Parental Post Traumatic Stress and Healthcare Utilization in infants with Complex Cardiac Defects

Abstract

Objective:

To examine the associations between post-traumatic stress of parents of infants with complex congenital heart defects, and their healthcare utilization for their infants during the early months of life.

Study design:

The current study is a secondary data analysis from a randomized controlled trail, in which 216 parent-infant dyads were recruited from 3 cardiac intensive care units of large pediatric centers in Northeastern America. The current sample included 153 dyads with post-traumatic stress data at hospital discharge and at 4-months followup. Poisson regressions were used to estimate the effect of post-traumatic stress change scores on number of emergency department (ED) visits, unscheduled cardiologist visits, and unscheduled pediatrician visits outcomes.

Results:

Infants whose parents gained PTSD over the study period were at increased risk for ED visits, and unscheduled cardiologist visits. Increased symptom severity predicted more unscheduled cardiologist visits and more unscheduled pediatrician visits. Increased symprom clusters (avoidance, arousal, re-experiencing) predicted more ED visits, more unscheduled cardiologist visits, and more unscheduled pediatrician visits.

Conclusions:

Parents of infants with cardiac conditions may experience post-traumatic stress following cardiac surgery, which can be linked to greater healthcare utilization. Findings highlight the importance of screening and treating post-traumatic stress to preserve parental mental health and prevent adverse outcomes.

Congenital heart disease (CHD) is among the top causes of deaths in infancy (1). The more complex conditions (lesions with hemodynamic consequences) require life sustaining surgeries, lengthy stays in a cardiac intensive care unit (CICU), and increased caretaking demands at home during infancy (2). The parental stress experienced in this population is well known, and in recent years parental post-traumatic stress has also been acknowledged.

Research on parental post-traumatic stress symptoms (PTSS) in the CHD population reported on prevalence rates ranging from 11% to 68% (3–5). A proportion of those parents also meet the DSM diagnostic criteria for post-traumatic stress disorder (PTSD). Parental poor mental health, including PTSS, has been linked to a greater pediatric healthcare utilization, attributed to the overall increased stress parents of children with pediatric conditions experience (6–11).

Research of healthcare utilization has been performed from various perspectives in the attempt to explain factors affecting individuals’ utilization behaviors. Dominant factors include, for example, socio-demographic characteristics, familial and community resources, organizational structure, and the individual’s perceived needs of the health condition (12–14). The latter are attributed to the psychosocial perspective, which posits that individuals seek healthcare based on factors beyond the actual medical need, such as knowledge, perceived resources (eg, social support), attitudes and illness beliefs. These factors are assumed to affect the perception of symptom severity.

Adopting this psychosocial approach, and in line with previous research, we suggest that increased distress may exacerbate parental perceptions regarding their child’s symptom-severity, and “boost” healthcare seeking behaviors. Healthcare services for infants with complex CHD typically begin immediately after birth and include long post-operative hospitalization in the CICU, post-discharge cardiologist condition-related care, pediatrician routine care, and other specialty-related visits. Our hypothesis states that parental post-traumatic stress will be associated with healthcare utilization, so that infants whose parents had increased levels of PTSS/ PTSD over the study period will have more ED visits, unscheduled pediatrician visits, and unscheduled cardiologist visits, than infants whose parents’ did not have post-traumatic stress.

