- Email: [email protected]
Costs of Prenatal Detection of Congenital Heart Disease
Costs of Prenatal Detection of Congenital Heart Disease Anusha Jegatheeswaran, MDa,b,†, Carol Oliveira, MDa,c,†, Constantine Batsos, DDSa, Anita J. Moon-Grady, MDd, Norman H. Silverman, MD, DSc (Med)e, Lisa K. Hornberger, MDf, Peter Coyte, PhDa,g, and Mark K. Friedberg, MDh,* Little information is available about the transportation costs incurred from the missed prenatal diagnosis of congenital heart disease (CHD). The objectives of the present study were to analyze the costs of emergency transportation related to the postnatal diagnosis of major CHD and to perform a cost/benefit analysis of additional training for ultrasound technicians to study the implications of improved prenatal detection rates. The 1-year costs incurred for emergency transportation of pre- and postnatally diagnosed infants with CHD in Northern California and North Western Nevada were calculated and compared. The prenatal detection rate in our cohort (n ⴝ 147) was 30.6%. Infants postnatally diagnosed were 16.5 times more likely (p <0.001) to require emergency transport. The associated emergency transportation costs were US$542,143 in total for all patients with CHD. The mean cost per patient was $389.00 versus $5,143.51 for prenatally and postnatally diagnosed infants, respectively (p <0.001). Assuming an improvement in detection rates after 1-day training for ultrasound technicians, the investment in training cost can be recouped in 1 year if the detection rate increased by 2.4% to 33%. Savings of $6,543,476 would occur within 5 years if the detection rate increased to 50%. In conclusion, CHD diagnosed postnatally results in greater costs related to emergency transportation of ill infants. Improving the prenatal detection rates through improved ultrasound technician training could result in considerable cost savings. © 2011 Elsevier Inc. All rights reserved. (Am J Cardiol 2011;108:1808 –1814) It is important for payers, insurers, health service providers, and government to understand congenital heart disease (CHD) economics when formulating policy around prenatal detection. Although CHD prenatal detection rates can be improved,1,2 the potential economic effect has not been defined. We hypothesized that a prenatal diagnosis leads to reduced costs associated with emergent transport of infants with major CHD. Using data from our initial study,3 our objective was to determine the incremental costs incurred in Northern California and North Western Nevada because of a missed prenatal diagnosis of major CHD, focusing on the costs of emergency transportation to a tertiary care center after
Table 1 Costs and guidelines Services
Ground transport (ambulance) Air transport Rotary wing Fixed wing Registered nurse and physician team Prostaglandin E1 Mechanical ventilation
Costs
Transport Guidelines (miles) ⬍75
$500 ($8/mile after first 20 miles)
75–149 $1,500 start up, then $35/mile $8,000–$12,000 (depending on distance) $3,000/hour
150–230
$225/ampoule/hour $60/hour
a
Department of Health Policy, Management and Evaluation, University of Toronto, Toronto, Ontario, Canada; bDivision of Cardiovascular Surgery, Labatt Family Heart Centre and cDivision of General and Thoracic Surgery, Hospital for Sick Children, Toronto, Ontario, Canada; dFetal Cardiovascular Program, University of California, San Francisco, San Francisco, California; eDivision of Pediatric Cardiology, Stanford University School of Medicine, Palo Alto, California; fFetal and Neonatal Cardiology Program, Pediatric Cardiology, Stollery Children’s Hospital, University of Alberta, Edmonton, Alberta, Canada; gInstitute for Clinical Evaluative Sciences, Toronto, Ontario, Canada; and hDivision of Cardiology, Labatt Family Heart Centre, Hospital for Sick Children, Toronto, Ontario, Canada. Manuscript received May 25, 2011; manuscript received and accepted July 15, 2011. The original study was supported by the Glaser Pediatric Research Network. *Corresponding author: Tel: (416) 813-7239; fax: (416) 813-7547. E-mail address: [email protected] (M.K. Friedberg). †
Drs. Jegatheeswaran and Oliveira contributed equally to this article.
