intelligence.

Table 1 Maternal and child demographics (n = dyads)




(e.g. bathing, feeding or dressing). No other direc-tions were given during this observation period. In addition, mothers also participated in a -min toy play interaction with their child. Two researchers observed both the naturalistic and the toy play ses-sions, and reliability checks were completed for % of the assessment sessions throughout the data col-lection phase. The two researchers were trained using continuous coding of both video and live models of maternal–child interactions. A minimum reliability coefficient of . on all maternal behaviours coded was obtained before data collection, and minimum inter-rater reliability of . was maintained through-out the data collection phase.

Maternal responsiveness was coded using a frequency count of maintaining and directive behaviours.• Directive behaviour, coded regardless of the child’s focus of attention, was defined as any verbal request that provided structured information about what was expected of the child.• Maintaining behaviour was defined as verbal or non-verbal behaviour that provided choices or feed-back regarding an activity or object to which the child was already visually or physically attending, or to which the child was attempting to attract the mother’s attention.

In addition to maintaining and directive behav-iours, scaffolding and restricting behaviours were coded. Scaffolding was coded if the mother provided a verbal or non-verbal link between objects, persons, activities or functions. A restriction was coded if the mother used a verbal or non-verbal cue or command limiting the child’s words or actions. Coding was continuous, with a -s time unit. If s elapsed, a separate interaction was coded. For example, if the mother made rapid or continuous requests, questions or comments, without giving the child a minimum of s to respond, this series was coded as one event. Scores were provided in the form of frequency counts of the specific behaviours (maintain, direct) and spe-cific characteristics of the behaviours (scaffolding, restrictions) relative to the total number of behaviour events observed. Therefore, time (i.e. min for the naturalistic and min for the toy play sessions) was used to standardize the observations across individu-als. Because the behaviours were coded in terms of frequency count, and a new behaviour was only counted if there was a -s time lapse (i.e. for some

mothers one behaviour lasted longer than for others), it was not possible to analyse the data by time. Rather, within the given time frame, the number of overall behaviours and the percentage of specific observed behaviours were coded as variables for anal-ysis. In previous studies, the interclass correlations between maintaining and directive behaviours have ranged from . (Smith et al. ) to . (Smith et al. ).

Warm sensitivity was measured using the Maternal Rating Scale (MRS; Dewitt et al. ; Landry et al. a). The MRS measures a number of constructs and behaviours based on the work of Ainsworth et al. (). Each of six constructs (maternal positive affect, maternal warmth, maternal flexibility, corpo-ral discipline, verbal content and maternal punitive-ness) is measured on a -point scale, yielding a score for each interval between and . Higher scores indicate more warm sensitivity and less punitiveness. Mothers were rated using this scale at three -min intervals during the -min naturalistic session and once following the toy play session. Ratings on each of the constructs were averaged across the three -min intervals, with the toy play interval analysed separately.

Maternal measures

The women were asked to complete a demographic form that asked for information regarding their ethnic background, maternal age, marital status and mater-nal education. The WASI was used to assess the cog-nitive functioning of the mothers following the observation sessions. In addition, four areas related to maternal mental health were explored. Parental stress was measured using the short version of the Parenting Stress Index (PSI; Abidin ). The PSI Short Form is a -item self-scoring questionnaire/profile, which yields a Total Stress score based on three scales: parental distress, parent–child dysfunc-tional interaction and difficult child. Depression and anxiety were measured with the Beck Depression Inventory-II (BDI-II; Beck et al. ) and the Beck Anxiety Inventory (BAI; Beck & Steer ). The BDI-II and BAI are self-report measures consisting of items which tap recent depression and anxiety symptoms. The BDI-II and BAI were chosen for their good psychometric properties, and because they are instruments used to measure current symptoms of




depression and anxiety. The women in this study were also asked questions about their mental health history and treatment in an interview following the assessment.

Finally, the Family Support Scale (FSS; Dunst et al. ) was completed by the mothers in order to examine perceived support each woman felt she received from others in her environment. The FSS consists of items rated on a -point scale ( = not at all helpful to = extremely helpful) that assess how helpful particular people or groups are to a family in raising a young child.

Child characteristics

Information about the developmental, behavioural and autistic status of all of the children was gathered during each assessment. The Mullen Scales of Early Learning (Mullen ) were used to assess the developmental status of the children. The mothers completed the Child Behaviour Checklist (CBCL; Achenbach & Rescorla ) in order to assess child problem behaviour. Finally, the Childhood Autism Rating Scale (CARS; Schopler et al. ) was used to assess autistic behaviours of the children. This scale, completed by the researcher following observa-tion of the child, assesses autistic characteristics in areas. It is relatively simple to use, and has been shown to have high agreement with psychiatric diag-nostic criteria for autism as outlined in the DSM-IV (Rellini et al. ).