Methods

We reported on a secondary analysis of data from a 3-centered randomized clinical trial conducted during 2012–2017 (NCT01941667). The parent study utilized home-monitoring intervention for parents of infants with complex CHD post-operational discharge. Infants and their parents were recruited from the CICUs of 3 large, university-affiliated pediatric cardiac centers in the American Northeastern region, and randomly assigned to intervention or control groups (N=216). Intervention included daily/weekly virtual monitoring for infant status for four months, and control group received usual care (For a detailed description of the parent study, see Medoff-Cooper et al¹⁵). Eligibility criteria for infants included a complex CHD diagnosis requiring cardiac surgery during the first 21 days of life. Eligibility criteria for caregivers included age ≥18 years, and the ability to speak and read English/Spanish. Exclusion criteria included gestational age <37 weeks, birthweight <2500 grams, Risk Adjustment in Congenital Heart Surgery Category of 1 (lowest risk) or associated congenital syndromes/anomalies (except DiGeorge syndrome). Infants who were listed for heart transplant, had cardiomyopathy, or significant neurologic insult, and/or were not discharged within two-and-a-half months of life were withdrawn from the study. Out of 216 subjects enrolled in the parent study, we included in our analysis all 153 subjects who had completed the post-traumatic stress measure at both hospital discharge and at end-of-study follow-up. No significant differences in participants’ characteristics were found between the parent study’s sample and the current sub-sample.

The study was approved by the centers’ institutional review boards and written informed consent was signed by parents of enrolled infants. Parents were approached for consent after their infant had undergone cardiac surgery and postoperative endotracheal extubation. Data for the parent study were collected at discharge, on a daily/weekly basis for the intervention group, and at the four-months follow-up, at the end of study. Baseline and clinical variables were obtained from medical records pre-discharge. Parents filled in self-report questionnaires at discharge and at the end of study, including demographic information, stress and quality of life measurements, and infantile feeding behaviors. Feeding and anthropometrics were also collected for the intervention group throughout the study period.

Outcomes for the current study were self-reported by parents and included number of ED visits number of unscheduled pediatrician visits, and number of unscheduled cardiologist visits. Number of outcomes were summarized for the period of initial post-operational discharge to the end of study, and treated as continuous.

Parents completed the Posttraumatic Diagnostic Scale (PTDS) (16) within two weeks from discharge, and again at the end of the study. The PTDS is a self-report measure of PTSS and PTSD designed for individuals who were exposed to high-magnitude stressors. Forty-nine items measure the number, type and severity of symptoms, corresponding to the 17 listed in the DSM-IV criteria over five continuous subscales (Number of Symptoms, Re-experiencing, Avoidance, Hyperarousal, Total symptom Severity). Higher scores on these subscales represent worse symptoms. Another subscale aids to diagnose PTSD according to the DSM- IV diagnostic criteria (PTSD Diagnosis scale, providing a binary score). The PTDS has been validated in various clinical and research setting and demonstrates good psychometric properties and a high internal consistency for the current study (α=.92). As the healthcare utilization outcomes in the parent study have been measured as counts over four months, it was essential to capture PTDS measures over the same period (as diminished symptoms could still relate to earlier utilization behaviors). Therefore, the independent variable of interest in the current study was the PTDS subscales change scores. PTDS change scores were calculated by subtracting end-of-study scores from initial scores at time of post-operational discharge. Positive change scores represent improved symptomology/diagnosis, whereas negative change scores represent worsened measures over time.

Clinical variables were abstracted from the medical records and included primary diagnosis, surgical and post-operative procedures, infant hospitalization length, nutritive assessment, anthropometrics, and discharge medications. CHD type was classified as single (SV), or biventricular (BV) postoperative cardiac physiology (17). CHD severity was determined according to the Risk Adjustment in Congenital Heart Surgery (RACHS-1) post-operative mortality risk (18) and classified as low risk (categories 2–3) or high risk (categories 4–6). Feeding mode at discharge was classified as oral or device-assisted feeding (19). Anthropometric measures were standardized to the World Health Organization’s growth-for-age z-scores (35,36). The potential confounding effect of the parent study’s intervention was examined as well.

Sex, ethnicity, parental age, parental education, insurance type, household income, and household members, were collected from patient records and via parental reporting. Familial social support is among the utilization behavior determinants according to the socio-psychological perspective. Social support was measured in the current study via the ENRICHED Social Support Index (ESSI), a 7-item self-report questionnaire for parents. Items are summed for a total score, with higher scores indicating greater social support. The reliability and validity of the ESSI have been shown in several cardiac populations (22–24).