0002-9149/11/$ – see front matter © 2011 Elsevier Inc. All rights reserved. doi:10.1016/j.amjcard.2011.07.052
delivery. In addition, a cost/benefit analysis was conducted to investigate the potential effects of a 1-day training program for ultrasound technicians (UTs) on costs. Methods The present cost analysis was performed at the Hospital for Sick Children and University of Toronto (Toronto, Ontario, Canada) using data collected prospectively at 3 referral centers in Northern California (Lucile Packard Children’s Hospital at Stanford University, University of California, San Francisco, and University of California, Davis). The study group consisted of infants ⬍6 months old with major CHD (i.e., requiring or expected to require intervention), admitted to 1 of these referral centers, during www.ajconline.org
Congenital Heart Disease/Cost/Benefit Ratio of Prenatal Detection
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Figure 1. Patient selection from initial study patient cohort, in addition to several important characteristics of prenatally and postnatally diagnosed patients. Category “Discharged” refers to infants who were initially discharged from hospital without a diagnosis of CHD and required subsequent transport and hospitalization once they became ill.
a 1-year period (2004 to 2005). The infants were either born in a referral center, transferred by air or ground ambulance from a regional hospital after birth, or transported by personal motorized vehicle within 6 months after birth. In some instances, the infants were transferred more than once. For example, some infants were first transferred to a regional center and then onward to the referral center. The infants were categorized into those diagnosed prenatally and those diagnosed postnatally from the parent’s response to questionnaires and information from prenatal care providers. Only infants with CHD expected to be detected by prenatal ultrasonography were included. We, therefore, excluded patients with isolated ventricular septal defect, atrial septal defect, or minor valve lesions. For the purposes of the cost analysis, and to better define the cost parameters using a conservative approach, we excluded infants transported ⬎230 miles, because they could have been transported to closer tertiary hospitals. The transport of infants with CHD requires a multidisciplinary team, the potential use of mechanical ventilation,
prostaglandin therapy in potentially ductal dependent lesions, and a ground or air transportation vehicle. The distances and associated costs were estimated for all emergency medical transfers to the tertiary care centers using the 2009 University of California San Francisco Children’s Hospital, private United States air ambulance service providers’ fees and transportation guidelines (Table 1).4 The cost of a fetal echocardiogram used in the sensitivity analysis was $3,642. Categorical data were compared using the chi-square test and the mean using the Student t test. The costs incurred by each group were aggregated and the average costs per patient calculated. For the calculation of the mean costs, if the total cost for a population can be extrapolated, parametric tests are appropriate.5,6 However, in acknowledgment of the non-normal distribution of the cost data, the median values were also calculated and compared using the Wilcoxon rank-sum test. There is debate regarding the appropriateness of parametric tests for cost data, and some investigators have suggested modeling the data before analysis.5,6 How-
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Figure 2. CHD umbrella diagnoses and their requirement for transport showing proportion of patients in each umbrella diagnosis for both prenatally and postnatally diagnosed patients. Categories: 0, all CHD diagnoses appearing ⬍5 times in frequency; 1, atrioventricular septal defect; 2, coarctation of aorta; 3, double outlet right ventricle; 4, transposition of great arteries; 5, heterotaxy, left or right; 6, hypoplastic left heart syndrome; 7, pulmonary atresia; 8, tetralogy of Fallot; and 9, tricuspid atresia. Table 2 Mean costs and resource use Resource per Patient Emergency transfer distance (miles) Costs (US$) Sensitivity analysis of costs ⫹20% ⫺20% Including 1 follow-up echocardiogram Including 2 follow-up echocardiograms
Prenatal Diagnosis (n ⫽ 45)
Postnatal Diagnosis (n ⫽ 102)
5.40 ⫾ 25 389 ⫾ 1,824
70 ⫾ 74 5,144 ⫾ 4,602
65 4,755
⬍0.0001 ⬍0.0001
467 ⫾ 2,189 311 ⫾ 1,459 4,031 ⫾ 1,824 7,673 ⫾ 1,824
6,172 ⫾ 5,522 4,115 ⫾ 3,682 5,144 ⫾ 4,602 5,144 ⫾ 4,603
5,705 3,804 1,113 ⫺2,530
⬍0.0001 ⬍0.0001 0.12; 0.59 0.0005; 0.09
