Acta Pædiatrica ISSN 0803-5253
REGULAR ARTICLE
Motor dysfunction and behavioural problems frequently coexist with congenital heart disease in school-age children R Liamlahi1, M von Rhein1, S B€uhrer2, E R Valsangiacomo Buechel3, W Knirsch3, M A Landolt4, B Latal ([email protected])1 1.Child Development Center, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland 2.Department of Anesthesiology, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland 3.Department of Cardiology, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland 4.Department of Psychosomatics and Psychiatry, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland
Keywords Behaviour, Child, Congenital heart disease, Development, Motor Correspondence Beatrice Latal, MD MPH, Child Development Center, University Children’s Hospital Zurich, Steinwiesstrasse 75, 8032 Zurich, Switzerland. Tel: +41442667751 | Fax: +41442667164 | Email: [email protected] Received 10 November 2013; revised 11 February 2014; accepted 18 March 2014. DOI:10.1111/apa.12639
ABSTRACT Aim: Patients with congenital heart disease are at risk of neurodevelopmental deficits. Impairments in motor and behavioural function occur frequently, but no information is available concerning the coexistence of deficits in these two developmental domains. This study explored the occurrence of motor and behavioural deficits and their coexistence in children with surgically corrected congenital heart disease. Methods: Outcome was assessed in 95 children with congenital heart disease who had undergone cardiopulmonary bypass surgery. Their mean age was 9.6 years (SD 2.5). Motor function was assessed with the Zurich Neuromotor Assessment and behaviour with the Strength and Difficulties Questionnaire. Results: Children with congenital heart disease performed poorer in all motor domains compared with the reference population (all p ≤ 0.001). Behaviour was affected in the domains ‘emotional symptoms’ and ‘hyperactivity/inattention’ (both p < 0.01), and 54% of the children with motor abnormalities showed behavioural deficits. Children with coexistent abnormalities in behaviour and motor function had higher rates of remedial school services and therapeutic support. Conclusion: Children with congenital heart disease are at risk of long-term motor and behavioural problems, and there is a high rate of coexistence of problems in both domains. Early and longitudinal assessment of all developmental domains is necessary to provide adequate early support.
INTRODUCTION Congenital heart disease occurs in about six per 1000 live births (1). As survival of children with congenital heart disease after bypass surgery has increased (2), the long-term outcome has become the focus of interest. It has been shown that patients with congenital heart disease are at increased risk of brain injury and subsequent neurodevelopmental deficits (3,4). Fine and gross motor impairments occur in 22.1% and 23.4%, respectively, of school-age children with surgically corrected transposition of the great arteries (5). There is evidence that motor difficulties may persist into adolescence (4). Furthermore, studies have described behavioural deficits such as hyperactivity and attention problems (6–8), which are reported to occur in 5% and 41% of children who receive surgery for congenital heart disease. Thus, impairments in motor function and behavioural difficulties seem to occur frequently in the population of patients operated on for congenital heart disease. This has also been shown for other at-risk populations. For example, around 40% of children born very preterm face motor difficulties at school age (9). These motor difficulties can be associated with poorer
752
performance in reading, spelling and arithmetic (10). Behavioural problems, such as attention deficit hyperactivity disorder, occur two to three times as often in the preterm population than in healthy controls (11). Despite the high occurrence of motor and behavioural deficits in children operated on for congenital heart disease, there is no information about a potential association of
Key notes
Children with congenital heart disease who undergo cardiopulmonary bypass surgery are at risk of neurodevelopmental impairments. Our study found that motor and behavioural problems are twice as frequent in children with congenital heart disease as in healthy controls. More than half of all children with congenital heart disease and impaired motor function showed coexisting behavioural problems and required significantly more educational and therapeutic support than children with just impaired motor function.
©2014 Foundation Acta Pædiatrica. Published by John Wiley & Sons Ltd 2014 103, pp. 752–758
Liamlahi et al.
deficits in those two developmental domains in patients with congenital heart disease. This is of significance as a coexistence of disturbances in motor function, and behaviour is known to occur in the normal population and, in particular, very frequently in children with attention deficit hyperactivity disorder (12). There is evidence that motor dysfunction is associated with a variety of behavioural and social difficulties (13). This is supported by a study on children with developmental coordination disorder, where over 50% of these children demonstrated behavioural problems (14). Furthermore, it has been shown that individuals with developmental coordination disorder and attention deficit hyperactivity disorder show a worse outcome when it comes to psychosocial functioning and educational level than individuals with just attention deficit hyperactivity disorder (15). We therefore aimed to characterise the occurrence of motor problems and behavioural deficits as well as their coexistence in a cohort of school-age children after bypass surgery for congenital heart disease. We hypothesised that children with congenital heart disease not only demonstrate motor problems or behavioural deficits, but that these impairments often coincide and are significantly associated with a higher need for therapeutic support.