Descriptive statistics were used to characterize demographic and clinical measures of parents and infants. Means, standard deviations, medians, and interquartile ranges were used to describe continuous variables. Distributions of continuous variables were visually examined to determine the need for transformations or variance stabilizations. Frequencies and percentages were used to describe categorical variables. Generalized linear regression models using the Poisson distribution for count outcomes were used to estimate the effect of PTDS change scores on healthcare utilization outcomes. Separate models were generated for each outcome including number of ED visits, number of cardiologist visits, and number of pediatrician visits. We considered covariates for the current analysis based on the theoretical framework and previous literature. Potential covariates were initially included in bivariate models based on their significance level with the outcome (p≤0.2), and further examined in multivariable models via the stepwise deletion procedure (p≤0.05). Final covariates for the various multivariable models included: ED visit models were adjusted for race and parental age; unscheduled cardiologist visits models were adjusted for post-operative physiology and insurance type; and unscheduled pediatrician visits models were adjusted for race and RACHS score. All models were also adjusted for baseline PTDS at discharge. Models were compared with Negative binomial and Multinomial logistic models to evaluate optimal model fit using the Akaike information criterion. When considering zero-inflated models over standard Poisson and negative binomial models because of excessive zeros, zero-inflated models demonstrated no differences in results. Sensitivity analyses to examine extreme values and missingness mechanisms were performed, determining that missingness was unrelated to healthcare utilization nor PTDS. Statistical significance for the multivariable models was considered at the 0.05 level. Multiplicity was not accounted for, given the limited sample size, and clinical significance was interpreted based on the overall results. All analyses were performed using STATA statistical software package Version 16 (25).

Results

Table I displays the sample’s demographic and clinical measures, as well as PTDS and healthcare utilization information (n=153). Most infants participating in the study were White (80%), non-Hispanic (92%), males (55%). Fifty-five percent of infants had a single-ventricle post-operative cardiac physiology, and 41% had RACHS scores between 4–6, classified as high post-operative mortality risk. At the time of hospital discharge, 52% of infants required tube-assisted feedings. Eighty percent of infants were diagnosed with the CHD condition prenatally. The hospital length of stay ranged from 6–73 days (Median=19). Half of the infants were discharged to home with three or more medications. The vast majority of parents were females (97%), with a mean age of 30 years (SD=5.43). Most parents were collage educated (58%), had private insurance (73%), and less than one-half (46%) had a house income higher than 100,000$. With regard to stress, 9.2% of parents met PTSD diagnostic criteria by the PTDS within two weeks from initial hospital discharge, with a mean of 6.5 symptoms (SD= 0.34). The mean total PTDS symptom severity in the sample was 9 (SD=1.34). With regard to study outcomes, distributions were right-skewed (Median=0, IQR=1), with ranges of 7, 10, and 3 for ED visits, pediatrician visits, and cardiologist visits, respectively.

Table 1.

Demographic and clinical characteristic of the study sample (N=153).