ever, for practical situations, where the true distribution is unknown, the sample mean remains the estimator of choice, particularly for a small sample size.5 To assess the robustness of the results, sensitivity analyses on the total costs, training costs, costs of follow-up fetal echocardiograms, and primary and improved secondary detection rates were performed. To account for variations in fuel prices, aircraft types, and transport team composition, a sensitivity analysis using ⫾20% of the costs was performed. We applied a high and low estimate of the UT training costs. Although we methodologically limited our analysis to the costs derived from our primary data set, which accrued solely by emergency transport, we assessed the effect of 1 or 2 follow-up fetal echocardiograms for patients with a prenatal diagnosis. The reported prenatal detection rates have varied widely.1 To account for the complete spectrum of possible prenatal detection rates, the sensitivity analysis included primary and secondary detection rates between 0 and 1 in intervals of 0.1. Statistical analysis used SAS, version 9.1 (SAS Institute, Cary, North Carolina), and p ⬍0.05 was considered significant. The cost/benefit analysis was conducted from a payer’s perspective to study the implications of UT training and improved detection rates on future incremental costs of
Mean Incremental Difference
p Value
postnatal CHD diagnosis. A 1- and 5-year prospective period was evaluated assuming the technology would be mostly unchanged throughout these periods. The total savings in the future was not discounted but calculated to today’s present value assuming that the time–value of money would offset the discount factor. The calculations were based on 2009 costs without adjustment for future inflation. The cost/benefit analysis was adjusted for the average population growth and birth rate in California (population growth 0.008%/year, extrapolating 15.1 births/ 1,000) and Nevada (population growth 0.024%/year, extrapolating 15.2 births/1,000) from 2006 to 2008.7,8 A full day UT program at the 2010 American Institute of Ultrasound in Medicine convention in San Diego cost $300.00 – $600.00 per person.9 A similar course at New York University costs $150 for pediatric cardiologists and $75 for UTs. As such, we adopted average 1-day training costs of $500 per person in our analysis and $250 and $750 for the sensitivity analysis. In 2006, there were 1,650 UTs employed in Northern California and North Western Nevada.10 Our analysis considered the cost of training of 1,815 UTs, an additional 10% of the 2006 number assuming 5% population growth and a 5% cushion to mitigate against a conservative estimate. Finally, we determined the mini-
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Figure 3. Total costs of CHD associated with transportation. Right vertical line indicates detection rate of our study. Total costs of CHD associated with transportation in Northern California and North Western Nevada presented. With increasing prenatal detection rate, total costs and those associated with postnatal diagnosis decreased. Expectedly, the costs of prenatal diagnosis increased with increased detection rate. Using estimates of our study, the total costs saved for different detection rates can be extrapolated using these data.
mally required detection rate improvement for a positive net benefit after 1 year and calculated the net benefit at 5 years for a broad range (10% to 100% in steps of 10%) of new detection rates after training. We considered improvements of 19.4% to 50% in the detection rates.1,11 Results The study population is shown in Figure 1. The CHD diagnostic categories for the prenatally and postnatally diagnosed groups and their requirement for transport are shown in Figure 2. A total of 45 infants (30.6%) with a prenatal diagnosis and 102 (69.4%) with a postnatal diagnosis were studied. In aggregate, infants with a prenatal diagnosis were transported 243 miles (mean 5.4) at a cost of $17,505.00 (mean US$389/infant). Infants diagnosed postnatally were transported a total of 7,181 miles (mean 70.4) at a cost of $524,638.00 (mean US$5,143.51/infant). Infants with a postnatal diagnosis were more likely to require emergency transport (relative risk 16.5, 95% confidence interval 4.25 to 64.44; p ⬍0.001). The mean distances and costs, including a ⫾20% sensitivity analysis for costs and follow-up fetal echocardiograms, are listed in Table 2. The mean emergency transport costs and distances traveled were significantly different between the groups ($389 vs $5,143.51; p ⬍0.0001, t test, and p ⬍0.0001, Wilcoxon test). The emergency transportation costs for postnatally diagnosed infants were 13.2 times greater than the costs for