All of the maternal and child measures used in this study are frequently used and have good psycho-metric properties, as reported in their respective manuals.

Procedures

Following Institutional Review Board (IRB) approval, families already enrolled in a larger longitudinal study of FXS whose children were between the ages of and were contacted via letters or postcards and asked to participate in the study. A recruitment announcement was posted on the FRAXA listserv and the Carolina Fragile X Project and National Fragile X Foundation web sites. Interested mothers were asked to call the project’s toll free number or send an email expressing a desire to learn more about the study. Following receipt of written informed

consent, a visit was scheduled. Each visit took place during the time of day each parent requested. Parents were asked to choose the time of day based on the family’s typical routines and the time of day they thought their child would be most attentive.

All families were visited at their homes by one or two researchers. Approximately – weeks prior to the visit, the CBCL, PSI and FSS were sent to the mother, and she was asked to complete them prior to the visit. At the beginning of each visit, the researchers spent approximately – min with the mother and her child, during which basic demo-graphic data were collected, the observation proce-dures were described, questions were answered, and rapport was established. The mother was then asked to continue with the family’s daily routine, while stay-ing in the same room with her child and engaging in typical daily activities (e.g. bathing, feeding and dressing). Following this -min observation period, the mother was asked to participate in a -min toy play session with her child. Using toys provided by the researchers (a puzzle, three puppets, a school bus, legos, a shape sorter, two books, stacking cups and an infant stimulation toy), the mother was asked to play with her child ‘the way she normally does’, and she selected the type and number of toys for play. This session provided a direct observation of a spe-cific, structured child-related interaction.

After the observation period, mothers were asked to complete the BDI-II and BAI, were assessed using the WASI, and completed a short interview on their mental health history. During this time, the child was assessed using the Mullen and CARS. The average length of time for the visit was . h.

Data analysis

Following each visit, data were entered into a data-base and independently verified by a second researcher. Preliminary analyses were used to assess the distributions and correlations of all variables.

Following examination of descriptive results, a series of Pearson correlations were run to identify relationships among the various sets of variables (e.g. maternal measures of depression, anxiety, stress, social support and IQ) for data reduction. Variables for the regressions were chosen based on the corre-lation results and which variables were theoretically most salient. Child age and gender served as control




variables for all correlation and regression models. The Benjamini-Hochberg method for adjusting P-values was calculated to control for type I error when running multiple tests (Benjamini & Hochberg ).

For observational data, the percent of total mater-nal behaviours was used for all analyses. For example, during the -min unstructured observation, a mother may have engaged in observed behav-iours. Of those behaviours, may have been main-taining behaviours. Therefore, that mother will have a maintaining ‘score’ of . (%). In this way, the specific observed behaviours were transformed into meaningful data that could be compared across subjects.

Results

Maternal behaviours

On average, the mothers in this study exhibited (SD = ; Range = –) interactive behaviours during the -min naturalistic observation. Table describes the percentage of observed behaviours the mothers engaged in during the specific target actions. On average, the majority of observed behaviours were maintaining behaviours (% during the naturalistic, % during toy play), indicating that mothers fol-lowed their child’s focus of attention more than they chose to direct the child. The percentage of observed scaffolding behaviours was much lower (% for the naturalistic, % during toy play), indicating that while the mothers generally maintained their child’s focus of attention during the interactions, they did not as frequently use those interactions to teach their

child. Maintaining scores ranged from . to . (SD = .) and Scaffolding scores ranged from . to . (SD = .), suggesting variability in the use of these interaction strategies.

On average, the mothers exhibited high levels of warm sensitivity on the MRS (M = ., SD = .) and displayed highly consistent warmth over both observation periods (r = ., P < .). Mothers exhibited little to no negative behaviours (corporal discipline, verbal content and maternal punitiveness). There was more variability on the positive behav-iours, especially positive affect (M = ., Range = .–.). The internal consistency for the MRS for this sample was good (toy play α = .; -min session α = .).

Maintaining behaviours were highly consistent across the two observation sessions (r = ., P < .); while directive, scaffolding and restrictive behaviours were not at all correlated between the two sessions. Therefore, in further analyses, only the MRS and maintaining behaviours from the hour-long session were used while directive, scaffolding and restrictive behaviours during the two observation ses-sions were examined separately.

