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J Thorac Cardiovasc Surg 2007;133:905-911
© 2007 The American Association for Thoracic Surgery

Surgery for Congenital Heart Disease

Early childhood health, growth, and neurodevelopmental outcomes after complete repair of total anomalous pulmonary venous connection at 6 weeks or younger

Gwen Y. Alton, RN, BSNa,c, Charlene M.T. Robertson, MD, FRCP(C)a,c,*, Reg Sauve, MD, FRCP(C)d, Abhay Divekar, MD, FRCP(C)f, Alberto Nettel-Aguirre, PhDd,e, Sharon Selzer, RNg, Ari R. Joffe, MD, FRCP(C)a, Ivan M. Rebeyka, MD, FRCS(C)a,b, David B. Ross, MD, FRCS(C)b Western Canadian Complex Pediatric Therapies Project Follow-Up Group*

a Department of Pediatrics, University of Alberta, Edmonton, Alberta, Canada
b Department of Surgery, University of Alberta, Edmonton, Alberta, Canada
c Neonatal and Infant Follow-up Clinic, Glenrose Rehabilitation Hospital, Edmonton, Alberta, Canada
d Department of Pediatrics, University of Calgary, Calgary, Alberta, Canada
e Research Methods Team, Faculty of Medicine, University of Calgary, Calgary, Alberta, Canada
f Division of Pediatric Cardiology, University of Manitoba, Winnipeg, Manitoba, Canada
g Department of Pediatrics, General Hospital, Regina, Saskatchewan, Canada.

Received for publication September 27, 2006; revisions received November 22, 2006; accepted for publication December 13, 2006.

* Address for reprints: Charlene M. T. Robertson, MD, FRCP(C), Room 242A Glen East, Glenrose Rehabilitation Hospital, 10230 111 Ave, Edmonton, Alberta, Canada T5G 0B7. (Email: croberts{at}cha.ab.ca).


   Abstract
Top
 Abstract
Introduction
 Materials and Methods
 Results
 Discussion
 Conclusions
 References
 
Objective: This interprovincial inception cohort study explores early childhood outcomes and their operative and perioperative predictors after total anomalous pulmonary venous connection repair, simple (patent ductus arteriosus, atrial septal defect, or both) or complex (any associated major cardiac anomalies).

Methods: From 1996 through 2004, a total of 41 consecutive neonates with total anomalous pulmonary venous connection underwent complete repair with deep hypothermic circulatory arrest. Multidisciplinary health and neurodevelopmental outcomes (Bayley Scales of Infant Development II [1993], Mental and Psychomotor Developmental Indices) were assessed at 18 to 24 months of age. Regression analyses explored potentially modifiable predictors of outcome.

Results: Survival after simple total anomalous pulmonary venous connection repair was 31 of 32 (97%); that after complex repair was 3 of 9 (33%). Relative risk of death was higher for complex (21.3) and obstructive (8.4) total anomalous pulmonary venous connections. Those who died had longer deep hypothermic circulatory arrest times (P < .001). For 34 survivors, Mental Developmental Index was 87 ± 16 and Psychomotor Developmental Index was 89 ± 13. Among survivors, 24.6% of variability in Mental Developmental Index was explained by its linear relationship with socioeconomic status, whereas 35.5% of variability in Psychomotor Developmental Index was explained by its linear relationship with weight at surgery and highest plasma lactate level after surgery. North American native peoples made up 38% of survivors, a higher than expected number.

Conclusion: Mean developmental scores were in the low average range of normative data. Socioeconomic factors predicted mental outcome. High postoperative lactate level on day 1 predicted low motor scores. Other potentially modifiable variables will need to be explored. Further epidemiologic study relating race to occurrence of total anomalous pulmonary venous connection is suggested. Early neurodevelopmental assessment is recommended.

