Journal of Pediatric Surgery 49 (2014) 700–705
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Prenatally-counseled congenital diaphragmatic hernia parents exhibit positive well-being before and after surgical repair Kate V. Dennett a, 1, Sarah Tracy a, 1, Laurie Oliver Taylor b, David Zurakowski c, d, Catherine Chen a, d,⁎ a b c d
Department of Surgery, Boston Children's Hospital, Boston, MA Advanced Fetal Care Center, Boston Children's Hospital, Boston, MA Department of Anesthesia, Boston Children's Hospital, Boston, MA Harvard Medical School, Boston, MA
a r t i c l e
i n f o
Article history: Received 1 February 2014 Accepted 13 February 2014 Key words: Congenital diaphragmatic hernia Pediatric surgery Prenatal counseling Parental well-being SF-36
a b s t r a c t Background/Purpose: The purpose of this paper was to examine the effect of prenatal counseling in the Advanced Fetal Care Center (AFCC) on the well-being of parents of infants with congenital diaphragmatic hernia (CDH). Methods: From 2008 through 2012, 26 mothers and fathers of surgically repaired CDH patients who received prenatal counseling at our institution, and 15 who received no prenatal counseling, each completed the ShortForm 36 version 1 (SF-36v1) at the appropriate time points: prenatal, two weeks, and six months postsurgery. Results: Parents in both groups did not differ by demographic characteristics. Patients who received prenatal counseling had significantly longer ventilatory time and length of stay (LOS) in the ICU and in the hospital compared to those who did not receive prenatal counseling (all P b .01). Mothers and fathers had similar SF-36v1 mental and physical component summary (MCS, PCS) post-surgery scores when compared by counseling status. Prenatal MCS scores for mothers and fathers (47 vs. 41; P = .24) were similar to those at six months post-surgery (47 vs. 47; P = .90). Conclusions: When hospital LOS was controlled between groups stratified by AFCC counseling status, MCS scores were comparable prenatally and were sustained at six months post-surgery for both parents. These findings may reflect the support services parents received beginning in the prenatal period. © 2014 Elsevier Inc. All rights reserved.
Congenital diaphragmatic hernia (CDH) is a surgical condition that can be diagnosed prenatally and has seen dramatic improvement in survival to over 80% in the past two decades [1,2]. We and others have reported significant morbidity associated with cardiac, pulmonary, gastrointestinal (GI), nutritional, and neurodevelopmental outcomes in CDH survivors [1–5], enabling CDH to be recognized as a pediatric surgical model of chronic illness in children. Much is known about the impact of pediatric disease on parental quality of life. Many studies have shown poorer health among caregivers of children with medical problems which can persist for many years and which are worsened with increasing complexity of child health problems [6,7]. These findings have been seen in families of children with conditions such as Abbreviations: AFCC, Advanced Fetal Care Center; BCH, Boston Children’s Hospital; CDH, congenital diaphragmatic hernia; CHRIs PR-Demographics, Child Health Ratings Inventory Parent Report- Demographics; ECMO, extracorporeal membrane oxygenation; GI, gastrointestinal; ICU, intensive care unit; LOS, length of stay; MCS, mental component summary; PCS, physical component summary; SF-36v1, Short-Form 36 Version 1. ⁎ Corresponding author at: Department of Surgery, Boston Children’s Hospital, 300 Longwood Avenue, Boston, MA 02115. Tel.: 1 617 355 0535; fax: +1 617 730 0298. E-mail address: [email protected] (C. Chen). 1 Co-first authors. http://dx.doi.org/10.1016/j.jpedsurg.2014.02.050 0022-3468/© 2014 Elsevier Inc. All rights reserved.
cancer [8], cardiac disease [9,10], septic shock [11], and immune thrombocytopenia [12]. A recent study focused on the quality of life of parents of children who required a gastrostomy tube and found that the gastrostomy improved the child’s physical health as well as the mental health of the child’s caregivers, especially after one year [13]. Since these illnesses are typically acquired, these studies all examined the parental impact of illness after birth and not beginning in the prenatal period. Many studies have also documented the gender difference in caregiver burden related to chronic illness and physical disabilities in children. Mothers demonstrated increased psychological distress and personal strain in many groups of children, including those born prematurely [14–17], those with cerebral palsy [18] or a physical disability [19], those with congenital heart disease [20–22], and those with a congenital anomaly [23] including anorectal malformations or CDH [24]. Because mothers are usually identified as the primary medical caregiver, fathers have been underrepresented in research on pediatric patients and parenting [25]. We have previously investigated the impact on family of CDH survivors at a median of 8 years after surgery and found that family impact is profound and long-standing, particularly for those survivors with more severe conditions and current clinical problems [26]. Our
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
current study addresses an area not previously studied: the parental well-being of mothers and fathers, individually, when faced with the diagnosis and surgical management of CDH beginning in the prenatal period, if applicable, and continuing until age 6 months post-surgery. 1. Methods 1.1. Study population We prospectively identified all patients (n = 58) diagnosed with a Bochdalek CDH who survived birth and were surgically repaired at Boston Children’s Hospital (BCH) from November 2008 through December 2012 (Fig. 1). Forty-one families consented to our institutional review board-approved study (X08-11-0538) and were enrolled. Twenty-six families were diagnosed prenatally and received counseling in our Advanced Fetal Care Center (AFCC). None of these families was enrolled at the initial AFCC consultation, but were enrolled at either their second, third, or fourth prenatal AFCC visit. Any family contemplating and eventually deciding on termination was not approached for the study. Fifteen families were not counseled in the AFCC prenatally and were admitted to BCH postnatally. These families were enrolled within 2 weeks of the time of CDH repair. 1.2. Demographic and clinical data collection One parent from each family completed the Child Health Ratings Inventory Parent Report- Demographics (CHRIs PR-Demographics) [27] during the prenatal period for those seen in the AFCC or at 2 weeks post-surgery for those not seen in the AFCC. This 41-item measure addresses the child’s background, family health care needs, and family characteristics, including employment status and health care insurance. We removed 15 questions from the prenatally acquired questionnaire and 5 questions from the postnatally acquired questionnaire that were not applicable at that time point depending on the timing of the counseling. For example, child’s date of birth was removed from the prenatal questionnaire and child’s school grade was removed from the postnatal questionnaire. We added a 5-item addendum to the CHRIs PR-Demographics, which addressed dates of birth of mother and father, any parental impairments, and the primary language spoken at home. Clinical data were collected for hospital encounters from the time of prenatal counseling (when applicable) to six months post-surgery by prospective review of hospital medical records. Patients with a genetic abnormality were diagnosed by a formal genetics consulta-
701
tion. Patients with a cardiac anomaly were defined as previously described by the CDH Study Group [28]. CDH defect size was determined according to previously defined criteria established by the CDH Study Group [29] with the following clarifications: no agenesis with primary repair (Type A), no agenesis with patch repair and defect size ≤4 cm by 4 cm (Type B), partial agenesis with patch repair and defect size N4 cm by 4 cm (Type C), complete agenesis with absent rim of anterior and posterior diaphragm requiring suturing of the patch to ribs anteriorly and posteriorly (Type D). Pulmonary, GI, and cardiac issues at discharge were defined as previously described [5]. Patients with neurologic issues at discharge were defined as having at least one of the following: a known seizure disorder, a ventriculoperitoneal shunt or hydrocephalus, any abnormal brain or spinal cord imaging study, or a need for antiepileptic medication. 1.3. Short-Form 36 Version 1 (SF-36v1) data collection For those patients who received prenatal counseling in the AFCC, mothers and fathers each completed the Short-Form 36 version 1 (SF36v1) questionnaire at three time points: prenatal, two weeks postsurgery, and six months post-surgery. Those families who did not receive prenatal counseling in the AFCC completed the SF-36v1 at the latter two time points. The SF-36v1 is a self-administered generic questionnaire that measures physical and mental health using 36 multiple-choice items [30,31]. Using a certified scoring software obtained from QualityMetric Incorporated (Lincoln, RI), raw scores were linearly transformed to ranges of 0 to 100, and eight health domain scores were obtained. In addition, two summary measure scales (physical component summary (PCS), mental component summary (MCS)) were generated, with higher scores indicating better functioning and well-being. 1.4. Statistical analysis All categorical variables were compared using Fisher’s exact test or chi-square. Continuous variables describing clinical characteristics and SF-36v1 scores that conformed to a normal distribution were expressed as mean ± SD with groups compared using the twosample Student t-test. Other continuous data including duration of ventilatory and extracorporeal membrane oxygenation (ECMO) support as well as intensive care unit (ICU) and hospital length of stay (LOS) were compared using the nonparametric Mann–Whitney U test and summarized by medians and interquartile ranges. Statistical
CDH patients surgically repaired at BCH from November 2008 through December 2012 n = 58
Enrolled n = 41
Counseled in the AFCC n = 26
Not counseled in the AFCC n = 15
Excluded n = 17
Declined to participate n = 10
Not approached due to parental social/medical reasons n=3
Postnatal death without study data n=4
Fig. 1. Schematic diagram of study cohort as a subset of the 58 congenital diaphragmatic hernia (CDH) patients surgically repaired at Boston Children’s Hospital (BCH) from November 2008 through December 2012.
