The associations between infant development and parenting stress in infants with congenital heart disease at six and twelve months of age

Journal of Pediatric Nursing 51 (2020) 1–7

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The associations between infant development and parenting stress in infants with congenital heart disease at six and twelve months of age Nadya Golfenshtein, PhD, MHA, RN a,⁎,1, Alexandra L. Hanlon, PhD a,2, Janet A. Deatrick, PhD, RN, FAAN a, Barbara Medoff-Cooper, PhD, RN, FAAN b,c a b c

University of Pennsylvania, School of Nursing, 418 Curie Blvd, Claire M. Fagin Hall, Philadelphia, PA 19104, United States of America University of Pennsylvania, School of Nursing, 418 Curie Blvd, Claire M. Fagin Hall, Philadelphia, PA 19104, United States of America Children's Hospital of Philadelphia, 3401 Civic Center Blvd., Philadelphia, PA 19104, United States of America

a r t i c l e

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Article history: Received 2 July 2019 Revised 24 November 2019 Accepted 24 November 2019 Available online xxxx

a b s t r a c t Purpose: Developmental delays are among the major morbidities of children with complex congenital heart disease. Parents of infants with complex congenital heart disease experience increased parenting stress levels, which can interfere with parenting processes during infancy. The current study examined associations between infant development and parenting stress in infants with complex congenital heart disease at six and twelve months of age. Design and methods: A secondary analysis of data examined cross-sectional associations between infant's mental and psychomotor development and parenting stress, using general linear regression modeling (N = 75). Data were obtained from a larger prospective cohort study. Results: Mental development was negatively associated with the Parent Domain at six months, and with the Parent Domain and Total Stress at twelve months. Psychomotor development was not significantly associated with parenting stress at six and twelve months. Conclusions: Parenting stress in parents of infant with complex congenital heart disease may be among the factors shaping the parent–child relationship during the first year of life, which plays an important role in infant development. A potential bidirectional relationship between parenting stress and infant development may fit a transactional model representing the phenomena. Practice Implications. Family interventions aiming to reshape illness perceptions may promote parental adaptive coping and productive parenting practices in populations at risk. © 2018 Elsevier Inc. All rights reserved.

Introduction With recent improvements in technology and healthcare, more infants with once fatal heart conditions are surviving into childhood (Cohen et al., 2011). Within just two decades, the survival rate among infants with complex congenital heart defects (CHD), the most prevalent group of congenital anomalies, has increased dramatically, and has become one of the most common pediatric chronic illnesses (Krasuski & Bashore, 2016). Children with complex CHD who have undergone cardiac surgery early in infancy (the first year of life) display a wide range of developmental delays, varying per defect type and ⁎ Corresponding author. E-mail addresses: ngolfi[email protected] (N. Golfenshtein), [email protected] (A.L. Hanlon), [email protected] (J.A. Deatrick), [email protected] (B. Medoff-Cooper). 1 Permanent Address: University of Haifa, Department of Nursing. 199 Abba Hushi Ave. 3498838. Haifa, Israel. 2 Permanent Address: Virginia Tech. Center for Biostatistics and Health Data Science.

https://doi.org/10.1016/j.pedn.2019.11.012 0882-5963/© 2018 Elsevier Inc. All rights reserved.

complexity, with the highest and severest impairments in infants with complex CHD (Marino et al., 2012). The multifactorial causes of the delays include condition-related factors such as genetic/chromosomal abnormalities, poor birth and growth parameters, and postoperative factors (Ballweg, Wernovsky, & Gaynor, 2007; Chang, Walker, Grantham-McGregor, & Powell, 2010; Fuller et al., 2009). The impairments include motor and speech delays, executive function deficits, inattention, and hyperactivity. These have long-term implications as children progress through school, often leading to academic disadvantage, poor social skills, low self-esteem, and behavioral disinhibition (Marino et al., 2012). Developmental delays have been associated with increased parenting stress across healthy and ill pediatric populations (Ben-Sasson, Soto, Martinez-Pedraza, & Carter, 2013; Molfese et al., 2010; Voigt et al., 2013). Similar associations have scarcely been investigated in the CHD population, despite the increased incidence of developmental delays. It is well established that parents of infants and children with CHD experience significantly higher parenting stress than the general