Abbreviations and Acronyms MDI = Bayley Scales of Infant Development II Mental Developmental Index; PDI = Bayley Scales of Infant Development II Psychomotor Developmental Index; TAPVC = total anomalous pulmonary venous connection


   Introduction
Top
 Abstract
 Introduction
Materials and Methods
 Results
 Discussion
 Conclusions
 References
 
Total anomalous pulmonary venous connection (TAPVC) accounts for approximately 2% of congenital heart disease.1-4Go Reported mortality for simple TAPVC is 4.9% to 6.8%,5,6Go whereas that for complex TAPVC with other associated cardiac anomalies is greater than 50%.7Go Survivors after simple TAPVC repair have good long-term school attendance and employment.8Go Parental reports suggest good general health, with 69% of survivors having average or above school performance.9Go School-aged assessments report full-scale and verbal intelligence to be similar to population norms, but survivors have reduced performance scores, visual–motor integration, and fine motor skills.10Go Little has been published about early childhood neurodevelopmental outcomes after TAPVC repair.

There is increasing awareness of the importance of assessing neurodevelopmental outcomes after neonatal cardiac surgery for complex congenital heart disease, and mechanisms for adverse outcomes have been proposed.11Go Assessments after neonatal cardiac surgery allow standardized results of neurodevelopmental outcomes and identification of those needing early childhood educational interventions.12Go

The objectives of this study were to describe the 18- to 24-month health, growth, and neurodevelopmental outcomes for all infants 6 weeks or younger undergoing intracardiac repair for simple (patent ductus arteriosus, atrial septal defect, or both) or complex (other associated major cardiac anomalies) TAPVC and to explore the proportions of variability in outcomes explained by demographic, operative, and perioperative variables.


   Materials and Methods
Top
 Abstract
 Introduction
 Materials and Methods
Results
 Discussion
 Conclusions
 References
 
This interprovincial inception cohort outcomes study was conducted in three provinces in Western Canada. As previously described,13,14Go from September 1996 through August 2004, infants 6 weeks or younger were identified at time of surgery for TAPVC (simple and complex). Surgery was performed at the Stollery Children’s Hospital, Edmonton, Alberta, Canada. Although reporting outcomes for those with simple TAPVC only would provide a cleaner study, there is need for information about those with complex abnormalities. Data for both simple and complex TAPVC are therefore given.

The operative management for the repair of TAPVC at our institution during this time was to cool the patient’s blood to 20°C or lower for a minimum of 20 minutes before deep hypothermic circulatory arrest. We report here both the lowest blood temperature immediately before deep hypothermic circulatory arrest (in °C) and the lowest rectal temperature on cardiopulmonary bypass for longer than 10 minutes (also in °C). A modified pH-stat cooling strategy was used. The hematocrit during cooling for this cohort was greater than 0.20L, whereas current levels are greater than 0.25L. Preoperative, intraoperative, and postoperative variables that had been agreed on previously were collected prospectively and have been described elsewhere.13,14Go Of particular interest was plasma lactate level as an indicator of outcome; levels were regularly measured twice daily as part of clinical management as well as obtained routinely with blood gas values. Long-term follow-up was discussed with parents or guardians once survival was probable. With appropriate consent, contact was made with the respective follow-up clinic at the tertiary site of origin.

Subjects
All 41 consecutive patients admitted with TAPVC from September 1996 through August 2004—28 (68%) boys, 28 (68%) referred from out of region, and 14 (34%) North American native peoples—were registered. There were no exclusions to enrollment. All 34 survivors (31 with simple TAPVC and 3 with complex) received multidisciplinary neurodevelopmental assessments through existing neonatal follow-up clinics: 25 (74%) in Calgary, Alberta, Regina and Saskatoon, Saskatchewan, or Winnipeg, Manitoba, and 9 (26%) at the Neonatal and Infant Follow-up Clinic at the Glenrose Rehabilitation Hospital, Edmonton, Alberta. Ethics board approvals were obtained from each site before onset of the study. All parents or guardians signed individual consent forms.