702
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
analysis was performed using the SPSS software package (version 19.0, SPSS Inc./IBM, Chicago, IL). Two-tailed values of P b .05 were considered statistically significant. 2. Results 2.1. Characteristics of study population From November 2008 through December 2012, 58 patients with Bochdalek CDH were surgically repaired at BCH. Forty-one families enrolled in this study and 17 families were excluded for the following reasons: 10 declined to participate, 3 were not approached due to parental social/medical reasons, and 4 had infants who died shortly after birth without study data (Fig. 1). Of the 17 excluded families, 10 were previously counseled in the AFCC and 7 were not. The excluded families did not differ from the enrolled families when compared by insurance type and several patient characteristics including sex, birth weight, presence of cardiac anomaly, and CDH defect size. Three of the 41 enrolled patients did not survive to hospital discharge.
Patients stratified by prenatal counseling did not differ by any of the clinical characteristics at birth described in Table 1, including sex, gestational age, birth weight, presence of genetic abnormality or cardiac anomaly, Apgar scores at 1 and 5 minutes, need for ECMO, or
Table 1 Clinical characteristics and hospital course of CDH patients categorized by AFCC prenatal counseling status.
Clinical characteristics Male sex Gestational age, weeks Birth weight, kg Congenital malformation Genetic abnormality Cardiac anomaly Apgar score at 1 min Apgar score at 5 min Defect size A B C D Hospital course Ventilator, days ECMO Days ICU LOS, days Hospital LOS, days Issues at discharge Pulmonary GI Cardiac Neurologic
2.3. Demographic and family characteristics Table 2 describes the demographic and family data of study participants. Data with percentages are calculated based on total number of responses per group. Eleven mothers who stated their primary daily activity as “Other” specified the following activities:
Table 2 Demographic and family data of study participants.
2.2. Characteristics of study participants
Variable
diaphragmatic defect size. Patients who received prenatal counseling in the AFCC had significantly longer days on the ventilator and longer ICU and hospital LOS when compared to those who did not receive AFCC counseling. There were no differences in frequency of medical issues (pulmonary, GI, cardiac, neurologic) at discharge when comparing patients in both groups (Table 1). Of the patients in the group who did not receive prenatal counseling in the AFCC, none of the families were known to have received prenatal counseling elsewhere, despite one of the patients in this group having a prenatal diagnosis of CDH.
Received AFCC counseling (n = 26)
Did not receive AFCC counseling (n = 15)
P
18 (69%) 37 ± 2
12 (80%) 38 ± 4
.72 .32
2.7 ± 0.5 9 (35%)
3.1 ± 0.9 1 (7%)
.10 .06
5 (19%)
0 (0%)
.14
5 (19%)
1 (7%)
.39
5±2
5±3
.33
7±2
7±2
.66 .13
9 (35%) 3 (12%) 9 (35%) 5 (19%)
8 (53%) 4 (27%) 3 (20%) 0 (0%)
24 (8–37) 7 (27%) 21 (10–29) 34 (13–54) 57 (33–107)
6 (2–20) 3 (20%) 10 (4–10) 8 (7–27) 24 (12–55)
b.01⁎ .72 .12 b.01⁎ b.01⁎
17 (74%) 22 (96%) 10 (44%) 10 (44%)
9 (60%) 14 (93%) 4 (27%) 3 (20%)
.48 1.00 .33 .18
Plus-minus data are mean ± SD with groups compared using the two-sample Student t-test. Percentages were compared by Fisher’s exact test or chi-square for defect size and issues at discharge. Days are expressed in terms of the median and interquartile range with groups compared using the Mann–Whitney U test. ⁎ Statistically significant.
Variable
Received AFCC Did not receive AFCC P counseling (n = 26) counseling (n = 15)
Mother age, years Father age, years Race White African American Hispanic/Latino Asian Other Insurance type Private Public Private and public Household Income Less than $19,000 $20,000–$39,999 $40,000–$59,999 $60,000–$79,999 $80,000 and over Marital status Married or living with partner Divorced, separated, or widowed Never married Engaged School grade of parent High School College Graduate/Professional School Language spoken at home English Non-English Siblings† People living in household† Mother’s primary daily activity Working full-time Working part-time Full-time homemaking Other Father’s primary daily activity Working full-time Working part-time Full-time homemaking Other Family members cut down on work hours for patient care Family members stopped working due to patient’s health conditions
31 ± 5 32 ± 6
31 ± 7 33 ± 7
18 (69%) 1 (4%) 2 (8%) 1 (4%) 4 (15%)
10 (67%) 0 (0%) 2 (13%) 1 (7%) 2 (13%)
19 (73%) 4 (15%) 3 (12%)
6 (50%) 4 (33%) 2 (17%)
3 (12%) 4 (16%) 4 (16%) 2 (8%) 12 (48%)
2 (15%) 2 (15%) 4 (31%) 0 (0%) 5 (39%)
20 (77%) 1 (4%)
11 (73%) 1 (7%)
5 (19%) 0 (0%)
2 (13%) 1 (7%)
4 (15%) 14 (54%) 8 (31%)
6 (43%) 3 (21%) 5 (36%)
21 (81%) 5 (19%) 1 (0–5) 4 (2–7)
10 (71%) 4 (29%) 1 (0–3) 4 (0–6)
9 (35%) 2 (8%) 8 (31%) 7 (27%)
4 (29%) 1 (7%) 5 (36%) 4 (29%)
19 (76%) 1 (4%) 1 (4%) 4 (16%) 17 (74%)
11 (79%) 2 (14%) 1 (7%) 0 (0%) 4 (29%)
.02⁎
10 (46%)
3 (23%)
.28
⁎ Statistically significant. † Median (full range).