the prenatally diagnosed group. The total emergency transportation costs for all study subjects, in the 1-year study period, were US$542,143 (mean US$3,688.05/infant). The effect of the varying detection rates along a continuum from 10% to 100% on the predicted total 1-year costs in Northern California and North Western Nevada are shown in Figure 3. The costs of prenatal detection increased by a relatively small degree with an increasing detection rate but with a greater decrease in the transport costs with prenatal detection. In 2004 to 2005, when the study cohort was born, there were an estimated 220,000 births in Northern California and North Western Nevada. Using our cohort of 147 infants, we obtained a 0.68/1000 birth rate with major CHD (likely underestimated secondary to excluding common CHD types, estimates of birth rates, and possible referral of infants to other centers). In 2009, the population of Northern California and North Western Nevada was estimated at 14.9 million. The 5-year prospective cost/benefit analysis accounted for population growth and birthrate (although not migration such as might occur in agricultural areas) and the incidence rate of CHD requiring intervention from our study sample (0.68/1,000 births). The cost/benefit analysis results are shown in Figure 4. In the worst-case scenario (with no follow-up echocardiograms), assuming an improvement in prenatal detection from 30.6% to 40% and $750.00 training costs for 1,815 UTs ($750.00 ⫻ 1,815 UT ⫽ $1,361,250.00),
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Figure 4. Cost/benefit analysis. Right vertical line indicates detection rate of our study. Graph shows cost benefit for UTs training at a cost of $250, $500, and $750 and 0, 1, and 2 follow-up fetal echocardiograms for continuous numbers of improved detection rates at a 1-year perspective. Although the number of fetal echocardiograms clearly affects the net cost benefit, the cost of UT training is of secondary importance. Table 3 Detection rates with positive net benefit Training Costs
$250 $500 $750
Follow-Up Echocardiograms for Prenatally Diagnosed Patients None
One
Two
0.32 0.32 0.33
0.35 0.39 0.43
0.71 0.73 0.75
would result in a cost benefit within 1 year and would save US$21.3 million within 5 years. Assuming a best-case scenario with high detection rates of 70%, as reported previously,1 and using $250 training costs, the cost benefit would be positive within 1 year and would be US$94.5 million within 5 years. Because prenatal detection usually leads to a fetal echocardiogram, as well as a variable number of follow-up fetal echocardiograms, we assessed their effect on the cost/benefit analysis (Table 3). The results are also given for the entire spectrum of prenatal detection from 10% to 100%, stratified by the number of fetal echocardiograms and training costs (Table 4). According to our study institutions’ practice, prenatal detection led to a fetal echocardiogram with 1 follow-up echocardiogram. This would affect the net benefit after 1 and 5 years as follows: with training costs of $500, the net benefit would be positive after 1 year, ranging from US$4.4 and 38.2 within 5 years. For training costs of $250 and $750, the net benefit would be positive after 1 year, with savings at 5 years of ⱕUS$37.8 million. For 2
follow-up fetal echocardiograms, the net benefit becomes positive only after 1 year at an improved detection rate of 71% for training costs of $250 and 75% for training costs of $750 (Table 3). In this optimistic scenario of high prenatal detection rates, after 5 years, the investment of UT training at $250 would be cost beneficial at an improved detection rate of 70%, yielding savings of US$0.3 million, and at an improved detection rate of 80%, savings of US$12.22 million (Table 4). Discussion Although previous studies have investigated the effect of a prenatal diagnosis of CHD on the postnatal course and outcomes, including the cost related to the initial hospitalization,11–19 our study is the first to estimate the costs associated with postnatal transportation of infants born with CHD. Our results showed that a missed prenatal diagnosis of infants with CHD results in greater costs related to emergency transportation and that improving the prenatal detection rates through improved UT training could reduce these costs.1,11,20 Our analysis provides a generalizable picture of the incremental costs associated with a prenatal versus a postnatal diagnosis of CHD for tertiary care centers, using patientlevel data that included the need for emergency transport and related ventilation. As such, we used cost values and adopted a payer’s perspective. However, a cost-effectiveness analysis could not be performed, because data on