METHODS Participants All patients with congenital heart disease who underwent full-flow cardiopulmonary bypass surgery at our institution between 1995 and 1998 were eligible for this retrospective cohort study. Patients younger than 7 years or older than 17 years at the time of assessment (2004–2006) were excluded. Further exclusion criteria were non-German speakers, premature birth ( > > < >
Normal population n = 930
p value*
Patients n = 95
7.7% 6.6% 9.8% 7.0% 7.1% 7.7%
0.003 0.76 0.35 0.18 0.77 0.001
24.2% 22.1% 24.2% 17.9% 12.6% 30.5%
> > > > < >
Normal population n = 930
p-value*
14.1% 15.3% 14.7% 13.3% 15.6% 18.4%
0.005 0.065 0.009 0.187 0.42 0.002
Bold values denote statistical significance. *Chi-square test. **The domains emotional symptoms, conduct problems, hyperactivity/inattention and peer relationship problems are added together to generate the total score.
Neuromotor performance of children with congenital heart disease was poorer than norms in all domains (Table 3). Children without cerebral palsy also performed poorer than the reference population; this was significant for the gross motor and static balance component (p < 0.001). The neurological severity score correlated significantly with all ZNA components (Spearman’s Rho and p value: pure motor 0.41, p < 0.001; adaptive fine motor 0.35, p = 0.001; adaptive gross motor 0.34, p = 0.001; static balance 0.23, p = 0.03). Twenty-four (25%) children performed below the 10th percentile in two or more ZNA components. This was significantly more than in the reference population where one per cent of all children performed below the 10th percentile in two or more components (p < 0.001) (19). When looking at parent-reported physical activities, there was no difference between those with motor difficulties (p < 10 in two or more components of the ZNA) compared with those without motor difficulties (64% versus 68%, p = 0.85). Association between behavioural and motor outcome We examined the percentage of children who performed below the 10th percentile in two or more ZNA components (n = 24) and who additionally were scored borderline or abnormal in the SDQ (Table 4). The rate of coexistence was highest for the domain ‘emotional symptoms’.
Table 3 Motor outcome assessed with the Zurich Neuromotor Assessment Component Pure motor Adaptive fine motor Adaptive gross motor Static balance
Z-Score Median (range)
p-value*
REGULAR ARTICLE
Motor dysfunction and behavioural problems frequently coexist with congenital heart disease in school-age children R Liamlahi1, M von Rhein1, S B€uhrer2, E R Valsangiacomo Buechel3, W Knirsch3, M A Landolt4, B Latal ([email protected])1 1.Child Development Center, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland 2.Department of Anesthesiology, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland 3.Department of Cardiology, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland 4.Department of Psychosomatics and Psychiatry, University Children’s Hospital Zurich and Children Research Center Zurich, Zurich, Switzerland
Keywords Behaviour, Child, Congenital heart disease, Development, Motor Correspondence Beatrice Latal, MD MPH, Child Development Center, University Children’s Hospital Zurich, Steinwiesstrasse 75, 8032 Zurich, Switzerland. Tel: +41442667751 | Fax: +41442667164 | Email: [email protected] Received 10 November 2013; revised 11 February 2014; accepted 18 March 2014. DOI:10.1111/apa.12639
ABSTRACT Aim: Patients with congenital heart disease are at risk of neurodevelopmental deficits. Impairments in motor and behavioural function occur frequently, but no information is available concerning the coexistence of deficits in these two developmental domains. This study explored the occurrence of motor and behavioural deficits and their coexistence in children with surgically corrected congenital heart disease. Methods: Outcome was assessed in 95 children with congenital heart disease who had undergone cardiopulmonary bypass surgery. Their mean age was 9.6 years (SD 2.5). Motor function was assessed with the Zurich Neuromotor Assessment and behaviour with the Strength and Difficulties Questionnaire. Results: Children with congenital heart disease performed poorer in all motor domains compared with the reference population (all p ≤ 0.001). Behaviour was affected in the domains ‘emotional symptoms’ and ‘hyperactivity/inattention’ (both p < 0.01), and 54% of the children with motor abnormalities showed behavioural deficits. Children with coexistent abnormalities in behaviour and motor function had higher rates of remedial school services and therapeutic support. Conclusion: Children with congenital heart disease are at risk of long-term motor and behavioural problems, and there is a high rate of coexistence of problems in both domains. Early and longitudinal assessment of all developmental domains is necessary to provide adequate early support.
