9
Early sensory over-responsivity in toddlers with autism spectrum disorders as a predictor of family impairment and parenting stress A. Ben-Sasson, 1 T. W. Soto, 2 F. Martı ´nez-Pedraza, 2 and A. S. Carter 2 1 Department of Occupational Therapy, University of Haifa, Haifa, Israel; 2 Department of Psychology, University of Massachusetts Boston, Boston, MA, USA Background: Sensory over-responsivity (SOR) affects many individuals with autism spectrum disorders (ASD), often leading to stressful encounters during daily routines. Methods: This study describes the associations between early SOR symptoms and the longitudinal course of restrictions in family life activities and parenting stress across three time-points in families raising a child with ASD (n = 174). Covariates were child diagnostic severity, emotional problems, and maternal affective symptoms. At time 1 mean chronological age was 28.5 months. Children were administered the Autism Diagnostic Observation Schedule (ADOS) and Mullen Scales of Early Learning (MSEL). Parents completed the Infant Toddler Sensory Profile (ITSP), Infant-Toddler Social Emotional Assessment (ITSEA), Beck Anxiety Index (BAI), and the Center for Epidemiologic Studies Depression Inventory (CES-D) at time 1; and the Parenting Stress Index (PSI) and Family Life Impairment Scale (FLIS) at the three annual time-points. Results: Latent Growth Curve Models indicated that higher SOR scores on the ITSP at time 1 were associated with higher initial levels of family life impairment and parenting stress and with a smaller magnitude of change over time. These associations were independent of severity of ADOS social- communication symptoms, MSEL composite score, ITSEA externalizing and anxiety symptoms, and maternal affective symptoms as measured by the BAI and CES-D. On average FLIS and PSI did not change over time, however, there was significant individual variability. Concurrently, SOR at time 1 explained 39–45% of the variance in family stress and impairment variables. Conclusions: An evaluation of SOR should be integrated into the assessment of toddlers with ASD considering their role in family life impairment and stress. Keywords: ASD, toddlers, sensory over-responsivity, family impairment, parenting stress. Introduction Higher rates of sensory over-responsivity (SOR) are observed in individuals across autism spectrum disorders (ASD), and SOR symptoms appear to per- sist from early childhood throughout the life span (see Ben-Sasson et al., 2009). SOR is a form of sen- sory modulation disorder manifested in intense and prolonged negative responses towards commonplace sounds, sights, tastes, smells, movements, and touch leading to negative emotionality, distractibil- ity, heightened stress, and avoidance of such input (e.g., avoiding eating soft textured foods, distress from sound of home appliances) as well as aggressive and defiant behavior (Reynolds & Lane, 2008). Rates of SOR in children with ASD vary from 56% to 79% (e.g., Baranek et al., 2006; Tomchek & Dunn, 2007), with a previously documented rate of 56% (>1 stan- dard deviation (SD) below norms) in the current full sample at the first assessment point when children were 18–33 months of age (Ben-Sasson et al., 2007). Sensory over-responsivity in children with ASD has been associated with compromised participation in activities of daily living (Kay, 2001) and social interactions (Hilton, Graver, & LaVesser, 2007) as well as internalizing problems (Pfeiffer, Kinnealey, Reed, & Herzberg, 2005). Consequently, caregivers of children with comorbid ASD and SOR are dealing with children who have elevated irritability and who often respond with emotional and behavioral dysre- gulation. In a previous paper on the present sample, maternal stress was concurrently associated with the child’s dysregulation problems, which included the following scales: sensory sensitivities, sleeping problems, eating problems, and negative emotional- ity (Davis & Carter, 2008). In this study, we sought to examine whether SOR in toddlers with ASD predicts the course of parenting stress and family life impairment above and beyond diagnostic severity and maternal affective symptoms using a standard- ized detailed measure of sensory behaviors. At time 1 the majority of children in this sample showed ex- treme under-responsivity and very few showed sen- sation seeking/craving symptoms (Ben-Sasson et al., 2007), which also can cause strain in family life. We focused on SOR rather than other types of sensory modulation symptoms (i.e., sensation seek- ing and sensory under-responsivity) as it (a) involves the most challenging behaviors such as tantrums and resistance during routine and nonroutine family activities; and (b) has the least measurement overlap Conflict of interest statement: Co-author Alice Carter receives royalties from Pearson Assessment. Journal of Child Psychology and Psychiatry 54:8 (2013), pp 846–853 doi:10.1111/jcpp.12035 Ó 2013 The Authors. Journal of Child Psychology and Psychiatry Ó 2013 Association for Child and Adolescent Mental Health. Published by John Wiley & Sons Ltd, 9600 Garsington Road, Oxford OX4 2DQ, UK and 350 Main St, Malden, MA 02148, USA

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  • Early sensory over-responsivity in toddlers withautism spectrum disorders as a predictor offamily impairment and parenting stress

    A. Ben-Sasson,1 T. W. Soto,2 F. Martnez-Pedraza,2 and A. S. Carter21Department of Occupational Therapy, University of Haifa, Haifa, Israel; 2Department of Psychology,