⫺77,478,764 ⫺64,664,368 ⫺51,849,972 ⫺39,035,575 ⫺26,221,179 ⫺13,406,783 ⫺592,386 12,222,010 25,036,407 37,850,803 ⫺77,025,014 ⫺64,210,618 ⫺51,396,222 ⫺38,581,825 ⫺25,767,429 ⫺12,953,033 ⫺138,636 12,675,760 25,490,157 38,304,553 ⫺76,571,264 ⫺63,756,868 ⫺50,942,472 ⫺38,128,075 ⫺25,313,679 ⫺12,499,283 315,113 13,129,510 25,943,906 38,758,303 ⫺12,971,360 ⫺7,335,384 ⫺1,699,409 3,936,567 9,572,543 15,208,518 20,844,494 26,480,470 32,116,445 37,752,421 ⫺12,517,610 ⫺6,881,634 ⫺1,245,659 4,390,317 10,026,293 15,662,268 21,298,244 26,934,220 32,570,195 38,206,171 ⫺12,063,860 ⫺6,427,884 ⫺791,909 4,844,067 10,480,043 16,116,018 21,751,994 27,387,970 33,023,945 38,659,921 ⫺50,525,376 ⫺26,439,029 ⫺2,352,681 21,733,667 45,820,015 69,906,362 93,992,710 118,079,057 142,165,405 166,251,753 10% 20% 30% 40% 50% 60% 70% 80% 90% 100%
⫺50,071,626 ⫺25,985,279 ⫺1,898,931 22,187,417 46,273,765 70,360,112 94,446,460 118,532,807 142,619,155 166,705,503
⫺50,979,126 ⫺26,892,779 ⫺2,806,431 21,279,917 45,366,264 69,452,612 93,538,960 117,625,307 141,711,655 165,798,003
$750 Training Costs $500 Training Costs $250 Training Costs $750 Training Costs $500 Training Costs $250 Training Costs $750 Training Costs $500 Training Costs $250 Training Costs
No Follow-Up Echocardiograms for Prenatally Diagnosed Patients
Prenatal Detection Rate
Table 4 Cost/benefit analysis for study region: savings at 5 years
One Follow-Up Echocardiogram for Prenatally Diagnosed Patients
Two Follow-Up Echocardiograms for Prenatally Diagnosed Patients
Congenital Heart Disease/Cost/Benefit Ratio of Prenatal Detection
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outcomes such as mortality, number of interventions, or days of hospital stay, were unavailable. Although older studies have suggested that a prenatal diagnosis improves morbidity, mortality, and length of stay;21–23 recent studies have found that only the preoperative status is affected.24,25 Despite a poorer preoperative status;25,26 the interval to surgery, postoperative ventilator days,27 and length of hospital stay26,27 could be similar between prenatally and postnatally diagnosed groups. This would suggest that the costs related to initial hospitalization are similar as intensive care and surgical techniques have improved. It is as yet unknown whether the observation that preoperative status is better in prenatally diagnosed infants with CHD will translate to better long-term neurodevelopmental and functional outcomes.28,29 An increased detection rate could also lead to a greater false-positive rate with associated costs and termination rates. Because no important data on false-positive rates have been published, we did not include this aspect in our analysis. We only included infants transported ⬍230 miles. Although in reality, 13% were transported longer distances, we chose a conservative approach in deeming that distant transfers might not be generalizable to other regions. Accounting for these infants would increase the costs of postnatal diagnosis. We increased the robustness of our study by accounting for the variability in CHD transport costs and detection rates in a sensitivity analysis. The effectiveness of UT training has been previously demonstrated.1,30 Training UTs to obtain outlet views in addition to the 4-chamber view1 increases the prenatal diagnosis of lesions such as transposition of the great arteries, which, in our cohort, was associated with low detection rates.3 The reported increases in detection rates through training have varied from 8% to 19%.1,30 To capture all possible detection improvements, we plotted the cost benefit against a continuous range of improved detection rates, although we realize that high detection rates might not be realistic after a 1-day course. Because we expect training to be effective for years, we calculated the cost benefit for a 5-year period. We did not adjust for potentially increased costs resulting from longer ultrasound examinations per patient (or a reduced number of ultrasound scans per day) or the potential need for refreshment training. UTs spend 2 to 6 minutes visualizing the heart.1 Because of this variability, we assumed that a more detailed cardiac examination by an experienced UT could be completed within a similar period, although we recognize that scanning is longer when abnormalities are found. In the best-case scenario, training would be cost beneficial for an increased detection rate of just 1% assuming