Child characteristics and maternal behaviours

Scores from the Mullen Early Learning Scales (com-posite and sub-scale) indicated that the children, on average, displayed significant delays across all devel-opmental domains. On average, the children in this study were reported to have internalizing behaviours and total problem behaviours in the borderline range on the CBCL. Most children exhibited some autistic behaviours, as measured by the CARS. However, only one child met criteria for autism. See Table for descriptive data on child variables.

Based on our correlation analysis, we selected the Receptive Language Age Equivalent (RLA) from the Mullen as a measure of child developmental status. Because of the high correlation between the RLA on the Mullen and the CARS scores, and because the CARS scores were not normally distributed in our sample, we chose to not use CARS as a variable in the regression analysis. Please see Table for all of the correlations among child characteristics. Based on the literature outlining the impact of child prob-lem behaviours on parental well-being and child out-comes, we chose to use the CBCL Total Problem

Table 2 Descriptive data for maternal behaviours (percent of totalnumber of observed behaviours)

n Mean SD Range

Maintaining (hour) 24 64 10 44–86Directive (hour) 24 27 7 12–38Scaffolding (hour) 24 19 8 4–31Zaps/restrictions (hour) 24 9 6 2–23Maintaining (toy play) 24 67 16 25–98Directive (toy play) 24 27 12 2–53Scaffolding (toy play) 24 20 10 3–44Zaps/restrictions (toy play) 24 7 6 0–22

SD, standard deviation.




Behaviour score in the regression analysis. Gender and child age were also included as covariates. Recep-tive language was found to be a significant predictor of maintaining behaviour (r2 = ., β = ., P < .) and scaffolding behaviour (r2 = ., β = ., P < .) during the naturalistic observa-tion. Following correction for multiple tests, no other

child characteristics were found to be related to any of the maternal behaviours.

Maternal mental health and behaviours

Information on mothers’ current treatment and men-tal health history was secured in order to further

n Mean SD Range

Mullen early learning composite* 24 58.83 15.68 49–108Mullen expressive language† 24 25.93 12.14 20–51Mullen receptive language† 24 27.00 11.81 20–59Mullen fine motor† 24 26.92 9.16 20–53Mullen visual reception† 24 27.38 12.17 20–63CBCL total†‡ 24 61.0 9.99 43–80CBCL internalizing†‡ 24 60.42 8.45 43–78CBCL externalizing†‡ 24 58.12 11.25 40–82CBCL emotional†§ 24 60.08 8.12 50–77CBCL anxious/depressed†§ 24 56.04 6.67 50–70CBCL somatic†§ 24 56.29 7.37 50–76CBCL withdraw†§ 24 64.63 8.93 51–82CBCL sleep problems†§ 24 60.17 13.84 50–100CBCL attention problems†§ 24 65.95 9.20 51–80CBCL aggression†§ 24 57.63 9.34 50–84CARS¶ 24 24.10 5.59 15–29.5

*Mean = , SD = .†Mean = , SD = .‡Clinically significant = ≥; Borderline = –.§Clinically significant = ≥; Borderline = –.¶Clinical cut-off = ; Severe autism +.CARS, Childhood Autism Rating Scale; CBCL, Child Behaviour Checklist; SD, standard deviation.

Table 3 Descriptive data for child characteristics

Table 4 Correlations among child characteristics

CBCLtotal CARS

MullenEL

MullenRL

MullenVR

MullenFM

MullenELC Gender

Childage

CBCL 0.077 0.220 0.185 0.119 0.147 −0.190 0.139 0.366CARS −0.503* −0.552** −0.463* −0.425* −0.646*** 0.509* 0.083Mullen EL 0.915*** 0.777*** 0.775*** 0.334 −0.166 0.704***Mullen RL 0.860*** 0.818*** 0.437* −0.280 0.621***Mullen VR 0.852*** 0.373 −0.361 0.617***Mullen FM 0.417* −0.328 0.615*Mullen ELC −0.587** −0.292Gender 0.177

*P < .; **P < .; ***P < ..CARS, Childhood Autism Rating Scale; CBCL, Child Behaviour Checklist; EL, expressive language; ELC, early learning composite; FM, fine motor; RL, receptive language; VR, visual reception.




describe the well-being of these mothers. Maternal reports of depression and anxiety are displayed in three ways in Fig. : () the percentage of mothers who obtained clinical scores on the BDI-II and BAI (i.e. mothers’ perceptions of depressed and anxious symptomology felt within weeks of the assessment); () the percentage of mothers who reported they were currently being treated for depression or anxiety through medication or therapy (i.e. those who were currently experiencing depression or anxiety which warranted treatment, but because of treatment may not have been experiencing acute symptomology); and () the percentage of mothers who reported any history of depression or anxiety.

