| Background:children with severe forms of congential heart disease (CHD) requiring intervention in the neonatal period are surviving at levels thought to be highly improbable just two decades ago. Heralded by the availability of prostaglandin E1 in the late 1970s, improvements in technology (echocardiography and color Doppler in particular), intensive care, and intra-operative support have recently allowed surgeons to reconstruct the hearts of many children with otherwise lethal forms of CHD. In the past, much of the published literature focused on the cardiac sequelae in this population. However, increasing attention has recently been given to the neuro-developmental outcomes in this population of patients. In the last 20 years there have been huge improvements in the survival of children with congenital cardiac disease, especially those with more complex conditions. While initial medical interest focused on mortality and progressed to functional capacity, there is now increasing interest in and awareness of the need to address the psychosocial issues that may affect these children as they develop.A considerable number of studies have been conducted to assess the impact of ConHD on children’s and adolescents’psychological and cognitive functioning. The outcome measure that is used in the majority of these studies assessing psychological functioning is the Child Behaviour Checklist parent form. This is a parent report measure that provides an estimate of overall emotional and behavioural problems and of internalizing (e.g., anxiety, depression, social withdrawal) and externalizing behaviour problems (e.g., hyperactivity, oppositional behaviour, aggression). The outcome measure that is used in the majority of studies assessing overall cognitive functioning, verbal (VIQ; e.g., verbal comprehensive abilities) and performance intelligence quotients (PIQ; e.g., perceptual organizational abilities) is the Wechsler Intelligence Tests for Children. Consensus among studies assessing psychological and cognitive functioning in CHD has not been reached. Authors report higher rates of behaviour problems and reduced cognitive functioning among children and adolescents with CHD, whereas others report no significant differences between patients with CHD and a comparison group.The main studies recently have been focused in the cognitive ability of children with complex congenital heart disease (CHD) such as patients with transposition of the great arteries (TGA), total anomalous pulmonary venous connection (TAPVC) and tetralogy of fallot (TOF). The results showed that the mean full-scale intelligence quote (IQ) in the patients were in normal range (90～110) while the performance IQ were significantly lower than the verbal IQ and the proportion of mental retardation (IQ<70) were higher than the normal children. One of famous studies was Boston hypothermic circulatory arrest by Bellinger, that prospective eight years for 171 patients with TGA who underwent an arterial-switch operation by 3 months of age. The results show that the prevalence of abnormalities on Magnetic Resonance Imaging scans were 15 percent with definite abnormalities, which were judged to be mild in 86 percent of these children at one year age, 45% with definite abnormalities involving neurocognitive functions (eg, language, attention) or motor functions (eg, balance, hopping),24% with full-scale IQ score proximately 0.5 standard deviate below the expected population mean and the group with marked difficulties on test of expressive language, visual-spatial and visual-motor skills and 24% with diagnostic criteria for oromotor apraxia at four years age. Above 60% patients with neurologic abnormalities,38% with lower performance IQ than verbal IQ scores,3% with mental retardation(IQ<70) and a part of patients with poorer attention ability, higher abnormal impulsive response, and more learning difficulty and behavioral problems at eight years age. The TGA patients with VSD had poorer cognitive abilities than the patients without VSD at four and eight years age respectively. Because the objective of the cohort researches were to study developmental and neurological status of patients with TGA after heart surgery with hypothermic circulatory arrest or low-flow cardiopulmonary bypass, all the results were compare to the norms, without compared to the same age normal children. Karl reported the neuro-cognitive ability in 74 patients with TGA. The results showed IQ were in normal range but were still lower than their best friends and part of patients had poor speech ability and heavier behavior problems.It has been suggested that patients with more severe heart disease are exposed to more detrimental factors such as diminished cerebral perfusion, difficult surgery, and physical impairments. Therefore, the different findings among studies could be explained by the fact that patients with severe heart disease are at increased risk for psychological and cognitive problems. In the literature, however, no consensus has been reached on the effect of disease severity on psychological and cognitive functioning. That is, some authors report worse psychological and cognitive functioning in more severe CHD whereas