.75 .83 .90
.35
.70
.55
.08
.69
.77 .59 .98
.83
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
maternity leave (n = 6), frequent visits to the hospital (n = 2), and one each of bed rest, student, and resignation of full-time job. Five fathers who stated their primary daily activity as “Other” specified the following activities: one each of unemployed, college student, disabled, and on leave from full-time job. For those families who received prenatal counseling in the AFCC, a significantly higher percentage of family members cut down on work hours for patient care (74% vs. 29%, P = .02). 2.4. Short-Form 36 Version 1 (SF-36v1) scores Tables 3 and 4 show SF-36v1 domain and component summary scores at each time point for patients prenatally counseled in the AFCC (Table 3) and for those who were not (Table 4). Eleven of the 26 parental pairs who were counseled in the AFCC were able to complete the SF-36v1 at the prenatal time point. We first examined the effect of prenatal AFCC counseling on SF-36v1 PCS and MCS scores stratified by sex. We found that at two weeks and six months post-surgery, there were no significant differences in PCS and MCS scores when comparing mothers and fathers, respectively, in each group (data not shown). These results are the same even after adjusting for differences in hospital LOS between the AFCC prenatally counseled group and the non-counseled group using ANOVA (data not shown). We next examined the SF-36v1 scores over time for parents who received prenatal counseling in the AFCC stratified by sex (Table 3). Mean PCS scores for mothers were significantly lower than those for fathers at both the prenatal (40 vs. 60, P b .01) and 2 weeks (48 vs. 58, P b .01) post-surgery time points, but were not different by 6 months post-surgery (56 vs. 54, P = .40). Although there were no significant differences in MCS scores at each of the time points between mothers and fathers, mothers appeared to have higher MCS scores prenatally (47 vs. 41, P = .24), and these scores were sustained at 6 months post-surgery for both parents (47 vs. 47, P = .90). 3. Discussion This is the first known study to examine the effect of prenatal counseling on parental well-being of CDH families. During our study time period, we identified two groups of CDH families that received all of their surgical care at our institution: one group was diagnosed prenatally and received counseling in our AFCC, and one group was transferred urgently to our institution for care in the postnatal period from an outside facility. All of the latter families were not known to have received any formal prenatal counseling, although one patient was diagnosed prenatally with CDH. We did note that the group who received AFCC counseling required more intensive care with longer
703
Table 4 SF-36v1 scores for parents who did not receive prenatal AFCC counseling. Domain
2 weeks post-surgery
PF RP BP GH VT SF RE MH PCS MCS
6 months post-surgery
Mother
Father
P
Mother
Father
P
89 59 54 77 45 67 69 63 50 42
98 90 92 83 64 83 75 75 59 47
.09 .05⁎ b.01⁎
87 90 75 71 54 94 94 76 52 52
97 93 94 75 55 86 97 79 57 50
.24 .74 .03⁎
± ± ± ± ± ± ± ± ± ±
19 48 30 20 26 31 42 16 8 12
± ± ± ± ± ± ± ± ± ±
5 25 15 18 21 22 38 17 4 12
.47 .05⁎ .14 .71 .08 b.01⁎ .36
± ± ± ± ± ± ± ± ± ±
23 23 24 26 18 11 19 14 10 9
± ± ± ± ± ± ± ± ± ±
9 17 10 13 18 30 11 9 4 9
.67 .90 .47 .75 .63 .14 .63
Data are means ± SD, scores range from 0–100. PF, physical functioning; RP, role limitations due to physical health; BP, bodily pain; GH, general health perceptions; VT, vitality; SF, social functioning; RE, role limitations due to emotional problems; MH, mental health; PCS, physical component summary; MCS, mental component summary. ⁎ Statistically significant.
duration of ventilation and longer LOS in the ICU and in the hospital. When we controlled for hospital LOS between the two groups, we were surprised that SF-36v1 mental and physical component summary scores were not different between mothers and fathers at 2 weeks and at 6 months post-surgery. In other words, despite the need for more intensive and longer supportive care in the hospital, parents who received prenatal AFCC counseling had comparable scores to those who did not. This could represent the beneficial effect of prenatal counseling on preparing families before birth for the difficult course ahead, or the detrimental effect of not having the opportunity to learn about CDH prenatally for the group not receiving AFCC counseling. This is also the first known study to examine parental well-being of CDH families beginning in the prenatal period. We know of no other studies of any pediatric condition where parental well-being of mothers and fathers was examined individually during the prenatal period. Not surprisingly, fathers had significantly better physical component summary scores than mothers at the prenatal and 2 week post-surgery time points in both groups. This difference in PCS scores was not noted at 6 months post-surgery in either group. When examining the scores for the AFCC counseled group, one interesting finding, though not significant, was the higher MCS scores in mothers compared to fathers at the prenatal time point. This was unexpected given the particular stressors that mothers carrying a fetus with CDH face and one wonders whether this may reflect a beneficial effect of prenatal counseling, particularly for mothers, during the prenatal period. We did not see a significant difference between MCS scores at
Table 3 SF-36v1 scores for parents who received prenatal AFCC counseling. Domain
PF RP BP GH VT SF RE MH PCS MCS
Prenatal
2 weeks post-surgery
6 months post-surgery
Mother
Father
P
Mother
Father
P
Mother
Father
P
52 30 45 81 42 64 67 66 40 47
93 98 86 82 59 77 61 66 60 41
b.01⁎ b.01⁎ b.01⁎
76 50 51 80 40 56 59 60 48 40
93 82 83 79 49 67 67 61 58 39
.01⁎ b.01⁎ b.01⁎
93 92 82 74 52 81 83 74 56 47
90 92 76 73 56 83 77 75 54 47
.74 .99 .49 .85 .58 .77 .65 .88 .40 .90
± ± ± ± ± ± ± ± ± ±
32 38 18 14 15 31 42 19 9 12
± ± ± ± ± ± ± ± ± ±
14 8 16 17 15 18 39 13 7 9
.83 .02⁎ .22 .73 1.00 b.01⁎ .24
± ± ± ± ± ± ± ± ± ±
28 46 31 17 22 32 40 22 12 14
± ± ± ± ± ± ± ± ± ±
15 31 19 16 20 30 40 22 8 15
.85 .16 .21 .50 .87 b.01⁎ .93
± ± ± ± ± ± ± ± ± ±
11 25 22 22 23 18 32 15 6 8
± ± ± ± ± ± ± ± ± ±
25 15 27 17 17 19 38 17 7 10
Data are means ± SD, scores range from 0–100. PF, physical functioning; RP, role limitations due to physical health; BP, bodily pain; GH, general health perceptions; VT, vitality; SF, social functioning; RE, role limitations due to emotional problems; MH, mental health; PCS, physical component summary; MCS, mental component summary. ⁎ Statistically significant.
704
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
2 weeks and 6 months post-surgery between mothers and fathers in the prenatally counseled group, although scores at 2 weeks for both parents were lower than those at 6 months post-surgery. This may reflect greater emotional strain for parents at a critically ill phase of their infant’s post-surgical life when they have to cope with their prenatal decision to continue with care. Our findings differ from those of Bevilacqua et al. who found that stress and depression levels were significantly higher in mothers than in fathers of children diagnosed either prenatally or postnatally with congenital heart disease when examined 2 weeks after hospitalization [32]. Caniano & Baylis have detailed the goals of prenatal consultation by pediatric surgeons caring for parents of a fetus with a congenital anomaly [33]. The authors describe three specific time periods, beginning with the initial prenatal consultation, which focuses on possible termination of the pregnancy, followed by two additional periods which focus on the location, timing, and mode of delivery, and the decisions that are required after birth surrounding operative intervention. At our institution, CDH families, in addition to receiving medical and surgical counseling by an interdisciplinary team of pediatric surgeons, radiologists, geneticists, subspecialists, and nurses, are provided with social work support during the AFCC visits (by coauthor LOT). Before 24 weeks gestation, CDH families are counseled on the risk category of their child’s CDH and pregnancy options. After 24 weeks gestation, AFCC counseling is focused on a reassessment of the percentage predicted lung volume and expected course of care. This level of support is continued in the post-delivery period while patients are in the ICU, including assistance with accommodations and orientation to the ICU environment. After discharge from the hospital, CDH patients are followed in our multidisciplinary CDH Clinic and currently do not receive any formal social work support, but the AFCC nurses provide continuity for social work issues. Interestingly, we found that a higher percentage of families who received prenatal AFCC counseling had family members who cut down on work hours for patient care (Table 2). This may reflect the ability of prenatally counseled families to plan in advance to reduce work hours, which may not be readily done by those families who learn about the diagnosis unexpectedly after birth. We were encouraged to see that MCS scores for mothers and fathers who received prenatal counseling were sustained 6 months post-surgery. Although our patients do not have formal Social Work referrals during the outpatient period, ongoing support is a high priority initiative. During this time interval, a social worker on the CDH care team could screen for residual post-traumatic stress or signs of parental depression, and aid with parental psychological mastery of the CDH experience by allowing parents to tell the story of their journey. Families of infants with ongoing medical issues often feel that the journey is prolonged and that they are unable to restore normalcy. A study by Hatzmann et al. in a cohort of parents of chronically ill children found that parental quality of life at greater than one year after diagnosis was positively influenced by factors including caredependency, emotional support, and increased number of days on holiday [34]. There are several limitations to our study. First, we do not have a comparison group for the prenatally counseled parents at the prenatal time point for obvious logistical reasons and therefore our explanations for the results at the prenatal time point are descriptive only. In addition, not all the prenatally counseled parents were able to provide us with data at the prenatal time point, potentially making the prenatal data not representative of the entire cohort. Second, each of our time points for data collection was broad in that particular clinical scenarios within each time period could have been associated with significant fluctuations in parental well-being which we are not able to capture. For instance, we were not able to obtain data at the first prenatal consultation when the diagnosis was explained to the family and this could have represented a time of higher stress compared to later in the prenatal period. Similarly, the time period after birth but