    University of Massachusetts Boston, Boston, MA, USA

    Background: Sensory over-responsivity (SOR) affects many individuals with autism spectrum disorders(ASD), often leading to stressful encounters during daily routines. Methods: This study describes theassociations between early SOR symptoms and the longitudinal course of restrictions in family lifeactivities and parenting stress across three time-points in families raising a child with ASD (n = 174).Covariates were child diagnostic severity, emotional problems, and maternal affective symptoms. Attime 1 mean chronological age was 28.5 months. Children were administered the Autism DiagnosticObservation Schedule (ADOS) and Mullen Scales of Early Learning (MSEL). Parents completed theInfant Toddler Sensory Profile (ITSP), Infant-Toddler Social Emotional Assessment (ITSEA), BeckAnxiety Index (BAI), and the Center for Epidemiologic Studies Depression Inventory (CES-D) at time 1;and the Parenting Stress Index (PSI) and Family Life Impairment Scale (FLIS) at the three annualtime-points. Results: Latent Growth Curve Models indicated that higher SOR scores on the ITSP attime 1 were associated with higher initial levels of family life impairment and parenting stress and with asmaller magnitude of change over time. These associations were independent of severity of ADOS social-communication symptoms, MSEL composite score, ITSEA externalizing and anxiety symptoms, andmaternal affective symptoms as measured by the BAI and CES-D. On average FLIS and PSI did notchange over time, however, there was significant individual variability. Concurrently, SOR at time1 explained 3945% of the variance in family stress and impairment variables. Conclusions: Anevaluation of SOR should be integrated into the assessment of toddlers with ASD considering their rolein family life impairment and stress. Keywords: ASD, toddlers, sensory over-responsivity, familyimpairment, parenting stress.

    IntroductionHigher rates of sensory over-responsivity (SOR) areobserved in individuals across autism spectrumdisorders (ASD), and SOR symptoms appear to per-sist from early childhood throughout the life span(see Ben-Sasson et al., 2009). SOR is a form of sen-sory modulation disorder manifested in intense andprolonged negative responses towards commonplacesounds, sights, tastes, smells, movements, andtouch leading to negative emotionality, distractibil-ity, heightened stress, and avoidance of such input(e.g., avoiding eating soft textured foods, distressfrom sound of home appliances) as well as aggressiveand defiant behavior (Reynolds & Lane, 2008). Ratesof SOR in children with ASD vary from 56% to 79%(e.g., Baranek et al., 2006; Tomchek & Dunn, 2007),with a previously documented rate of 56% (>1 stan-dard deviation (SD) below norms) in the current fullsample at the first assessment point when childrenwere 1833 months of age (Ben-Sasson et al., 2007).Sensory over-responsivity in children with ASD

    has been associated with compromised participationin activities of daily living (Kay, 2001) and social

    interactions (Hilton, Graver, & LaVesser, 2007) aswell as internalizing problems (Pfeiffer, Kinnealey,Reed, & Herzberg, 2005). Consequently, caregiversof children with comorbid ASD and SOR are dealingwith children who have elevated irritability and whooften respond with emotional and behavioral dysre-gulation. In a previous paper on the present sample,maternal stress was concurrently associated withthe childs dysregulation problems, which includedthe following scales: sensory sensitivities, sleepingproblems, eating problems, and negative emotional-ity (Davis & Carter, 2008). In this study, we sought toexamine whether SOR in toddlers with ASD predictsthe course of parenting stress and family lifeimpairment above and beyond diagnostic severityand maternal affective symptoms using a standard-ized detailed measure of sensory behaviors. At time 1the majority of children in this sample showed ex-treme under-responsivity and very few showed sen-sation seeking/craving symptoms (Ben-Sassonet al., 2007), which also can cause strain in familylife. We focused on SOR rather than other types ofsensory modulation symptoms (i.e., sensation seek-ing and sensory under-responsivity) as it (a) involvesthe most challenging behaviors such as tantrumsand resistance during routine and nonroutine familyactivities; and (b) has the least measurement overlap

    Conflict of interest statement: Co-author Alice Carter receives

    royalties from Pearson Assessment.

    Journal of Child Psychology and Psychiatry 54:8 (2013), pp 846853 doi:10.1111/jcpp.12035

    2013 The Authors. Journal of Child Psychology and Psychiatry 2013 Association for Child and Adolescent Mental Health.Published by John Wiley & Sons Ltd, 9600 Garsington Road, Oxford OX4 2DQ, UK and 350 Main St, Malden, MA 02148, USA