While % (n = ) indicated mild to moderate lev-els of depressive symptomology on the BDI, % (n = ) reported being treated for depression at the time of the assessment, and % (n = ) reported a personal history of depression. Eight (%) of the mothers reported experiencing mild to severe anxiety at the time of the assessment, % (n = ) were being treated for anxiety with medication, and % (n = ) had a personal history of an anxiety disorder.

Overall, % (n = ) of the mothers who com-pleted the PSI reported experiencing clinically signif-icant levels of overall parenting stress. When the sub-scales indicating the types of stress being experienced were examined, % (n = ) reported clinically sig-nificant stress specific to raising a difficult child, % (n = ) reported clinically significant stress specific to

the dysfunction in the parent–child interactions, and % (n = ) reported clinically significant stress due not to the target child, but to parenting stress in general.

On the FSS, the mothers reported moderate levels of support (M = ; SD = .). In general, the moth-ers reported that they received the most support from their spouse or partner and professional helpers. Social groups, church members and coworkers were reported as providing the least support on average.

Depression was highly correlated with both stress (r = .; P < .) and anxiety (r = .; P < .). IQ, maternal age and social support were not as highly correlated with each other or the other mental health variables (see Table ). Although maternal education has been found to be a predictor of child outcome in past studies, we had very little variability in education status in our sample. There-fore, we did not include it in further analyses. In order to reduce the number of variables, and because of its high correlation with depression, we did not include anxiety in further analyses. Both stress and depression were retained because of their importance for maternal behaviours in the literature. Results of this regression analysis indicated that stress was a significant predictor (r2 = ., β = −., P < .) of the total number of behaviours exhibited by the mother during the hour-long interaction. Following adjustment for multiple tests, no other maternal char-acteristics were found to predict any of the maternal behaviours.

Figure 1 Percent of mothers with depres-sion and anxiety. BAI, Beck Anxiety Inventory; BDI, Beck Depression Inventory.

21n = 5

32n = 8

56n = 14

32n = 8

36n = 9

52n = 13

0

10

20

30

40

50

60

70

History History Current treated

Current treated

IABIDB

(%)




Child characteristics and maternal mental health

Because many of the mothers in this study reported high levels of depression, anxiety and stress, and because the mental health status of the mothers was related to the number of interactive behaviours they engaged in with their child, we examined the relations among maternal mental health and child develop-ment and behaviour. Child problem behaviours was a significant predictor of maternal stress (r2 = ., β = ., P < .). No other significant relations were found after controlling for multiple tests.

Discussion

The goals of this study were to describe maternal behaviours during mother–child interactions and to identify factors associated with maternal behaviour within dyads affected by FXS in order to elucidate within-group variability in child outcomes. We found that mothers of children with FXS displayed high levels of depression, anxiety and stress. Child prob-lem behaviour was related to maternal stress, and stress was related to the number of interaction behav-iours displayed during the hour-long naturalistic observation.

Maternal behaviours

There was considerable variability among the moth-ers in this study; however, they generally used main-taining and non-scaffolding behaviours more frequently than directive and scaffolding behaviours. While the mothers generally were responsive to their children’s actions and focus of attention, they less

frequently used those opportunities to provide struc-tured information to their children about their envi-ronment. This pattern of behaviours could be due to the structure of the study, in that it was an ambigu-ous, somewhat contrived situation in which mothers were being observed. Many of the mothers com-mented that they normally did not spend that much time alone with their children. However, all of the women were given the same information and instruc-tions. Some mothers took the opportunity to play with and teach their children, whereas others went about their daily activities, attending to and maintain-ing their children’s focus of attention, but not spend-ing extra time guiding their children in play.

Scaffolding and directive behaviours were not con-sistent across the structured and unstructured ses-sions, whereas maintaining and warmth were consistent across sessions. Mothers may have felt a greater need to direct their child during the struc-tured session. Also, they may have seen direct play-based interactions as opportunities for teaching. In contrast, when mothers were instructed to go about daily activities, they may have not felt the need to direct their child or engage their child in teaching opportunities. These results demonstrate the impor-tance of context in observing and measuring charac-teristics of mother–child interactions.