others do not.ventricular septal defects are the most common form of congenital heart disease and the most frequently seen congenital abnormalities. Surgical closure of a VSD is the most common open-heart procedure performed in paediatrics, Approximately 1 of 500 infants is born with a VSD, approximately 25% of all cases of congenital heart disease, The postoperative course is relatively free of complications or residual hemo-dynamic problems and generally leaves patients with normal cardiac physiology and expectations for typical development.Surgical repair was the traditional treatment, which was considered as the gold standard. But it was associated with morbidity and mortality, discomfort, sternotomy, and skin scar. Therefore, trans-catheter approach has been adopted as an alternative approach to surgery. Recently, this approach has been more promising for a routine use with the introduction of the Amplatzer family devices and complications are limited. One of the most complications seems to be complete atrioventricular block (cAVB) related to the device in peri-membranous VSD.There were fewer reports of cognitive ability in patients with VSD. Jonathan’s study found the patients who underwent repair of VSD at less than 6 months had mental development IQ scores 92.6 with 3% patients scoring 70 or less and psychomotor development index 86.1 with 18% patients scoring 70 or less.18% patients had microcephaly at the time of testing, of these patients,40% had microcephaly at birth.By now, intelligence quote test were main research way in cognition ability of patients with VSD.Objective:1. The effect on event-relate potentials (ERP) in patients with VSD after surgical or transcatheter different intervention ways.2. The effect on continuous performance test (CPT) in patients with VSD after surgical or transcatheter different intervention ways.3. The influence on Halstead-Reitan Battery Test (HRB) in patients with VSD after surgical or trans-catheter different intervention ways.4. The results of Benton Visual Retention Test (BVRT) in patients with VSD after surgical or trans-catheter different intervention ways.5. The behavior problems and relation factors in patients with VSD after surgical or trans-catheter different intervention ways.6. The results of IQ in patients with VSD after surgical or trans-catheter different intervention ways.Methods:We recruited patients who underwent surgery or trans-catheter closure of VSD at Provincial Hospital affiliated with Shandong University (Jinan, Shandong Province) between January 2002 and December 2005. Eligibility criteria included 1) birth between January 1995 and December 2002 for study age range 6-13 years; 2) restrictive VSD and left-to-right shunt <50%; 3) defect in muscular or peri-membranous part of the septum; 4) similar family demographic characteristics (socioeconomic status, parents’education level) in both patient groups (the Hollingshead Four Factor Index of Social Statuswas used to determine family socioeconomic status according to parental occupation and education) and 5) no associated anomalies, congestive heart failure or pulmonary hypertension before surgery. We screened 47 patients in the surgery group and 56 patients in the trans-catheter group; 31 patients in the surgery group were included and 35 in the trans-catheter group. Two children in the surgery group did not finish the test because of poor cooperation. Every patient was matched with their best friend in age and sex, but 6 subjects in the trans-catheter group and 4 in the surgery group did not bring their best friend for an interview. In all,56 "best-friend" control subjects were available for this study. All subjects returned to the Provincial Hospital affiliated with Shandong University for a day-long series of assessments, including ERP, echocardiography(ECG), CPT, BVRT, HRB, IQ, CBCL (parents report), and neurological examination, between June 2008 and January 2009.Statistical analysis was performed using SPSS 16.0 for windows with a significance level of 0.05 and presented as mean±SD. The differences between groups were analysed using ANOVA with post hoc testing. Comparisons between two groups were performed by Student’s t test for parametric data.Results:1. Patient characteristics47 patients in the surgery group and 56 patients in the trans-catheter group were screened; 31 patients in the surgery group were included and 35 patients in the trans-catheter group. The children who returned for evaluation did not differ significantly from those who did not return with respect to sociodemographic factors, intraoperative perfusion variables, or preoperative and postoperative neurological status in the surgery and trans-catheter groups. The 3 groups (surgery, trans-catheter and healthy controls) did not differ in demographic characteristics. The 2 treatment groups did not differ in VSD size or location or median age at intervention. The duration of repair time and hospitalization was significantly greater for surgical than trans-catheter patients.2. Intellectual Ability (IQ Scores)At IQ assessment, the patients with VSD were more likely than their best friends. Mean Full-scale IQ (surgery group vs their best friends 105.2±16.2 