Developmental Psychology

Developmental Psychology Adaptation to the Birth of a Child With a Congenital Anomaly: A Prospective Longitudinal Study of Maternal Well-Being and Psychological Distress Ragnhild B. Nes, Espen Røysamb, Lars J. Hauge, Tom Kornstad, Markus A. Landolt, Lorentz M. Irgens, Leif Eskedal, Petter Kristensen, and Margarete E. Vollrath Online First Publication, March 3, 2014. http://dx.doi.org/10.1037/a0035996

CITATION Nes, R. B., Røysamb, E., Hauge, L. J., Kornstad, T., Landolt, M. A., Irgens, L. M., Eskedal, L., Kristensen, P., & Vollrath, M. E. (2014, March 3). Adaptation to the Birth of a Child With a Congenital Anomaly: A Prospective Longitudinal Study of Maternal Well-Being and Psychological Distress. Developmental Psychology. Advance online publication. http://dx.doi.org/10.1037/a0035996

Developmental Psychology 2014, Vol. 50, No. 4, 000

© 2014 American Psychological Association 0012-1649/14/$12.00 DOI: 10.1037/a0035996

This document is copyrighted by the American Psychological Association or one of its allied publishers. This article is intended solely for the personal use of the individual user and is not to be disseminated broadly.

Adaptation to the Birth of a Child With a Congenital Anomaly: A Prospective Longitudinal Study of Maternal Well-Being and Psychological Distress Ragnhild B. Nes

Espen Røysamb

Norwegian Institute of Public Health, Oslo, Norway

Norwegian Institute of Public Health, Oslo, Norway, and University of Oslo

Lars J. Hauge

Tom Kornstad

Norwegian Institute of Public Health, Oslo, Norway

Norwegian Institute of Public Health, Oslo, Norway, and Statistics Norway, Oslo, Norway

Markus A. Landolt

Lorentz M. Irgens

University Children’s Hospital, Zürich, Switzerland

Norwegian Institute of Public Health, Oslo and Bergen, Norway, and University of Bergen

Leif Eskedal

Petter Kristensen

Sørlandet Hospital, Kristiansand, Norway

National Institute of Occupational Health, Oslo, Norway, and University of Oslo

Margarete E. Vollrath Norwegian Institute of Public Health, Oslo, Norway, and University of Oslo This study explores the stability and change in maternal life satisfaction and psychological distress following the birth of a child with a congenital anomaly using 5 assessments from the Norwegian Mother and Child Cohort Study collected from Pregnancy Week 17 to 36 months postpartum. Participating mothers were divided into those having infants with (a) Down syndrome (DS; n ⫽ 114), (b) cleft lip/palate (CLP; n ⫽ 179), and (c) no disability (ND; n ⫽ 99,122). Responses on the Satisfaction With Life Scale and a short version of the Hopkins Symptom Checklist were analyzed using structural equation modeling, including latent growth curves. Satisfaction and distress levels were highly diverse in the sample, but fairly stable over time (retest correlations: .47–.68). However, the birth of a child with DS was associated with a rapid decrease in maternal life satisfaction and a corresponding increase in psychological distress observed between pregnancy and 6 months postpartum. The unique effects from DS on changes in satisfaction (Cohen’s d ⫽ ⫺.66) and distress (Cohen’s d ⫽ .60) remained stable. Higher distress and lower life satisfaction at later assessments appeared to reflect a persistent burden that was already experienced 6 months after birth. CLP had a temporary impact (Cohen’s d ⫽ .29) on maternal distress at 6 months. However, the overall trajectories did not differ between CLP and ND mothers. In sum, the birth of a child with DS influences maternal psychological distress and life satisfaction throughout the toddler period, whereas a curable condition like CLP has only a minor temporary effect on maternal psychological distress. Keywords: psychological distress, depression, life satisfaction, Down syndrome, congenital anomalies

Oslo, Norway; Institute of Health and Society, University of Oslo, Oslo, Norway. Margarete E. Vollrath, Division of Mental Health, Norwegian Institute of Public Health, Oslo, Norway; Department of Psychology, University of Oslo, Oslo, Norway. The Norwegian Mother and Child Cohort Study is supported by the Norwegian Ministry of Health and the Ministry of Education and Research, National Institutes of Health (NIH)/National Institute of Environmental Health Sciences Contract N01-ES-75558), NIH/National Institute of Neurological Disorders and Stroke Grant 1 UO1 NS 047537-01 and Grant 2 UO1 NS 047537-06A1. We are grateful to all the participating families in Norway who take part in this ongoing cohort study. We also thank Kristin Gustavson at the Norwegian Institute of Public Health for valuable insights and contributions. Correspondence concerning this article should be addressed to Ragnhild B. Nes, Division of Mental Health, Norwegian Institute of Public Health, Pb 4404 Nydalen, Oslo, Norway N-0403. E-mail: [email protected]

Ragnhild B. Nes, Division of Mental Health, Norwegian Institute of Public Health, Oslo, Norway. Espen Røysamb, Division of Mental Health, Norwegian Institute of Public Health, Oslo, Norway; Department of Psychology, University of Oslo, Oslo, Norway. Lars J. Hauge, Division of Mental Health, Norwegian Institute of Public Health, Oslo, Norway. Tom Kornstad, Division of Mental Health, Norwegian Institute of Public Health, Oslo, Norway; Statistics Norway, Oslo, Norway. Markus A. Landolt, University Children’s Hospital, Zürich, Switzerland. Lorentz M. Irgens, Division of Mental Health, Norwegian Institute of Public Health, Oslo, Norway; Medical Birth Registry of Norway, Norwegian Institute of Public Health, Bergen, Norway; Department of Public Health and Primary Health Care, University of Bergen, Bergen, Norway. Leif Eskedal, Department of Pediatrics, Sørlandet Hospital, Kristiansand, Norway. Petter Kristensen, Department of Occupational Medicine and Epidemiology, National Institute of Occupational Health, 1