Early Childhood Assessments
Outcomes assessment was completed at 18 to 24 months (mean 21 months). At assessment, a research nurse recorded history of hospitalizations, illnesses, medication use, and need for supplemental oxygen. Physical measurements were obtained as described previously.13,14Go The family socioeconomic status was determined according to the Blishen Index, a formula that considers the relative income, education requirement, and prestige factor of employment with a population mean (± SD) of 43 ± 13.15Go Maternal education was indicated by years of schooling. Race was coded according to National Institutes of Health specifications.16Go Pediatricians experienced in neurodevelopmental follow-up examined each child for evidence of cerebral palsy17Go or visual impairment, defined as corrected visual acuity in the better eye worse than 20/60.13,14Go Hearing was evaluated by experienced certified pediatric audiologists in soundproof environments as described elsewhere.13,14Go Hearing impairment was defined as binaural or bilateral sensorineural hearing loss greater than 40 dB at any frequency from 250 to 4000 Hz.13,14Go Certified pediatric psychologists and psychometrists administered The Bayley Scales of Infant Development II,18Go a widely accepted standardized outcome measure used in neonatal follow-up clinics that yields separate Mental Developmental Index (MDI) and Psychomotor Developmental Index (PDI) scores, each with a mean of 100 and SD of 15. Developmental indices lower than 70 (2 SD below mean) indicated mental or motor delay.

Statistical Analysis
Descriptive variables for outcomes were analyzed with the univariate t test, {chi}2 test, and Fisher exact test (2-sided) analyses. Bonferroni correction was applied. The relative risk for death was calculated. Sequential stepwise multiple regressions for variables from each of the five stages—preoperative, operative, postoperative day 1, postoperative days 2 to 5, and postoperative days 6 and after—were used to explore the overall greatest proportion of outcome explained by a combination of predictors to a significance level of .05. Because numbers of survivors with complex TAPVC were small, analysis of variables predictive of outcome for this group could not be completed separately. SPSS version 12.0 for Windows (SPSS, Inc, Chicago, Ill) was used for analyses.


   Results
Top
 Abstract
 Introduction
 Materials and Methods
 Results
Discussion
 Conclusions
 References
 
Of the 41 children, 32 had simple TAPVC (14 supracardiac, 15 infracardiac, 1 cardiac, and 2 mixed; 11 obstructive and 21 not obstructed) and 9 had complex TAPVC (3 supracardiac, 4 infracardiac, and 2 mixed; 6 obstructive and 3 not obstructed). Table E1 gives the anatomic diagnoses of the 9 infants with complex TAPVC.


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  		TABLE E1 Anatomic diagnoses of the 9 infants with complex total anomalous pulmonary venous connection
 
The anatomic and operative variables for all 41 infants undergoing repair (32 simple, 9 complex) at 6 weeks or younger are given in Table 1. The relative risks for death among the 41 children were as follows: complex TAPVC, 21.3, and obstructive anatomy, 8.4. Of the 7 children who died, all were boys, 6 had complex TAPVC, 6 had obstruction, 2 had supracardiac TAPVC, 3 had mixed TAPVC, and 6 received postoperative extracorporeal membrane oxygenation. The child with simple TAPVC who died had associated bilateral pulmonary artery stenosis and disseminated intravascular coagulation. Of the patients with complex TAPVC, 4 died with multiple organ dysfunction, 1 with septic shock, and 1 with pulmonary hypertension. Six died in the hospital at 8 to 144 days post-operatively; the single late out-of-hospital death occurred at 63 postoperative days.


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  		TABLE 1 Descriptive anatomic and operative characteristics of 41 infants 6 weeks or younger undergoing complete repair for total anomalous pulmonary venous connection
 