  • with ASD diagnostic symptoms in the current DSMcriteria (American Psychiatric Association, 2000),particularly with regards to sensory under-respon-sivity. Previous research from this sample indicatedstability in SOR over time based on the Infant-Tod-dler Social Emotional Assessments (ITSEA: Carter &Briggs-Gowan, 2006) 6-item Sensory Sensitivityscale (Green, Ben-Sasson, Soto, & Carter, 2012).Studying the course of family impairment and par-enting stress in relation to SOR can elucidate factorsthat challenge families early on and over time, andmay require early intervention. Investigating theimpairing nature of SOR in children with ASD istimely given the proposal to include unusual sensorybehaviors as a subdomain of stereotyped behaviorsin the DSM-V. Prior to examining the role of earlySOR symptoms on parenting stress and familyimpairment we first examined the course of bothfamily impairment and parenting stress among par-ents raising young children with ASD. Several stud-ies have addressed parenting stress but there hasbeen limited attention to family impairment, or theextent to which a childs symptoms disrupt routinefamily activities. Parents of children with ASD expe-rience greater levels of parenting stress, depression,and anxiety than parents raising children with otherdevelopmental disabilities (Olsson & Hwang, 2001;Seltzer, Kraus, Orsmond, & Vestal, 2001; Weiss,2002), suggesting that these stress levels are notmerely associated with the challenges of raising achild with a disability. Evidence from one- (Herring,Taffe, Tonge, Sweeney, & Einfled, 2006; Lecavalier,Leone, & Witz, 2006) and 2-year (Peters-Scheffer,Didden, & Korzilius, 2012) follow-ups indicate rela-tive stability in parenting stress in families of indi-viduals with ASD. Several attempts have been madeto identify child factors that predict stress in parentsof individuals with ASD. Among these identifiedfactors in cross-sectional studies are: the severity ofASD symptoms (Benson & Dewey, 2008; Osborne &Reed, 2009) and in particular, social-communicationabnormalities (Hastings & Johnson, 2001; Kon-stantareas & Homatidis, 1989; Werner DeGrace,2004); an uneven intellectual profile (Konstantareas& Homatidis, 1989); and executive dysfunction (Ep-stein, Saltzman-Benaiah, OHare, Goll, & Tuck,2008). Concurrent evidence from this sample at theinitial assessment indicated that maternal parentingstress was associated with the childs dysregulationand social relatedness but not with child cognitiveand communication skills (Davis & Carter, 2008).Thus, when predicting changes in parenting stressand family impairment variables, we controlled forchild diagnostic and behavioral features. Behaviorproblems in children with ASD have been consis-tently linked to lower parental well-being (Bromleyet al., 2004; Hastings & Brown, 2002; Konstantare-as & Homatidis, 1989; Lecavalier et al., 2006). Spe-cifically, parenting stress in families raising a childwith ASD has been positively correlated with hyper-

    activity, stereotyped behaviors, self-injuriousbehaviors, and sensory over-sensitivity (Lecavalieret al., 2006), as well as hyper-irritability(Konstantareas & Homatidis, 1989). In a comparisonof toddlers with ASD and those with developmentaldelays, initial and follow-up child emotional andbehavioral problems contributed significantly moretowards maternal stress, parental mental healthproblems, and perceived family dysfunction thantype of diagnosis (Herring et al., 2006). In a longi-tudinal study of individuals with ASD and intellec-tual disability, emotional and behavioral problemsaccounted for 34% and behavioral inflexibility to-wards objects added an additional 13.6%, of thevariance in maternal stress whereas developmentalage and ASD severity were not associated withmaternal stress (Peters-Scheffer et al., 2012). Somebehavior problems in individuals with ASD are likelyassociated with sensory symptoms and reflect effortsto avoid or control sensory input. For example,engaging in maladaptive forms of repetitive behav-iors such as head banging and biting can function asways for controlling vestibular or tactile input. It isimportant to note that not all sensory symptomsresult in problem behaviors, and that repetitivebehaviors can occur in the absence of SOR.In an epidemiological study of children without

    ASD, SOR was associated with concurrent familyimpairment independent of the contribution of DSMpsychopathologies and sociodemographic risk (Carter,Ben-Sasson, & Briggs-Gowan, 2012). Amongfamilies raising a child with Asperger syndrome(512 years old), Epstein et al. (2008) found thatchildrens sensory symptoms were strongly associatedwith maternal parenting stress (r = .56). We wereinterested in examining the relation between earlychild SOR symptoms and the course of parentingstress and family impairment as a window into theimpact of early emerging sensory symptoms onfamily climate.In this study, SOR, parenting stress, and family life

    impairment were measured using parent-reporttools. Decreased maternal well-being could influencethe parents perceptions of her childs behavior.Maternal depression and anxiety have been associ-ated with greater discrepancies in reporting childpsychopathology symptoms compared with otherinformants (Briggs-Gowan, Carter, & Schwab-Stone,1996). Including indicators of maternal anxiety anddepression symptoms in these trajectory models wasimportant to account for this response bias, whichcould also impact ratings of family impairment andparenting stress. In addition, evidence from the cur-rent sample at time 1 for relations between SOR,child anxiety, and externalizing symptoms, cognitivelevel, social-communication symptoms, and mater-nal anxiety (Green et al., 2012) supported the inclu-sion of these parameters as covariates in the currentresearch. This study sought to examine whether earlySOR in children with ASD is associated with initial

    doi:10.1111/jcpp.12035 Sensory over-responsivity, family impairment, and autism 847

    2013 The Authors. Journal of Child Psychology and Psychiatry 2013 Association for Child and Adolescent Mental Health.

  • and later rates of change in family life impairmentand maternal parenting stress. To better interpretthese associations, families of children with andwithout clinically significant levels of SOR werecompared on family impairment, parenting stress,and child and parent factors.