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Motherhood is generally expected to be one of the most fulfilling experiences for the majority of women. A sizable literature indicates, however, that mothers are at excess risk of psychological distress, mental disorders, and reduced life satisfaction during the postpartum period and in the years thereafter (Dyrdal & Lucas, 2013; Dyrdal, Roysamb, Nes, & Vitterso, 2011; Eberhard-Gran, Eskild, Tambs, Samuelsen, & Opjordsmoen, 2002; Munk-Olsen, Laursen, Pedersen, Mors, & Mortensen, 2006). The birth of a child with a disability might increase the risk even further (Bailey, Golden, Roberts, & Ford, 2007; Singer, 2006). Approximately 8 million children worldwide are born with a serious birth defect annually (Christianson, Howson, & Modell, 2006). Mothers of these children commonly experience an overload of their adaptive capacities, with a majority reporting an initial reaction characterized by acute grief, disbelief, and a period of intense emotional disruption (Drotar, Baskiewicz, Irvin, Kennell, & Klaus, 1975). When lasting or chronic, illness and disability introduce uncertainty about the future and numerous caretaking challenges, particularly to the mother, who is often the primary caretaker. Although many adapt quite successfully to the increasing demands introduced by the disability, and parents of disabled children generally do not report fewer positive outcomes than parents of healthy children (Hastings & Taunt, 2002), an abundance of studies have shown mothers of disabled children to experience more physical health problems (Brehaut et al., 2009), more psychological distress and adjustment problems, and to be at excess risk of clinical depression (Bailey et al., 2007; Singer, 2006) regardless of the timing of the diagnosis (prenatal or postnatal) (Brosig, Whitstone, Frommelt, Frisbee, & Leuthner, 2007). The preponderance of studies on maternal disability-related stress has explored adjustment by means of negative indicators of mental health, such as depression and psychological distress, and their impact on positive indicators, such as life satisfaction (LS), has received comparatively limited attention. This is despite the fact that positive and negative emotions constitute important dimensions of mental health and subjective well-being that appear to be relatively independent (Bradburn, 1969; Knutson et al., 1998). Outside pediatrics, a number of studies have explored the stability and change in both affective (positive emotion, positive affect) and cognitive (life satisfaction) indicators of positive mental health following major life stressors. These studies have often shown the impact of life stressors to be of relatively short duration, mainly causing a temporary displacement from stable affective levels (Suh, Diener, & Fujita, 1996). Adaptation to individual affective baselines (“set point”) thus commonly occurs fairly soon following an event. This general adaptation process is assumed to operate as a protective buffer against the potentially adverse consequences of prolonged and intense emotional states. The adaptation process appears in large part to be due to genes (Nes, Roysamb, Reichborn-Kjennerud, Harris, & Tambs, 2007; Nes, Roysamb, Tambs, Harris, & Reichborn-Kjennerud, 2006) that are partly shared with personality (Weiss, Bates, & Luciano, 2008), and cognitive factors like positive reappraisal, response shift, and benefit finding might constitute important mechanisms. Most people have, for example, been shown to experience benefit finding (Davis, Nolen-Hoeksema, & Larson, 1998) or psychological growth (Frazier et al., 2009) in the aftermath of severe stress. In a previous study of mothers of acutely ill newborns (Affleck & Tennen, 1996), 75% cited at least one benefit from their child’s

hazardous birth and hospitalization. At 6 and 18 months postpartum, these mothers reported brighter mood and less distress. Longitudinal reports have indicated that some major life stressors such as divorce (Lucas, 2005), unemployment (Lucas, Clark, Georgellis, & Diener, 2004), and becoming disabled (Lucas, 2007) might have long-lasting and even permanent impact on well-being levels, and a recent meta-analysis has shown the magnitude of the effects to depend on the stressor considered (Luhmann, Hofmann, Eid, & Lucas, 2012). However, most studies are not prospective and have not examined stressors that occur independent of personality. People create their environments partly due to personality and genes (the nature of nurture). Exposure to life stressors such as unemployment or divorce might therefore partly reflect personality. Previous studies have, for example, shown stressful events to be causally linked to the onset of major depression. Approximately one third of this association appears to reflect the fact that individuals with a genetic vulnerability to depression select themselves into high-risk environments (Kendler & KarkowskiShuman, 1997). In addition, changes in well-being or distress might result from normative, developmental processes rather than the experience of a given life stressor or event, and most studies to date have not controlled well for such normative changes. This is quite problematic, as conclusions about the impact of stressors often depend on whether appropriate control groups were compared (Yap, Anusic, & Lucas, 2012). Most studies have also explored only single indicators, thus not being able to examine the degree to which separate components of mental health are differentially affected and how levels and changes in multiple indicators are related. Overall, Luhmann, Hofmann, et al. (2012) underscore the need for more longitudinal research on less conventional life events, assessing multiple components of mental health and wellbeing conjointly.

Congenital Anomalies Collectively, congenital anomalies, or major “birth defects,” include structural defects (malformations, deformations, dysplasias) and chromosomal anomalies with a total prevalence estimated to 23.9 per 1,000 births in Europe between 2003 and 2007 (of which 80% are live births), thus constituting a relatively rare event (Dolk, Loane, & Garne, 2010). Down syndrome (DS) and orofacial clefts (i.e., cleft lip and/or palate [CLP]) are two of the most frequently occurring congenital anomalies in humans with a prevalence estimated to be 0.97 (DS) and 1.30 (CLP) per 1,000 live births (Dolk et al., 2010). Children with DS and CLP are usually born to healthy mothers, and the event is usually unanticipated, causing an initial shock and prolonged caretaking challenges. The two conditions vary considerably in severity, chronicity, and the degree of physical, intellectual, and functional impairment, thereby posing quite different challenges over time. CLP is a treatable craniofacial birth defect, with corrective surgery commonly conducted 3–12 months after birth, has a good prognosis, and does not usually confer excess risk of death or developmental delay with the exception of speech-related problems. By contrast, DS is a chromosomal anomaly (Trisomy 21) that involves irreversible intellectual impairment that substantially affects future development with risk of reduced life span and is often complicated with congenital heart disease, hypothyroidism, and recurrent respiratory infections. The foreseen limits in mental development,

MATERNAL WELL-BEING AND PSYCHOLOGICAL DISTRESS

social stigma associated with intellectual deficiency, and the anticipated level of future parental care might all contribute to aggravate the distress that DS mothers experience relative to mothers of children with a curable condition such as CLP and mothers of healthy children (Pelchat et al., 1999).


The Present Study The purpose of the present paper was twofold. The most proximate aim concerns examination of maternal adaptation to the birth of a child with a congenital anomaly, namely, CLP or DS. The more general, theoretical aim was to explore the stability and change in both positive (LS) and negative (psychological distress) maternal mental health following two major life stressors that occur fairly evenly with respect to personality, one mainly reflecting temporary stress (i.e., CLP), the other more chronic (i.e., DS). The two conditions are fairly prevalent and commonly correctly diagnosed, lending themselves as suitable conditions for the purpose of this study. As individuals might vary substantially in their reaction and adaptation to different life stressors, we explore adaptation within individuals as well as between groups over time. To the best of our knowledge, this is the first large-scale prospective study exploring stability and change in both LS and psychological distress among mothers having children with disabilities across pregnancy, infancy, and toddlerhood simultaneously. We use data from a Norwegian prospective pregnancy cohort, including more than 99,000 mothers and multiple assessments conducted over 3.5 years, thus allowing for in-depth investigation of the mothers’ mental health and well-being during this particular life stage while accounting for maternal mental health prior to birth and demographic variables (age, education, marital status, parity) obtained from national registers.