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population (Mullen et al., 2014; Sarajuuri, Lonnqvist, Schmitt, Almqvist, & Jokinen, 2012; Uzark & Jones, 2003). Parenting stress, the distress experienced while trying to meet the parenting role demands (DeaterDeckard, 2004), is linked to numerous adverse outcomes in both parents and children, including decreased well-being, anxiety, depression, parental role strains, and poor illness adjustment (Fonseca, Nazaré, & Canavarro, 2011; Semke, Garbacz, Kwon, Sheridan, & Woods, 2010). The current study sought to examine the associations between infant development and parenting stress in infants with complex CHD at six and twelve months of age. The parenting stress model Abidin (1995) identified specific stress-evoking factors in the parenting role and categorized them into several domains. The Child domain includes child's behavioral/temperamental characteristics (e.g. demandingness, adaptability, distractibility, mood) and parental expectations and perceptions of the child (e.g. acceptability, parental reinforcement). The Parent Domain includes parental functionality and personality components (e.g. depression, parental competence, feelings of attachment) and additional circumstantial components (e.g. parental health, role restriction, isolation, relationship with spouse). The model also includes situational circumstances outside the parent–child system. Although Abidin's model is widely used in the CHD population (DeMaso et al., 1991; Phipps & Drotar, 1990; Uzark & Jones, 2003) it lacks unique, illness-related stressors. Research has mainly attributed the increased early parenting stress in the complex CHD pediatric populations to illness-related factors, such as the multiple medical procedures, the lengthy stay in the cardiac intensive care unit (CICU), the transition home from the hospital, and the increased caretaking burden (Lisanti, Allen, Kelly, & Medoff-Cooper, 2017; Sarajuuri et al., 2012). These factors may have implications for the relationships being formed between the parents and the infant (Årestedt, Benzein, & Persson, 2015). Studies investigating family relations within the context of pediatric chronic or life-threatening illnesses have reported strains in the parental role (Goldberg, Morris, Simmons, Fowler, & Levison, 1990; Miles, Carter, Hennessey, Eberly, & Riddle, 1989). Specifically, the quality of the parent–child relationship and the parental practices are altered due to parental maladjustment to the illness (Årestedt et al., 2015). The developmental literature states that disturbed parent–child relationships and child-rearing malpractices can negatively impact the child's development (Bowlby, 1988; Hintermair, 2006; Molfese et al., 2010; Voigt et al., 2013). We hypothesize that higher parenting stress scores will be associated with lower infant mental and psychomotor developmental scores at six and twelve months of age. Demonstrating such associations beyond the early infancy period, during which most medical interventions occur, and after parents have begun adjusting to the illness, may illuminate additional stressors later in infancy in a population with high incidence of developmental delays.

for their heart defect within their first six weeks of life; who were born after 35 gestational weeks; and who weighed above 2000 g at birth. Infants with other congenital or acquired lesions, potentially affecting feeding, growth, or development, were excluded from the parent study. The total enrollment for the original study was 241 infants. The sample for the current analysis included 75 infants. Study procedures and data collection Both the original and the current studies were approved by the institutional review board. Informed consent was signed by all parents or legal guardians. Data were obtained at hospital discharge, and during participants' subsequent visits to the outpatient clinic. Parents completed self-reporting questionnaires for parenting stress at three, six, nine, and twelve months of age; and infant development was evaluated by medical professionals at six and twelve months of age. Medical information was obtained from medical charts. Study variables and instruments Parenting stress Parenting stress was assessed via the Parenting Stress Index (PSI) Long Form, a validated, standardized, self-reporting questionnaire for parents. The Long Form consists of 120 items, yielding scores over 17 subscales on the domains identified in Abidin's model. Forty-seven 5-point Likert-scale items measure stress over the Child Domain subscales. An example item is: “My child seems to cry or fuss more often than most children.” Fifty-four 5-point Likert-scale items measure the Parent Domain subscales. An example item is: “I often feel guilty about the way I feel towards my child.” Scores from the Parent and Child domains are summed to an overall Total Stress score. Subscales' alpha reliability coefficients range from 0.70 to 0.90 (Abidin, 1995). Individual interpretation of subscales permits analyzing specific aspects of the parent–child system (Abidin, 1995). PSI scores are analyzed on a continuum; higher scores indicate higher stress levels.

A repeated cross-sectional analysis of data obtained from a larger prospective cohort study was performed to examine associations between infant development and parenting stress at six and twelve months of age.

