Children subjected to cardiac surgery for congenital heart disease. Part 1 Emotional and psychological outcomes,

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1 doi: /icvts Interactive CardioVascular and Thoracic Surgery 7 (2008) Institutional report - Congenital Children subjected to cardiac surgery for congenital heart disease. Part 1 Emotional and psychological outcomes, Samuel Menahem *, Zeffie Poulakis, Margot Prior a,b, b c a Department of Paediatric Cardiology, Monash Medical Centre and Departments of Paediatrics and Psychological Medicine, Monash University, Murdoch Children s Research Institute, The Royal Children s Hospital, Melbourne, Victoria, Australia b Centre for Community Child Health, Murdoch Children s Research Institute, The Royal Children s Hospital, Melbourne, Victoria, Australia c Department of Psychology, University of Melbourne, Melbourne, Victoria, Australia Received 14 November 2007; received in revised form 8 April 2008; accepted 10 April 2008 Abstract This study investigated the psychological and emotional functioning of children with congenital heart disease (CHD) subjected to surgery. Children aged 2 12 years with CHD who underwent cardiac surgery were enrolled. Information was collected prior to surgery and 12 months or later following surgery. Measures included assessment of the child s receptive vocabulary, adaptive behaviour skills, emotional and behavioural development, temperament and parent quality of life, as well as surgical data. Similar information was collected from a control group prior to undergoing non-cardiac surgery. Of the 69 children contacted to enrol, completed pre- and post-surgical data were obtained from 39 children, and pre-surgical data from 12 controls. Children with CHD subjected to surgery displayed psychological and emotional functioning indistinguishable from normative populations or the control group. These findings persisted at reassessment months after surgery. Psychological functioning at follow-up was most closely related to functioning prior to surgery. Significant residual defects and the need for further surgery were associated with poorer functioning. The results suggest an optimistic psychological and emotional outcome following cardiac surgery. This study may assist in identifying children most at risk of adverse outcomes after cardiac surgery and help guide therapeutic interventional programmes Published by European Association for Cardio-Thoracic Surgery. All rights reserved. Keywords: Congenital heart disease; Paediatric; Psychological; Emotional outcomes 1. Introduction Congenital heart disease (CHD) affects 1% of newborns. Of these, the majority are minor lesions that do not require surgery. However, some are serious and may result in significant morbidity and remains the largest cause of neonatal mortality second to prematurity. While CHD can be accurately identified pre-natally, many remain to be diagnosed post-natally. Major advances in surgical treatment have improved outcomes for children with CHD. These advances have resulted in dramatic increases in survival rates, with a reduction in the morbidity and complications arising from such interventions. More attention is now being placed on the developmental and psychological outcomes of these children, realizing that any form of cardiac surgery is a major stress for children and their families w1x. Presented in part to the World Congress of Paediatric Cardiology and Cardiac Surgery, Buenos Aires, September 2005, and at the World Congress of the International Association of Child and Adolescent Psychiatry and Allied Professions, Melbourne, September Seeding Grants from the Murdoch Children s Research Institute, Heart Kids Victoria & Dube Fund. *Corresponding author. Department of Paediatric Cardiology, Monash Medical Centre, 246 Clayton Road, Clayton South, Victoria 3168, Australia. Tel.: q ; fax: q address: samuel.menahem@southernhealth.org.au (S. Menahem) Published by European Association for Cardio-Thoracic Surgery Children with CHD are at risk of suboptimal developmental outcomes for a number of reasons. Firstly, they are at higher risk from the physiological consequences resulting from the cardiac abnormality itself and include cyanosisy hypoxia, cardiac failure andyor collapse, and cerebral insults w2, 3x. To that may be added the more chronic effects of malnutrition, failure to thrive and recurrent infections. Secondly, there is a body of evidence suggesting that cardiac surgery may influence psychological neurodevelopmental functioning. Much attention has focused on their developmental and cognitive functioning following surgery and the contributions from the nature of the cardiopulmonary bypass and the variable methods of cardioplegia w4x. More recently, there has been increasing evidence to suggest that neurological deficits may be evident in infants with CHD prior to surgical intervention, arising from the possible reduced oxygenation to the growing fetal brain, for example in transposition of the great vessels or hypoplastic left heart syndrome w3x. Such defects may pre-date the birth of the infant and are not necessarily primarily related to the sequelae of bypass or other cerebral insults that may occur during surgery w5x. With improved outcomes, there has been growing interest in the emotional impact of cardiac surgery on children, a