Table E2 shows the descriptive variables for the 34 surviving children, 31 with simple TAPVC and 3 with complex. The mean ± SD levels of plasma lactate for all surviving children for the time periods were as follows: preoperatively, 2.5 ± 2.0 mmol/L (range 0.8-8.4 mmol/L); first 24 hours postoperatively, 6.2 ± 3.3 mmol/L (range 1.8-14.2 mmol/L); days 2 through 5 postoperatively, 3.5 ± 3.1 mmol/L (range 1.0-12.6 mmol/L); and day 6 and later postoperatively, 1.3 ± 0.45 mmol/L (range 0.1-2.5 mmol/L). The highest plasma lactate level for each child occurred within 24 hours after surgery, except for 1 child with a level of 8.4 mmol/L before surgery, normal levels postoperatively, and a normal outcome. There were 7 of 34 children (21%) with plasma lactate levels on day 1 elevated by at least 1 SD for that period (>9.5 mmol/L). After Bonferroni correction, no differences were seen for demographic, preoperative, operative, or postoperative variables. Children with a racial background described by National Institutes of Health classification16Go as of North American native peoples made up 13 (38%) of the 34 assessed. Of these 13 children, infracardiac anatomy occurred in 8 (61.5%) and obstruction in 5 (38.5%). There were no statistically significant differences, however, in anatomic, operative, or perioperative variables between North American native and white children.


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  		TABLE E2 Demographic, operative, and perioperative descriptors for surviving 34 infants after surgery at 6 weeks or younger for total anomalous pulmonary venous connection mean (SD), n (%)
 
Growth and health outcomes for 34 survivors are described in Table 2. No child had ongoing hypoxia or known episodes of desaturation. The majority of hospitalizations and doctor visits were for pulmonary illnesses. Race, socioeconomic status, mother’s schooling, and location of family home did not affect growth and health outcomes (data not shown).


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  		TABLE 2 Growth and general health outcomes at 18 to 24 months of 34 survivors in relation to anatomic complexity of heart defect after total anomalous pulmonary venous connection repair
 
There were no cases of cerebral palsy, visual impairment, or sensorineural hearing loss among the 34 survivors. No survivors were considered medically fragile, defined as requiring long-term hospitalization or in-home nursing care. One child with simple TAPVC had cat-eye syndrome; growth and developmental scores were not in the delayed range. No other child had a chromosomal abnormality or neurologic syndrome. Mean scores for the 34 survivors were as follows: MDI 87 ± 16 and PDI 89 ± 13. MDI scores did not differ in relation to TAPVC anatomic complexity (simple 87 ± 16 vs complex 84 ± 9, t = 0.362, P > .2). Although there were almost 14 points of difference on PDI scores between the 31 children with simple TAPVC and the 3 with complex TAPVC, the small number of children in the latter group resulted in no significant difference in the scores (simple 91 ± 13.1 vs complex 77 ± 8, t = 1.642, P = .110). Scores show a shift to the left relative to population norms of 100 ± 15. The distributions of neurodevelopmental scores for survivors of simple and complex TAPVC repair are seen in Figure 1.


Figure 1
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  		Figure 1. Distribution of individual 18- to 24-month mental and psychomotor developmental indices for 34 survivors (open circles for 31 simple and closed circles for 3 complex) after total anomalous pulmonary venous connection repair (TAPVC) at 6 weeks or younger, with broken line indicating mean scores after simple repair and solid line indicating mean scores after complex repair. MDI, Bayley Scales of Infant Development II (1993) Mental Developmental Index; PDI, Bayley Scales of Infant Development II (1993) Psychomotor Developmental Index.

 
Among the 34 survivors with TAPVC, 5 had delays (scores <70) according to the MDI, PDI, or both. In the simple TAPVC group, 4 of 31 infants (13%) had MDI scores lower than 70, and 2 (7%) had PDI scores lower than 70. None of the 3 children in the complex TAPVC group had scores lower than 70. With all variables listed in Table E2, comparisons were made between children with scores of at least 70 and children with delays. The only significant differences (P < .05) found were as follows: weight at surgery, not delayed 3.6 ± 0.62 versus delayed 2.9 ± 0.37; weight in kilograms, t = 2.372, P = .024; and lowest flow on cardiopulmonary bypass for longer than 10 minutes, not delayed 127.9 ± 27.6 versus delayed 145 ± 11.2 mL/(kg · min), t = –2.708, P = .019. With Bonferroni correction, there were no longer any significance differences. Similarly, considering only the 31 patients with simple TAPVC, there were no significant differences between children with scores above and below 70.