Maternal behaviour and child development

Maintaining and scaffolding behaviours during the naturalistic session were significantly correlated with child-receptive language skills. This finding is not surprising and is consistent with much of the litera-ture (Bornstein & Tamis-LeMonda , ;

Table 5 Correlations among maternal characteristics

PSI total score BDI BAI FSIQ-2 FSS Mother age Maternal education

PSI 0.531** 0.424* 0.278 −0.197 −0.071 0.314BDI 0.708*** 0.346 −0.411* 0.029 0.047BAI 0.200 −0.239 −0.013 −0.076FSIQ-2 −0.200 0.450* −1.07FSS 0.391 0.237Age 0.404

*P < .; **P < .; ***P < ..BAI, Beck Anxiety Inventory; BDI, Beck Depression Inventory; FSIQ-, Full Scale IQ- subtest; FSS, Family Support Scale; PSI, Parenting Stress Index.




Landry et al. , a, ; McCathren et al. ; Yoder et al. ; Tamis-LeMonda et al. ; Murphy & Abbeduto ). In this study, children who had higher language skills (as determined by the Mullen scores) had mothers who, during this obser-vation, used more maintaining and scaffolding behav-iours. Although it is possible that the relationship is entirely driven by the child (i.e. higher functioning children elicit the specific behaviours from the mother), previous researchers (Bornstein & Tamis-LeMonda , ; McCathren et al. ; Landry et al. , a, ; Yoder et al. ; Tamis-LeMonda et al. ) who have explored this rela-tionship suggest that the opposite is more likely (i.e. maternal behaviours assist in the development of skills in the child). This finding has implications for intervention in that mothers could be taught to increase their maintaining, scaffolding and warm sen-sitivity to increase child language. More research in the relations between maternal behaviours and child language is needed, however, to describe the direc-tion of this relationship and better understand the mechanisms for which intervention could be involved.

Maternal well-being

The scores on the mental health variables indicate that many of the women in this study were experi-encing emotional distress. Whereas –% of women in the general population are expected to experience depressive symptomology in their lifetime (NIMH ), % of this sample had experienced some depression in their lives, % were being treated for depression at the time of the assessment, and % indicated significant levels of depression on the BDI-II. In addition, half of the mothers in this study reported a clinically significant level of stress.

Studies that have explored mental health issues among mothers of children with disabilities have found prevalence rates that range from % to % (Dumas et al. ; Blacher et al. ; Hoare et al. ; Veisson ; Mobarak et al. ; Olsson & Hwang ; Baker et al. ; Baker et al. ; Emerson ; Glidden & Schoolcraft ; Hastings ; Saloviita et al. ; Abbeduto et al. ). The reason for this wide range may be due in part to measurement, with more women scoring in

the clinical range on some instruments than on others.

However, the nature of the child’s disability and degree of behaviour problems associated with the disability is likely a greater contributor to the wide range of mental health issues reported in these stud-ies. Several researchers have found that behavioural difficulties, rather than developmental or intellectual delays, are more likely to predict parental stress (Ols-son & Hwang ; Baker et al. , ; Hastings ; Saloviita et al. ). Olsson & Hwang () found that, in a sample of mothers raising a child with intellectual disabilities and autism, % had elevated scores on the BDI; and Thompson et al. () found a prevalence rate for depression of % for mothers of children with FXS. Similarly, studies using the PSI with caregivers of children with autism (Tomanik et al. ), attention deficit hyperactivity disorder (Baker & McCal ) and oppositional defiant dis-order (Ross et al. ) report significant levels of stress. In contrast, several studies examining children with Down syndrome, who generally do not display as many problem behaviours as children with autism or other developmental delays, have found that their mothers do not report levels of stress or depression that are different from those of mothers of typically developing children (Dumas et al. ; Van Riper et al. ; Scott et al. ). In general, parents of children whose developmental or physical disabilities do not result in increased challenging behaviours have not been found to report such high levels of stress (Dumas et al. ; von Gontard et al. ; Britner et al. ).

In this sample, % of the women who reported significant levels of stress reported that their stress was related to raising a difficult child. Mothers in this study who rated their children as having significant behaviour problems on the CBCL tended to feel the most stress in relation to having a difficult child. This result is similar to other studies exploring stress in parents of children with FXS (Sarimski ; von Gontard et al. ; Johnston et al. ; Abbeduto et al. ). In the current study, the mothers also reported during the interviews that aggressive behav-iours were the most challenging aspect of interacting with their children. This finding seems to suggest that the problem behaviours exhibited by children with FXS may have contributed directly to the increased levels of depression and stress in their mothers.