2 S. Menahem et al. / Interactive CardioVascular and Thoracic Surgery 7 (2008) Table 1 What do we already know about children with CHD? Construct Intellectual functioning (IQ) Expressive Language Behaviour problems Adaptive behaviour Academic achievement When measured after surgery Generally in low average norm average range w2 4, 14x Generally show a delay w14, 15x Generally show more difficulties than normsycontrol groups w14, 15x Some evidence of delay Lower than controls major stressor for the child and hisyher family w6x. Previously documented in other surgery, Connolly et al. w7x, claimed that children who had surgery for CHD had features of post-traumatic stress disorder when reviewed 4 8 weeks following surgery. Various domains of psychological and emotional functioning have been investigated in children with CHD and are outlined in Table 1. Two other studies have compared children with CHD prior to and after surgery in relation to psychological and emotional functioning. Haneda et al. w8x found that children s adaptive behaviour skills and IQ were similar prior to and after surgery. Wray and Sensky w9x reported that children with CHD showed an improvement in self-concept following surgery, having rated themselves prior to surgery as weaker, more frightened and more ill when compared with normal children. Our study was set up to prospectively examine the emotional and psychological functioning of children with CHD prior to and following cardiac surgery, reviewing their behaviour and emotional status, adaptive behaviour skills and receptive vocabulary. The additional aims were to determine whether apparent poorer functioning on measures of behaviour or emotional development (see Table 1) pre-dated or occurred consequent to surgery. Parental concerns and reactions to their children who have undergone cardiac surgery will be reported in a companion paper w10x. 2. Materials and methods Following institutional ethics approval, children aged 2 12 years who underwent cardiac surgery were prospectively and consecutively enrolled once parental informed consent was obtained. The children and their parents were interviewed and assessed by a post-graduate psychology student (ZP) prior to surgery, and at least 12 months post-surgery. A control group who underwent non-cardiological, nonneurological surgery were also enrolled and followed the same protocol prior to surgery, but with no follow-up review as they seemed to differ little from normative values. The measures used in this study are outlined in Table 2 the age group selected allowing for reasonable objective testing following the rapid changes occurring during infancy and prior to the onset of adolescence. Other measures included standardised tools for measuring receptive language and adaptive behaviour functioning which included communication, socialisation and daily living skills. The New York Heart Association (NYHA) scores were modified in view of the age group being studied (see Table 4). 3. Statistical analysis Following data entry and screening (cleaning), descriptive statistics were generated for all data collected. Where norms and standardised data were available, comparisons were made with data collected from both the CHD and control groups. Pearson product-moment correlations were calculated to ascertain status between pre-surgery and post-surgery. Student t-tests were used when samples were compared (e.g. case group vs. control group). 4. Results Over a 3 year period, there were 103 children with CHD proceeding to surgery who met the study criteria for enrollment. Because of limited resources and other logistic issues, only 69 were able to be contacted to enrol. Of these, 16 could not be interviewed early enough to have their measures completed prior to surgery. In total, 4 had Table 2 Measures used in this study Measure Method of collection of data Construct Peabody picture vocabulary test Direct child assessment Receptive vocabulary Vineland adaptive behaviour scales w11x Parent interview Adaptive behaviour skills Behaviour screening questionnaire Parent interview Behaviour and emotional problems Short temperament scale for toddlersy Parent questionnaire Temperament children PedsQL (only collected at follow-up) Parent questionnaire and child questionnaire Health-related quality of life Medical and surgical data questionnaire Completed by cardiologist Clinical information about (study designed questionnaire) the nature of the CHD the intervention required bypass and cross-clamp times length of ICU stay requirements for ventilatory and inotropic support short- and medium-term complications after surgery, etc. *Available on request.