Method Sample and Procedure The present study is based on data from the Norwegian Mother and Child Cohort Study (MoBa) conducted by the Norwegian Institute of Public Health (Magnus et al., 2006). The MoBa is a prospective pregnancy cohort study following more than 100,000 pregnancies. Since 1999, the MoBa has been inviting a majority of pregnant women in Norway, and the goal of recruiting 100,000 pregnancies was reached in 2008. Recruitment occurred through a postal invitation as women were scheduled for a routine ultrasound examination at 17–18 weeks of gestation (see www.fhi.no/tema/ morogbarn). The study was approved by the Regional Committee for Medical Research Ethics in southeastern Norway, and informed consent was obtained from each participant. Participants have reported medical, sociodemographic, and psychological information by completing six questionnaires: three during pregnancy (Questionnaire 1 [Q1] at the time of recruitment, Q2 at Week 22, and Q3 at Week 30) and three after birth (Q4 at 6 months, Q5 at 18 months, and Q6 at 36 months postpartum). No compensation was given for participation. The participation rate at the first assessment was 40.6%. During pregnancy, the response rate was 92%–95%, dropping to 87% at Q4, 77% at Q5, and to 62.5% at 36 months (for further detail about the MoBa, see Magnus et al., 2006).

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Data from Q1, Q3, Q4, Q5, and Q6 were used in the present study, hereafter referred to as Pregnancy Week 17, Pregnancy Week 30, 6 months, 18 months, and 36 months postpartum, respectively. The Q2 did not include the relevant measures (i.e., satisfaction and distress). The MoBa releases updated data files every spring. The present analyses are based on Version 7 of the quality-assured data files released to the researcher in 2012 and comprise 106,930 pregnancies recruited between 1999 and 2008. Pregnancies not ending in live births were excluded (n ⫽ 706), leaving 106,224 eligible pregnancies. Respondents having children with severe congenital anomalies other than DS and CLP were then excluded from the analyses (n ⫽ 2,502) as were responders with missing responses on all relevant variables (n ⫽ 3,522). Due to the nature of the MoBa and the continued data collection process, the number of participants included at the different time points varies. As such, the difference in the number of data entries at each wave is due not only to attrition but also to the continuous enrollment, resulting in participants reaching the data collection points at different times. Altogether, 49,499 (49.8%) mothers participated at all assessments. Information on congenital malformations and relevant birth status variables (i.e., gestational age, birth weight) were added to the file from the Medical Birth Registry of Norway (MBRN), which was established in 1967 and contains standardized data regarding all pregnancies in Norway from 12 weeks of gestation (Irgens, 2000). Participants were divided into mothers having infants with (a) DS (n ⫽ 114),1 (b) CLP (n ⫽ 179), and (c) no disability (ND; n ⫽ 99,122). Data on maternal age, educational attainment, and parity were provided by the MBRN, the Norwegian National Education Database of Statistics Norway, and the Central Population Register. Mean maternal age at birth was 30.2 years (SD ⫽ 4.6), and 44.6% were expecting their first child. Approximately 96% had a marital or equivalent partner at the time of birth. Altogether, 11.6% of the mothers had not completed high school, 26.0% had completed high school or the equivalent, 48.0% held an undergraduate university or vocational degree, 14.3% held a graduate degree, and 9% were students (0.3% had missing information).

Measures LS. LS was measured with the Satisfaction With Life Scale (SWLS; Diener, Emmons, Larsen, & Griffin, 1985). This five-item scale measures people’s perception of their life as a whole, thus constituting a cognitive well-being measure and includes items such as “The conditions of my life are excellent” and “I am satisfied with my life,” rated on a 1–7 Likert scale (1 ⫽ completely disagree, 7 ⫽ completely agree). Respondents completing a minimum of three items were included in the analyses. The SWLS is the most commonly used measure of LS worldwide and shows good psychometric properties including validity, internal consistency, test–retest reliability, and adequate invariance across gender and age (Clench-Aas, Nes, Dalgard, & Aaro, 2011; Diener et al., 1985; Lucas, Diener, & Suh, 1996). Overall, the scale showed excellent internal consistency, with Cronbach’s alphas estimated to 1 Number refers to mothers/cases at the first assessment (i.e., Gestation Week 17).


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be .89, .89, .89, and .91 at Time Points 1, 2, 3, and 5, respectively (the SWLS was not administered at 18 months). Individual mean satisfaction scores were used in the descriptive analyses. In the modeling analyses, the SWLS was analyzed as (a) a sum score standardized on the sample mean and standard deviation at the first assessment (latent growth curve analyses) and (b) a latent variable (cholesky). Psychological distress. Psychological distress was measured by a short version of the 25-item Hopkins Symptom Checklist (SCL-25; Hesbacher, Rickels, Morris, Newman, & Rosenfeld, 1980). The SCL-25 is a widely used self-administered screening instrument for detecting psychological problems, specifically symptoms of anxiety and depression, in nonpsychiatric settings and includes highly correlated scales for anxiety (10 items) and depression (15 items). The SCL scales have similar reliability and validity and figures of sensitivity and specificity as the General Health Questionnaire and the Short Form 36 (Strand, Dalgard, Tambs, & Rognerud, 2003). The SCL-5 used here includes five items empirically selected from the SCL-25 that explain the maximally highest proportion of the variance of the two subscales and the total scores (Tambs & Moum, 1993) and has been shown to correlate .92 with the global SCL-25 score. The SCL-5 has been shown to perform similar to the longer version, and alpha reliabilities for the present sample were good, reaching 0.80, 0.81, 0.82, 0.83, and 0.85 for the five assessments, respectively. When completing the SCL-5, the mother was asked to indicate whether during the last 14 days she was (a) not bothered or distressed at all, (b) was a little bit, (c) quite much, or (d) very much bothered by such problems as “Feeling blue” and “Worrying too much about things.” Respondents completing a minimum of three items were included in the analyses. Responses were scored using a 1– 4 scale. In the descriptive analyses, we used mean scores as well as a cutoff point of 2.0 to indicate the prevalence of high distress levels. This cutoff point is commonly used as a valid indicator of mental disorder, with prediction being somewhat better for depression than other disorders (Strand et al., 2003). In the modeling analyses, the SCL-5 was modeled as (a) a sum score standardized on the sample mean and standard deviation at the first assessment (latent growth curve analyses) and as (b) a latent variable (cholesky decomposition). Covariates. Data on education, parity, and civil status were provided by the Norwegian National Education Database of Statistics Norway and the Central Population Register.