Demographical characteristics (categorical)	N (%)
Parental sex (female)	147 (96.7)
Parental Education (N=131)
High School Graduate	21 (16)
Partial College	23 (17.6)
College Graduate	87 (66.4)
Infant sex (female)	69 (45.1)
Infant Race
White	123 (80.4)
Black	17 (11.1)
Asian	10 (6.5)
Mixed/other	3 (2.0)
Infant Ethnicity
Hispanic	13 (8.5)
Non-Hispanic	140 (91.5)
Insurance Type
Private	112 (73.2)
Medicaid	30 (19.6)
Other	17 (7.1)
Household Income (N= 120)
$0–24,999	20 (16.7)
$25,000–49,999	22 (18.3)
$50,000–99,999	40 (33.4)
>$100,000	38 (31.6)
Demographical characteristics (continiuos)	Mean (SD)	Median(IQR)
Parental Age	30.31 (5.43)	31 (8)
Number of Children in Household	2 (1.03)	2 (2)
Clinical parameters (categorical)	N (%)
Ventricular Physiology (Single Ventricle)	84 (55.0)
Prenatal Cardiac Diagnosis (Yes)	123 (80.4)
RACHs-1 score (N=117)
Low risk (categories 1–3)	69 (59.0)
High risk (categories 4–6)	48 (41.0)
Tube assisted Feeding at discharge	80 (52.3)
Clinical parameters (continious)	Mean (SD)	Median(IQR)
Infants’ gestation age (weeks)	38.82 (0.89)	39 (1)
Infants’ Birthweight (g)	3335 (444.0)	3315 (657)
Infants’ Birthlength (cm)	49 (2.0)	49 (3)
Number of Medications at discharge	2.82 (1.55)	3 (2)
Length of initial hospitalization (days)	23.06 (14.16)	19 (67)
Parental PSTD Scores (within 2w from discharge)
Positive PTSD diagnosis, n (%)	14 (9 .2%)
Total Symptom Severity	8.97 (1.34)	6 (10)
Number of Symptoms	6.45 (0.34)	5 (7)
Re-experiencing	3.39 (0.99)	3 (4)
Avoidance	2.96 (0.48)	2 (5)
Arousal	2.52 (0.74)	2 (4)
Total Symptom Severity Delta* n (%)	−0.38 (6.61)	0 (7)
No change	21 (13.9%)
Decrease	65 (42.2%)
Increase	67 (43.9%)
Healthcare Utilization Outcomes
Emergency Department Visits		0 (1)
Unscheduled Pediatrician Visits		0 (1)
Unscheduled Cardiologist Visits		0 (1)

Note. SD=Standard Deviation; IQR=Inter Quartile Range; RACHS-1= Risk Adjustment in Congenital Heart Surgery (Al-Radi et al., 2007); PTDS=Post-traumatic Stress Disorder Scale (Foa et el, 1997);

^*PTSD diagnosis delta represents the change in PTSD Symptom Severity over the study period, and is defined as subscale score at the end of study minus the subscale score at initial hospital discharge.

Table 2 displays the results of bivariate Poisson regression models for health care utilization in infants with CHD, regressed on parental PTDS change scores. Findings indicate that negative change scores in PTDS subscales (representing worsening symptoms/health condition over the study period) are linked to increased healthcare utilization. Specifically, acquisition of PTSD diagnosis over the study period was linked to increased risk for ED visits (RR=1.92, 95% CI=1.08–3.41, P=0.026), and unscheduled cardiologist visits (RR=2.14, 95% CI=1.70–3.05, P=0.002), compared with those without diagnosis. Having a PTSD diagnosis at both time points, was also linked to increased risk for unscheduled cardiologist visits (RR=2.33, 95% CI=1.45–2.90, P=0.009), compared with those without PTSD diagnosis. Negative Symptom Severity change score, was linked to more ED visits (RR=0.95, 95% CI=0.92–0.97, P = .001), more unscheduled cardiologist visits (RR=0.89, 95% CI=0.85–0.94, P<0.001), and more pediatrician visits (RR=0.95, 95% CI=0.92–0.97, P<0.001). Worsening symptom clusters were linked to more healthcare utilization (See detailed estimates in Table 2).

Table 2.

Unadjusted Poisson regression model results for health care utilization and change in PTDS parameters from baseline to end of study in parents of infants with CHD.

	ED visits (N=129)			Unscheduled cardiologist visits (N=129)			Unscheduled pediatrician visits (N=121)
Change in PTSD	RR	95% CI	P-value	RR	95% CI	P-value	RR	95% CI	P-value
PTSD Diagnosis (No)*	REF			REF			REF
Diminished Diagnosis	1.51	0.61–3.74	0.376	2.60	0.59–3.31	0.203	1.02	0.38–2.77	0.967
Gained Diagnosis	1.92	1.08–3.41	0.026	2.14	1.70–3.05	0.002	1.29	0.74–2.27	0.360
Diagnosis (yes)*	1.01	0.37–2.75	0.993	2.33	1.45–2.90	0.009	0.51	0.16–1.61	0.252
Total Symptom Severity	0.95	0.92–0.97	0.001	0.89	0.85–0.94	<0.001	0.95	0.92–0.97	<0.001
Number of Symptoms	0.76	0.54–1.06	0.112	0.68	0.42–1.11	0.125	0.82	0.58–1.13	0.226
Re-experiencing	0.92	0.84–0.99	0.045	0.79	0.68–0.91	0.002	0.91	0.85–0.98	0.016
Avoidance	0.86	0.79–0.92	<0.001	0.76	0.67–0.86	<0.001	0.90	0.84–0.96	0.003
Arousal	0.91	0.83–0.97	0.013	0.76	0.66–0.87	<0.001	0.87	0.81–0.93	<0.001