3 602 S. Menahem et al. / Interactive CardioVascular and Thoracic Surgery 7 (2008) their surgery postponed, 4 did not respond to the invitation to participate and 6 declined participation. Data were therefore obtained for 39 children. A total of 12 children were enrolled in the control group, the numbers being limited for logistic reasons. They underwent surgical procedures with an anticipated length of hospital stay that was comparable to the CHD group. The children in the control group completed the same assessments as the CHD group prior to surgery. They included children admitted for bowel or urological procedures. No follow-up review was undertaken as their pre- and postsurgery data were very similar to those obtained from the CHD children and standardised norms. The median age at the time of cardiac surgery was 4.46 years (range years). Nearly half of the children were male. Their diagnoses are outlined in Table 3. Two children had closed procedures while the other 37 had cardiopulmonary bypass. In total, 8 children had their Fontan completed. All children except 1 were considered to have an optimal or very good technical outcome; two-thirds excluding those that had a Fontan palliation had no residual cardiac defect. In total, 2 children were thought to possibly require further surgery. The median hospital stay was 6 days (range 4 36 days). Table 4 outlines the functional status of the children prior to and following surgery. Table 5 summarises the psychological and emotional outcomes for the CHD group prior to and subsequent to cardiac surgery. There were no significant differences between the CHD group and the control group on any of the measures, both at the pre-surgery and post-surgery data collection Table 3 Diagnoses in the CHD group Diagnosis Surgery n VSD Closure 6 Multiple VSDs Closure 2 VSDyAVP Closure 1 Secundum ASD Closure 6 Sinus venosus ASD Closure 3 Scimitar syndrome ASD closure redirection of 1 anomalous pulmonary veins DORVyVSDyPA Fontan 1 Post-op DORVyVSDyIAAyPS Conduit replacement 1 Post-op DORVyVSDyMitral valve ring Resection 1 AVSD Closure, mitral valve repair 2 Unbalanced AVSD PA band 1 Fontan 1 Post-op pulmonary atresiay Conduit replacement 1 VSDyMAPCAs Post-op pulmonary atresiayvsdy Resection of aneurysm, 1 MAPCAsyRV outflow aneurysm conduit replacement Truncus arteriosusyps Conduit replacement 1 Truncus arteriosusyiaayps Conduit replacement 1 DILV Fontan 1 Left AV valve atresia Fontan 1 Tricuspid atresia Fontan 4 Patent ductus arteriosus Ligation 1 Coarctation of abdominal aorta Interpositional graft 1 Tetralogy of fallot Repair 1 VSD, ventricular septal defect; AVP, aortic valve prolapse; ASD, atrial septal defect; DORV, double outlet right ventricle; Post-op, post-operative; IAA, interrupted aortic arch; PS, pulmonary stenosis; AVSD, atrio-ventricular septal defect; MAPCAs, multiple aorto-pulmonary collateral arteries; RV, right ventricle; DILV, double inlet left ventricle; AV, atrio-ventricular. Table 4 Functional status pre-surgery and post-surgery NYHA functional status scale (modified) Pre-surgery Post-surgery (%) (%) NYHA1 symptoms in the normal range NYHA2 symptoms with moderate exertion NYHA3 symptoms with mild exertion 15 0 NYHA4 symptoms at rest 6 0 points. Similarly, the CHD group did not differ from normative data on any of the measures except for the socialisation domain. However, 14% of the children with CHD had Vineland Adaptive Behaviour Scores (VABS) w11x Maladaptive Behaviour Scores in the significant category but dropped to 9% on follow-up. In addition, the year subgroups were found to have an easier temperament compared to norms. For the CHD group, Pearson product-moment correlations indicated that there was no correlation between the psychological and emotional measures at follow-up and the nature of the cardiac abnormality, the type of surgery done, duration of bypass and cross-clamp time, and the duration of intensive care stay. However, children with significant persistent lesions tended toward poorer behavioural profiles when assessed after surgery (rs0.70, P-0.001). Children requiring further surgery were more likely to have lower scores on the VABS Adaptive Behavior Composite w11x (rs 0.55, Ps0.006), and on the VABS Communication Domain score w11x (rs 0.55, Ps0.007). NYHA scores at follow-up were significantly correlated with scores on the VABS Adaptive Behavior Composite score at follow-up (rs 0.51, Ps0.007). The strongest correlations with the emotional and psychological measures at followup were with the same measures pre-surgery. There were 3 children and 1 of whom had a VSD closed, who did not do well on the psychological and emotional measures especially in adaptive functioning following cardiac surgery, 2 of whom required multiple operations. Also, 2 of the 3 children also showed difficulties in emotional and psychological functioning prior to surgery. 