Statistical Analyses Preliminary data analyses were conducted with SPSS version 20 and Mplus version 7 (Muthén & Muthén, 2007). Means and standard deviations were calculated using full information maximum likelihood (FIML) estimation in Mplus. Two complementary sets of structural equation models (SEMs) were specified using Mplus to examine stability and change, including latent growth curve (LGC) models and cholesky decomposition models. LGC modeling constitutes one of the main methods for exploring individual differences in intraindividual change (Duncan & Duncan, 2004). The LGC framework is highly flexible, allowing for exploration of individual levels and changes over time (i.e., adaptation) and accommodating both linear and nonlinear change patterns. The LGC models were conducted on SCL-5 and SWLS

sum scores standardized on the sample means and standard deviations at the first assessment. Missing data were estimated using the FIML procedure in Mplus, which permits use of all available information at the different assessments and provides improved estimation relative to a casewise deletion procedure (Graham, 2009). In accordance with standard recommendations (Bollen & Curran, 2004), we first specified separate univariate LGC models for SWLS and SCL-5 with one intercept factor and subsequently added slope factors (Muthén & Muthén, 1998 –2012). The latent intercept factor was fixed to one at each assessment and the latent slope factors were fixed to zero at birth, with the different assessments corresponding to time in years from birth. The first slope factor was specified to account for individual differences in linear change from Pregnancy Week 17–36 months postpartum and the second to account for nonlinear change. A piecewise model with two partly overlapping linear slopes was also tested to examine possible differences before and after birth. One slope factor was estimated for the period from Pregnancy Week 17 to 6 months postpartum, another for development from 6 to 36 months postpartum. Best fitting univariate models were then included in a full bivariate model to (a) examine changes in LS and psychological distress simultaneously and (b) explore how levels and changes in the two variables were related. The two indicators were combined by systematically correlating the intercepts and slope factors. DS and CLP were specified to impact on the intercept and slope factors in the full bivariate model, as were the covariates age, education, and parity. To complement the LGC analyses and examine stable and time-specific influences at each specific time point, we parameterized an extended cholesky, or triangular decomposition model (Loehlin, 1996), which is illustrated in Figure 1. Previous research has shown that retest correlations for the SWLS to a considerable extent reflect stable dispositions (Lucas & Donnellan, 2007; Schimmack, Krause, Wagner, & Schupp, 2010). A range of studies have likewise demonstrated that common psychological symptoms display considerable temporal stability (Nes et al., 2007). Thus, scales such as the SWLS and the SCL-5 reflect both statelike (time-specific) and traitlike (stable) aspects. The cholesky model is fundamentally atheoretical and basically decomposes the variance and covariance at each specific time point. Figure 1A illustrates a standard cholesky model for time series data consisting of three assessments. Here, a first latent factor (F1) impacts on all successive assessments (V1 – V3). The second latent factor (F2) is uncorrelated with the first factor (F1) and has effects on the second and the third assessments (V2 – V3), whereas the third latent factor (F3) is uncorrelated with the first two (F1 and F2) and is specific to the last assessment (V3) only. Thus, F1 represents the causes present at time1 that affect the observed variable (e.g., SWLS) at time1 as well as at subsequent assessments (i.e., stable variance). F2 represents additional causes (new variance) that arise at time2 and whose effects are added to the effects from F1 from time2 and onwards. Figure 1B shows this standard model extended to include a predictor with unique effects on the latent factors (F1 – F3). This model enables separation of the unique effects from the predictor at each specific time point while accounting for the stable trait variance. In addition, the new variance is separated into (a) time-specific influences and (b) lasting influences. In Figure 1C, the model is extended even further by exchanging the three observed variables (i.e., sum scores) for three within-time latent constructs, reflecting the shared variance between the individual items at each assessment.


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Predictor

Predictor


F1

V1

F2

V2

F3

V3

F1

V1

F2

V2

A

B

F1

F2

F3

V1

V2

V3

F3

V3

i1

i2

i1

i2

i1

i2

C

Figure 1. Cholesky decomposition model. F ⫽ factor; V ⫽ variable, i ⫽ item. Subscripts for V and F refer to time point.

The cholesky decomposition analyses were run using raw data and the robust maximum likelihood estimator in Mplus. Satisfaction with life and psychological distress were specified as latent factors (i.e., see Figure 1C). Influences from age, parity, and education were specified to impact on F1. The error variance of the individual items was allowed to correlate across time (e.g., first SWLS item at Pregnancy Week 17–36 months postpartum). In addition, the item loadings were constrained to be equal across assessments. As the modeling analyses were run regressing a standardized outcome (F1) on dichotomous predictor variables (DS and CLP), the unstandardized path coefficients constitute direct measures of Cohen’s d. Commonly, an effect size ⬍ 0.2 is considered small, 0.5 medium, and 0.8 large (Cohen, 1988). Model fit for both sets of models (i.e., LGC and cholesky decomposition) was assessed using the chi-square statistic, comparative fit index (CFI), Tucker-Lewis Index (TLI), root-meansquare error of approximation (RMSEA), Bayesian information criterion (BIC), and Akaike’s information criterion (AIC).

Results Attrition Altogether, 49,499 mothers (49.8%) participated at all five time points. Regressing the SWLS and SCL-5 scores on participation (dichotomous) showed that mothers who did not participate at all assessments (n ⫽ 49,934) scored significantly lower on the SWLS and higher on the SCL-5 than mothers participating at all time points. The effects were very small, however, with Cohen’s d ranging between 0.13 and 0.16 across the different assessments.

Prevalence of Severe Psychological Distress In total, 7.0%, 6.7%, 6.1%, 7.4%, and 7.9% of mothers of typically developing children scored above the clinical cutoff (⬎2.0) on the SCL-5 at Assessments 1–5, respectively. Corresponding estimates for CLP mothers were 6.9%, 6.8%, 8.8%, 7.2%, and 7.3% and for DS mothers, 8.3%, 7.8%, 13.3%, 12.5%, and 21.2%, respectively. There were no significant differences

between the three groups of mothers during pregnancy, or between ND and CLP mothers postpartum. Odds ratios between DS and ND mothers at the postpartum assessments were estimated to be 2.33 (95% CI [1.24, 4.40], p ⫽ .01), 1.79 (95% CI [0.89, 3.60], p ⫽ .08), and 3.12 (95% CI [1.60, 6.10], p ⫽ .01) at 6, 18, and 36 months, respectively.