training costs of $250 to $500 and no follow-up fetal echocardiograms. In the “worst” case of $750 training costs and 2 follow-up echocardiograms, a 44% improvement would be necessary for a positive net cost benefit. These findings are related to the crude number of UTs in the study region. However, not all UTs perform prenatal screening, reducing the training costs. Furthermore, training could be organized locally to reduce costs and tele-echocardiography might be an option in some areas. We did not account for the indirect costs associated with training such as transportation, lodging, and loss of revenue from the UTs attending training programs. These costs vary, depending on the location of
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the training, and are often borne by the UT, making it difficult to accurately account for. In general, these would increase the costs of improving prenatal detection. Our study was susceptible to selection bias. However, because a randomized design is not applicable, our analysis was appropriately based on a prospective cohort study. Our data might be applicable to other centers with similar referral patterns. However, because healthcare systems and costs vary, the generalization of our data must be done prudently. Including comprehensive outcome data would allow a costeffectiveness analysis and would decrease the number of assumptions needed. This requires additional study. A cost analysis is based on several assumptions from published data and billing agencies. Although we accounted for uncertainty by using sensitivity analyses, the results are macroeconomic estimates and not absolute true values. A microeconomic costing approach might allow a more exact calculation of the costs of a postnatal diagnosis of CHD. We were unable to account for the possibility of increased clinical visits, testing, and caesarean sections secondary to prenatal diagnosis, because these data were not available. Likewise, pregnancy termination rates would affect the costs because postnatal transportation would become irrelevant. 1. Carvalho JS, Mavrides E, Shinebourne EA, Campbell S, Thilaganathan B. Improving the effectiveness of routine prenatal screening for major congenital heart defects. Heart 2002;88:387–391. 2. Sharland GK, Allan LD. Screening for congenital heart disease prenatally: results of a 2 1/2-year study in the South East Thames Region. Br J Obstet Gynaecol 1992;99:220 –225. 3. Friedberg MK, Silverman NH, Moon-Grady AJ, Tong E, Nourse J, Sorenson B, Lee J, Hornberger LK. Prenatal detection of congenital heart disease. J Pediatr 2009;155:26 –31. 4. UCSF Children’s Hospital – Transport Services. Available at: http:// www.ucsfchildrenshospital.org/services/transport_service/index.html. Accessed on November 30, 2009. 5. Briggs AH, O’Brien BJ. The death of cost-minimization analysis? Health Econ 2001;10:179 –184. 6. Briggs A, Nixon R, Dixon S, Thompson S. Parametric modelling of cost data: some simulation evidence. Health Econ 2005;14:421– 428. 7. U.S. Census Bureau – Annual Estimates of the Resident Population for the United States, Regions, States, and Puerto Rico. Available at: http://www.census.gov/popest/states/tables/NST-EST2008-01.xls. Accessed on November 30, 2009. 8. Births, Birth Rates, and Fertility Rates. Available at: http://www.infoplease.com/ipa/A0763849.html. Accessed November 30, 2009. 9. 2010. AIUM Annual Convention and Preconvention Program—Preliminary Program. Available at: http://www.aium.org/cme/events/ ann2010/programFull.pdf. Accessed on November 30, 2009. 10. Job Bank USA – Ultrasound Technicians Salaries, Locations, and Schools. Available at: http://www.jobbankusa.com/career-profiles/ ultrasound-technicians.shtml. Accessed on November 30, 2009. 11. Bahtiyar MO, Copel JA. Improving detection of fetal cardiac anomalies: a fetal echocardiogram for every fetus? J Ultrasound Med 2007; 26:1639 –1641. 12. Ewigman BG, Crane JP, Frigoletto FD, LeFevre ML, Bain RP, McNellis D. Effect of prenatal ultrasound screening on perinatal outcome. RADIUS Study Group. N Engl J Med 1993;329:821– 827.