5. Discussion No firm conclusion can be drawn from this study as the total number of children reviewed was small, the nature of their cardiac lesions varied and the surgery required differed, making a multivariant analysis very difficult. Nevertheless, our study seemed to suggest that children with CHD were comparable to norms on a number of objective measures of emotional and psychological functioning both prior to and following cardiac surgery in the medium term. The best predictor of psychological and emotional functioning at follow-up was their functioning in the same domain prior to surgery. In other words, those doing well prior to surgery showed the most resilience at follow-up. Likewise, if psychological or emotional problems were evident prior to surgery, these persisted after surgery. Although the numbers were small, those children who had significant residual lesions or who required further surgery, or who had lower NYHA modified scores, did less well on

4 S. Menahem et al. / Interactive CardioVascular and Thoracic Surgery 7 (2008) Table 5 t-test comparison of CHD group pre-surgery mean scores and follow-up results with control group and normative data Measure Pre-surgery Follow-up CHD group CHD group Control group CHD group CHD group CHD group CHD group Comparison of Mean (S.D.) Mean (S.D.) compared to compared to Mean (S.D.) compared to pre-surgery vs. ns39 ns12 control group norms norms follow-up VABS Adaptive behaviour* Composite (14.22) (15.54) ns (Ps0.68) ns (Ps0.21) (13.23) ns (Ps0.46) ns (Ps0.72) Communication (17.35) (17.80) ns (Ps0.88) ns (Ps0.97) (15.73) ns (Ps0.21) ns (Ps0.40) Daily living skills (13.92) (15.53) ns (Ps0.62) ns (Ps0.46) (11.64) ns (Ps0.14) ns (Ps0.07) Socialization (18.04) (15.87) ns (Ps0.86) Ps (10.98) ns (Ps0.06) ns (Ps0.07) Motor skills (17.33) (6.36) ns (Ps0.22) ns (Ps0.66) (17.02) ns (Ps0.40) ns (Ps0.68) Receptive vocabulary* (11.52) (10.69) ns (Ps0.82) ns (Ps0.25) (16.61) ns ns Temperament q 2.47 (0.97) 2.33 (0.87) ns (Ps0.70) ns (except (0.96) ns ns p 4.5 years Ps0.001 ) Behaviour and (2.66) (1.66) ns (Ps0.82) nya (2.39) nya ns emotional status q p S.D., standard deviation; ns, not significant; vs., versus; *ms100; ss15; ms ; ss ; Easier temperament. adaptive behaviour composite scores. The CHD children were also very similar to a small control group who were about to have non-cardiac surgery. This study included a prospective design, with collection of data prior to and after surgery w8, 9x. Assessment following surgery was conducted 1 4 years later, enabling more medium term outcomes to be measured. Our findings were in contrast to an earlier study w7x which found that children assessed 4 8 weeks following cardiac surgery displayed signs of post-traumatic stress disorder. It is possible that these emotional difficulties may subside with time especially if the results of the surgery are good w12x. In addition, the tenuous nature of diagnosis of post-traumatic stress disorder at a young age should be noted. The enrolment of participants was whenever possible continuous and unbiased in terms of identifying potential candidates. A wide range of child functioning measures were employed (receptive vocabulary, adaptive behaviour, emotional functioning), as opposed to focusing on intellectual functioning and neuro-developmental outcomes (see above). The age of the participants was specifically selected so that instruments were available to reliably assess the constructs of interest in children without introducing the complexities of assessment of infants or adolescents. One should note, however, that most cardiac surgery, including more complex surgery, is now being carried out on infants and this is the focus of a current prospective study w13x. Previous reports have suggested that children with CHD do not perform as well as control groups on measures of intellectual functioning. In contrast, children with CHD examined in this study showed psychological and emotional profiles that were comparable to norms both preceding and following surgery. Unlike the current study, many of the earlier studies did not assess functioning prior to surgery, and as suggested earlier, any deficits may have preceded surgical intervention w3x. Although overall this group of children was functioning at levels comparable to normative groups when assessed in multiple