INTRODUCTION Congenital heart disease occurs in about six per 1000 live births (1). As survival of children with congenital heart disease after bypass surgery has increased (2), the long-term outcome has become the focus of interest. It has been shown that patients with congenital heart disease are at increased risk of brain injury and subsequent neurodevelopmental deficits (3,4). Fine and gross motor impairments occur in 22.1% and 23.4%, respectively, of school-age children with surgically corrected transposition of the great arteries (5). There is evidence that motor difficulties may persist into adolescence (4). Furthermore, studies have described behavioural deficits such as hyperactivity and attention problems (6–8), which are reported to occur in 5% and 41% of children who receive surgery for congenital heart disease. Thus, impairments in motor function and behavioural difficulties seem to occur frequently in the population of patients operated on for congenital heart disease. This has also been shown for other at-risk populations. For example, around 40% of children born very preterm face motor difficulties at school age (9). These motor difficulties can be associated with poorer
752
performance in reading, spelling and arithmetic (10). Behavioural problems, such as attention deficit hyperactivity disorder, occur two to three times as often in the preterm population than in healthy controls (11). Despite the high occurrence of motor and behavioural deficits in children operated on for congenital heart disease, there is no information about a potential association of
Key notes
Children with congenital heart disease who undergo cardiopulmonary bypass surgery are at risk of neurodevelopmental impairments. Our study found that motor and behavioural problems are twice as frequent in children with congenital heart disease as in healthy controls. More than half of all children with congenital heart disease and impaired motor function showed coexisting behavioural problems and required significantly more educational and therapeutic support than children with just impaired motor function.
©2014 Foundation Acta Pædiatrica. Published by John Wiley & Sons Ltd 2014 103, pp. 752–758
Liamlahi et al.
deficits in those two developmental domains in patients with congenital heart disease. This is of significance as a coexistence of disturbances in motor function, and behaviour is known to occur in the normal population and, in particular, very frequently in children with attention deficit hyperactivity disorder (12). There is evidence that motor dysfunction is associated with a variety of behavioural and social difficulties (13). This is supported by a study on children with developmental coordination disorder, where over 50% of these children demonstrated behavioural problems (14). Furthermore, it has been shown that individuals with developmental coordination disorder and attention deficit hyperactivity disorder show a worse outcome when it comes to psychosocial functioning and educational level than individuals with just attention deficit hyperactivity disorder (15). We therefore aimed to characterise the occurrence of motor problems and behavioural deficits as well as their coexistence in a cohort of school-age children after bypass surgery for congenital heart disease. We hypothesised that children with congenital heart disease not only demonstrate motor problems or behavioural deficits, but that these impairments often coincide and are significantly associated with a higher need for therapeutic support.
METHODS Participants All patients with congenital heart disease who underwent full-flow cardiopulmonary bypass surgery at our institution between 1995 and 1998 were eligible for this retrospective cohort study. Patients younger than 7 years or older than 17 years at the time of assessment (2004–2006) were excluded. Further exclusion criteria were non-German speakers, premature birth ( > > < >
Normal population n = 930
p value*
Patients n = 95
7.7% 6.6% 9.8% 7.0% 7.1% 7.7%
0.003 0.76 0.35 0.18 0.77 0.001
24.2% 22.1% 24.2% 17.9% 12.6% 30.5%
> > > > < >
Normal population n = 930
p-value*
14.1% 15.3% 14.7% 13.3% 15.6% 18.4%
0.005 0.065 0.009 0.187 0.42 0.002
Bold values denote statistical significance. *Chi-square test. **The domains emotional symptoms, conduct problems, hyperactivity/inattention and peer relationship problems are added together to generate the total score.
Neuromotor performance of children with congenital heart disease was poorer than norms in all domains (Table 3). Children without cerebral palsy also performed poorer than the reference population; this was significant for the gross motor and static balance component (p < 0.001). The neurological severity score correlated significantly with all ZNA components (Spearman’s Rho and p value: pure motor 0.41, p < 0.001; adaptive fine motor 0.35, p = 0.001; adaptive gross motor 0.34, p = 0.001; static balance 0.23, p = 0.03). Twenty-four (25%) children performed below the 10th percentile in two or more ZNA components. This was significantly more than in the reference population where one per cent of all children performed below the 10th percentile in two or more components (p < 0.001) (19). When looking at parent-reported physical activities, there was no difference between those with motor difficulties (p < 10 in two or more components of the ZNA) compared with those without motor difficulties (64% versus 68%, p = 0.85). Association between behavioural and motor outcome We examined the percentage of children who performed below the 10th percentile in two or more ZNA components (n = 24) and who additionally were scored borderline or abnormal in the SDQ (Table 4). The rate of coexistence was highest for the domain ‘emotional symptoms’.
Table 3 Motor outcome assessed with the Zurich Neuromotor Assessment Component Pure motor Adaptive fine motor Adaptive gross motor Static balance
Z-Score Median (range)
p-value*