before surgery may also have been more stressful for parents than one week after surgery. Third, our study had a limited sample size and we did not note any significant differences in clinical characteristics between our two groups based on prenatal counseling status. However, with a larger sample size, some statistically significant differences may arise between patients in both groups, such as larger defect sizes or higher rates of congenital or cardiac malformations in the group receiving prenatal counseling. This may help to explain why we noted a significant difference in number of days on the ventilator, and ICU and hospital LOS, where these measures were longer in the group that received prenatal counseling. Finally, continuing our assessments longitudinally over several years could provide insight into issues that may be more relevant for families as the children pass into toddlerhood. Our previous work provides evidence that the impact of CDH on patients and families is profound and long-standing years later [26]. Future work should continue this data collection in a longitudinal fashion to help guide appropriate services long-term. A compelling initiative similar to an early intervention program to reduce parenting stress after preterm birth described by Kaaresen et al. [15] could be created for our CDH families, most of whom have children with ongoing medical conditions. In addition, differences between mothers and fathers can be examined further by conducting focus groups aimed at understanding specific stressors for each parent during the prenatal period. Finally, understanding factors that are associated with increased marital stress and identifying ways for parents to cope with these stressors would be vital to the integrity of the family unit. Acknowledgments This study was supported by the Boston Children's Hospital Surgical Foundation. The authors would like to thank Donna Morash, RN, Robin Scott, BA and the Boston Children’s Hospital Advanced Fetal Care Center for help with patient recruitment and scheduling. References [1] Danzer E, Hedrick HL. Neurodevelopmental and neurofunctional outcomes in children with congenital diaphragmatic hernia. Early Hum Dev 2011;87:625–32. [2] Chiu PP, Ijsselstijn H. Morbidity and long-term follow-up in CDH patients. Eur J Pediatr Surg 2012;22:384–92. [3] Muratore CS, Kharasch V, Lund DP, et al. Pulmonary morbidity in 100 survivors of congenital diaphragmatic hernia monitored in a multidisciplinary clinic. J Pediatr Surg 2001;36:133–40. [4] Muratore CS, Utter S, Jaksic T, et al. Nutritional morbidity in survivors of congenital diaphragmatic hernia. J Pediatr Surg 2001;36:1171–6. [5] Friedman S, Chen C, Chapman JS, et al. Neurodevelopmental outcomes of congenital diaphragmatic hernia survivors followed in a multidisciplinary clinic at ages 1 and 3. J Pediatr Surg 2008;43:1035–43. [6] Hatzmann J, Heymans HS, Ferrer-i-Carbonell A, et al. Hidden consequences of success in pediatrics: parental health-related quality of life – results from the Care Project. Pediatrics 2008;122:e1030–8. [7] Brehaut JC, Garner RE, Miller AR, et al. Changes over time in the health of caregivers of children with health problems: growth-curve findings from a 10-year Canadian population-based study. Am J Public Health 2011;101:2308–16. [8] Klassen AF, Klaassen R, Dix D, et al. Impact of caring for a child with cancer on parents’ health-related quality of life. J Clin Oncol 2008;26:5884–9. [9] Goldbeck L, Melches J. The impact of the severity of disease and social disadvantage on quality of life in families with congenital cardiac disease. Cardiol Young 2006;16:67–75. [10] Arafa MA, Zaher SR, El-Dowaty AA, et al. Quality of life among parents of children with heart disease. Health Qual Life Outcomes 2008;6:91. [11] Buysse CM, Raat H, Hazelzet JA, et al. Surviving meningococcal septic shock: health consequences and quality of life in children and their parents up to 2 years after pediatric intensive care unit discharge. Crit Care Med 2008;36:596–602. [12] Zilber R, Bortz AP, Yacobovich J, et al. Analysis of health-related quality of life in children with immune thrombocytopenia and their parents using the kids' ITP tools. J Pediatr Hematol Oncol 2012;34:2–5. [13] Pemberton J, Frankfurter C, Bailey K, et al. Gastrostomy matters – the impact of pediatric surgery on caregiver quality of life. J Pediatr Surg 2013;48:963–70. [14] Miles MS, Funk SG, Kasper MA. The stress response of mothers and fathers of preterm infants. Res Nurs Health 1992;15:261–9. [15] Kaaresen PI, Rønning JA, Ulvund SE, et al. A randomized, controlled trial of the effectiveness of an early-intervention program in reducing parenting stress after preterm birth. Pediatrics 2006;118:e9-19.
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705 [16] Correia LL, Linhares MB. Maternal anxiety in the pre- and postnatal period: a literature review. Rev Lat Am Enfermagem 2007;15:677–83. [17] Singer LT, Fulton S, Kirchner HL, et al. Parenting very low birth weight children at school age: maternal stress and coping. J Pediatr 2007;151:463–9. [18] Rentinck IC, Ketelaar M, Jongmans MJ, et al. Parents of children with cerebral palsy: a review of factors related to the process of adaptation. Child Care Health Dev 2007;33:161–9. [19] Tong HC, Kandala G, Haig AJ, et al. Physical functioning in female caregivers of children with physical disabilities compared with female caregivers of children with a chronic medical condition. Arch Pediatr Adolesc Med 2002;156:1138–42. [20] Goldberg S, Morris P, Simmons RJ, et al. Chronic illness in infancy and parenting stress: a comparison of three groups of parents. J Pediatr Psychol 1990;15:347–58. [21] Lawoko S, Soares JJ. Quality of life among parents of children with congenital heart disease, parents of children with other diseases and parents of healthy children. Qual Life Res 2003;12:655–66. [22] Helfricht S, Latal B, Fischer JE, et al. Surgery-related posttraumatic stress disorder in parents of children undergoing cardiopulmonary bypass surgery: a prospective cohort study. Pediatr Crit Care Med 2008;9:217–23. [23] Hunfeld JA, Tempels A, Passchier J, et al. Brief report: parental burden and grief one year after the birth of a child with a congenital anomaly. J Pediatr Psychol 1999;24:515–20. [24] Poley MJ, Brouwer WB, van Exel NJ, et al. Assessing health-related quality-of-life changes in informal caregivers: an evaluation in parents of children with major congenital anomalies. Qual Life Res 2012;21:849–61. [25] Bonner MJ, Hardy KK, Willard VW, et al. Brief Report: psychosocial functioning of fathers as primary caregivers of pediatric oncology patients. J Pediatr Psychol 2007;32:851–6.
705
[26] Chen C, Jeruss S, Terrin N, et al. Impact on family of survivors of congenital diaphragmatic hernia repair: a pilot study. J Pediatr Surg 2007;42: 1845–52. [27] Parsons SK, Shih MC, Mayer DK, et al. Preliminary psychometric evaluation of the Child Health Ratings Inventory (CHRIs) and Disease-Specific Impairment Inventory-Hematopoietic Stem Cell Transplantation (DSII-HSCT) in parents and children. Qual Life Res 2005;14:1613–25. [28] Tsao K, Lally PA, Lally KP, et al. Minimally invasive repair of congenital diaphragmatic hernia. J Pediatr Surg 2011;46:1158–64. [29] Congenital Diaphragmatic Hernia Study Group, Lally KP, Lally PA, et al. Defect size determines survival in infants with congenital diaphragmatic hernia. Pediatrics 2007;120:e651–7. [30] Ware Jr JE, Sherbourne CD. The MOS 36-item short-form health survey (SF-36). I. Conceptual framework and item selection. Med Care 1992;30:473–83. [31] McHorney CA, Ware Jr JE, Raczek AE. The MOS 36-Item Short-Form Health Survey (SF-36): II. Psychometric and clinical tests of validity in measuring physical and mental health constructs. Med Care 1993;31:247–63. [32] Bevilacqua F, Palatta S, Mirante N, et al. Birth of a child with congenital heart disease: emotional reactions of mothers and fathers according to time of diagnosis. J Matern Fetal Neonatal Med 2013;26:1249–53. [33] Caniano DA, Baylis F. Ethical considerations in prenatal surgical consultation. Pediatr Surg Int 1999;15:303–9. [34] Hatzmann J, Maurice-Stam H, Heymans HS, et al. A predictive model of Health Related Quality of life of parents of chronically ill children: the importance of caredependency of their child and their support system. Health Qual Life Outcomes 2009;7:72.