Note.

^*At both time points, according to the PTDS diagnostic criteria; PTDS= Posttraumatic Diagnostic Scale (Foa, et al., 1997); RR= Relative Rate [exp(β)]; 95% CI=95% Confidence Intervals.

Table 3 further displays results from the multivariable Poisson regression models for the associations between the change in parental PTDS over the study period and healthcare utilization in infants with complex CHD. Findings indicate that negative change scores in PTDS subscales (indicating worsening symptoms/condition over time) are linked to increased healthcare utilization. Specifically, acquisition of PTSD diagnosis over the study period was linked to increased risk for ED visits (RR=2.36, 95% CI=1.31–4.25, P=0.004), and unscheduled cardiologist visits (RR=2.95, 95% CI=1.06–3.22, P=0.039), compared with those without diagnosis. Having PTSD diagnosis at both time points, was also linked to increased risk for unscheduled cardiologist visits (RR=3.96, 95% CI=1.14–4.68, P=0.030), compared with those without PTSD diagnosis. Negative Symptom Severity change score was associated with more ED visits (RR=0.94, 95%CI=0.93–0.98, P=0.001), more unscheduled cardiologist visits (RR=0.90, 95%CI=0.89–0.94, P<0.001), and more unscheduled pediatrician visits (RR=0.95, 95%CI=0.92–0.97, P=0.013). Negative Symptom Cluster change scores were linked to increased healthcare utilization as follows: 1) Negative Re-experiencing change score was linked to more unscheduled cardiologist visits (RR=0.86, 95%CI=0.72–0.96, P=0.009). 2) Negative Avoidance change score was associated with more ED visits (RR=0.84, 95%CI=0.79–0.92, P<0.001), more unscheduled cardiologist visits (RR=0.81, 95%CI=0.71–0.88, P=0.004), and more unscheduled pediatrician visits (RR=0.91, 95%CI=0.85–0.99, P=0.025). 3) Negative Arousal change score was linked to more ED visits (RR=0.86, 95%CI=0.82–0.93, P=0.006), more unscheduled cardiologist visits (RR=0.78, 95%CI=0.67–0.83, P<0.001), and more unscheduled pediatrician visits (RR=0.90,95%CI=0.85–0.95, P=0.003).

Table 3.

Adjusted Poisson model results for health care utilization and change in PTDS subscales from baseline to end of study in parents of infants with CHD.

	ED visits* (N=120)			Unscheduled cardiologist visits** (N=129)			Unscheduled pediatrician visitsϮ (N=93)
Change in PTSD	RR	95% CI	P-value	RR	95% CI	P-value	RR	95% CI	P-value
PTSD Diagnosis (No)±	REF			REF			REF
Diminished Diagnosis	1.24	0.45–3.41	0.673	2.92	0.66– 3.88	0.157	1.09	0.39–2.98	0.869
Gained Diagnosis	2.36	1.31–4.25	0.004	2.95	1.06–3.22	0.039	1.53	0.86–2.71	0.145
Diagnosis (yes)±	1.04	0.37–2.75	0.990	3.96	1.14–4.68	0.030	0.45	0.11–1.81	0.261
Total Symptom Severity	0.94	0.93–0.98	0.001	0.90	0.89–0.94	<0.001	0.95	0.92–0.97	0.013
Number of Symptoms	0.71	0.55–1.02	0.077	0.72	0.53–1.23	0.427	0.76	0.56–1.08	0.135
Re-experiencing	0.94	0.86–1.01	0.078	0.86	0.72–0.96	0.009	0.97	0.89–1.03	0.281
Avoidance	0.84	0.79–0.92	<0.001	0.81	0.71–0.88	0.004	0.91	0.85–0.99	0.025
Arousal	0.86	0.82–0.93	0.006	0.78	0.67–0.83	<0.001	0.90	0.85–0.95	0.003

Note.