    MethodsParticipants

    The sample includes 174 toddlers with ASD and theirmothers who participated in a longitudinal studyexamining trajectories of child development andparental adjustment among families of children newlydiagnosed with an ASD. Toddlers had a mean chrono-logical age of 28.5 months (SD = 3.9, range = 1833 months of age) at time 1, 40.97 months (SD = 4.23)at time 2, and 53.17 (SD = 4.57) at time 3. Their meanEarly Learning Composite score (ELCS) was 66.5(SD = 16.4, range = 49126) based on the MullenScales of Early Learning (MSEL: Mullen, 1995).Approximately three-fourths of the sample (78%,n = 136) were boys and 80% (n = 139) of toddlers wereidentified by their mothers as Non-Hispanic/LatinoWhite. At time 1, 55 (33.13%) toddlers were classified inthe SOR group based on having ITSP SOR scores thatwere at least 2 SD below age norms versus 111 classi-fied in the non-SOR group. Eight toddlers were missingITSP scores at time 1; see Table 1.Mothers of toddlers in this sample were primarily the

    biological parent (99%) with an average age of36.17 years (range = 1958; SD = 5.03) at the time ofparticipation. Eighty-four percent of mothers self-identified as Non-Hispanic/Latino White. Most mothersreported middle to upper class household incomes

    (88%) and had at least 2 years of college (79%). Themajority of mothers were married or cohabiting (91%);fewer mothers were separated/divorced (4%), or singleand had never been married (5%).Inclusion criteria were: (a) childs chronological age

    between 18 to 33 months; and (b) a diagnosis of autisticdisorder or a pervasive developmental disorder basedupon meeting research criteria on the Autism Diag-nostic Interview-Revised (ADI-R; Rutter, LeCouteur, &Lord, 2003) and the Autism Diagnostic ObservationalSchedule-Generic (ADOS-G; Lord, Rutter, DiLavore, &Risi, 2000), as well as based on the clinical impressionof an experienced clinical psychologist. Children with aphysical disability, known genetic disorder, or neuro-logical disorder were excluded. Only one child perfamily was included in these analyses to meet the sta-tistical assumption of independence of observations.

    Procedures

    Participants were recruited through early interventionproviders; specialty autism services programs, pedia-tricians, and word of mouth. Following a telephonescreening for eligibility, mothers were sent consentforms, and a booklet that included the Infant ToddlerSensory Profile (ITSP; Dunn, 2002), Infant-ToddlerSocial and Emotional Assessment (Carter & Briggs-Gowan, 2006), Parenting Stress Inventory (PSI; Abidin,1995), Family Life Impairment Scale (FLIS; Briggs-Go-wan, Horwitz, & Carter, 1997), Center for EpidemiologicStudies -Depression Inventory (CES-D; Radloff, 1977),and Beck Anxiety Index (BAI; Beck, Epstein, Brown, &Steer, 1988). A child visit took place in a laboratorysetting, was videotaped, and included the ADOS-G(Lord et al., 2002) and MSEL (Mullen, 1995). A care-giver visit took place either at the childs home or at the

    Table 1 Descriptive statistics for main study variables between SOR groups at Time 1 and (where applicable) Times 2 and 3

    Time 1 Time 2 Time 3

    SOR(n = 55)M (SD)

    Non-SOR(n = 111)M (SD)

    Waldtest

    SOR(n = 51)M (SD)

    Non-SOR(n = 96)M (SD)

    Waldtest

    SOR(n = 33)M (SD)

    Non-SOR(n = 52)M (SD)

    Waldtest

    ADOS (social +communicationstandardized)a

    51.23 (18.84) 58.48 (17.51) 5.80*

    Mullencomposite

    68.95 (19.26) 65.03 (13.77) 1.82

    ITSEA anxiety 0.43 (0.3) 0.25 (0.19) 16.05** ITSEAexternalizing

    0.67 (0.3) 0.42 (0.82) 27.78**

    BAIa 9.07 (7.65) 5.70 (5.48) 8.56* CES-Db 16.18 (11.01) 12.81 (9.47) 3.78 FLIS 9.23 (5.13)a 5.65 (3.98)b 20.69** 8.85

    (4.64)a

    6.46(4.19)b

    9.45* 8.87(4.59)a

    6.39(4.34)b

    6.16*

    PSI 91.96 (21.17)a 79.96 (19.83)b 12.29** 92.53(19.67)a

    83.982(20.00)b

    5.82* 92.64(20.46)a

    85.78(23.32)b

    4.38*

    ADOS, Autism Diagnostic Observation Schedule; BAI, Beck Anxiety Index; CES-D, Center for Epidemiologic Studies DepressionInventory; FLIS Family Life Impairment Scale; ITSEA, Infant-Toddler Social Emotional Assessment; PSI, Parenting Stress Index;SOR, sensory over-responsivity.a20% above moderate cutoff of 22 in SOR group versus 7% in non-SOR.b44% above cutoff of 16 in SOR group versus 32% in non-SOR.p < .06. *p < .05. **p < .001 for between group comparisons. Same subscripts in a row indicate nonsignificant differences betweenmeans using the Wald test of parameter constraints.