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Prenatally-counseled congenital diaphragmatic hernia parents exhibit positive well-being before and after surgical repair Kate V. Dennett a, 1, Sarah Tracy a, 1, Laurie Oliver Taylor b, David Zurakowski c, d, Catherine Chen a, d,⁎ a b c d
Department of Surgery, Boston Children's Hospital, Boston, MA Advanced Fetal Care Center, Boston Children's Hospital, Boston, MA Department of Anesthesia, Boston Children's Hospital, Boston, MA Harvard Medical School, Boston, MA
a r t i c l e
i n f o
Article history: Received 1 February 2014 Accepted 13 February 2014 Key words: Congenital diaphragmatic hernia Pediatric surgery Prenatal counseling Parental well-being SF-36
a b s t r a c t Background/Purpose: The purpose of this paper was to examine the effect of prenatal counseling in the Advanced Fetal Care Center (AFCC) on the well-being of parents of infants with congenital diaphragmatic hernia (CDH). Methods: From 2008 through 2012, 26 mothers and fathers of surgically repaired CDH patients who received prenatal counseling at our institution, and 15 who received no prenatal counseling, each completed the ShortForm 36 version 1 (SF-36v1) at the appropriate time points: prenatal, two weeks, and six months postsurgery. Results: Parents in both groups did not differ by demographic characteristics. Patients who received prenatal counseling had significantly longer ventilatory time and length of stay (LOS) in the ICU and in the hospital compared to those who did not receive prenatal counseling (all P b .01). Mothers and fathers had similar SF-36v1 mental and physical component summary (MCS, PCS) post-surgery scores when compared by counseling status. Prenatal MCS scores for mothers and fathers (47 vs. 41; P = .24) were similar to those at six months post-surgery (47 vs. 47; P = .90). Conclusions: When hospital LOS was controlled between groups stratified by AFCC counseling status, MCS scores were comparable prenatally and were sustained at six months post-surgery for both parents. These findings may reflect the support services parents received beginning in the prenatal period. © 2014 Elsevier Inc. All rights reserved.
Congenital diaphragmatic hernia (CDH) is a surgical condition that can be diagnosed prenatally and has seen dramatic improvement in survival to over 80% in the past two decades [1,2]. We and others have reported significant morbidity associated with cardiac, pulmonary, gastrointestinal (GI), nutritional, and neurodevelopmental outcomes in CDH survivors [1–5], enabling CDH to be recognized as a pediatric surgical model of chronic illness in children. Much is known about the impact of pediatric disease on parental quality of life. Many studies have shown poorer health among caregivers of children with medical problems which can persist for many years and which are worsened with increasing complexity of child health problems [6,7]. These findings have been seen in families of children with conditions such as Abbreviations: AFCC, Advanced Fetal Care Center; BCH, Boston Children’s Hospital; CDH, congenital diaphragmatic hernia; CHRIs PR-Demographics, Child Health Ratings Inventory Parent Report- Demographics; ECMO, extracorporeal membrane oxygenation; GI, gastrointestinal; ICU, intensive care unit; LOS, length of stay; MCS, mental component summary; PCS, physical component summary; SF-36v1, Short-Form 36 Version 1. ⁎ Corresponding author at: Department of Surgery, Boston Children’s Hospital, 300 Longwood Avenue, Boston, MA 02115. Tel.: 1 617 355 0535; fax: +1 617 730 0298. E-mail address: [email protected] (C. Chen). 1 Co-first authors. http://dx.doi.org/10.1016/j.jpedsurg.2014.02.050 0022-3468/© 2014 Elsevier Inc. All rights reserved.
cancer [8], cardiac disease [9,10], septic shock [11], and immune thrombocytopenia [12]. A recent study focused on the quality of life of parents of children who required a gastrostomy tube and found that the gastrostomy improved the child’s physical health as well as the mental health of the child’s caregivers, especially after one year [13]. Since these illnesses are typically acquired, these studies all examined the parental impact of illness after birth and not beginning in the prenatal period. Many studies have also documented the gender difference in caregiver burden related to chronic illness and physical disabilities in children. Mothers demonstrated increased psychological distress and personal strain in many groups of children, including those born prematurely [14–17], those with cerebral palsy [18] or a physical disability [19], those with congenital heart disease [20–22], and those with a congenital anomaly [23] including anorectal malformations or CDH [24]. Because mothers are usually identified as the primary medical caregiver, fathers have been underrepresented in research on pediatric patients and parenting [25]. We have previously investigated the impact on family of CDH survivors at a median of 8 years after surgery and found that family impact is profound and long-standing, particularly for those survivors with more severe conditions and current clinical problems [26]. Our
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
current study addresses an area not previously studied: the parental well-being of mothers and fathers, individually, when faced with the diagnosis and surgical management of CDH beginning in the prenatal period, if applicable, and continuing until age 6 months post-surgery. 1. Methods 1.1. Study population We prospectively identified all patients (n = 58) diagnosed with a Bochdalek CDH who survived birth and were surgically repaired at Boston Children’s Hospital (BCH) from November 2008 through December 2012 (Fig. 1). Forty-one families consented to our institutional review board-approved study (X08-11-0538) and were enrolled. Twenty-six families were diagnosed prenatally and received counseling in our Advanced Fetal Care Center (AFCC). None of these families was enrolled at the initial AFCC consultation, but were enrolled at either their second, third, or fourth prenatal AFCC visit. Any family contemplating and eventually deciding on termination was not approached for the study. Fifteen families were not counseled in the AFCC prenatally and were admitted to BCH postnatally. These families were enrolled within 2 weeks of the time of CDH repair. 1.2. Demographic and clinical data collection One parent from each family completed the Child Health Ratings Inventory Parent Report- Demographics (CHRIs PR-Demographics) [27] during the prenatal period for those seen in the AFCC or at 2 weeks post-surgery for those not seen in the AFCC. This 41-item measure addresses the child’s background, family health care needs, and family characteristics, including employment status and health care insurance. We removed 15 questions from the prenatally acquired questionnaire and 5 questions from the postnatally acquired questionnaire that were not applicable at that time point depending on the timing of the counseling. For example, child’s date of birth was removed from the prenatal questionnaire and child’s school grade was removed from the postnatal questionnaire. We added a 5-item addendum to the CHRIs PR-Demographics, which addressed dates of birth of mother and father, any parental impairments, and the primary language spoken at home. Clinical data were collected for hospital encounters from the time of prenatal counseling (when applicable) to six months post-surgery by prospective review of hospital medical records. Patients with a genetic abnormality were diagnosed by a formal genetics consulta-
701
tion. Patients with a cardiac anomaly were defined as previously described by the CDH Study Group [28]. CDH defect size was determined according to previously defined criteria established by the CDH Study Group [29] with the following clarifications: no agenesis with primary repair (Type A), no agenesis with patch repair and defect size ≤4 cm by 4 cm (Type B), partial agenesis with patch repair and defect size N4 cm by 4 cm (Type C), complete agenesis with absent rim of anterior and posterior diaphragm requiring suturing of the patch to ribs anteriorly and posteriorly (Type D). Pulmonary, GI, and cardiac issues at discharge were defined as previously described [5]. Patients with neurologic issues at discharge were defined as having at least one of the following: a known seizure disorder, a ventriculoperitoneal shunt or hydrocephalus, any abnormal brain or spinal cord imaging study, or a need for antiepileptic medication. 1.3. Short-Form 36 Version 1 (SF-36v1) data collection For those patients who received prenatal counseling in the AFCC, mothers and fathers each completed the Short-Form 36 version 1 (SF36v1) questionnaire at three time points: prenatal, two weeks postsurgery, and six months post-surgery. Those families who did not receive prenatal counseling in the AFCC completed the SF-36v1 at the latter two time points. The SF-36v1 is a self-administered generic questionnaire that measures physical and mental health using 36 multiple-choice items [30,31]. Using a certified scoring software obtained from QualityMetric Incorporated (Lincoln, RI), raw scores were linearly transformed to ranges of 0 to 100, and eight health domain scores were obtained. In addition, two summary measure scales (physical component summary (PCS), mental component summary (MCS)) were generated, with higher scores indicating better functioning and well-being. 1.4. Statistical analysis All categorical variables were compared using Fisher’s exact test or chi-square. Continuous variables describing clinical characteristics and SF-36v1 scores that conformed to a normal distribution were expressed as mean ± SD with groups compared using the twosample Student t-test. Other continuous data including duration of ventilatory and extracorporeal membrane oxygenation (ECMO) support as well as intensive care unit (ICU) and hospital length of stay (LOS) were compared using the nonparametric Mann–Whitney U test and summarized by medians and interquartile ranges. Statistical
CDH patients surgically repaired at BCH from November 2008 through December 2012 n = 58
Enrolled n = 41
Counseled in the AFCC n = 26
Not counseled in the AFCC n = 15
Excluded n = 17
Declined to participate n = 10
Not approached due to parental social/medical reasons n=3
Postnatal death without study data n=4
Fig. 1. Schematic diagram of study cohort as a subset of the 58 congenital diaphragmatic hernia (CDH) patients surgically repaired at Boston Children’s Hospital (BCH) from November 2008 through December 2012.