13. Germanakis I, Sifakis S. The impact of fetal echocardiography on the prevalence of liveborn congenital heart disease. Pediatr Cardiol 2006; 27:465– 472. 14. Bull C. Current and potential impact of fetal diagnosis on prevalence and spectrum of serious congenital heart disease at term in the UK. British Paediatric Cardiac Association. Lancet 1999;354:1242–1247 ik. 15. Jaeggi ET, Fouron JC, Silverman ED, Ryan G, Smallhorn J, Hornberger LK. Transplacental fetal treatment improves the outcome of prenatally diagnosed complete atrioventricular block without structural heart disease. Circulation 2004;110:1542–1548. 16. Simpson JM, Sharland GK. Fetal tachycardias: management and outcome of 127 consecutive cases. Heart 1998;79:576 –581. 17. Johnson BA, Ades A. Delivery room and early postnatal management of neonates who have prenatally diagnosed congenital heart disease. Clin Perinatol 2005;32:921–946, ix. 18. Verheijen PM, Lisowski LA, Stoutenbeek P, Hitchcock JF, Brenner JI, Copel JA, Kleinman CS, Meijboom EJ, Bennink GB. Prenatal diagnosis of congenital heart disease affects preoperative acidosis in the newborn patient. J Thorac Cardiovasc Surg 2001;121:798 – 803. 19. Hornberger LK, Barrea C. Diagnosis, natural history, and outcome of fetal heart disease. Semin Thorac Cardiovasc Surg Pediatr Card Surg Annu 2001;4:229 –243. 20. Garne E, Stoll C, Clementi M; Euroscan Group. Evaluation of prenatal diagnosis of congenital heart diseases by ultrasound: experience from 20 European registries. Ultrasound Obstet Gynecol 2001;17:386 –391. 21. Copel JA, Tan AS, Kleinman CS. Does a prenatal diagnosis of congenital heart disease alter short-term outcome? Ultrasound Obstet Gynecol 1997;10:237–241. 22. Kumar RK, Newburger JW, Gauvreau K, Kamenir SA, Hornberger LK. Comparison of outcome when hypoplastic left heart syndrome and transposition of the great arteries are diagnosed prenatally versus when diagnosis of these two conditions is made only postnatally. Am J Cardiol 1999;83:1649 –1653. 23. Tworetzky W, McElhinney DB, Reddy VM, Brook MM, Hanley FL, Silverman NH. Improved surgical outcome after fetal diagnosis of hypoplastic left heart syndrome. Circulation 2001;103:1269 –1273. 24. Mahle WT, Clancy RR, McGaurn SP, Goin JE, Clark BJ. Impact of prenatal diagnosis on survival and early neurologic morbidity in neonates with the hypoplastic left heart syndrome. Pediatrics 2001;107: 1277–1282. 25. Sivarajan V, Penny DJ, Filan P, Brizard C, Shekerdemian LS. Impact of antenatal diagnosis of hypoplastic left heart syndrome on the clinical presentation and surgical outcomes: the Australian experience. J Paediatr Child Health 2009;45:112–117. 26. Levey A, Glickstein JS, Kleinman CS, Levasseur SM, Chen J, Gersony WM, Williams IA. The impact of prenatal diagnosis of complex congenital heart disease on neonatal outcomes. Pediatr Cardiol 2010; 31:587–597. 27. Bartlett JM, Wypij D, Bellinger DC, Rappaport LA, Heffner LJ, Jonas RA, Newburger JW. Effect of prenatal diagnosis on outcomes in D-transposition of the great arteries. Pediatrics 2004;113:e335– e340. 28. Mahle WT, Wernovsky G. Long-term developmental outcome of children with complex congenital heart disease. Clin Perinatol 2001;28: 235–247. 29. Tabbutt S, Nord AS, Jarvik GP, Bernbaum J, Wernovsky G, Gerdes M, Zackai E, Clancy RR, Nicolson SC, Spray TL, Gaynor JW. Neurodevelopmental outcomes after staged palliation for hypoplastic left heart syndrome. Pediatrics 2008;121:476 – 483. 30. Tegnander E, Eik-Nes SH. The examiner’s ultrasound experience has a significant impact on the detection rate of congenital heart defects at the second-trimester fetal examination. Ultrasound Obstet Gynecol 2006;28:8 –14.
Table 1 Costs and guidelines Services
Ground transport (ambulance) Air transport Rotary wing Fixed wing Registered nurse and physician team Prostaglandin E1 Mechanical ventilation
Costs
Transport Guidelines (miles) ⬍75
$500 ($8/mile after first 20 miles)
75–149 $1,500 start up, then $35/mile $8,000–$12,000 (depending on distance) $3,000/hour
150–230
$225/ampoule/hour $60/hour
a
Department of Health Policy, Management and Evaluation, University of Toronto, Toronto, Ontario, Canada; bDivision of Cardiovascular Surgery, Labatt Family Heart Centre and cDivision of General and Thoracic Surgery, Hospital for Sick Children, Toronto, Ontario, Canada; dFetal Cardiovascular Program, University of California, San Francisco, San Francisco, California; eDivision of Pediatric Cardiology, Stanford University School of Medicine, Palo Alto, California; fFetal and Neonatal Cardiology Program, Pediatric Cardiology, Stollery Children’s Hospital, University of Alberta, Edmonton, Alberta, Canada; gInstitute for Clinical Evaluative Sciences, Toronto, Ontario, Canada; and hDivision of Cardiology, Labatt Family Heart Centre, Hospital for Sick Children, Toronto, Ontario, Canada. Manuscript received May 25, 2011; manuscript received and accepted July 15, 2011. The original study was supported by the Glaser Pediatric Research Network. *Corresponding author: Tel: (416) 813-7239; fax: (416) 813-7547. E-mail address: [email protected] (M.K. Friedberg). †
Drs. Jegatheeswaran and Oliveira contributed equally to this article.