    848 A. Ben-Sasson et al. J Child Psychol Psychiatry 2013; 54(8): 84653

    2013 The Authors. Journal of Child Psychology and Psychiatry 2013 Association for Child and Adolescent Mental Health.

  • research center and included the ADI-R (Rutter et al.,2003). Families were followed annually for 2 years afterbaseline. At each time, mothers completed the ques-tionnaire booklet.

    MeasuresChild measures

    Infant/Toddler Sensory Profile (ITSP; Dunn, 2002). TheITSP (736 months) is a 48-item caregiver question-naire which measures sensory processing behaviors indaily experiences. Parents rate the frequency of eachbehavior from 1 almost always to 5 almost never.Scores are grouped into four scores: Low Registration,Sensation Seeking, Sensory Sensitivity, and SensationAvoiding, and are interpreted relative to age norms. ALow Threshold score (referred to as SOR in this study) iscomputed as the sum of Sensitivity and Avoidingscores. This composite score was applied in this studyto classify children as presenting with SOR or not basedon a cutoff of 2 SDs below norms. Lower scores indicatea higher frequency of over-responsivity. In the norma-tive sample the reliability of the composite scores ran-ged from 0.69 to 0.85, and validity was good (Dunn,2002). For this sample Cronbachs alpha for ITSP Sen-sitivity items was 0.79, and 0.77 for Avoidance items.Autism Diagnostic Observation Schedule-Generic

    (ADOS-G; Lord et al., 2000). The ADOS-G is a semi-structured observation used to measure social andcommunicative functioning and repetitive behaviors inindividuals suspected of having anASD.Most toddlers inthis study (n = 161) were administered Module 1 (pre-verbal or have single words), and six toddlers wereadministered Module 2 (phrase speech). A diagnosticalgorithm, based upon DSM-IV and ICD-10 criteria, canbe computed from the Reciprocal Social Interaction andCommunication scores, resulting in a diagnosis of aut-ism, autism spectrum, or neither (Lord et al., 2002). Inthis study, we used the ADOS-G to confirm diagnosticsymptoms and the Social and Communication compositestandardized score as an indicator of autism symptoms.Mullen Scales of Early Learning (MSEL; Mullen, 1995).

    The MSEL provide a direct assessment of cognitivefunctioning. It yields five scales (Fine Motor, GrossMotor, Visual Reception, Expressive Language, andReceptive Language) and an ELCS, a standard score thataggregates all scales except the Gross Motor Scale.Infant-Toddler Social and Emotional Assessment

    (ITSEA; Carter & Briggs-Gowan, 2006). The ITSEA is a166-item parent-report questionnaire that assesses thesocial and emotional competencies and problembehaviors of children 1- to 3-years old. Parents rate thedegree to which their child exhibited certain behaviorsin the past month (0 not true/rarely, 1 somewhattrue/sometimes, 2 very true/often). The ITSEA iscomprised of four domains (Competence, Externalizingand Internalizing problem behaviors, and Dysregula-tion), and can be broken down into 17 individual scales,including a General Anxiety scale. The ITSEA hasacceptable reliability, with alphas for the four domainsranging from 0.85 to 0.90, and for the 17 scales from0.52 to 0.82. The testretest reliability of ITSEA is goodto excellent over a 6-day period (alpha = 0.760.91)(Carter & Briggs-Gowan, 2006). In this sample, internal

    consistency ranged from 0.770.86 for the fourdomains, and from 0.520.86, with 2 scales below 0.6,for the 17 scales.

    Family measures

    Parenting Stress Index/Short Form (PSI/SF; Abidin,1995). The PSI is a parent self-report measure designedto assess parenting stress focusing on the parent-childsystem. It included 36 items comprising four scales:Parental Distress, Parent-Child Dysfunctional Interac-tion, Difficult Child Characteristics, and DefensiveResponding. Parents rated each item on a 5-point Likerttype scale ranging from 1 (strongly agree) to 5 (stronglydisagree). The PSI Total Score was derived and higherscores indicated higher levels of stress. The scales showhigh internal consistency, with alphas of 0.800.91 andtestretest reliability ranged from 0.680.85 for the fourscales over a 6-month period. In this study, internalconsistency alphas ranged from 0.780.89 at time 1,from 0.790.89 at time 2, and from 0.830.92 at time 3.Family Life Impairment Scale (FLIS; Briggs-Gowan

    et al. 1997). The FLIS is a parent-report scale that as-sesses the extent to which a childs behavior, personal-ity, or special needs limit participation in typical familyactivities (e.g., taking the child to visit family or runningerrands) or negatively affect the parent. It is comprisedof 19 items rated on a 3-point scale from not true tovery true, which yield a Global Impairment score. Thisscale has acceptable internal consistency (Cronbachalpha = 0.85) and 10- to 45-day testretest reliability(n = 119, M = 26.4, SD = 8.14, ICC = 0.71). Internalconsistency for the subscales in this sample rangedfrom Cronbachs alphas of 0.610.83 at time 1, from0.65 to 0.79 at time 2, and from 0.620.80 at time 3.Center for Epidemiologic Studies Depression Inventory