All variables from Table E2 with a correlation (P < .10) with MDI or PDI for all 34 survivors are shown in Table 3, first for all 34 survivors and then for the 31 survivors with simple TAPVC. Stepwise multiple regressions were used to determine combinations of variables predictive of MDI and PDI for all 34 survivors. Only those variables of significance (P < .05) from Table 3 were entered for each regression. The only variable to contribute to MDI was socioeconomic status, which explained 24.6% of the variability (slope 0.535, SE 0.166, t = 3.227, P = .003). The only variables to contribute to the variability of PDI were weight at surgery, which explained 25.1% (slope 8.600, SE 3.746, t = 2.296, P = .029), and highest plasma lactate on day 1, which contributed another 10.4% to the variability of PDI, for a combined 35.5% explained variability (slope 1.432, SE 0.675, t = 2.121, P = .043). Repeating the correlations of predictor variables with MDI and PDI for the 31 survivors after simple TAPVC resulted in the same variables showing significance (P < .1; Table 3). The results of the stepwise multiple regressions were also similar. Socioeconomic status explained 24.3% of the variability of MDI (slope 0.654; SE 0.156; t = 4.196; P < .001). For PDI, weight at surgery explained 22.6% of the variability; highest plasma lactate on day 1 contributed another 8.5%, for a total of 31.1% of the variability explained (slope 1.416; SE 0.645; t = 2.196; P = .036).


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  		TABLE 3 Two-tailed Pearson product–moment correlations (r) with P values of less than .10 for predictor variables in relation to developmental indices for all 34 survivors after complete repair at 6 weeks or older for total anomalous pulmonary venous connection and for those 31 with simple anatomy
 
Because of the large proportion of the variability in MDI explained by socioeconomic status, correlations between this predictor variable and other demographic variables were completed for 34 children. Significant univariate correlations of socioeconomic status were mother’s schooling (r = 0.776, P < .001) and race (r = 0.616, P < .001).


   Discussion
Top
 Abstract
 Introduction
 Materials and Methods
 Results
 Discussion
Conclusions
 References
 
This is the first report of early childhood outcomes of standardized neurodevelopmental assessment for survivors after early infant TAPVC repair. This indication in early childhood of the need for early developmental and educational intervention supports previous recommendations that early intervention would benefit some children with TAPVC repair.10Go Kirshbom and colleagues9Go initially reported that most school-aged survivors of simple TAPVC can expect good functional outcomes, with 69% having average to above average school performance according to parent questionnaires. The need for educational support of TAPVC survivors was shown in a later study that used age-appropriate standardized tests; Kirshbom and associates10Go reported that 58% of the 6- to 19 year- old- children had delays in one or more domains tested: performance intelligence, mathematics achievement, attention, fine motor function, and visual motor integration. Through the early identification of children at risk for these school-related difficulties and the provision of educational intervention as indicated, we hope to be able to reduce adverse outcomes.10,12Go

Direct comparisons between a developmental test for 18- to 24-month-old children, as we have done, and formal intellectual testing for school-aged children, as reported by Kirshbom and associates,10Go cannot be completed because of the different ages of the children and the low socioeconomic status found in our population. For those surviving after repair of simple TAPVC, Kirshbom found the verbal IQ at a median of 11 years for 30 children to be 98.6 ± 20.2, not differing from population norms. For the comparable anatomic lesion, the MDI mean score for our 31 children after repair of simple TAPVC was 87 ± 16. Mean motor score for our 31 survivors of simple TAPVC, 91 ± 13, was below population norms. Although we cannot compare our data directly with those of Kirshbom and associates,10Go the performance IQ of children in that study was also below population normative data.