Infant development The Bayley Scales of Infant Development–2nd Edition (BSID-II; Bayley, 1993) was used to assess infant development. Studies using the later version of the tool— the BSID-III—in the CHD population reported lower detection percentages of delays compared with similar samples who were tested with the BSID-II (Acton et al., 2011; Long, Galea, Eldridge, & Harris, 2012). The BSID-II is a validated, standardized measure of children's development between 1 and 42 months of age. One hundred seventy-eight items measure mental capacity and cognitive skills on the Mental Scale; 111 items measure gross and fine motor skills on the Motor Scale; and 30 subjective-rating items assess emotional, behavioral, social, and environmental orientation on the Behavior Rating Scale (BRS). Raw Mental and Motor scores are transformed into the Mental Development Index (MDI) and the Psychomotor Development Index (PDI), with scores ranging from 50 to 150 (Mean = 100, SD = 15; Bayley, 1993). The BRS produces a subjective, percentile ranking score (Bayley, 1993). Alpha reliability coefficients range from 0.64 to 0.93, with the lowest coefficients for the BRS (Bayley, 1993). In accordance with previous research (Medoff-Cooper et al., 2016), only the MDI and the PDI were used in the current study. Infant development scores are measured on a continuum; lower scores indicate developmental delays.

Setting and participants

Covariates considered for the analysis

A convenience sample of infants with complex CHD was recruited from the CICU of a large hospital in the northeast mid-Atlantic region of the US. The original study aimed to explore feeding behaviors, growth, and development in infants with complex CHD. Therefore, the sample included infants who underwent corrective or palliative surgery

Clinical parameters Clinical parameters representing illness severity, which may confound the relationship of interest, included postoperative cardiac physiology (single- vs. bi-ventricle; Khairy, Poirier, & Mercier, 2007), hospital length of stay (in days; Ballweg et al., 2007), feeding mode

Methods Study design

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(oral vs. device-assisted feeding; Medoff-Cooper & Irving, 2009), gestational age (Best, Tennant, & Rankin, 2017; Steurer et al., 2017), birthweight, and infant growth (WHO growth Z-scores; MedoffCooper et al., 2016). Demographics Demographics were collected from the medical records and via parents' self-reporting, and included infant gender, race, ethnicity, parental education, and whether the CHD was diagnosed prenatally. Data analyses Descriptive statistics were generated to characterize all demographic and clinical variables. Means, standard deviations, medians, and ranges were used to describe continuous variables. Frequencies and percentages were used to describe categorical variables. Multivariable linear regression modeling was used to examine the associations between parenting stress (Parent Domain, Child Domain, and Total Stress subscales) and infant neurodevelopment (MDI and PDI) at 6 and 12 months, separately (Cohen & Cohen, 1983). Covariates were selected based on the 0.2 significance level in bivariate models, and further by backward deletion process at the 0.2 significance level. MDI and PDI were regressed over PSI subscales in separate multivariable models, adjusting for infant-length Z-scores at the measured timepoint, gestational age, and hospital length of stay. Estimates of R2 values are provided to quantify the variation in outcome explained by each model (Cohen, 1988). Because of the secondary nature of the study and the fixed sample size, multiplicity was not taken into account. Specifically, statistical significance was set at the 0.05 level, and all analyses were conducted using STATA Version 13 (StataCorp, 2013). Interpretations of the models are made in the context of both clinical meaningfulness and statistical significance. Results Table 1 depicts the demographic and clinical characteristics of the sample (n = 75). The final sample was mostly White (n = 70; 93%), non-Hispanic (n = 50; 67%), and male (n = 54; 72%). Thirty-five infants (47%) had single-ventricle (SV) post-op physiology, and almost half (49%) were diagnosed with CHD prenatally. The infants' median hospital length of stay was 15 (Range = 2–159) days, with 31 infants (41%) requiring device-assisted feeding (i.e. nasogastric/gastric tube) at discharge, and nine (12%) still requiring device-assisted feeding at three months. Infant developmental scores were below the population's average (Mean = 100, SD = 15) at six and twelve months of age, with psychomotor development (PDI scores) more severely delayed (Mean = 79.79, SD = 14.31, and Mean = 79.13, SD = 16.38 respectively) than MDI scores (Mean = 91.24, SD = 10.78, and Mean = 94.21, SD = 12.26 respectively). Table 3 summarizes results from the multivariable linear regression models for parenting stress and infant development (MDI, PDI) at six and twelve months (see Table 2 for univariable results). MDI was negatively associated with Parent Domain scores at six (p = 0.044) and twelve months (p = 0.024) of age, and with Total Stress scores at twelve months of age (p = 0.046), adjusting for infant length, gestational age, and hospital length of stay. The percentage variation for these models ranged between 27 and 40. No significant associations were found between parenting stress and PDI scores. Discussion The current study sought to examine associations between parenting stress and infant development at six and twelve months of age in infants with complex CHD. Consistent with previous reports (Long et al., 2012; Mussatto et al., 2014), both PDI and MDI scores in our sample are below standardized means, with MDI being less severely affected.