Levels of Maternal LS and Psychological Distress FIML estimated correlations between the SCL-5 and SWLS sum scales were negative and moderate, ranging from ⫺.37 (Pregnancy Week 30) to ⫺.52 (36 months). Estimated means (FIML) for SWLS and SCL-5 with 95% confidence intervals and standard deviations are shown in Table 1. Overall, the mothers in our study reported fairly high scores on the SWLS throughout participation in the study. However, SWLS scores decreased somewhat over time, reaching their lowest level at 36 months postpartum (M ⫽ 5.45, SD ⫽ 1.11), whereas the SCL-5 scores remained largely stable. Decrease in satisfaction scores in the overall sample is assumed to reflect a natural decrease in LS following the birth of a child (Dyrdal et al., 2011; Dyrdal & Lucas, 2013). There were significant main effects (p ⬍ .05) of age, education, and parity, with higher satisfaction and lower psychological distress associated with higher education and being a first-time mother. Comparisons of means showed that DS mothers scored significantly lower on the SWLS than did ND and CLP mothers at 6, F(2, 84,908) ⫽ 11.46, p ⫽ .00, and 36 months, F(2, 53,590) ⫽ 3.06, p ⫽ .05. On the SCL-5, DS mothers scored significantly higher than ND and CLP mothers at 6 months, F(2, 85,563) ⫽ 10.33, p ⫽ .00; 18 months, F(2, 72,340) ⫽ 4.13, p ⫽ .02; and 36 months, F(2, 54,854) ⫽ 5.02, p ⫽ .01. CLP mothers scored significantly higher than ND mothers on SCL-5 at 6 months, F(1, 85,482) ⫽ 6.76, p ⫽ .02, but not at later time points. To investigate the relative increase and decrease in LS and psychological distress, SWLS and SCL-5 scores were standardized on the sample mean and standard deviation at the first assessment. Figures 2 and 3 show the standardized SWLS and SCL-5 scores for the three groups across the entire study period. As seen in Figure 2, SWLS scores for DS mothers decreased with 0.40 and

NES ET AL.

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Table 1 Estimated Means With 95% Confidence Intervals (in Brackets) and Standard Deviations for SWLS and SCL-5 Across Time (FIML Estimation Using Mplus) No disability (n ⫽ 99,139)

Down syndrome (n ⫽ 114)

Measure

Time

M

SD

M

SD

M

SD

Satisfaction With life (SWLS)

1 2 3 5 1 2 3 4 5

5.66 [5.65, 5.66] 5.72 [5.72, 5.73] 5.74 [5.74, 5.75] 5.45 [5.44, 5.46] 1.26 [1.26, 1.26] 1.26 [1.26, 1.26] 1.24 [1.24, 1.24] 1.28 [1.28, 1.28] 1.28 [1.28, 1.28]

1.07 0.97 0.99 1.11 0.40 0.39 0.39 0.42 0.44

5.65 [5.52, 5.77] 5.65 [5.53, 5.77] 5.66 [5.53, 5.80] 5.42 [5.23, 5.60] 1.30 [1.25, 1.35] 1.26 [1.21, 1.31] 1.33 [1.26, 1.40] 1.31 [1.25, 1.36] 1.30 [1.23, 1.37]

1.04 0.97 1.00 1.06 0.40 0.38 0.50 0.39 0.38

5.60 [5.43, 5.78] 5.74 [5.60, 5.88] 5.27 [5.07, 5.46] 5.06 [4.80, 5.31] 1.28 [1.20, 1.36] 1.27 [1.21, 1.34] 1.42 [1.34, 1.51] 1.45 [1.34, 1.55] 1.48 [1.36, 1.59]

1.15 0.87 1.13 1.38 0.47 0.41 0.47 0.55 0.59

Psychological distress (SCL-5)


Cleft lip/palate (n ⫽ 179)

Note. SWLS ⫽ Satisfaction With Life Scale; SCL-5 ⫽ five-item Hopkins Symptom Checklist; FIML ⫽ full information maximum likelihood; Time 1 ⫽ Pregnancy Week 17; Time 2 ⫽ Pregnancy Week 30, Time 3 ⫽ 6 months; Time 4 ⫽ 18 months; Time 5 ⫽ 36 months.

0.50 standard deviations from pregnancy to 6 and 36 months postpartum, respectively. By contrast, satisfaction scores for CLP and ND mothers increased with 0.05 and 0.10 standard deviations until 6 months, but subsequently decreased, reaching its lowest level at 36 months. SCL-5 scores were higher for both DS (0.34 SDs) and CLP (0.13 SDs) mothers 6 months after birth. However, at later assessments, the CLP mothers’ distress level was not significantly different from mothers of typically developing children, whereas the DS mothers’ distress level remained high (0.35 SD18months; 0.47 SD36months).

LGC Analyses The fit of the growth curve models are reported in Table 2. First, we explored the form of growth for the SWLS and SCL-5 data separately, excluding child condition and background variables. The univariate intercept-only models fit the data moderately well. Adding a linear growth factor improved the fit to the SWLS (⌬AIC ⫽ ⫺3790.53, ⌬BIC ⫽ ⫺3762.01) as well as the SCL-5 (⌬AIC ⫽ ⫺3421.00, ⌬BIC ⫽ ⫺3392.48) data. Inclusion of an additional quadratic growth term further improved the fit for both the SWLS (⌬AIC ⫽ ⫺2428.21, ⌬BIC ⫽ ⫺2390.19) and the SCL (⌬AIC ⫽ ⫺1463.42, ⌬BIC ⫽ ⫺1425.40). A piecewise model consisting of two linear growth functions was then tested. This

model fit the SWLS (␹12 ⫽ 405.63, RMSEA ⫽ 0.00, CFI ⫽ 1.00, TLI ⫽ 1.00) and the SCL-5 (␹62 ⫽ 202.53, RMSEA ⫽ 0.18, CFI ⫽ 1.00, TLI ⫽ 0.99) data excellently and turned out to be the best fitting univariate model for the SWLS (⌬AIC ⫽ ⫺130.45) as well as the SCL-5 (⌬AIC ⫽ ⫺406.31). The full bivariate model was therefore specified to include an intercept and two linear growth factors for both SWLS and SCL-5. 2 This model fitted the data very well (␹14 ⫽ 413.37, RMSEA ⫽ 0.017, CFI ⫽ 1.00, TLI ⫽ 0.99) when allowing the residuals to correlate. Examination of intercept variances showed that the intercepts differed significantly between participants (ISWLS ⫽ 0.519, p ⬍ .00; ISCL ⫽ 0.549, p ⬍ .00), indicating that the initial levels of satisfaction and distress were highly diverse among the sample. The slope (S) variance estimates also differed significantly (S1SWLS ⫽ 0.101, p ⬍ .00; S2SWLS ⫽ 0.056, p ⬍ .00; S1SCL ⫽ 0.268, p ⬍ .00; S2SCL ⫽ 0.041, p ⬍ .00). Correlations between intercepts (r ⫽ ⫺.59) and the first growth factors accounting for change between pregnancy and 6 months postpartum (r ⫽ ⫺.51) were substantial, suggesting that the two characteristics did not develop independently. Next, child conditions (i.e., CLP and DS) and background variables were added to the model and the intercepts and growth factors regressed on the predictors. This final 2 model fitted the data well (␹29 ⫽ 578.38, RMSEA ⫽ 0.013, CFI ⫽

Figure 2. Maternal satisfaction with life from Pregnancy Week 17 to 3 years after birth. Scores are standardized at the first assessment.