The lack of prediction of low mental scores in our study beyond unmodifiable variables related to socioeconomic status suggests that further exploration of other variables might be of benefit. Modifiable predictive variables of mental scores after TAPVC may yet be identified and should be a priority for future research. Our study identifies the potentially modifiable predictor of PDI to be higher plasma lactate within the first 24 hours after surgery. We have previously shown that postoperative plasma lactate may help to differentiate developmental outcomes.19Go Delayed motor scores occurred in the simple and complex TAPVC groups, supporting the literature that documents lower fine motor and performance scores in this population.10Go We have had similar findings in survivors after surgery for transposition of the great arteries.14Go The particularly low mean motor score in our 3 children with complex anatomy, 77 ± 8, is similar to our findings in a previous study of 16 children after surgery for hypoplastic left heart syndrome (68 ± 19).13Go

The important role of socioeconomic status in the outcomes of infants with congenital heart surgery has been documented previously.20-23Go We sought factors that contributed to the lower socioeconomic status in this study: maternal education level and race were associated; however, we have not identified all contributing variables. The significant predictive value of socioeconomic status for the MDI in this study is in contrast to our previous publication on development after arterial switch operation, in which socioeconomic status and mother’s education did not predict outcome.14Go Although it is common in the literature for socioeconomic status to relate to the developmental outcome of young children, it is unusual for it to be the only predictor. The usual method of addressing the effect of wide variations of socioeconomic status affecting outcome is to control for this variable.24,25Go Such control was not possible in this outcome study. A combination of postsurgical TAPVC and low socioeconomic status strongly predicted neurodevelopmental delay. We recommend early intervention and education for all children with delays. Early individual assessment helps to identify children in need of additional neurodevelopmental or educational help.

We used The Bayley Scales of Infant Development II18Go to assess the developmental level of these survivors at 18 to 24 months because this commonly used tool provides benefit to the children by outlining developmental strengths and weaknesses, thus assisting in planning individual developmental intervention as required. With this test, correlations with later outcome are best for the oldest children within the range of the test, which is up to 42 months of age. It is important to point out that The Bayley Scales of Infant Development II18Go is not an intelligence test. Severe delay of more than 3 SD below mean on mental developmental tests is associated with a persistent mental delay.26Go After neonatal cardiac surgery, there have been few long-term studies done comparing developmental testing with intelligence testing at school age, but one study after repair of transposition of the great arteries suggested 1-year developmental testing had only modest correlation with later performance. We plan to assess these children at 5 years. Thus far, only 13 have been assessed. The Pearson product correlation (2-sided) of the MDI with full-scale intelligence is 0.663. Further testing is needed, however, to determine the utility of developmental tests in young children after neonatal intracardiac repair.

Fifty-eight percent of our 34 children with TAPVC repair required rehospitalization, whereas 9% of 82 children after the arterial switch operation,14Go from this site and under the same system of medical care, required rehospitalization. In this study, these hospitalizations were largely for pulmonary illnesses regardless of demographic background, suggesting that children after TAPVC repair require close medical follow-up for pulmonary complications. These patients’ general health was good, as supported by other studies.8-10Go There was no microcephaly reported in our study, in contrast to 28% reported by Kirshbom and associates10Go and 30% in a population of young children of similar age after complex heart surgery of several types.27Go One child with simple TAPVC and a weight less than the 3rd percentile had a gastrostomy tube.

Our study of consecutive admissions to our regional heart center with TAPVC shows a high incidence of children of native peoples (14 of 41 children, 34%). This proportion of native children is 3.6 times higher than that among our population of children 6 weeks or younger with all types of severe complex cardiac anomalies cared for in 1996 through 1999 and previously published.13Go This finding of a higher than expected incidence of TAPVC among native Canadians has been previously described.28Go We were unable to show that native children had more complicated operative or perioperative periods.

One of the major strengths of this study is its 100% follow-up of all survivors and accountability for all 41 children that underwent surgery. The major limitation to our study is the small number of survivors with complex TAPVC. This precluded the ability to test modifiable variables in that group. In addition, the significantly large number of subjects with low socioeconomic status may have acted to limit the determination of predictive variables.


   Conclusions
Top
 Abstract
 Introduction
 Materials and Methods
 Results
 Discussion
 Conclusions
References
 
An increased survival of children with TAPVC during recent decades suggests a need to enhance our understanding of their neurodevelopmental outcomes. We strongly recommend that children who have been treated for this life-threatening condition undergo early developmental assessments that may assist in planning early intervention programs to enhance their future skills. In this study, after evaluation of a wide variety of predictor variables, we found few modifiable predictors, suggesting that alternative predictors should be sought. The observation of a high prevalence of TAPVC among North American native peoples warrants further epidemiologic study.