vs 110.5±10.9; transcatheter group vs theirbest friends 104.2±15.3 vs 108.6±14.6), Verbal IQ (surgery group vs their best friends 99.3±12.8 vs 106.4±16.2; transcatheter group vs theirbest friends 100.6±12.4 vs 110.5±18.2)and Performance IQ(surgery group vs their best friends 108.2±11.6 vs 115.2±13.6; transcatheter group vs theirbest friends 106.5±14.9 vs 106.7±14.6) were not significantly different between the VSD patients and their best friends in the operation group and tans-catheter group respectively. Also there were not difference in patients between the surgery operation group and the trans-catheter group.3. P300 auditory-evoked potentials3.1 P300 peak latency and amplitude in VSD patients and controlsAll subjects could concentrate on target stimuli. The mean P300 latencies in Fz and Cz leads was longer for patients with VSD (329.2±24.8 and 335±20.0 ms) than for healthy controls (319.1±20.6 and 313±18.2 ms). However, the P300 peak latency in the Pz lead was comparable between patients and controls (322.2±33 vs 318±26.6 ms). The P300 amplitude in Fz and Cz leads was lower for patients (9.6±2.90 and 10.0±3.0μv) than controls (11.0±3.92 and 13.00±2.66μv [P<0.05]). Patients and controls did not differ in P300 amplitude in the Pz lead (10.6±2.97 vs 11.2±3.38μv, P>0.05).3.2. P300 latency and amplitude between two group patientsP300 peak latency in Fz (339±29.0 vs 315±175ms, P<0.05) and Cz leads (345±27.3 vs 312±17.3ms, P<0.05) was longer and amplitude in the Cz lead (10.1±3.31 vs 13.1±2.54 uv, P<0.05) was lower for surgery patients than for healthy controls (p <0.05). In the Cz lead, P300 peak latency was longer (328±30.8 vs 314±10.8, P<0.05) and amplitude lower (9.9±2.7 6 vs 13.0±2.66 uv,P<0.05)for trans-catheter patients than healthy controls. Patients who underwent surgery had longer P300 latency in Fz and Cz leads than did patients with trans-catheter closure (p<0.05). However, the 2 groups did not differ in amplitudes (p>0.05).3.3 Univariate and multivariate regression analysis of factors related to P300 latency and amplitudeBy univariate analysis, increased test age was associated with long P300 peak latency in Fz, Cz and Pz leads in patients (P<0.05) (Table 3). Long duration of CPB and aortic cross-clamping was associated with long P300 peak latency in Fz and Cz leads (P<0.05). Use of CPB and aortic cross-clamping was associated with long P300 peak latency in the Fz lead (P<0.05). Long stay in the intensive care unit (ICU) was associated with long P300 peak latency in Fz and Cz leads. However, VSD size, age at surgery and surgery time were not associated with P300 peak latencies or amplitudes (P>0.05). On multivariate analysis, long duration of CPB and aortic cross-clamping predicted long P300 peak latency in Fz and Cz leads. However, the use of aortic cross-clamping and duration of ICU stay was not associated with P300 peak latency or amplitude.4. Continuous performance test in patients with VSDWhen the patients as a cohort, the VSD patients had lower auditory persistence quote (93.8±11.2 vs 104±12.1, P<0.05) and visual persistence quote (96.9±10.3 vs 104±11.9, P<0.05) than the control group, the patients in trans-catheter intervention group had lower auditory persistence quote (93.3±12.4 vs 104.6±18.4, P<0.05) and visual persistence quote (97.8±18.4 vs 112.3±13.5, P<0.05) than control group, while patients in surgery groups had lower auditory persistence quote(94.4±16.3 vs 103.2±17.0, P<0.05), auditory speed quote (92.8±16.3 vs 99.9±8.11, P<0.05), visual persistence quote (95.3±27.4 vs 110.0±12.4, P<0.05) and, visual persistence speed (97.4±9.3 vs 104.0±11.0, P<0.05) than their best friends. The difference were significant in auditory sense motor quote(93.6±21.2 vs 100.9±5.7, P<0.05), visual prudence quote (89.4±22.7 vs 99.3±16.5, P<0.05) between the two group patients.5. Halstead-Reitan Battery Test in VSD patients.5.1 Category testThe error scores were neither significantly different between the patients and the control group, nor between two group patients.5.2 Tactual performance Test (TPT) in VSD patientsThe time of TPT has not significant different by dominant hand between the patients and controls, while the time of TPT were more in the surgery patients (468.4±223.4s) than the trans-catheter patients (380.9±153.1s) and the control groups (336.4±218.4 s) by non-dominant hand, the difference were significance.The data of memory location in TPT were significantly poorer in the surgery patients (3.26±2.5) than the trans-catheter patients (4.8±1.7) and the control groups (4.7±2.3), while there was no difference between the trans-catheter patients and the controls.5.3 Rhythm Test in VSD patientsThe test had 30 items and divided into A, B, C three parts. All the patients finished the music rhythm test. The numbers of correct in the test of part A were not significant difference between the patients and the control groups. But in part B and C, the correct data were significantly lower in VSD patients (6.67±1.7,4.58±3.4) underwent surgery operation than in patients suffered trans-catheter intervention (7.69±1.8,6.04±2.1) and the control groups (7.30±1.1,7.78±1.3), the trans-catheter patients had lower number of correct than their best