^±At both time points, according to the PTDS diagnostic criteria; PTDS= Posttraumatic Diagnostic Scale (Foa, et al., 1997); RR= Relative Rate [exp(β)]; 95% CI=95% Confidence Intervals;

^*=Models are adjusted for baseline PTDS, race and parental age;

^**=Models are adjusted for baseline PTDS, cardiac physiology and insurance type;

^Ϯ=Models are adjusted for baseline PTDS, race and RACHS-1 score.

Table 4 presents additional sub-sample analyses results for the associations between PTDS subscales change scores and health care utilization outcomes by cardiac physiology, and by RACHS-scores. Separate adjusted Poisson regression models for the SV and the BV groups, as well as for the Low RACHS and High RACHS groups indicate that the relationships under investigation differ between groups. In general, parents of infants with SV physiology or High RACHS demonstrate more significant relationships between the PTSD subscales of PTSD diagnosis, and the Total Severity score than parents of infants with BV physiology or Low RACHS score. Moreover, The associations in focus in the sicker infants groups (SV, High RACHS) appear to be significant for the ED and the Cardiologist visits outcomes, whereas such associations in the less complex infants groups are significant for the pediatrician visits outcome. For all detailed estimates see Table 4.

Table 4.

Adjusted Poisson model results for health care utilization and change in PTDS subscales change scores- Sub-sample analyses by cardiac physiology, and RACHS-scores.

Healthcare utilization outcomes
ED visits*
By Cardiac Physiology	Single ventricle (n=69)			Bi-ventricle (n=57)
	RR	95% CI	P	RR	95% CI	P
PTSD Diagnosis (No)±	REF			REF
Diminished Diagnosis	3.87	0.48 , 10.17	0.989	2.05	0.49 , 10.82	0.865
Gained Diagnosis	2.98	1.04 , 8.56	0.042	1.87	0.87 , 1.91	0.106
Diagnosis (yes)±	2.34	1.22 , 4.51	0.011	1.54	0.34 , 6.82	0.567
Total Symptom Severity	0.97	0.93 , 1.01	0.173	0.91	0.85 , 1.96	<0.001
	Unscheduled Cardiologist Visits**
	Single ventricle (n=69)			Bi-ventricle (n= 57)
PTSD Diagnosis (No)±	REF			REF
Diminished Diagnosis	1.71	1.29 , 12.98	0.991	18.32	2.78 , 20.45	0.002
Gained Diagnosis	3.45	0.97 , 12.28	0.055	6.10	0.58 , 63.28	0.129
Diagnosis (yes)±	3.44	1.34 , 8.80	0.010	8.06	0.32 , 9.91	0.607
Total Symptom Severity	0.89	0.84 , 0.94	<0.001	0.98	0.86 , 1.11	0.788
	Unscheduled pediatrician visitsϮ
	Single ventricle (n=48)			Bi-ventricle (n= 43)
PTSD Diagnosis (No)±	REF			REF
Diminished Diagnosis	0.39	0.05 , 2.92	0.362	3.09	0.91 , 10.48	0.069
Gained Diagnosis	0.47	0.06 , 3.49	0.463	.297	0.04 , 2.16	0.232
Diagnosis (yes)±	0.66	0.19 , 2.24	0.514	2.617	1.29 , 5.29	0.007
Total Symptom Severity	1.04	0.99 , 1.09	0.057	.862	0.81 , 0.90	<0.001
	ED visits*
By RACHS-score	RACHS High risk (n=57)			RACHS Low Risk (n=34)
PTSD Diagnosis (No)±	REF			REF
Diminished Diagnosis	5.77	0.16 , 0.93	0.935	1.42	0.22 , 3.48	0.861
Gained Diagnosis	1.68	0.39 , 7.21	0.482	1.42	0.90 , 1.10	0.974
Diagnosis (yes)±	3.95	1.34 , 8.80	0.001	1.43	0.01 , 36.84	0.837
Total Symptom Severity	0.93	0.84 , 0.94	0.003	0.97	0.93 , 1.06	0.438
	Unscheduled Cardiologist Visits**
	RACHS High risk (n=61)			RACHS Low Risk (n=36)
PTSD Diagnosis (No)±	REF			REF
Diminished Diagnosis	3.38	0.01 , 0.80	0.995	7.34	1.17 , 45.68	0.033
Gained Diagnosis	10.79	2.67 , 43.56	<0.001	1.83	0.20 , 16.09	0.584
Diagnosis (yes)±	4.48	1.48 , 13.54	0.008	1.11	0.09 , 2.61	0.411
Total Symptom Severity	.890	0.83 , 0.95	<0.001	0.98	0.86 , 1.11	0.757
	Unscheduled pediatrician visitsϮ
	RACHS High risk (n=57)			RACHS Low Risk (n=34)
PTSD Diagnosis (No)±	REF			REF
Diminished Diagnosis	0.31	0.04 , 2.31	0.257	2.59	0.79 , 8.51	0.115
Gained Diagnosis	0.47	0.06 , 3.46	0.464	0.28	0.03 , 2.11	0.222
Diagnosis (yes)±	1.43	0.73 , 2.78	0.292	1.73	0.41 , 7.25	0.451
Total Symptom Severity	0.98	0.95 , 1.02	0.439	0.91	0.86 , 0.96	0.002