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Table 1 Demographic and clinical characteristics of the study sample, N = 75. Categorical variables

Frequency (%)

Infant gender Male Female Ethnicity Hispanic Non-Hispanic Unknown Race White Black Other Unknown Mother's education High school Collage Post-graduate degree Unreported Post-op cardiac physiology Single ventricle Bi-ventricle Prenatal diagnosis Yes No Feeding mode at discharge Oral Tube assisted Missing Feeding mode at 3 months Oral Tube assisted Missing

54 (72) 21 (28) 7 (9) 50 (67) 18 (24) 70 (93) 3 (4) 1 (1) 1 (1) 4 (5) 27 (36) 10 (13) 34 (45) 35 (47) 40 (53) 36 (49) 38 (51) 35 (47) 31 (41) 9 (22) 53 (71) 9 (12) 13 (17)

Continuous variables

Mean (SDa)

Median (range)

Birth weight, gms (n = 75) Gestational age, wks (n = 74) Weight at 6mo, z-scoreb (n = 44)

3395 (517) 38.9 (1.3) −0.77 (1.31)

Length at 6mo, z-score (n = 44)

−0.41 (1.41)

Head circumference at 6mo, z-score (n = 44) Hospital length of stay, days (n = 75) MDIc at 6mo (n = 46)

−0.12 (1.04)

3430 (2409–4900) 39 (35–42) −0.44 (−4.74 1.14) −0.32 (−6.19 1.99) −0.14 (−2.59 1.55) 15 (2–159) 92.5 (60–111)

PDIc at 6mo (n = 47) MDI at 12 months (n = 63) PDI at 12 months (n = 63) a b c

24.2 (26.17) 91.24 (10.78) 79.79 (14.31) 94.21 (12.26) 79.13 (16.38)

79 (50–111) 93 (66–117) 77 (50–113)

Standard deviation. WHO growth Z-scores. Bayley's Mental and Psychomotor Development Index scores.

Findings indicate that lower mental development scores are linked to higher parenting stress related to parental characteristics at both time points, and to higher total stress at twelve months. Therefore, these findings only partly support our hypothesis with regard to MDI. Low MDI scores represent cognitive delays including higher-order mental processing, language acquisition, reasoning, memory, and integration of these processes (Aylward, 1997). Although neurodevelopmental delays might be expected to be less influenced by environment than by biological processes, studies have demonstrated that causes of these delays are multifactorial, with familial and socioeconomic factors being perhaps central predictors in children with CHD (Ballweg et al., 2007; Fuller et al., 2009; Wernovsky, 2006). McCusker et al. (2007) demonstrated the importance of family processes etiologically on neurodevelopmental outcomes of children with CHD. In their analysis, parenting style, marital status, maternal mental health difficulties, and maternal worry had greater predictive importance than the disease and surgical factors. Examination of the

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Table 2 Univariable regression models for MDIb and PDI on PSI at 6 and 12 months. PSI subscalea

PDI

MDI

β

SEc

95% CId

P

R2

β

SE

95% CId

P

R2

6 months (N = 47) Child domain Parent domain Total stress

−0.10 −0.11 −0.07

0.14 0.11 0.07

(−0.37, 0.18) (−0.33, 0.10) (−0.15, 0.14)

0.491 0.288 0.304

0.01 0.03 0.05

−0.14 −0.12 −0.08

0.10 0.08 0.05

(−0.35, 0.67) (−0.28, 0.41) (−0.18, 0.02)

0.180 0.143 0.107

0.04 0.05 0.06

12 months (N = 63) Child domain Parent domain Total stress

0.12 −0.14 −0.05

0.14 0.08 0.06

(−0.15, 0.40) (−0.30, 0.02) (−0.16, 0.07)