702
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
analysis was performed using the SPSS software package (version 19.0, SPSS Inc./IBM, Chicago, IL). Two-tailed values of P b .05 were considered statistically significant. 2. Results 2.1. Characteristics of study population From November 2008 through December 2012, 58 patients with Bochdalek CDH were surgically repaired at BCH. Forty-one families enrolled in this study and 17 families were excluded for the following reasons: 10 declined to participate, 3 were not approached due to parental social/medical reasons, and 4 had infants who died shortly after birth without study data (Fig. 1). Of the 17 excluded families, 10 were previously counseled in the AFCC and 7 were not. The excluded families did not differ from the enrolled families when compared by insurance type and several patient characteristics including sex, birth weight, presence of cardiac anomaly, and CDH defect size. Three of the 41 enrolled patients did not survive to hospital discharge.
Patients stratified by prenatal counseling did not differ by any of the clinical characteristics at birth described in Table 1, including sex, gestational age, birth weight, presence of genetic abnormality or cardiac anomaly, Apgar scores at 1 and 5 minutes, need for ECMO, or
Table 1 Clinical characteristics and hospital course of CDH patients categorized by AFCC prenatal counseling status.
Clinical characteristics Male sex Gestational age, weeks Birth weight, kg Congenital malformation Genetic abnormality Cardiac anomaly Apgar score at 1 min Apgar score at 5 min Defect size A B C D Hospital course Ventilator, days ECMO Days ICU LOS, days Hospital LOS, days Issues at discharge Pulmonary GI Cardiac Neurologic
2.3. Demographic and family characteristics Table 2 describes the demographic and family data of study participants. Data with percentages are calculated based on total number of responses per group. Eleven mothers who stated their primary daily activity as “Other” specified the following activities:
Table 2 Demographic and family data of study participants.
2.2. Characteristics of study participants
Variable
diaphragmatic defect size. Patients who received prenatal counseling in the AFCC had significantly longer days on the ventilator and longer ICU and hospital LOS when compared to those who did not receive AFCC counseling. There were no differences in frequency of medical issues (pulmonary, GI, cardiac, neurologic) at discharge when comparing patients in both groups (Table 1). Of the patients in the group who did not receive prenatal counseling in the AFCC, none of the families were known to have received prenatal counseling elsewhere, despite one of the patients in this group having a prenatal diagnosis of CDH.
Received AFCC counseling (n = 26)
Did not receive AFCC counseling (n = 15)
P
18 (69%) 37 ± 2
12 (80%) 38 ± 4
.72 .32
2.7 ± 0.5 9 (35%)
3.1 ± 0.9 1 (7%)
.10 .06
5 (19%)
0 (0%)
.14
5 (19%)
1 (7%)
.39
5±2
5±3
.33
7±2
7±2
.66 .13
9 (35%) 3 (12%) 9 (35%) 5 (19%)
8 (53%) 4 (27%) 3 (20%) 0 (0%)
24 (8–37) 7 (27%) 21 (10–29) 34 (13–54) 57 (33–107)
6 (2–20) 3 (20%) 10 (4–10) 8 (7–27) 24 (12–55)
b.01⁎ .72 .12 b.01⁎ b.01⁎
17 (74%) 22 (96%) 10 (44%) 10 (44%)
9 (60%) 14 (93%) 4 (27%) 3 (20%)
.48 1.00 .33 .18
Plus-minus data are mean ± SD with groups compared using the two-sample Student t-test. Percentages were compared by Fisher’s exact test or chi-square for defect size and issues at discharge. Days are expressed in terms of the median and interquartile range with groups compared using the Mann–Whitney U test. ⁎ Statistically significant.
Variable
Received AFCC Did not receive AFCC P counseling (n = 26) counseling (n = 15)
Mother age, years Father age, years Race White African American Hispanic/Latino Asian Other Insurance type Private Public Private and public Household Income Less than $19,000 $20,000–$39,999 $40,000–$59,999 $60,000–$79,999 $80,000 and over Marital status Married or living with partner Divorced, separated, or widowed Never married Engaged School grade of parent High School College Graduate/Professional School Language spoken at home English Non-English Siblings† People living in household† Mother’s primary daily activity Working full-time Working part-time Full-time homemaking Other Father’s primary daily activity Working full-time Working part-time Full-time homemaking Other Family members cut down on work hours for patient care Family members stopped working due to patient’s health conditions
31 ± 5 32 ± 6
31 ± 7 33 ± 7
18 (69%) 1 (4%) 2 (8%) 1 (4%) 4 (15%)
10 (67%) 0 (0%) 2 (13%) 1 (7%) 2 (13%)
19 (73%) 4 (15%) 3 (12%)
6 (50%) 4 (33%) 2 (17%)
3 (12%) 4 (16%) 4 (16%) 2 (8%) 12 (48%)
2 (15%) 2 (15%) 4 (31%) 0 (0%) 5 (39%)
20 (77%) 1 (4%)
11 (73%) 1 (7%)
5 (19%) 0 (0%)
2 (13%) 1 (7%)
4 (15%) 14 (54%) 8 (31%)
6 (43%) 3 (21%) 5 (36%)
21 (81%) 5 (19%) 1 (0–5) 4 (2–7)
10 (71%) 4 (29%) 1 (0–3) 4 (0–6)
9 (35%) 2 (8%) 8 (31%) 7 (27%)
4 (29%) 1 (7%) 5 (36%) 4 (29%)
19 (76%) 1 (4%) 1 (4%) 4 (16%) 17 (74%)
11 (79%) 2 (14%) 1 (7%) 0 (0%) 4 (29%)
.02⁎
10 (46%)
3 (23%)
.28
⁎ Statistically significant. † Median (full range).