friends (6.04±2.1 vs 7.33±2.0, P<0.05) only in the C parts, the difference were significant.5.4 finger tapping test in VSD patientsThe finger tapping test had five ten-second parts. The results showed that the mean tapping times in whole test were not significantly different between the VSD patients and the controls. In the last ten-second part, the surgery patients’tapping times were less than the control group (34.4±20.7 vs43.7±17.5, P<0.05)5.5 Grip strength test in VSD patientsThe result showed that children with VSD and without VSD patients had similar grip strength of the dominant and non-dominant hand. Also it is between the two group patients.6. Benton Visual Retention Test (BVRT) in VSD Patients6.1 The raw scores for sum correct of Benton Visual Retention TestThe raw scores for sum correct of BVRT of form B, form C and form D were not significantly different among the surgery patients (8.9±2.8,8.5±3.2 and 5.8±4.7),, trans-catheter patients (8.6±3.1,7.8±4.1 and 7.6±7.3) and the control group (9.0±2.1,8.1±4.5 and 7.0±4.3), by ANOVA analyse.6.2 The raw scores for sum error of BVRTThe results showed that it were neither significantly different in the scores for sum error of BVRT of form B and C between the surgery patients and their best friends nor the trans-catheter patients and their best friends. But the error scores of omission (1.20±0.90 vs 0.80±0.50, P<0.05), degeneration(2.30±0.99 vs 1.45±0.76, P<0.05) and rotation(2.02±1.02 vs 1.50±0.92, P<0.05) in form D were significantly higher in the surgery patients than their best friends. The difference did not exist between the trans-catheter patient and their friends.7. Achenbanch’s Child Behavior Checklist by patents in VSD patients.Sixty-four VSD patients and their fifty-five best friend as the control group were interested with Achenbanch’s Child Behavior Checklist. The checklist was answered by mother (66%) and the father (34%). There was not significantly different between the patients and controls.7.1 The raw score of CBCL in VSD patientsThe subscale scores of depression(4.45±2.1 vs 2.18±1.07, P<0.05), somatic problems(3.95±1.32 vs 1.03±0.78, P<0.05) and internalizing(13.12±11.26 vs 6.02±5.56, P<0.05) were higher in the VSD boys than their best friends, the girls’scores of depression(5.64±2.30 vs 2.57±1.42, P<0.05), compulsory (3.85±1.67 vs 1.76±1.13, P <0.05), social withdrawal(4.23±1.23 vs 2.11±1.65, P<0.05)and internalizing(14.2±10.53 vs 7.80±5.69, P<0.05)were higher in the VSD patients than their best friends. Neither total scores nor externalizing scores were significantly different between in the patients and the controls.7.2 The rate of behavior problems in VSD patientsThe patient had abnormal behavior if his raw scores of subscale were higer than the 98 percents of cheses norm data of CBCL. The rate of behavior problems in the subscales of depression; social regression and somatic problems were significantly higher in VSD patients underwent surgery operation than the controls, but it was not existed between the tans-catheter intervention patients and the controls.7.3 Logistic regression analysis the behavior problems and related factorsThe result showed that the factors of age of intervention, the scar of post-operation, CAVB and surgery operation were influenced on the rate of behavior problems in VSD patients.Conclusion:1. The VSD patients had normal IQ scores.2. When doing the average works, there were not difference between the patients and the controls. But the ability of VSD patients was poorer than the controls when doing the complex or difficult work. Some respects of cognitive ability were poorer in the patients underwent surgery operation than the trans-catheter patients, these maybe interrelated to the longer CPB times and aortic-clamp times.3. The raw scores of internalizing behavior (depression; social withdrawal) were higer in VSD patients. The VSD patients in surgical operation had higher behavioural problems.4. The surgery operation patients had poorer cognitive function in some aspects and behavioural problems than the transcatheter VSD patients. The treatment way of transcatheter instead of sugery operation can alleviate the patients’cognitive impairment and had less behavior problems.Innovations and meanings1. Ventricular septal defect is one of the most common congenital heart defects. But the studies of cognitive function in VSD patients were few. In the area, most study focus on the complicated heart defects or a cohort of congenital heart defects, not merely a kind of congenital heart defects, all kind of confounding factors led to the bias of results. Our study was focus on the patients with ventricular septal defect.2. It is the first time to explore the cognitive functions in patients with ventricular defect by the transcatheter closure or surgery operation. The different intervention ways (tans-catheter closure or surgery operation) influenced on the cognitive function in VSD patients.3. Various aspects of cognitive function in VSD patients were test including intelligence test, event related potentials, continuous performance test, Benton visual retention test and Halstead-Reitan Battery Test... |
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