   Acknowledgments
 
We thank the families of these children for their active participation in the developmental sites across Western Canada and for their commitment to this project. We sincerely thank the research coordinators who made this research study possible: H. Christianson and D. Anseeuw-Deeks, Calgary, Alberta, V. Debooy, Winnipeg, Manitoba, T. Martindale, Saskatoon, Saskatchewan, and L. Sanders, Edmonton, Alberta.


   Footnotes
 
Supported initially by the Glenrose Rehabilitation Hospital Research Trust Fund, with ongoing operational funding from The Registry and Follow-up of Complex Pediatric Therapies Project, Alberta Health and Wellness.

* D. Moddemann, Winnipeg, Manitoba, P. Blakley, Saskatoon, Saskatchewan, and A. Ninan, Regina, Saskatchewan. Back


   References
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 Abstract
 Introduction
 Materials and Methods
 Results
 Discussion
 Conclusions
 References
 

  1. Rowe RD. Anomalies of venous return. In: Keith JD, Rowe RD, Vlad PK, editors. Heart disease in infancy and childhood. 3rd ed.. New York: Macmillan; 1978. pp. 554.
  2. Raisher BD, Grant JW, Martin TC, Strauss AW, Spray TL. Complete repair of total anomalous pulmonary venous connection in infancy. J Thorac Cardiovasc Surg 1992;104:443-448.[Abstract]
  3. Sinzobahamvya N, Arenz C, Brecher AM, Blaschczok HC, Urban AE. Early and long-term results for correction of total anomalous pulmonary venous drainage (TAPVD) in neonates and infants. Eur J Cardiothorac Surg 1996;10:433-438.[Abstract/Free Full Text]
  4. Rordam S, Abdelnoor M, Sorland S, Tjonneland S. Factors influencing survival in total anomalous pulmonary venous drainage. Scand J Thorac Cardiovasc Surg 1994;28:55-60.[Medline]
  5. Sano S, Brawn WJ, Mee RB. Total anomalous pulmonary venous drainage. J Thorac Cardiovasc Surg 1989;97:886-892.[Abstract]
  6. Lupinetti FM, Kulik TJ, Beekman 3rd RH, Crowley DC, Bove EL. Correction of total anomalous pulmonary venous connection in infancy. J Thorac Cardiovasc Surg 1993;106:880-885.[Abstract]
  7. Caldarone CA, Najm HK, Kadletz M, Smallhorn JF, Freedom RM, Williams WG, et al. Surgical management of total anomalous pulmonary venous drainage: impact of coexisting cardiac anomalies. Ann Thorac Surg 1998;66:1521-1526.[Abstract/Free Full Text]
  8. Boger AJ, Baak R, Lee PC, Boersma E, Meijboom FJ, Witsenburg M. Early results and long-term follow-up after corrective surgery for total anomalous pulmonary venous return. Eur J Cardiothorac Surg 1999;16:296-299.[Medline]
  9. Kirshbom PM, Myung RJ, Gaynor JW, Ittenbach RF, Paridon SM, DeCampli WM, et al. Preoperative pulmonary venous obstruction affects long-term outcome for survivors of total anomalous pulmonary venous connection repair. Ann Thorac Surg 2002;74:1616-1620.[Abstract/Free Full Text]
  10. Kirshbom PM, Flynn TB, Clancy RR, Ittenbach RF, Hartman DM, Paridon SM, et al. Late neurodevelopmental outcome after repair of total anomalous pulmonary venous connection. J Thorac Cardiovasc Surg 2005;129:1091-1097.[Abstract/Free Full Text]
  11. Wernovsky G, Shillingford AJ, Gaynor JW. Central nervous system outcomes in children with complex congenital heart disease. Curr Opin Cardiol 2005;20:94-99.[Medline]
  12. Majnemer A, Limperopoulos C, Shevell M, Rosenblatt B, Rohlicek C, Tchervenkov C. Long-term neuromotor outcome at school entry of infants with congenital heart defects requiring open-heart surgery. J Pediatr 2006;148:72-77.[Medline]