.75 .83 .90
.35
.70
.55
.08
.69
.77 .59 .98
.83
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
maternity leave (n = 6), frequent visits to the hospital (n = 2), and one each of bed rest, student, and resignation of full-time job. Five fathers who stated their primary daily activity as “Other” specified the following activities: one each of unemployed, college student, disabled, and on leave from full-time job. For those families who received prenatal counseling in the AFCC, a significantly higher percentage of family members cut down on work hours for patient care (74% vs. 29%, P = .02). 2.4. Short-Form 36 Version 1 (SF-36v1) scores Tables 3 and 4 show SF-36v1 domain and component summary scores at each time point for patients prenatally counseled in the AFCC (Table 3) and for those who were not (Table 4). Eleven of the 26 parental pairs who were counseled in the AFCC were able to complete the SF-36v1 at the prenatal time point. We first examined the effect of prenatal AFCC counseling on SF-36v1 PCS and MCS scores stratified by sex. We found that at two weeks and six months post-surgery, there were no significant differences in PCS and MCS scores when comparing mothers and fathers, respectively, in each group (data not shown). These results are the same even after adjusting for differences in hospital LOS between the AFCC prenatally counseled group and the non-counseled group using ANOVA (data not shown). We next examined the SF-36v1 scores over time for parents who received prenatal counseling in the AFCC stratified by sex (Table 3). Mean PCS scores for mothers were significantly lower than those for fathers at both the prenatal (40 vs. 60, P b .01) and 2 weeks (48 vs. 58, P b .01) post-surgery time points, but were not different by 6 months post-surgery (56 vs. 54, P = .40). Although there were no significant differences in MCS scores at each of the time points between mothers and fathers, mothers appeared to have higher MCS scores prenatally (47 vs. 41, P = .24), and these scores were sustained at 6 months post-surgery for both parents (47 vs. 47, P = .90). 3. Discussion This is the first known study to examine the effect of prenatal counseling on parental well-being of CDH families. During our study time period, we identified two groups of CDH families that received all of their surgical care at our institution: one group was diagnosed prenatally and received counseling in our AFCC, and one group was transferred urgently to our institution for care in the postnatal period from an outside facility. All of the latter families were not known to have received any formal prenatal counseling, although one patient was diagnosed prenatally with CDH. We did note that the group who received AFCC counseling required more intensive care with longer
703
Table 4 SF-36v1 scores for parents who did not receive prenatal AFCC counseling. Domain
2 weeks post-surgery
PF RP BP GH VT SF RE MH PCS MCS
6 months post-surgery
Mother
Father
P
Mother
Father
P
89 59 54 77 45 67 69 63 50 42
98 90 92 83 64 83 75 75 59 47
.09 .05⁎ b.01⁎
87 90 75 71 54 94 94 76 52 52
97 93 94 75 55 86 97 79 57 50
.24 .74 .03⁎
± ± ± ± ± ± ± ± ± ±
19 48 30 20 26 31 42 16 8 12
± ± ± ± ± ± ± ± ± ±
5 25 15 18 21 22 38 17 4 12
.47 .05⁎ .14 .71 .08 b.01⁎ .36
± ± ± ± ± ± ± ± ± ±
23 23 24 26 18 11 19 14 10 9
± ± ± ± ± ± ± ± ± ±
9 17 10 13 18 30 11 9 4 9
.67 .90 .47 .75 .63 .14 .63
Data are means ± SD, scores range from 0–100. PF, physical functioning; RP, role limitations due to physical health; BP, bodily pain; GH, general health perceptions; VT, vitality; SF, social functioning; RE, role limitations due to emotional problems; MH, mental health; PCS, physical component summary; MCS, mental component summary. ⁎ Statistically significant.
duration of ventilation and longer LOS in the ICU and in the hospital. When we controlled for hospital LOS between the two groups, we were surprised that SF-36v1 mental and physical component summary scores were not different between mothers and fathers at 2 weeks and at 6 months post-surgery. In other words, despite the need for more intensive and longer supportive care in the hospital, parents who received prenatal AFCC counseling had comparable scores to those who did not. This could represent the beneficial effect of prenatal counseling on preparing families before birth for the difficult course ahead, or the detrimental effect of not having the opportunity to learn about CDH prenatally for the group not receiving AFCC counseling. This is also the first known study to examine parental well-being of CDH families beginning in the prenatal period. We know of no other studies of any pediatric condition where parental well-being of mothers and fathers was examined individually during the prenatal period. Not surprisingly, fathers had significantly better physical component summary scores than mothers at the prenatal and 2 week post-surgery time points in both groups. This difference in PCS scores was not noted at 6 months post-surgery in either group. When examining the scores for the AFCC counseled group, one interesting finding, though not significant, was the higher MCS scores in mothers compared to fathers at the prenatal time point. This was unexpected given the particular stressors that mothers carrying a fetus with CDH face and one wonders whether this may reflect a beneficial effect of prenatal counseling, particularly for mothers, during the prenatal period. We did not see a significant difference between MCS scores at
Table 3 SF-36v1 scores for parents who received prenatal AFCC counseling. Domain
PF RP BP GH VT SF RE MH PCS MCS
Prenatal
2 weeks post-surgery
6 months post-surgery
Mother
Father
P
Mother
Father
P
Mother
Father
P
52 30 45 81 42 64 67 66 40 47
93 98 86 82 59 77 61 66 60 41
b.01⁎ b.01⁎ b.01⁎
76 50 51 80 40 56 59 60 48 40
93 82 83 79 49 67 67 61 58 39
.01⁎ b.01⁎ b.01⁎
93 92 82 74 52 81 83 74 56 47
90 92 76 73 56 83 77 75 54 47
.74 .99 .49 .85 .58 .77 .65 .88 .40 .90
± ± ± ± ± ± ± ± ± ±
32 38 18 14 15 31 42 19 9 12
± ± ± ± ± ± ± ± ± ±
14 8 16 17 15 18 39 13 7 9
.83 .02⁎ .22 .73 1.00 b.01⁎ .24
± ± ± ± ± ± ± ± ± ±
28 46 31 17 22 32 40 22 12 14
± ± ± ± ± ± ± ± ± ±
15 31 19 16 20 30 40 22 8 15
.85 .16 .21 .50 .87 b.01⁎ .93
± ± ± ± ± ± ± ± ± ±
11 25 22 22 23 18 32 15 6 8
± ± ± ± ± ± ± ± ± ±
25 15 27 17 17 19 38 17 7 10
Data are means ± SD, scores range from 0–100. PF, physical functioning; RP, role limitations due to physical health; BP, bodily pain; GH, general health perceptions; VT, vitality; SF, social functioning; RE, role limitations due to emotional problems; MH, mental health; PCS, physical component summary; MCS, mental component summary. ⁎ Statistically significant.
704
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705
2 weeks and 6 months post-surgery between mothers and fathers in the prenatally counseled group, although scores at 2 weeks for both parents were lower than those at 6 months post-surgery. This may reflect greater emotional strain for parents at a critically ill phase of their infant’s post-surgical life when they have to cope with their prenatal decision to continue with care. Our findings differ from those of Bevilacqua et al. who found that stress and depression levels were significantly higher in mothers than in fathers of children diagnosed either prenatally or postnatally with congenital heart disease when examined 2 weeks after hospitalization [32]. Caniano & Baylis have detailed the goals of prenatal consultation by pediatric surgeons caring for parents of a fetus with a congenital anomaly [33]. The authors describe three specific time periods, beginning with the initial prenatal consultation, which focuses on possible termination of the pregnancy, followed by two additional periods which focus on the location, timing, and mode of delivery, and the decisions that are required after birth surrounding operative intervention. At our institution, CDH families, in addition to receiving medical and surgical counseling by an interdisciplinary team of pediatric surgeons, radiologists, geneticists, subspecialists, and nurses, are provided with social work support during the AFCC visits (by coauthor LOT). Before 24 weeks gestation, CDH families are counseled on the risk category of their child’s CDH and pregnancy options. After 24 weeks gestation, AFCC counseling is focused on a reassessment of the percentage predicted lung volume and expected course of care. This level of support is continued in the post-delivery period while patients are in the ICU, including assistance with accommodations and orientation to the ICU environment. After discharge from the hospital, CDH patients are followed in our multidisciplinary CDH Clinic and currently do not receive any formal social work support, but the AFCC nurses provide continuity for social work issues. Interestingly, we found that a higher percentage of families who received prenatal AFCC counseling had family members who cut down on work hours for patient care (Table 2). This may reflect the ability of prenatally counseled families to plan in advance to reduce work hours, which may not be readily done by those families who learn about the diagnosis unexpectedly after birth. We were encouraged to see that MCS scores for mothers and fathers who received prenatal counseling were sustained 6 months post-surgery. Although our patients do not have formal Social Work referrals during the outpatient period, ongoing support is a high priority initiative. During this time interval, a social worker on the CDH care team could screen for residual post-traumatic stress or signs of parental depression, and aid with parental psychological mastery of the CDH experience by allowing parents to tell the story of their journey. Families of infants with ongoing medical issues often feel that the journey is prolonged and that they are unable to restore normalcy. A study by Hatzmann et al. in a cohort of parents of chronically ill children found that parental quality of life at greater than one year after diagnosis was positively influenced by factors including caredependency, emotional support, and increased number of days on holiday [34]. There are several limitations to our study. First, we do not have a comparison group for the prenatally counseled parents at the prenatal time point for obvious logistical reasons and therefore our explanations for the results at the prenatal time point are descriptive only. In addition, not all the prenatally counseled parents were able to provide us with data at the prenatal time point, potentially making the prenatal data not representative of the entire cohort. Second, each of our time points for data collection was broad in that particular clinical scenarios within each time period could have been associated with significant fluctuations in parental well-being which we are not able to capture. For instance, we were not able to obtain data at the first prenatal consultation when the diagnosis was explained to the family and this could have represented a time of higher stress compared to later in the prenatal period. Similarly, the time period after birth but