Note.

^±At both time points, according to the PTDS diagnostic criteria; PTDS= Posttraumatic Diagnostic Scale (Foa, et al., 1997); RR= Relative Rate [exp(β)]; 95% CI=95% Confidence Intervals; RACHS Low risk=categories 2–3; RACHS High risk=categories 4–6;

^*Models are adjusted for baseline PTDS, race and parental age;

^**Models are adjusted for baseline PTDS and insurance type;

^ϮModels are adjusted for baseline PTDS, and race.

Discussion

Our main findings support our hypotheses by showing that parents who experienced worsening PTDS measures over the study’s period were more likely to utilize healthcare services compared with parents with no change in PTSS. Results also suggest that parents whose PTDS measures improved over time were more likely to utilize healthcare services to a lesser degree. Although potentially exposed to type I error due to multiple testing our results demonstrate a coherent trend which aligns with previous reports on pediatric populations (and other populations suffering from post traumatic stress) (26–28). For example, Marsac et al found that the more severe the traumatic stress parents experience within the first month following their child’s injury, the greater number of subsequent medical visits observed for their children. (29) Similarly, Thompson et al showed that parent PTSS severity at 7 months following their child’s diagnosis of a life threatening illness predicted higher utilization at 12 months. (6) Apparently, such relationships are not solely limited to post traumatic stress, as greater healthcare utilization has been observed with regard to various stress-experiences and psychological adversities (parenting stress, anxiety, depression) of parents whose children are ill (10,30). Studies examining physical biomarkers in their attempt to explain health seeking behaviors among individuals suffering from adversities, demonstrate increased somatic symptoms and hyperarousal of the immune system as a response to stress (31). Such clinical evidence may explain why individuals suffering from PTSS / PTSD are prone to seek more medical care for themselves, but do not fully explain such behaviors in parents who care for their sick child.