7

Figure 3. Maternal psychological distress from Pregnancy Week 17 to 3 years after birth. Scores are standardized at the first assessment.

1.00, TLI ⫽ 0.99). The effect of DS on the intercepts was significant for both LS (Cohen’s d ⫽ ⫺0.31) and psychological distress (Cohen’s d ⫽ 0.26), indicating that the predicted means differed between DS and cohort mothers at birth. The slope factors accounting for change from pregnancy until 6 months postpartum were substantially predicted by DS, but not by CLP. Those who experienced having a child with DS experienced rapidly decreasing LS (Cohen’s d ⫽ ⫺1.35) and increasing psychological distress (Cohen’s d ⫽ 0.71) after birth, whereas the growth factors of CLP and ND mothers were not significantly different. DS did not predict the two growth terms accounting for changes after 6 months, indicating no differences in growth trajectories across groups after 6 months (i.e., difference remained stable). Parity and education predicted the intercept and slopes of both SWLS and SCL-5, whereas age predicted both the intercept and slope of SCL-5, but only the slope factor of SWLS.

cholesky factors (i.e., F3 and V3) at 6 months, and thus to have a substantial unique effect on change in both SWLS (Cohen’s d ⫽ ⫺0.66) and SCL-5 (Cohen’s d ⫽ 0.60) scores (bold paths from DS in Figure 4) at 6 months postpartum. There was no unique impact of DS on changes in satisfaction and distress scores emerging at later time points. Lower LS and higher distress at later time points thus appear to reflect stable influences from the new variance measured at 6 months (reflected in the path coefficients representing stable influences from 6 months and onward). CLP was found to have a small but significant effect on the SCL-5 (Cohen’s d ⫽ 0.29) at 6 months, but not on the SWLS (bold path from CLP in Figure 4). The model-estimated cross-time correlations for the latent SWLS factors ranged between 0.56 (between Pregnancy Weeks 17 and 30) and 0.47 (Pregnancy Week 17 and 36 months), indicating considerable, although decreasing, stability in SWLS scores over time. Modelestimated correlations between the latent SCL-5 factor at the first and the successive assessments were .68 (between Pregnancy Weeks 17 and 30), .58 (Pregnancy Week 17 and 6 months), .54 (Pregnancy Week 17 and 18 months), and .51 (Pregnancy Week 17 and 36 months), thus decreasing somewhat over time, but indicating considerable cross-time stability also in psychological distress.

Bivariate Cholesky Decomposition Results from the bivariate cholesky decomposition model are illustrated in Figure 4. The full cholesky model fitted the data 2 moderately well, ␹1030 (N ⫽ 99,433) ⫽ 78,289.83, p ⫽ .00, CFI ⫽ .95, TLI ⫽ 0.94, RMSEA ⫽ .027. DS was found to impact on both

Table 2 Fit Measures From Latent Growth Curve Modeling Variable

Model

␹2

df

p

RMSEA

CFI

TLI

AIC

BIC

Psychological distress

Intercept only ⫹ Linear slope ⫹ Quadratic slope Piecewise Intercept only ⫹ Linear slope ⫹ Quadratic slope Piecewise Full modela ⫹ Predictors

2610.81 1050.02 394.54 202.53 3696.61 1532.18 118.28 405.63 413.37 578.38

13 10 6 6 8 5 4 1 14 29

.00 .00 .00 .00 .00 .00 .00 .00 .00 .00

0.045 0.032 0.026 0.018 0.068 0.055 0.034 0.000 0.017 0.013

0.94 0.98 0.99 1.00 0.85 0.94 1.00 1.00 1.00 1.00

0.95 0.98 0.99 1.00 0.88 0.92 0.97 1.00 0.99 0.99

1012005.85 1008584.85 1007121.43 1006715.12 794573.37 790782.84 788354.63 788224.18 1748032.10 1707546.32

1012072.39 1008679.91 1007254.51 1006848.20 794630.40 790868.39 788478.20 788347.75 1748412.39 1708210.41

Life satisfaction

Bivariate

Note. RMSEA ⫽ root-mean-square error of approximation; CFI ⫽ comparative fit index; TLI ⫽ Tucker–Lewis Index; AIC ⫽ Akaike’s information criterion; BIC ⫽ Bayesian information criterion. a Correlated error terms.

NES ET AL.

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Birth

1.00

SWLS SWLS

SWLS

SWLS SWLS

.56

SWLS

66 months months

Week 30

Week Week 17 17

.47

.76

.54

.83

36 months

.76

.37 .32


.31

F2

F1

F3

F5

-.66 Down Syndrome

-.37

-.22

-.45

-.43

.60

Cle Lip/palate

.29 V2

V1

V3

V4

.23 .22 .77 .34

1.00

.68

.76

.58

.24

.28

V5

.73

.28

.73

.54 .51

SCL-5

SCL-5

SCL-5

SCL-5

SCL-5

Week 17

Week 30

6 months

18 months

36 months

Birth

Figure 4. Bivariate Cholesky decomposition for maternal life satisfaction (SWLS) and psychological distress (SCL-5) Pregnancy Week 17 to 36 months postpartum. Two-headed arrows ⫽ correlations between the Satisfaction With Life Scale (SWLS) and the five-item Hopkins Symptom Checklist (SCL-5); bold paths ⫽ significant influences on new variance in the SWLS and SCL-5; F ⫽ latent factor representing new variance in SWLS; V ⫽ latent factor representing new variance in SCL-5; subscripts ⫽ time points.

Correlations between the latent SWLS and SCL-5 factors were estimated to be ⫺.45, ⫺.22, ⫺.37, and ⫺.43 at Pregnancy Week 17, Pregnancy Week 17, and 6 and 36 months postpartum.

Discussion In this study, we examined the reaction and adaptation to the birth of a child with a congenital anomaly in a large population-based prospective study, aiming to explore how major life stress affects stability and change in both LS and psychological distress more generally, and the impact of having a child with DS or CLP more specifically. Overall, the LGC analyses and cholesky decomposition indicated that having a child with DS, but not with CLP, causes substantial changes in maternal LS and psychological distress. The effects are observed as occurring between pregnancy and 6 months postpartum, and seem to persist up to 3 years after birth.