    (CES-D; Radloff, 1977). The CES-D is a self-reportmeasure of adult depression symptoms that includes20 items rated on a 4-point Likert scale ranging from 0(rarely or none of the time) to 3 (most or all of the time).Higher total scores in the CES-D indicate greater riskfor depression. This measure has shown high internalconsistency (coefficient alpha values between 0.840.90) and good testretest reliability (ranging from0.510.67) for 2- and 4-week intervals (Radloff, 1977).In the current sample, the internal consistency alpharanged from 0.880.92.Beck Anxiety Inventory (BAI; Beck et al., 1988). The

    BAI is a self-reportmeasure of physiological and cognitivesymptoms of anxiety. It consists of 21 itemswith answersgiven on a 4-point scale of 0 (not at all) to 3 (severely). Therespondent is asked to rate the degree to which he or shehas been bothered by these feelings (e.g., nervous; fearof losing control) in the past week. Internal consistency(alpha) is 0.92 and 1-week testretest correlation is 0.75(Beck et al., 1988). For the current sample, Cronbachsalphas ranged from 0.86 to 0.91.

    Data analysis

    Missing data analysis indicated that data were missingcompletely at random (Littles MCAR test v2[87] =108.98, p > .05). For all of the time 1 predictor variables(including covariates) less than 5% of data weremissing.For the outcome variables, more data were missing as

    doi:10.1111/jcpp.12035 Sensory over-responsivity, family impairment, and autism 849

    2013 The Authors. Journal of Child Psychology and Psychiatry 2013 Association for Child and Adolescent Mental Health.

  • fewer families participated over time. Missing data per-centages for families with fewer than 2 data points forFLIS and PSI were 13.2% for each variable. Full Infor-mationMaximumLikelihood (FIML)wasused toestimatemissingdata inall statisticalmodeling.Despite relativelyhigh levels of missing data, these cases were included inthe analyses as FIML has been proven to be a robustestimator in SEMmodels (Enders & Bandalos, 2001).A structural equation approach employing latent

    growth curve modeling (LGCM) was used to determinethe rate of change in FLIS and PSI across three timepoints. Two LGCMs (one for FLIS and one for PSI) werecomputed to characterize the slope of each of the factorsover time. SOR, as defined by continuous scores on theITSP at time 1, was used as a time-invariant predictorvariable, predicting to both the intercepts and slopes inFLIS and PSI models. In addition, scores on the CES-Dand BAI at time 1 were used to create the time-invariantlatent variable termed maternal affective symptoms.Initial exploratory models also included time 1 childcovariates that were thought to potentially contribute tothe trajectories of family impairment and parentingstress. These variables were ITSEA Anxiety, ITSEAExternalizing, ADOS Social-Communication, andMullen ELCS.Follow-up analyses were conducted to examine the

    clinical significance of SOR in family life. A clinicallyhigh SOR group (2 SD below the standardization sam-ples mean) and a non-SOR group were compared onfamily impairment and parenting stress over time, aswell as on time 1 child and maternal variables that wereentered into LGCM. The equality of the estimatedparameters for SOR and non-SOR groups was tested bya Wald test. All models were estimated using maximumlikelihood methods with MPlus 5.0 software (Muthen &Muthen, 2007).

    ResultsLatent growth curve models

    Determining if early SOR in children with ASD isassociated with later parenting stress and familyimpairment above and beyond baseline diagnosticcharacteristics, child anxiety and externalizingsymptoms, and maternal affective symptoms was theprimary goal of these analyses. Results of the LGCMsdemonstrated good fit to the data. The baseline(theoretical) FLIS model, with SOR and maternalaffective symptoms as predictors, demonstrated goodfit, x2(8) = 18.155, p = .020, CFI = .97, TLI = .943RMSEA = .086. Similarly, the baseline (theoretical)PSI model, with SOR and maternal affective symp-toms in the model as predictors, demonstrated goodfit, x2(8) = 18.319, p = .019, CFI = .975, TLI = .952,RMSEA = .087.To report the most parsimonious FLIS and PSI

    models, covariates were individually added to thebaseline models and subsequently trimmed if theydid not significantly predict to outcome variables ordid not significantly improve overall model fit. Childexternalizing, child anxiety, and ADOS Social-Com-munication were trimmed from both final models.