domains, there were nonetheless a few children who displayed delays and deficits in their functioning. Identifying those children most at risk of adverse emotional outcomes after cardiac surgery may well be possible prior to the surgery itself and may allow for referral for early therapeutic interventions. Successful correction of the cardiac lesion itself did not seem to improve such children s emotional and psychological well-being though the numbers involved were quite limited. A further study is underway assessing if that translates to the adolescent. Anecdotal observations suggested that parents were enthusiastic to describe what their children were able to do, rather than to focus on their difficulties or areas where they were experiencing delays or had developed a disability. Parents of children with CHD, who underwent cardiac surgery, also generally did well at follow-up except in a couple of areas and is the subject of a companion paper w10x. It would appear that despite the life-threatening nature of the surgical intervention, and the stresses that this may present to a child and his or her family, provided a child was functioning within age-appropriate levels psychologically and emotionally prior to surgery, heyshe continued to function age-appropriately in these areas 1 year or more following cardiac surgery. This finding applied especially if there were no significant residual cardiac defects and augers well for the future emotional well-being of the operated child. Acknowledgments Special thanks is given to Prof Elizabeth Wertheim, School of Psychological Science, La Trobe University, Melbourne, for her helpful comments on this research, and to the Cardiologists and Cardiac Surgeons at the Royal Children s Hospital, Melbourne, who allowed their patients to be studied. References w1x Menahem S. Counselling strategies for parents of infants with congenital heart disease. Cardiol Young 1998;8: w2x Mahle WT. Neurologic and cognitive outcomes in children with congenital heart disease. Curr Opin Pediatr 2001;13: w3x Wernovsky G, Shillingford AJ, Gaynor JW. Central nervous system outcomes in children with complex congenital heart disease. Curr Opin Cardiol 2005;20:94 99.

5 604 S. Menahem et al. / Interactive CardioVascular and Thoracic Surgery 7 (2008) w4x Bellinger DC, Wypij D, du Plessis AJ, Rappaport LA, Riviello D, Jonas RA, Newburger JW. Developmental and neurological effects of alphastat versus ph-stat strategies for deep hypothermic cardiopulmonary bypass in infants. J Thorac and Cardiovasc Surg 2001;121: w5x Chang T, Jonas RA. Neurological complications of cardiovascular surgery. Curr NeuroyNeurosci Rep 2006;6: w6x Ben-Amitay G, Korov I, Reiss A, Toren P, Yoran-Hagesh R, Kotler M, Mozes T. Is elective surgery traumatic for children and their parents? J Pediatr Child Health 2006;42: w7x Connolly D, McClowry S, Hayman L, Mahony L, Artman M. Post traumatic stress disorder in children after cardiac surgery. J Pediatr 2004;144: w8x Haneda K, Itoh T, Togo T, Ohmi M, Mohri H. Effects of cardiac surgery on intellectual function in infants and children. Cardiovasc Surg 1996; 4: w9x Wray J, Sensky T. How does the intervention of cardiac surgery affect the self-perception of children with congenital heart disease? Child: Care, Health Develop 1998;24: w10x Menahem S, Poulakis Z, Prior M. Children subjected to cardiac surgery for congenital heart disease. Part 2 Parental emotional experiences. Interact CardioVasc Thorac Surg 2008;7: w11x Sparrow SS, Balla DA, Cicchetti DV. Vineland adaptive behavior scales: interview edition, survey form. Manual. Minnesota American Guidance Service, Mineapolis, Minnesota w12x Kassam-Adams N. Introduction to the special issue: post traumatic stress related pediatric illness and surgery. J Pediatr Psychol 2006;31: w13x Franich-Ray C, Al-Farqani A, Menahem S, Al-Farqani A, Anderson V, Northam E, Cochrane A. Impact on parents and families of infants subjected to cardiac surgery preliminary findings. In: Fourth World Congress Paediatric Cardiology and Cardiac Surgery 2005:261 (Abstract). w14x Bellinger DC, Wypij D, Kuban KC, Rappaport LA, Hickey PR, Wernovsky G, Jonas RA, Newburger JW. Development and neurological status of children at 4 years of age after heart surgery with hypothermic circulatory arrest or low-flow cardiopulmonary bypass. Circulation 1999; 10: w15x O Dougherty M, Wright FS, Loewenson RB, Torres F. Cerebral dysfunction after chronic hypoxia in children. Neurology 1985;35: Appendix A: Supplementary data Supplementary data associated with this article can be found in the online version at doi: /icvts