The LGC analyses indicated that the initial levels and growth trajectories of both satisfaction and distress were highly diverse among the sample and that satisfaction and distress levels were considerably stable over time. However, the birth of a child with DS was associated with a rapid decrease in LS and a corresponding increase in psychological distress. Controlling for relevant family factors such as parity, age, and education, along with stable levels of satisfaction and distress, the complementary cholesky analyses showed a considerable reduction in LS (Cohen’s d ⫽ ⫺0.66) and a corresponding increase in psychological distress (Cohen’s d ⫽ 0.60) among DS mothers 6 months after birth (see Figure 4). By contrast, CLP mothers reported higher distress scores (Cohen’s d ⫽ 0.29) 6 months after birth, but their satisfaction scores remained similar to cohort mothers throughout the study. The type of diagnosis—probably reflecting the severity and chronicity of the



child’s condition—thus appears to have different impact on overall LS and psychological distress after birth. The results corroborate findings from a previous cross-sectional study reporting mothers of a 6-month-old infant with DS or congenital heart disease (CHD) to experience more stress and adaptation difficulties than mothers of infants with CLP or nondisabled babies (Pelchat et al., 1999). In this previous study, parents of children with DS and CHD were shown to struggle more to accept their child, felt more threatened by their parental situation, and perceived their situation as more stressful and uncontrollable. The irreversibility of the disability, the foreseen limits in the child’s development, health and future, and the anticipated caregiving burden probably contribute to elevate distress and reduce LS in DS mothers. The present study did not allow for exploration of such factors. However, it supports previous reports indicating a range of responses to having a child with disabilities. These individual differences are likely dependent on a number of biological (e.g., genetic factors), socioeconomic, psychosocial (e.g., coping strategy, social support network, previous stressors), and child-related (e.g., adaptive behavior) factors and the interactions between them. Rather than merely a response to a disabled child, adaptation is a response to the child mediated by the coping resources available to the mother and her family. Parents of children with chronic conditions probably function much like others who experience stressful circumstances, with some developing maladjustment, others showing positive coping and resilience, seeking out new sources of purpose and recognizing that children with a disability might have a positive impact on the family. Such positive coping, stressrelated growth, or benefit finding, has been indicated to be strong for DS parents (Corrice & Glidden, 2009; King, Scollon, Ramsey, & Williams, 2000). Still, despite acknowledgment of individual variability and positive coping, the present study convincingly shows that the birth of a child with DS is associated with substantial increase in maternal psychological distress and reduced LS from birth and throughout the toddler stage.

Stability and Change The mothers varied significantly in their initial levels of both LS and distress as well as in their direction and rate of change. In line with previous studies (Dyrdal et al., 2011; Yap et al., 2012), having a healthy child was associated with a minor short-term boost in overall LS, whereas having a child with DS was associated with a considerable reduction. Mothers of DS children had significantly lower predicted means at the time of birth, their observed satisfaction scores were significantly lower 6 months postpartum, and there was no indication of improvement in their satisfaction scores over time. As the normative trend was toward lower LS 3 years postpartum, the difference in satisfaction scores between DS and cohort mothers remained stable between 6 and 36 months. Overall, our findings are at variance with studies indicating that satisfaction levels commonly are reactive to short-term changes in external circumstances, but generally adapt relatively quickly to long-term ones (Suh et al., 1996) and in line with studies reporting long-term effects of major life stressors such as divorce (Lucas, 2005), unemployment (Lucas et al., 2004), and becoming disabled (Lucas, 2007). Complementing the satisfaction findings, psychological distress levels were significantly higher among DS mothers at all postpar-

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tum assessments with no reduction observed over time, indicating stability in symptom levels throughout the toddler period. Higher levels at 18 and 36 months appear to reflect a persistent burden emerging after birth and measured from 6 months postpartum, rather than psychological responses to new challenges arising over time. Additionally, DS mothers were shown to have excess risk of severe psychological distress indicative of a clinical diagnosis at 6 and 36 months, evidencing that many mothers of young DS children face challenges that clearly impact on their mental health and overall well-being. The SWLS and SCL-5 were moderately and negatively correlated throughout the study, and LGC analyses indicated that satisfaction and distress levels did not develop independently. Nevertheless, the normative pattern of stability and change differed somewhat across the outcome measures in the overall sample. Whereas maternal LS increased from late pregnancy to 6 months postpartum and subsequently decreased, psychological distress levels remained largely stable. Additionally, CLP was shown to constitute a stronger predictor of recent negative affect than overall LS 6 months after birth, whereas DS predicted satisfaction and distress equally well. These findings are in accordance with previous studies indicating that different aspects of subjective wellbeing constitute distinct components (Lucas et al., 1996; Luhmann et al., 2012), and might show differential stability (Eid & Diener, 2004) and different associations with measures of health and longevity (Wiest, Schuz, Webster, & Wurm, 2011). Negative and positive indicators of mental health and well-being probably reflect divergent motivational functions (Luhmann, Hawkley, Eid, & Cacioppo, 2012). Previous research indicates that people tend to consider global life circumstances (e.g., financial situation, employment status) when making cognitive well-being judgments and to consider specific activities and recent events when making affective judgments. As the SWLS asks respondents to evaluate their life as a whole while the SCL queries about specific recent symptoms of negative affect, the latter is probably more likely to reflect the mothers’ immediate life situation and daily events and, to a greater extent, reveal the worry, sadness, and daily hassles that mothers of disabled children experience. Daily hassles are also commonly shown to be better predictors of psychological symptoms than discrete life events (Kanner, Coyne, Schaefer, & Lazarus, 1981). The research literature indicates that major life stressors commonly affect both cognitive and affective indicators quite instantly. However, long-term effects on global cognitive measures (i.e., SWLS) are to be expected only when stressors cause changes in global life circumstances. Likewise, long-term effects on affective measures (i.e., SCL) result when the stressor remains a source of daily stress. In the present study, the birth of a child with DS, but not CLP, predicted reduced LS and increased psychological distress at all postpartum assessments. Our findings therefore suggest that having an infant with DS is associated with persistent stress that is changing life circumstances sufficiently to alter maternal LS overall. Throughout the toddler stage, DS mothers appraise their lives as less good and feel worse in emotional terms because their child is a source of concern. By contrast, CLP mothers appraise their lives as overall good, but feel worse in emotional terms some months postpartum as their child is a source of temporary concern. In sum, the results show that global LS and psychological distress are uniquely and similarly affected by the birth of a child with DS,

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and differentially affected by the birth of a child with CLP. They contrast studies finding cognitive well-being to generally improve relatively soon following a life stressor and extend the literature by examining these processes conjointly in a prospective populationbased sample for a major life stressor occurring independent of personality, namely the birth of a child with a chronic disability.