    MSEL ELCS was retained only in the FLIS model as itsignificantly predicted to the FLIS slope.The final FLIS model, with SOR included as a

    predictor variable and maternal affective symptomsand MSEL ELCS as covariates, with slope loadingsrestricted to 0, 1, 2, (a linear increase over time),demonstrated good fit, x2(10) = 19.391, p = .036,CFI = .973, TLI = .945, RMSEA = .073 (see Fig-ure 1). The final PSI model, with SOR included as apredictor variable and maternal affective symptomsas a covariate, with slope parameters restricted to 0,1, 2, also demonstrated good fit, x2(8) = 18.319,p = .019, CFI = .975, TLI = .952, RMSEA = .087 (seeFigure 2). Although the RMSEA for both modelssuggested marginal fit, since the relative chi-squaresand the CFIs indicated good fit and sample size wassmall, results support an adequate fitting model(Brown, 2006).In these models, SOR significantly predicted the

    intercept for family impairment (b = )0.43,p < 0.001)and parenting stress (b = 0.39, p < 0.05). Theprediction of SOR to the slope of parenting stress wassignificant (b = 0.36, p < 0.01) and approachedsignificance to the slope of family impairment(b = 0.326, p = 0.07). Maternal affective symptomspredicted the intercept of family impairment(b = 0.398, p < 0.01) and parenting stress (b = 0.57,

    FLIS intercept

    FLIS slope

    SORMaternal affective

    symptoms

    BAI CES-D

    FLIS time 1

    FLIS time 2

    FLIS time 3

    1 11

    01

    2

    .942**.725**

    .398** .015.453a** .326

    1.0

    .112.474**

    .570** .744**

    .256**.280**.270**

    .088

    .187

    .028

    .083.149

    Mullen

    .339*

    Figure 1 A latent growth curve model of SOR predicting toFLIS with a latent maternal affective symptoms covariate.Note. p < .06. *p < .05. **p < .001. aThe negative correlationresults from the lower SOR ITSP score denoting a higherfrequency and the higher FLIS score denoting more restrictions

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  • p < 0.01), but not change (i.e., slope) in these vari-ables (FLIS: b = 0.015, p > 0.05; PSI: b = 0.009,p > 0.05). The MSEL ELCS predicted the slope offamily impairment (b = )0.339, p < 0.01), but not theintercept. LGCMS demonstrated significant individ-ual variability around the slope for both FLIS and PSI.

    Clinical-group comparisons on family lifeimpairment and parenting stress

    We were interested in elucidating the clinical signifi-cance of the association between SOR and familyvariables by comparing FLIS and PSI scores over timefor families of children with versus without clinicallysignificant SOR scores. In this sample, 55 childrenwere identified at time 1 with elevated SOR based onhaving ITSP Low Threshold scores that were morethan 2 SD below age norms. Wald tests of ParameterConstraints showed that mothers in the SOR group,contrasted tomothers in theNon-SORgroup, reportedsignificantly higher levels of parenting stress,and overall family impairment at each time-point (seeTable 1). Moreover, mothers within each group (SORvs. Non-SOR) did not show significant changes inscores from time 1 to Time 3, in family impairment(SOR: Walds test = 0.19, df = 2, p = .909; Non-SOR:

    Walds test = 2.36, df = 2, p = .307) or parentingstress scores (SOR: Walds test = 0.86, df = 2,p =.651;Non-SOR:Walds test = 3.24,df = 2,p =.198).

    Group comparisons of child and maternal factors

    The SOR group showed a distinct clinical profilecompared with the Non-SOR group at baseline withsignificantly lower concurrent social-communicationscores on the ADOS-G, and higher ITSEA external-izing and general anxiety symptoms. In addition,mothers of the SOR group reported higher anxietysymptoms on the BAI and marginally higherdepressive symptoms as measured by the CES-Dcompared with the Non-SOR group (see Table 1).

    DiscussionAbove and beyond the challenge of raising a toddlerwith ASD, SOR appears to increase maternal stressand the degree to which family life routines are dis-rupted. Primary findings from this longitudinalstudy of toddlers with ASD showed that parents oftoddlers with higher SOR symptoms were more likelyto have higher levels of initial parenting stress andfamily life restrictions, which persisted over a 2-yearperiod. The association of SOR with family lifeimpairment and parenting stress surpassed childdiagnostic, anxiety, and externalizing symptoms, aswell as maternal affective symptoms, which wasconsidered a proxy for shared reporting bias. Find-ings also indicate that toddlers with ASD and SORwere more likely to have lower severity of social-communication symptoms and higher levels of anx-iety and externalizing problems than those withoutSOR. These findings underscore the need to assessthe practical and psychological impact that sensorysymptoms have on children and families to designmore effective early intervention plans. This is par-ticularly important given findings indicating thatparenting stress may reduce parents effectiveness inapplying early intervention techniques (Osborne,McHugh, Saunders, & Reed, 2008).The tested models, with SOR being associated

    with concurrent family life impairment and parent-ing stress, explained 4358% of the variance inthese outcomes, respectively. It is plausible thatprevious reports of the contribution of objectinflexibility and emotional/behavioral problems inchildren with ASD to parenting stress (e.g., Peters-Scheffer et al., 2012) are actually measuring conse-quences of SOR for some children. The magnitude ofthese effects are consistent with an earlier reportrelating sensory symptoms to maternal stress inolder children with Aspergers syndrome (Epsteinet al., 2008) and between SOR and family impair-ment in school-aged children with internalizingand/or externalizing psychopathologies and no ASD(Carter et al., 2012).