before surgery may also have been more stressful for parents than one week after surgery. Third, our study had a limited sample size and we did not note any significant differences in clinical characteristics between our two groups based on prenatal counseling status. However, with a larger sample size, some statistically significant differences may arise between patients in both groups, such as larger defect sizes or higher rates of congenital or cardiac malformations in the group receiving prenatal counseling. This may help to explain why we noted a significant difference in number of days on the ventilator, and ICU and hospital LOS, where these measures were longer in the group that received prenatal counseling. Finally, continuing our assessments longitudinally over several years could provide insight into issues that may be more relevant for families as the children pass into toddlerhood. Our previous work provides evidence that the impact of CDH on patients and families is profound and long-standing years later [26]. Future work should continue this data collection in a longitudinal fashion to help guide appropriate services long-term. A compelling initiative similar to an early intervention program to reduce parenting stress after preterm birth described by Kaaresen et al. [15] could be created for our CDH families, most of whom have children with ongoing medical conditions. In addition, differences between mothers and fathers can be examined further by conducting focus groups aimed at understanding specific stressors for each parent during the prenatal period. Finally, understanding factors that are associated with increased marital stress and identifying ways for parents to cope with these stressors would be vital to the integrity of the family unit. Acknowledgments This study was supported by the Boston Children's Hospital Surgical Foundation. The authors would like to thank Donna Morash, RN, Robin Scott, BA and the Boston Children’s Hospital Advanced Fetal Care Center for help with patient recruitment and scheduling. References [1] Danzer E, Hedrick HL. Neurodevelopmental and neurofunctional outcomes in children with congenital diaphragmatic hernia. Early Hum Dev 2011;87:625–32. [2] Chiu PP, Ijsselstijn H. Morbidity and long-term follow-up in CDH patients. Eur J Pediatr Surg 2012;22:384–92. [3] Muratore CS, Kharasch V, Lund DP, et al. Pulmonary morbidity in 100 survivors of congenital diaphragmatic hernia monitored in a multidisciplinary clinic. J Pediatr Surg 2001;36:133–40. [4] Muratore CS, Utter S, Jaksic T, et al. Nutritional morbidity in survivors of congenital diaphragmatic hernia. J Pediatr Surg 2001;36:1171–6. [5] Friedman S, Chen C, Chapman JS, et al. Neurodevelopmental outcomes of congenital diaphragmatic hernia survivors followed in a multidisciplinary clinic at ages 1 and 3. J Pediatr Surg 2008;43:1035–43. [6] Hatzmann J, Heymans HS, Ferrer-i-Carbonell A, et al. Hidden consequences of success in pediatrics: parental health-related quality of life – results from the Care Project. Pediatrics 2008;122:e1030–8. [7] Brehaut JC, Garner RE, Miller AR, et al. Changes over time in the health of caregivers of children with health problems: growth-curve findings from a 10-year Canadian population-based study. Am J Public Health 2011;101:2308–16. [8] Klassen AF, Klaassen R, Dix D, et al. Impact of caring for a child with cancer on parents’ health-related quality of life. J Clin Oncol 2008;26:5884–9. [9] Goldbeck L, Melches J. The impact of the severity of disease and social disadvantage on quality of life in families with congenital cardiac disease. Cardiol Young 2006;16:67–75. [10] Arafa MA, Zaher SR, El-Dowaty AA, et al. Quality of life among parents of children with heart disease. Health Qual Life Outcomes 2008;6:91. [11] Buysse CM, Raat H, Hazelzet JA, et al. Surviving meningococcal septic shock: health consequences and quality of life in children and their parents up to 2 years after pediatric intensive care unit discharge. Crit Care Med 2008;36:596–602. [12] Zilber R, Bortz AP, Yacobovich J, et al. Analysis of health-related quality of life in children with immune thrombocytopenia and their parents using the kids' ITP tools. J Pediatr Hematol Oncol 2012;34:2–5. [13] Pemberton J, Frankfurter C, Bailey K, et al. Gastrostomy matters – the impact of pediatric surgery on caregiver quality of life. J Pediatr Surg 2013;48:963–70. [14] Miles MS, Funk SG, Kasper MA. The stress response of mothers and fathers of preterm infants. Res Nurs Health 1992;15:261–9. [15] Kaaresen PI, Rønning JA, Ulvund SE, et al. A randomized, controlled trial of the effectiveness of an early-intervention program in reducing parenting stress after preterm birth. Pediatrics 2006;118:e9-19.
K.V. Dennett et al. / Journal of Pediatric Surgery 49 (2014) 700–705 [16] Correia LL, Linhares MB. Maternal anxiety in the pre- and postnatal period: a literature review. Rev Lat Am Enfermagem 2007;15:677–83. [17] Singer LT, Fulton S, Kirchner HL, et al. Parenting very low birth weight children at school age: maternal stress and coping. J Pediatr 2007;151:463–9. [18] Rentinck IC, Ketelaar M, Jongmans MJ, et al. Parents of children with cerebral palsy: a review of factors related to the process of adaptation. Child Care Health Dev 2007;33:161–9. [19] Tong HC, Kandala G, Haig AJ, et al. Physical functioning in female caregivers of children with physical disabilities compared with female caregivers of children with a chronic medical condition. Arch Pediatr Adolesc Med 2002;156:1138–42. [20] Goldberg S, Morris P, Simmons RJ, et al. Chronic illness in infancy and parenting stress: a comparison of three groups of parents. J Pediatr Psychol 1990;15:347–58. [21] Lawoko S, Soares JJ. Quality of life among parents of children with congenital heart disease, parents of children with other diseases and parents of healthy children. Qual Life Res 2003;12:655–66. [22] Helfricht S, Latal B, Fischer JE, et al. Surgery-related posttraumatic stress disorder in parents of children undergoing cardiopulmonary bypass surgery: a prospective cohort study. Pediatr Crit Care Med 2008;9:217–23. [23] Hunfeld JA, Tempels A, Passchier J, et al. Brief report: parental burden and grief one year after the birth of a child with a congenital anomaly. J Pediatr Psychol 1999;24:515–20. [24] Poley MJ, Brouwer WB, van Exel NJ, et al. Assessing health-related quality-of-life changes in informal caregivers: an evaluation in parents of children with major congenital anomalies. Qual Life Res 2012;21:849–61. [25] Bonner MJ, Hardy KK, Willard VW, et al. Brief Report: psychosocial functioning of fathers as primary caregivers of pediatric oncology patients. J Pediatr Psychol 2007;32:851–6.
705
[26] Chen C, Jeruss S, Terrin N, et al. Impact on family of survivors of congenital diaphragmatic hernia repair: a pilot study. J Pediatr Surg 2007;42: 1845–52. [27] Parsons SK, Shih MC, Mayer DK, et al. Preliminary psychometric evaluation of the Child Health Ratings Inventory (CHRIs) and Disease-Specific Impairment Inventory-Hematopoietic Stem Cell Transplantation (DSII-HSCT) in parents and children. Qual Life Res 2005;14:1613–25. [28] Tsao K, Lally PA, Lally KP, et al. Minimally invasive repair of congenital diaphragmatic hernia. J Pediatr Surg 2011;46:1158–64. [29] Congenital Diaphragmatic Hernia Study Group, Lally KP, Lally PA, et al. Defect size determines survival in infants with congenital diaphragmatic hernia. Pediatrics 2007;120:e651–7. [30] Ware Jr JE, Sherbourne CD. The MOS 36-item short-form health survey (SF-36). I. Conceptual framework and item selection. Med Care 1992;30:473–83. [31] McHorney CA, Ware Jr JE, Raczek AE. The MOS 36-Item Short-Form Health Survey (SF-36): II. Psychometric and clinical tests of validity in measuring physical and mental health constructs. Med Care 1993;31:247–63. [32] Bevilacqua F, Palatta S, Mirante N, et al. Birth of a child with congenital heart disease: emotional reactions of mothers and fathers according to time of diagnosis. J Matern Fetal Neonatal Med 2013;26:1249–53. [33] Caniano DA, Baylis F. Ethical considerations in prenatal surgical consultation. Pediatr Surg Int 1999;15:303–9. [34] Hatzmann J, Maurice-Stam H, Heymans HS, et al. A predictive model of Health Related Quality of life of parents of chronically ill children: the importance of caredependency of their child and their support system. Health Qual Life Outcomes 2009;7:72.