Strengths and Limitations The present study has a number of important strengths, including its large sample and prospective design with baseline assessments of both positive and negative components of mental health conducted prior to birth. Along with the linking of cohort data to relevant demographic information (e.g., socioeconomic status, parity, and maternal age) from national registries, this allows for examination of differential and unique effects of DS and CLP on separate components of mental health simultaneously. An additional strength concerns all children having the same age. As different stages of the child’s development are likely to introduce varying caretaking challenges, particularly for children with special health care needs, the use of an age homogenous sample allows for in-depth examination of the particular developmental stage stretching from birth to 36 months postpartum. However, the results need to be interpreted in the context of some methodological limitations. First, the response rate in the MoBa study is lower than optimal (initial recruitment rate of 40.6%), and there is an overall selection bias leading to overrepresentation of mothers with higher education, with more living in stable relationships and having positive birth outcomes. When compared with all women giving birth in Norway, associations of the various exposures and outcomes in the study have not been shown to be considerably affected (Nilsen et al., 2009). However, a minor difference in satisfaction and distress scores was observed between mothers participating at all assessments and mothers missing one or more assessments (Cohen’s d ⫽ 0.13– 0.16). The presence of some sample selection bias in terms of healthier respondents thus allows for the suggestion that our findings underestimates, rather than overestimates, the level of psychological distress and overestimates overall LS. Second, the sample size of the DS group was rather small due to the infrequency of the event and included only 52 cases at the last assessment. The analyses were conducted using the FIML procedure in Mplus, however, which permits use of all available information at the different assessments and provides improved estimation relative to a casewise deletion procedure (Graham, 2009). Third, the actual number of DS cases in our sample is somewhat on the low side. The diagnosis of DS was based on the MBRN, and several studies have been conducted to validate the DS registration in the MBRN. One report estimated the prevalence to 1.25 per 1,000 live births (Bjerkedal & Kristensen, 2007). A more recent study reported the prevalence to be 2.0 per 1,000 based on all laboratory-verified cases among 288,213 births (live births, stillbirths, and terminations) reported to the MBRN during 2001–2005 (Melve et al., 2008). Sensitivity in the MBRN was estimated to be 81.6% in this latter study, with sensitivity being higher for live births than for stillbirths. False positives were estimated to less than 10% and were often pregnancy terminations (13 out of a total of 51 cases). Our sample is thus not likely to include many false positives. Additionally, having a child with DS did not predict

participation in our sample over time (i.e., no indication of selective dropout among DS mothers). Fourth, information regarding prenatal diagnosis was not available, and some mothers might have been prepared for their child’s condition before birth, particularly mothers of children with CLP. The prenatal detection rate for CLP has improved significantly over time, and a Norwegian prospective study on prenatal ultrasound detection of facial clefts (49,314 deliveries) reported that 45% of cases with CLP were detected (Offerdal et al., 2008). The majority of these cases (69%) were detected at the routine ultrasound examination at Pregnancy Week 17. No cases with only cleft palate were detected prenatally. DS has been shown to be detected prenatally in 43% of cases based on karyotyping (available for expecting mothers over 38 years only) and ultrasound examination in a Norwegian study (Ogston, Mackintosh, & Myers, 2011). However, the termination rate of prenatally diagnosed DS cases in Norway has been estimated to range between 73% and 96% during the years relevant to the present study (Mo, Seliussen, Irgens, & Gåsemyr, 2006). As all pregnancy terminations were excluded in our study, the majority of DS mothers were probably not informed about their child’s diagnosis before birth. However, 30%– 40% of CLP mothers might have been informed. Fifth, the SCL-5 is a self-report measure, and its sensitivity and specificity to formal DSM anxiety and mood disorders are probably only moderate. A previous Finnish study of young adults has shown the specificity and sensitivity of the HSCL-25 for any present DSM–III–R psychiatric disorder to be 48% and 87%, respectively, and the positive and negative predictive power to be 34% and 92% (Veijola et al., 2003). The longer versions of the Hopkins SCL are usually recommended for clinical purposes due to their superior discriminative abilities (Müller, Postert, Beyer, Furniss, & Achtergarde, 2010). Nevertheless, despite not constituting an ideal indicator of formal psychiatric disorder, high levels of anxiety and depressive symptoms represent significant risk factors for subsequent major depression and are considered important targets for prevention as well as intervention in their own right (Kessler, Zhao, Blazer, & Swartz, 1997). The higher percentage of DS mothers scoring above the clinical cutpoint on the SCL-5 is also in accordance with findings from past longitudinal studies, which indicate that depressive levels commonly are high at the time of diagnosis. However, the higher percentage observed at later time points contrasts previous findings of declining levels over time (Bailey et al., 2007; Glidden & Schoolcraft, 2007; Singer, 2006). Of note, although previous studies have shown that relatively few report clinical levels of parent-related stress scores as their child ages, many report child-related stress scores in the clinical referring range (Hauser-Cram et al., 2001). Hauser-Cram and colleagues (2001) have, for example, reported that DS mothers’ child-related stress scores increase over time, and by child age 10 years, DS mothers report considerably more stress than mothers of children with developmental delay or motor impairment.

Concluding Remarks Mental health problems and diminished LS are linked to poorer individual, familial, and societal outcomes, and even mild levels of



distress might have detrimental effects on individuals and their families. Maternal mental health represents a significant public health concern, and mothers of children with health problems and disabilities are particularly vulnerable. The cumulative effect of multiple daily caregiving stressors has been shown to promote elevations in inflammatory markers associated with depression, cardiovascular disorders, frailty, and mortality (Gouin, Glaser, Malarkey, Beversdolf, & Kiecolt-Glaser, 2012; Mausbach, Patterson, Rabinowitz, Grant, & Schulz, 2007). Caregivers of children with health problems have been found to have excess risk of health problems in general (Brehaut et al., 2009) and mental health problems like depression more specifically (Bailey et al., 2007; Singer, 2006). Maternal depression is also related to negative child behavior outcomes across a wide age range, including later child internalizing and behavior problems and a child’s first year might represent a particularly sensitive period (Bagner, Pettit, Lewinsohn, & Seeley, 2010). This underscores the importance of early identification and intervention. Parents and the disability service system need to know how families of disabled children compare with families of typically developing children. Likewise, they need information about the likely changes in care demands and their consequences over time. Most experiences comprise both positive and negative aspects, and the dominant quality might change with varying circumstances and demands. The present study followed the mothers until child age 3 years only, and further longitudinal, prospective, and large-scale studies of both positive and negative outcomes are needed for advancing our understanding of the complexity of parental adjustment over time.

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Received January 2, 2013 Revision received October 25, 2013 Accepted January 8, 2014 䡲