0.372 0.083 0.410

0.01 0.04 0.01

−0.11 −0.14 −0.09

0.10 0.06 0.04

(−0.32, 0.09) (−0.26, −0.03) (−0.17, −0.01)

0.275 0.017 0.035

0.02 0.09 0.07

a b c d

Parenting Stress Index subscales. Bayley Mental Development Index scores. Standard error. 95% Confidence intervals.

development research clearly indicates that parenting stress does interfere with both the formation and the quality of parent–child relationships, by altering the important parenting practices (Carey, Nicholson, & Fox, 2002; Ello & Donovan, 2005; Stelter & Halberstadt, 2011). Children lacking secure attachment are at risk for severe emotional deficits, maladjusted behaviors, and disturbed interaction skills as they grow up (Howe, 2005; Thompson, 2008). McCusker et al. (2010) demonstrated clinically and statistically significant gains in MDI scores after a program of psychosocial interventions with mothers of infants born with complex CHD. Whereas studies in other pediatric populations provided temporal evidence for parenting stress being a predictor of child behavior problems and neurodevelopmental delays (Goldberg et al., 1997; Grunau et al., 2009), such direct causal associations have yet to be demonstrated in infants with CHD. The few cross-sectional studies in the CHD pediatric population (Goldberg, Simmons, Newman, Campbell, & Fowler, 1991; DeMaso, Beardslee, Silbert, & Fyler, 1990; DeMaso et al., 1991) posited transactional effects. Indeed, it is most likely to assume a reciprocal/bidirectional model of the relationship between parenting stress and infant development (Baker, Blacher, Crnic, & Edelbrock, 2002; Neece, Green, & Baker, 2012; Zaidman-Zait et al., 2014), as delayed infants may not meet parental expectations, or provide adequate parental reinforcement, raising a parent's stress level in turn (Brosig, Whitstone, Frommelt, Frisbee, & Leuthner, 2007). The lack of associations between stress and PDI, despite the lower PDI scores in our sample, may be explained by the fact that models were adjusted for medical risk, which is a central determinant in motor development (Feldman, Eidelman, & Rotenberg, 2004). Evidence points to psychomotor impairments in medically fragile infants, including infants with complex CHD. Psychomotor impairments are currently attributed to neurological damage resulting from nutritional and

hormonal deficits, prolonged ventilation/oxygenation, and surgical procedures/medical complications (Moffatt, Longstaffe, Besant, & Dureski, 1994; Pop et al., 1999). Specifically, in children with severe CHD, factors related to illness severity, and palliative surgery, as measured by prolonged and repeated hospital admissions, had the worst psychomotor developmental outcomes. Psychomotor differences were demonstrated between infants who had undergone palliative surgery and those who had undergone corrective surgery, indicating delayed performance on all assessments in the former group of infants (Bellinger et al., 1995). Dittrich et al. (2003) suggested that the inconsistent human and physical environment encountered during prolonged and repeated hospital admissions may compromise psychomotor development. Limitations The relatively homogenic sample (93% White), which was recruited from a single institute, may limit the generalizability of the findings. The secondary nature of the study limited its sample size and power, allowing detection of a minimal R2 of 0.09 to 0.13. It seems possible that PDI and PSI could have a weaker, but still meaningful, relationship that cannot be identified because of the sample size. The power issue also limited our ability to adjust for multiplicity (e.g. Bonferroni/Holm's procedures; see Proschan & Waclawiw, 2000). Therefore, the clinical significance and interpretation of results was performed with caution. The sample included mostly mothers, as the infants' primary caregivers. Whereas prior parenting stress research has primarily included mothers (Carey, Nicholson, & Fox; Phipps & Drotar, 1990; Rimmerman & Stanger, 2001), studies that examined both parents showed differences in parenting stress patterns (Dudek-Shriber, 2004; Goldberg et al., 1990; Sarajuuri et al., 2012). Furthermore, the limited socioeconomic data and family functioning parameters prevent us from drawing

Table 3 Multivariablea regression models for MDIc and PDI on PSI at 6 and 12 months. PSI subscaleb

PDI (N = 42)

MDI (N = 41)

β

SEd

95% CIe

P

R2

β

SE

95% CIe

P

R2

6 months Child domain Parent domain Total stress

0.11 −0.06 −0.00

0.15 0.11 0.07

(−2.00, 0.41) (−0.28, 0.16) (−0.15, 0.14)