  13. Robertson CM, Joffe AR, Sauve RS, Rebeyka IM, Phillipos EZ, Dyck JD, et al. Outcomes from an interprovincial program of newborn open heart surgery. J Pediatr 2004;144:86-92.[Medline]
  14. Freed DH, Robertson CM, Sauve RS, Joffe AR, Rebeyka IM, Ross DB, et al. Intermediate-term outcomes of the arterial switch operation for transposition of great arteries in neonates: alive but well?. J Thorac Cardiovasc Surg 2006;132:845-852.[Abstract/Free Full Text]
  15. Blishen BR. The 1981 socioeconomic index for occupations in Canada. Can Rev Sociol Anthropol 1987;24:465-488.
  16. NIH policy on reporting race and ethnicity data: subjects in clinical research [cited 2006 Aug 17]. Available from: URL: http://grants2.nih.gov/grants/guide/notice-files/not-od-01-053.html.
  17. Bax MC. Terminology and classifications of cerebral palsy. Dev Med Child Neurol 1964;6:259-297.
  18. Bayley N. Bayley scales of infant development manual. 2nd ed.. San Antonio: Psychological Corp; 1993.
  19. Cheung PY, Chui N, Joffe AR, Rebeyka IM, Robertson CM, Western Canadian Complex Pediatric Therapies Project, Follow-up Group Postoperative lactate concentrations predict the outcome of infants aged 6 weeks or less after intracardiac surgery: a cohort follow-up to 18 months. J Thorac Cardiovasc Surg 2006;130:837-843.
  20. Forbess JM, Visconti KJ, Hancock-Friesen C, Howe RC, Bellinger DC, Jonas RA. Neurodevelopmental outcome after congenital heart surgery: results from an institutional registry. Circulation 2002;106(12 Suppl 1):I95-I102.[Medline]
  21. Grabitz RG, Joffres MR, Collins-Nakai RL. Congenital heart disease: incidence in the first year of life. The Alberta Heritage Pediatric Cardiology Program. Am J Epidemiol 1988;128:381-388.[Abstract/Free Full Text]
  22. Wernovsky G, Stiles KM, Gauvreau K, Gentles TL, duPlessis AJ, Bellinger DC, et al. Cognitive development after the Fontan operation. Circulation 2000;102:883-889.[Abstract/Free Full Text]
  23. Goldberg CS, Schwartz EM, Brunberg JA, Mosca RS, Bove EL, Schork MA, et al. Neurodevelopmental outcome of patients after the Fontan operation: A comparison between children with hypoplastic left heart syndrome and other functional single ventricle lesions. J Pediatr 2000;137:646-652.[Medline]
  24. Ricci M, Elliott M, Cohen GA, Catalan G, Stark J, de Leval MR, et al. Management of pulmonary venous obstruction after correction of TAPVC: risk factors for adverse outcome. Eur J Cardiothorac Surg 2003;24:28-36.[Abstract/Free Full Text]
  25. Forbess JM, Visconti KJ, Bellinger DC, Howe RJ, Jonas RA. Neurodevelopmental outcomes after biventricular repair of congenital heart defects. J Thorac Cardiovasc Surg 2002;123:631-639.[Abstract/Free Full Text]
  26. Honzik MP. Value and limitations of infant tests: an overview. In: Lewis M, editor. Origins of intelligence: infancy and early childhood. New York: Plenum Press; 1976. pp. 46-67.
  27. Limperopoulos C, Majnemer A, Shevell MI, Rohlicek C, Rosenblatt B, Tchervenkov C, et al. Predictors of developmental disabilities after open heart surgery in young children with congenital heart defects. J Pediatr 2002;141:51-58.[Medline]
  28. McCrindle BW, Wood MM, Collins GF, Wheatley B, Rowe RD. An increased incidence of total anomalous pulmonary drainage among aboriginal Canadians. Can J Cardiol 1996;12:81-85.[Medline]



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