However, currently there is no one risk score that assess correctly the mortality of these procedures. Thus, we analyse the risk scores used at our institution.
We used area under the receiver operating characteristic curve c-index to measure model discrimination, and Hosmer-Lemeshow H-L statistic along with calibration plots to measure calibration. At present, there are few tools to address the case-mix adjustment for congenital cardiac surgery in adults.
The 2 former scores were designed to compare the complexity of different congenital cardiac surgeries. However, the latter is designed to evaluate in-hospital mortality for adults with acquired cardiac pathology.
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The purpose of this study is to validate these risk scoring systems in a Spanish cohort of adult patients with CHD. Individual patient consent was not necessary. The primary outcome was in-hospital mortality defined as death occurring during the surgical procedure or the first 30 days or postoperative hospitalization stay. Normal distribution of quantitative variables were analysed with the Kolmogorov-Smirnov test.
Continuous variables with normal distribution were expressed with mean and standard deviation.
Variables that did not have a normal distribution were expressed with median and interquartile range. Statistical significance was considered when the probability for a type I error alpha was lesser than 0. The performance of the 3 risk scores was compared using discrimination and calibration. Discrimination refers to the ability of the model to distinguish patients with the endpoint dead from patients without alive.
This is quantified with the concordance c statistic. Calibration on the other hand refers to agreement between observed endpoints and predictions, in other words, it measures the accuracy of a prediction.
Traditionally, Hosmer-Lemeshow H-L goodness-of-fit tests were performed to assess calibration. However, this test has several disadvantages; it doesn't measure the magnitude of miscalibration, deviations in calibration are sensitive to sample size and the test is dependant to arbitrary groupings. In our case the standard H-L test was complemented with calibration plots of observed events plotted against expected events.
These tests were repeated with the first half and the second half validation period 13 years of the cohort to detect Hawthorn effect. Missing data for calculation of the different risk score was considered as missing completely at random as these were due to inability to access patient history. Multiple imputation was used to handle missing data.
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Table 1 shows our demographic data. Six hundred and eight procedures were performed from May till June The overall in-hospital mortality was of 20 deaths 3. Nearly half About half Table 2 Description of in-hospital mortality cases Click for larger image Click for full table Download as Excel file. Table 3 Frequency of re-sternotomies Click for larger image Click for full table Download as Excel file. The different types of congenital anomalies needing surgical procedures are represented in Table 4.
The most frequent procedure were septal defect repairs However, the highest risk scores were in the thoracic arteries or veins, single ventricle and miscellaneous groups. A large proportion of our cohort had concomitant procedures, which increased the complexity of the cases and this is directly related to the surgical risk.
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Table 4 Main lesions needing surgical treatment with average risk scores Click for larger image Click for full table Download as Excel file. There were no missing data regarding mortality. In Figure 1 , we compare the AUC of the 3 risk scores. All 3 risk scores showed a fair discrimination without any statistically significant differences. Click for larger image Download as PowerPoint slide.
Figure 2 presents the calibration plots for the 3 scores. A perfect calibration has a calibration slope of 1, when the risk is overestimated the value is greater than 1 and vice versa in cases of underestimation.
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In Figure 3 , the Aristotle and Euroscore I are analysed stratified in risk categories, rather than the point system. RACHS-1 seems to slightly underestimate the in-hospital mortality, while Aristotle slightly overestimates it. Euroscore-I shows an important miscalibration. Figure 3 Calibration plots of Aristotle 1—5 and Euroscore I low, medium, and high stratified by risk categories.
When the Aristotle and Euroscore-I are grouped in risk categories both show an important miscalibration. No difference in calibration was detected in the first and second half 13 years of the validation period. However, there was an increase in the average risk scores during these 2 time spans.
During the last 13 years, despite the increase in risk complexity the mortality decreased. Our institution is a tertiary centre and since it is one of the national referral centres for CHD.
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We have a consolidated multidisciplinary unit taking care of adults with CHD with over two decades of experience. The volume load of this specific aspect of our speciality has been steadily increasing, from 20—25 cases per year in the first decade to over 50 cases per year in the last few years.
The complexity of the cases has also started to increase, 19 in the first decade septal defects were the most frequent cases referred to our centre. Nowadays, right sided lesions in reoperative scenarios like PVR and tricuspid valve repair after a Fallot repair is our daily bread and butter. There are around , adults with CHD in Spain. As our counterparts in acquired heart disease, it is necessary for us to have a specific risk score in order compare our outcomes with other institutions.
To our knowledge this is the first validation study in a retrospective Spanish cohort of adults with CHD. All three scoring systems fared marginally good for discrimination. When the scores were further stratified into risk categories, both Aristotle and Euroscore I performed poorly in calibration.
The Society of Thoracic Surgeons-European Association for Cardio-Thoracic Surgery mortality score was not used in this study because the number of missing values was too large to be addressed by imputation methods. Approximately half 10 cases of our hospital mortality were due to left cardiac lesions, with average RACHS-1 score of 3, Aristotle score of 7. All of the cases had concomitant procedures, either the aorta, septal defects or right side procedures and half of these cases were resternotomies.
The second most frequent in-hospital mortality group were right cardiac lesions 4 cases , with an average RACHS 1 score of 2. The case of a risk scoring system has always been a challenge in congenital heart surgery. Diagnosis and procedures are numerous, with small patient samples.
The situation of adult CHD has the added complexity of treating acquired cardiac diseases in patients with CHD of long evolution which has not been repaired or patients with prior surgeries which now also require a further intervention in order to repair residual lesions, complications or sequels derived from the first surgery. Thanks to the herculean efforts of scholars and data collectors do we have the RACHS-1 and Aristotle scoring systems in the paediatric arena.
But, variables contemplated in these score such as low weight, or prematurity do not account for the risk in these adult patients with acquired pathologies. On the other, hand Euroscore I fails to appreciate the nature of these patients, many with genetic disorders, which are prone to higher mortality and morbidity. The limitations of this study are its retrospective nature which spreads over 26 years.
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Although it is of a single institution and the surgical team has not changed, but management policies have varied and so has the case-mix.
In conclusion, there is a growing necessity of a specific risk score for adults with CHD, 1 , 3 modelled with the unique case-mix of this special cohort of patients in a prospective manner. Conflict of Interest: The authors have no financial conflicts of interest.
Korean Circ J. Published online May 07, Correspondence to Bunty K. Ramchandani, MD. Email: bunty.
Go to:. Congenital heart defect ; Validation studies. Outcomes The primary outcome was in-hospital mortality defined as death occurring during the surgical procedure or the first 30 days or postoperative hospitalization stay. Statistical analysis Normal distribution of quantitative variables were analysed with the Kolmogorov-Smirnov test.
Demographics Table 1 shows our demographic data. Missing data There were no missing data regarding mortality. Project administration: Ramchandani BK. Writing - original draft: Ramchandani BK. Chance of surgery in adult congenital heart disease. Eur J Prev Cardiol ;— Surgery in adults with congenital heart disease.
Circulation ;— Kogon B, Oster M. Assessing surgical risk for adults with congenital heart disease: are pediatric scoring systems appropriate?
J Thorac Cardiovasc Surg ;— Fuller S, Jacobs JP. Congenital heart surgery in adults: the challenge of estimation of risk of mortality. Mortality following congenital heart surgery in adults can be predicted accurately by combining expert-based and evidence-based pediatric risk scores.
Estimating mortality risk for adult congenital heart surgery: an analysis of the society of thoracic surgeons congenital heart surgery database. Ann Thorac Surg ;— Consensus-based method for risk adjustment for surgery for congenital heart disease.