0.483 0.568 0.961

0.21 0.21 0.20

−0.04 −0.14 −0.07

0.10 0.07 0.05

(−0.25, 0.17) (−0.28, −0.00) (−0.16, 0.02)

0.692 0.044 0.136

0.31 0.40 0.35

12 months Child domain Parent domain Total stress

0.12 −0.11 −0.03

0.14 0.08 0.06

(−0.17, 0.40) (−0.27, 0.05) (−0.15, 0.08)

0.412 0.174 0.544

0.18 0.20 0.18

−0.11 −0.13 −0.08

0.10 0.06 0.04

(−0.31, 0.09) (−0.25, −0.02) (−0.16, −0.00)

0.286 0.024 0.046

0.23 0.29 0.27

a b c d e

All models are adjusted for infant length Z-scores at the measured PSI time point, gestational age, and length of hospital stay Parenting Stress Index subscales scores, each represents a separate multivariate model. Bayley Mental Development Index scores. Standard error. 95% Confidence intervals.

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further conclusions regarding other familial influences on the examined relationships. The PSI is a reliable general parenting stress measure, validated in multiple populations, including the CHD population (DeMaso et al., 1991; Goldberg et al., 1990; Uzark & Jones, 2003). Nonetheless, sources of parenting stress vary in different pediatric populations per specific illness-related factors and family needs (Golfenshtein, Srulovici, & Medoff-Cooper, 2016). Such illness-related factors are currently not included in Abidin's model. Future research should consider the use of more illness-specific parenting stress measures to provide information beyond that obtained from the general measure (Chan & Sigafoos, 2001; Streisand, Kazak, & Tercyak, 2003).

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Conclusions The current study examined the associations between infant development and parenting stress in a population of critically ill infants. The associations between parenting stress and infant development may be transactional, suggesting potential mediating effects of parental perceptions and the parent–infant relationship on infant development and maternal adjustment to the parenting process. Nursing practice should use Family Systems interventions in stressful periods. Early interventions to educate family members about the expected developmental trajectory of their infant and to reshape illness perceptions may potentially reduce parenting stress and promote illness adaptation. Funding

Implications and future directions for research, practice, and policy The current study introduces additional psychological and familial aspects of parenting infants with increased incidence of developmental delays. Surprisingly, findings indicate that the stress associated with developmental delays in this population revolves around parental characteristics, perceptions, attachment issues, and parental competence, as reflected in the Parent Domain. This is contra-intuitive to the perception of stress revolving around child-related factors in a population with cognitive delays. Zeanah, Boris, and Larrieu (1997) suggested that infant development is best appreciated within the context of caregiving relationships. Pediatric nurses may be better prepared to attend to what lower scores on the MDI may herald for a mother's level of stress with respect to the issues included in the Parent Domain of the PSI. Studies suggest moving forward from linear models of cause and effect towards multidimensional biopsychosocial models in the attempt to explain developmental psychopathology (Burgess, Marshall, Rubin, & Fox, 2003; Simmons, Goldberg, Washington, Fischer-Fay, & Maclusky, 1995; Wachs, 2009; Zeanah et al., 1997). The mediating/moderating role of parenting stress should be examined in such models as a potential key indicator of dysfunctional interactions in the parent–child relationship (Gordon & Hinshaw, 2015). With recent shifts towards the Family Systems Nursing paradigm (Anderson, 2000; Östlund & Persson, 2014; Wright & Leahey, 1990), it becomes clearer that focusing nursing research and nursing practice on one family member limits the ability to comprehensively refer to parenting stress without accounting for other familial dynamics. Knafl et al. (2013) showed fewer behavior problems in children cared for by Family-Focused caregivers than in children cared for by ConditionFocused caregivers. Brosig et al. (2007) highlighted the need for practitioners working with these children and families to ask about parental stress, family functioning, and behavioral expectations for the child in the context of routine medical/cardiac follow-up. The Illness Belief Model emphasizes the role of illness beliefs held by individuals, families, and healthcare providers in shaping the process of adjusting to illness (Bell & Wright, 2015). Parental perceptions of the illness and expectations regarding the infant's developmental trajectory may be linked to feelings of parental competency. Therefore, such factors should be considered in the contexts of both stress and development as potential key indicators of parental coping with and adjustment to illness (Gordon & Hinshaw, 2015). Studies recommended establishing a comprehensive and long-term system to care for families facing severe pediatric illness (Sung, GiDo, & Park, 2012). A recent literature review indicated that interventions that strived to change families' illness perceptions and coping mechanisms in response were most effective in reducing parenting stress (Golfenshtein, Srulovici, & Deatrick, 2016). In addition to regular stress screening in families of infants with developmental delays, interventions may also include early education and support to promote adaptive parental mechanisms for coping with illness (Jackson, Frydenberg, Liang, Higgins, & Murphy, 2015; Sira, Desai, Sullivan, & Hannon, 2014).