The genetic status of these women could also be related to the levels of stress and depression reported. Previous studies have suggested an increased risk of affective disorders for premutation carriers (Franke et al. , ; Hagerman & Hagerman ). However, premutation carriers who were mothers of affected children reported more symptoms of depres-sion than did premutation carriers who did not have children or whose children were unaffected (Franke et al. ). Those with the premutation, regardless of their children’s status, had rates of depression higher than their non-carrier siblings (Franke et al. ). This would suggest that symptoms of depres-sion in these women may be due in part to having the premutation, but is likely primarily be due to having a child with FXS. Unfortunately, in this study, we were not able to examine the mothers’ mental health history prior to their children’s diagnosis.

Maternal well-being and maternal behaviours

Identifying factors related to individual differences in maternal behaviour was one of the main goals of this study. Given the high levels of depression and stress found in these women, we expected to find some effects for these mental health variables on the observed maternal behaviours. The only mental health variable that was related to maternal behav-iours was stress. In this study, overall stress was negatively related to the number of behaviours the mothers exhibited. This result suggests that mothers who feel more overwhelmed by their children may choose to interact less with them.

Maternal report of stress, depression and anxiety were strongly correlated in this study. It is likely that shared method variance accounts for the strong rela-tionship between these variables. Those women who reported higher levels of stress, depression and anxi-ety have decreased psychological well-being and fewer resources which possibly led to a decreased interest or willingness in interacting with their children.

Surprisingly, we did not find any direct relations between depression and maternal interactive behav-iours. The small number of participants and large number of variables in this study may not have pro-vided enough statistical power to provide many inter-pretable results. With a larger, less homogeneous sample, there may have been more significant rela-

tionships among these variables. However, there are other possible explanations for these results. For example, when being observed, the mothers may not have been behaving in the way they might normally act. Also, given that female carriers of FXS have been shown to experience a higher rate of social anxiety (Sobesky et al. ), they may have been more sen-sitive about the impressions they were making during the observations and when responding to the questionnaires.

Limitations and implications

This study was preliminary and exploratory in nature. Because of the small, highly educated, homo-geneous sample and one time observation, the results only provide a glimpse into the complex world of interactions in dyads affected by FXS. Knowing whether mothers had permutation or full mutation FXS would have strengthened this study. Because mothers with the full mutation may be more affected by FXS and because women with the permutation FXS may be at-risk for depression and anxiety, the genetic status could be an important factor. Examin-ing the specific behaviours of the children would have provided more information about the appropriate-ness of maternal behaviours and our understanding of these complex reciprocal mother–child relation-ships. In addition, the lack of a control group in this study limits the ability to make syndrome-specific conclusions, as many of the findings could be related to the child having a disability, and may not be related to FXS specifically. However, despite these limita-tions, this study provides important information that can guide interventions and future studies. The impact of maternal depression, anxiety and stress on the mothers’ physical health (Bright & Hayward ), family relationships (Costigan et al. ), and on child development (Baker et al. ; Saloviita et al. ) and child behaviour should be examined in detail in the future to identify interventions that promote optimal maternal, family and child outcomes.

Mothers who reported higher levels of stress inter-acted less with their children, and problem behaviour in children with FXS predicted maternal stress. This pattern may result in a negative cycle in which both the parent’s stress level and the child’s behaviours interact with each other to cause chronic strain on




the parent–child relationship. Longitudinal research examining these issues is necessary to determine whether this pattern does indeed lead to such a neg-ative cycle. The acquisition of adaptive coping skills has been shown to be effective in reducing depression and the impact of negative life events. Intervention to develop these skills can have a positive impact on child outcomes (Lee ).

The relationship of maternal behaviour to child language provides evidence that, despite the known delays inherent in FXS, environmental influences also impact children’s development. While we could not determine whether mother or child characteris-tics were responsible for this finding, there is evidence to support the theory that maternal behaviours have a direct influence on the development of language skills (McCathren et al. ; Kaiser & Hemmeter ; Yoder et al. ; Tamis-LeMonda et al. ; Fewell & Deutscher ). Not only have previous researchers found that maternal behaviours can have an impact on child development, they have demon-strated that interventions that teach mothers to be more responsive can have a positive influence on chil-dren. The current study provides a foundation for future work examining the impact of similar interven-tions within the FXS population. It also provides a starting point for examining family variables and child outcomes.

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Accepted June

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Correlates of maternal behaviours in mothers of children with fragile X syndrome