    PSI intercept

    PSI slope

    SORMaternal affective

    symptoms

    BAI CES-D

    PSI time 1

    PSI time 2

    PSI time 3

    11

    1 01

    2

    .896**.763**

    .574**.009

    .391**.358

    .268

    .224

    1

    .873**.416**

    .205**.235**.246**

    .418** .198*

    Figure 2 A latent growth curve model of SOR predicting toPSI with a latent maternal affective symptoms covariate.Note. p < .06. *p < .05. **p < .001. aThe negative correlationresults from the lower SOR ITSP score denoting a higherfrequency and the higher FLIS score denoting more restrictions

    doi:10.1111/jcpp.12035 Sensory over-responsivity, family impairment, and autism 851

    2013 The Authors. Journal of Child Psychology and Psychiatry 2013 Association for Child and Adolescent Mental Health.

  • Although average levels of family impairment andparenting stress did not change over time, there wassignificant variation around their slopes, suggestingthat individualshadvarying rates of changeover time.Interestingly, higher SOR scores were associatedwithless change in family variables. These findingsmay beexplained by the fact that families with elevated SORstarted and stayed higher on both measures thanthose familieswith toddlerswith lowerSOR. Thus, thepersisting initially high levels of parenting stress andfamily impairment in theSORgroupmayhave left lessroom for change. The low mean magnitude of changein family variables may be different in a more socio-economically diverse sample. This sample was a fairlydemographically homogeneous group of families frompredominantly white, upper to upper-middle class,and high education backgrounds.Differences in maternal stress levels and family life

    restrictions between SOR and Non-SOR groups werenoteworthy. What appears to be unique to families ofchildren with ASD and SOR is the need to adaptactivities and routines to cope with/avoid the childssensitivities during self-care activities, feeding, andoutdoor activities. SOR can lead to tantrums andirritability in children, as evidenced in this sample byelevated externalizing and anxiety scores. Mothersmay search for ways to minimize their childs dis-ruptive behavior and/or distress using strategiessuch as redesigning routines (e.g., vacuum whenchild is at school), social activities (e.g., avoid outingsto malls or family events), and home environments(e.g., lock closets, place trampoline in yard) to pre-vent encounters with the sensory stimuli that triggertheir childs over-response (Werner DeGrace 2004).In the current study, SOR predicted family vari-

    ables above and beyond most child diagnostic (i.e.,ADOS social-communication scores), emotional, andbehavioral (i.e., ITSEA externalizing and anxietyscores) scales. This is in contrast with evidenceshowing that ASD symptoms contribute to parentingstress and family functioning in young children(Herring et al., 2006; Osborne & Reed, 2009). Thefact that SOR appears to contribute to familyimpairment and parenting stress more proximallythan other child factors may relate to the practicalimplications a childs sensitivity poses to family dailyactivities and routines. The young age of the currentsample compared with previous family studies andthe relatively low levels of clinically significant emo-tional and behavioral problems (see Table 1) mayexplain, in part, why behavior problems were notmore strongly associated with family impairmentand parenting stress. These problems may increasewith age (Barker et al., 2011) and become moredominant in predicting interference in family activi-ties. Further research is needed to address laterfamily outcomes. In addition, relying upon a single-parent informant for both child and family charac-teristics is a limitation of this report. Although we inpart controlled for reporting bias by including

    maternal report of affective symptoms as a covariate,there is a need to validate current findings utilizingmultisource, multimethod assessments for bothchild and family factors.Although the tested models investigated how SOR

    predicted changes in family life, we must acknowl-edge the possible contribution of parenting stressand restrictions of activities to the display of SORbehaviors. Parenting stress may lead the parent toover-respond to their childs behavior, respond in aless co-regulated and sensitive manner, and be lesseffective in applying self- and child-directed copingstrategies. The manner in which parents set up thephysical and social environment will determine thesensory experiences of the child (e.g., choose loudouting places as opposed to quieter ones, organizetoys in an orderly vs. chaotic manner, buy clothingwithout seams and tags). Thus, there is need forfuture research to examine how family factors suchas stress, routines, and the coping strategies parentsapply to prevent or reduce their childrens sensorysymptoms influence the severity of sensory symptoms.

    ConclusionsSensory over-responsivity contributes to family lifeimpairment above and beyond ASD symptoms andmaternal affective symptoms. This study supportsthe evaluation of family well-being and SOR duringthe assessment of toddlers with ASD. It is importantto monitor maternal stress particularly for youngchildren with ASD who show extreme SOR. As not allmothers of toddlers with SOR showed elevated stressand family restrictions, further research can explainthe resilience of those families. This study calls forthe design of early interventions that emphasizefamily education and partnership to address sensorysymptoms in a way that matches the familys psy-chological and practical context.

    AcknowledgementThis work was supported by Studies to Advance AutismResearch and Treatment (STAART) grant from the NIMH(U54MH066398). We wish to thank the families andchildren who participated in this study.

    CorrespondenceAyelet Ben-Sasson, Department of Occupational Ther-apy, University of Haifa, Haifa 30905, Israel; Email:[email protected]

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    Accepted for publication: 1 November 2012Published online: 21 January 2013

    doi:10.1111/jcpp.12035 Sensory over-responsivity, family impairment, and autism 853

    2013 The Authors. Journal of Child Psychology and Psychiatry 2013 Association for Child and Adolescent Mental Health.

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