This work was supported by the National Institutes of Health R01 NR002093. CRediT authorship contribution statement Nadya Golfenshtein: Conceptualization, Methodology, Formal analysis, Writing - original draft, Investigation. Alexandra L. Hanlon: Conceptualization, Methodology, Supervision, Writing - review & editing. Janet A. Deatrick: Conceptualization, Supervision, Writing - review & editing. Barbara Medoff-Cooper: Conceptualization, Methodology, Supervision, Writing - review & editing, Data curation, Funding acquisition. Declaration of competing interest None. References Abidin, R. R. (1995). Parenting stress index: Professional manual (3rd ed.). Lutz, FL: Psychological Assessment Resources, Inc. Acton, B. V., Biggs, W. S., Creighton, D. E., Penner, K. A., Switzer, H. N., Thomas, J. H. P., ... Robertson, C. M. (2011). Overestimating neurodevelopment using the Bayley-III after early complex cardiac surgery. Pediatrics, 128(4), e794–e800. https://doi.org/ 10.1542/peds.2011-0331. Anderson, K. H. (2000). The family health system approach to family systems nursing. Journal of Family Nursing, 6(2), 103–119. Årestedt, L., Benzein, E., & Persson, E. (2015). Families living with chronic illness: Beliefs about illness, family, and health care. Journal of Family Nursing, 21(2), 206–231. https://doi.org/10.1177/1074840715576794. Aylward, G. P. (1997). What is infant and early childhood neuropsychology? New York: Springer, 1–12. Baker, B. L., Blacher, J., Crnic, K. A., & Edelbrock, C. (2002). Behavior problems and parenting stress in families of three-year-old children with and without developmental delays. American Journal on Mental Retardation, 107(6), 433–444 Retrieved from http:// proxy.library.upenn.edu:5397/doi/full/10.1352/0895. Ballweg, J. A., Wernovsky, G., & Gaynor, J. W. (2007). Neurodevelopmental outcomes following congenital heart surgery. Pediatric Cardiology, 28(2), 126–133. https://doi.org/ 10.1007/s00246-006-1450-9. Bayley, N. (1993). Bayley scales of infant development (2nd ed.). San Antonio, TX: The Psychological Corporation. Bell, J. M., & Wright, L. M. (2015). The illness beliefs model: Advancing practice knowledge about illness beliefs, family healing, and family interventions. Journal of Family Nursing, 21(2), 179–185. https://doi.org/10.1177/1074840715586889. Bellinger, D. C., Jonas, R. A., Rappaport, L. A., Wypij, D., Wernovsky, G., Kuban, K. C., & Walsh, A. Z. (1995). Developmental and neurologic status of children after heart surgery with hypothermic circulatory arrest or low-flow cardiopulmonary bypass. New England Journal of Medicine, 332(9), 549–555. https://doi.org/10.1056/ NEJM199503023320901. Ben-Sasson, A., Soto, T. W., Martinez-Pedraza, F., & Carter, A. S. (2013). Early sensory over responsivity in toddlers with autism spectrum disorders as a predictor of family impairment and parenting stress. Journal of Child Psychology and Psychiatry, 54(8), 846–853. https://doi.org/10.1111/jcpp.12035; 10.1111/jcpp.12035. Best, K. E., Tennant, P. W., & Rankin, J. (2017). Survival, by birth weight and gestational age, in individuals with congenital heart disease: A population-based study. Journal of the American Heart Association, 6(7), e005213. https://doi.org/10.1161/JAHA.116. 005213. Bowlby, J. (1988). A secure base: Parent-child attachment and healthy human development. London, UK: Basic Books (AZ). Brosig, C. L., Whitstone, B. N., Frommelt, M. A., Frisbee, S. J., & Leuthner, S. R. (2007). Psychological distress in parents of children with severe congenital heart disease: The

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