Alternativly, both the parental post-traumatic stress and the increased healthcare utilization may have been affected by a third, medical emergency or an illness related variable which is likely to occur in this population. Indeed, our sub-sample analyses suggest that illness severity may play a role in the relationship between parental PTSS/PTSD and healthcare utilization. The associations differ between illness severity groups so that parents of sicker infants (SV physiology , Higher RACHS scores) demonstrated more significant associations between PTSS/ PTSD and healthcare utilization, and this was specifically evident with regard to the ED, and Cardiologist visits. Whereas parents of less medicaly-complex infants showed fewer significant associations, and mostly with regard to pediatrician visits. Such differences may suggest that among the sicker infants, the actual existing medical needs were driving parental utilization behaviors, and these may have also served as the stressors.

Nevertheless, illness severity indicators rarely predict PTSS / PTSD among individuals and caretakers coping with an illness (4,32,33). The more recognized predictors of PTSS/PTSD include individual’s characteristics (personality traits, cognitive processes, functional status), environmental and social factors (34,35). For example, Manne et al concluded that in mothers of children undergoing bone marrow transplantation it was the cognitive interpretations of the threat for the child’s future functioning, and lack of social support that placed them at risk for post-traumatic stress symptomatology. (36) For parents of childhood cancer survivors, PTSS were predicted by the perceived threat to the child’s life and the perceived intensity of treatment rather than the objective medical ratings of treatment intensity or life threat (37).

Similarily, a decent percentage of variance in healthcare utilization studies accounts for psychosocial and cognitive variables, such as parental perceptions of their role demands, child behavior problems, and perceptions of self-efficacy to cope. These often go above and beyond the influence of child health status (38,39). This may suggest that parental perceptions of the pediatric condition, and their capabilities to manage it may be as important as the objective medical necessity when seeking healthcare. This evidence may lead to a the line of thought that the psychosocial factors (and especially the subjective experience of the pediatric illness), rather than the medical objective factors, seem to much contribute to the development of parental post traumatic stress and shape health seeking behaviors (3,35,40). Hence, these factors should be prioritized whenever screening and intervening in population at-risk.

There is strong consensus that mental health screening for parents of children with CHD and providing them psychological care are urgently needed. Among populations at risk, such as in parents of infants with CHD, screening should be conducted early before surgery, and prospectively assess personality characteristics, illness perceptions, threat appraisal, coping styles, and support networks, in order to identify parents at risk for psychosocial adversities and illness maladjustment (31). Beneficial treatments in populations suffering from post-traumatic stress following childhood illness have included antidepressant drugs and cognitive behavioral therapy (41,42).

Our sample size was limited by the secondary nature of the analysis, and as such presents several limitations. First, the change in PTSS/PTSD has been measured at the same time frame as the number of healthcare services, and therefore, no cause-effect relationship can be established by the data. Second, the fixed sample size prevented us from correcting our models for the Multiple Testing Problem (43)(e.g. Bonferroni correction), and clinical significance was interpreted based on the overall results (44). Third, the study relies on parents’ self-report of healthcare utilization, and as such lacks indicators for the appropriatness or “excessiveness” of the utilization behaviors. This is a very complex group of infants and it is likely that the healthcare utilization was indeed warrented. Further limitations are derived from the sample’s homogeneity, which included only mothers from specific SES and ethnical backgrounds. Future studies should include more diverse samples with both parents, from of a wide range of race, ethnicity and socioeconomic status.

Parents of infants with CHD may experience post-traumatic stress at the time of hospital discharge and throughout the first months following cardiac surgery, which can be linked to increased utilization of healthcare services for their infants. This relationship highlights the importance of promptly screening and treating PTSS in those parents to preserve their competency as caregivers and prevent the negative consequences of post-traumatic stress.

List of Abbreviations and Acronyms

CHD

Congenital heart disease

CICU

cardiac intensive care unit

PTSS

Post-traumatic Stress Symptoms

PTSD

Post-traumatic Stress Disorder

ED

Emergency Department

RACHS

Risk Adjustment in Congenital Heart Surgery

PTDS

Posttraumatic Diagnostic Scale

SV

Single ventricle physiology

BV

Bi-ventricular physiology

ESSI

ENRICHED Social Support Index