0002-9149/11/$ – see front matter © 2011 Elsevier Inc. All rights reserved. doi:10.1016/j.amjcard.2011.07.052
delivery. In addition, a cost/benefit analysis was conducted to investigate the potential effects of a 1-day training program for ultrasound technicians (UTs) on costs. Methods The present cost analysis was performed at the Hospital for Sick Children and University of Toronto (Toronto, Ontario, Canada) using data collected prospectively at 3 referral centers in Northern California (Lucile Packard Children’s Hospital at Stanford University, University of California, San Francisco, and University of California, Davis). The study group consisted of infants ⬍6 months old with major CHD (i.e., requiring or expected to require intervention), admitted to 1 of these referral centers, during www.ajconline.org
Congenital Heart Disease/Cost/Benefit Ratio of Prenatal Detection
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Figure 1. Patient selection from initial study patient cohort, in addition to several important characteristics of prenatally and postnatally diagnosed patients. Category “Discharged” refers to infants who were initially discharged from hospital without a diagnosis of CHD and required subsequent transport and hospitalization once they became ill.
a 1-year period (2004 to 2005). The infants were either born in a referral center, transferred by air or ground ambulance from a regional hospital after birth, or transported by personal motorized vehicle within 6 months after birth. In some instances, the infants were transferred more than once. For example, some infants were first transferred to a regional center and then onward to the referral center. The infants were categorized into those diagnosed prenatally and those diagnosed postnatally from the parent’s response to questionnaires and information from prenatal care providers. Only infants with CHD expected to be detected by prenatal ultrasonography were included. We, therefore, excluded patients with isolated ventricular septal defect, atrial septal defect, or minor valve lesions. For the purposes of the cost analysis, and to better define the cost parameters using a conservative approach, we excluded infants transported ⬎230 miles, because they could have been transported to closer tertiary hospitals. The transport of infants with CHD requires a multidisciplinary team, the potential use of mechanical ventilation,
prostaglandin therapy in potentially ductal dependent lesions, and a ground or air transportation vehicle. The distances and associated costs were estimated for all emergency medical transfers to the tertiary care centers using the 2009 University of California San Francisco Children’s Hospital, private United States air ambulance service providers’ fees and transportation guidelines (Table 1).4 The cost of a fetal echocardiogram used in the sensitivity analysis was $3,642. Categorical data were compared using the chi-square test and the mean using the Student t test. The costs incurred by each group were aggregated and the average costs per patient calculated. For the calculation of the mean costs, if the total cost for a population can be extrapolated, parametric tests are appropriate.5,6 However, in acknowledgment of the non-normal distribution of the cost data, the median values were also calculated and compared using the Wilcoxon rank-sum test. There is debate regarding the appropriateness of parametric tests for cost data, and some investigators have suggested modeling the data before analysis.5,6 How-
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Figure 2. CHD umbrella diagnoses and their requirement for transport showing proportion of patients in each umbrella diagnosis for both prenatally and postnatally diagnosed patients. Categories: 0, all CHD diagnoses appearing ⬍5 times in frequency; 1, atrioventricular septal defect; 2, coarctation of aorta; 3, double outlet right ventricle; 4, transposition of great arteries; 5, heterotaxy, left or right; 6, hypoplastic left heart syndrome; 7, pulmonary atresia; 8, tetralogy of Fallot; and 9, tricuspid atresia. Table 2 Mean costs and resource use Resource per Patient Emergency transfer distance (miles) Costs (US$) Sensitivity analysis of costs ⫹20% ⫺20% Including 1 follow-up echocardiogram Including 2 follow-up echocardiograms
Prenatal Diagnosis (n ⫽ 45)
Postnatal Diagnosis (n ⫽ 102)
5.40 ⫾ 25 389 ⫾ 1,824
70 ⫾ 74 5,144 ⫾ 4,602
65 4,755
⬍0.0001 ⬍0.0001
467 ⫾ 2,189 311 ⫾ 1,459 4,031 ⫾ 1,824 7,673 ⫾ 1,824
6,172 ⫾ 5,522 4,115 ⫾ 3,682 5,144 ⫾ 4,602 5,144 ⫾ 4,603
5,705 3,804 1,113 ⫺2,530
⬍0.0001 ⬍0.0001 0.12; 0.59 0.0005; 0.09
ever, for practical situations, where the true distribution is unknown, the sample mean remains the estimator of choice, particularly for a small sample size.5 To assess the robustness of the results, sensitivity analyses on the total costs, training costs, costs of follow-up fetal echocardiograms, and primary and improved secondary detection rates were performed. To account for variations in fuel prices, aircraft types, and transport team composition, a sensitivity analysis using ⫾20% of the costs was performed. We applied a high and low estimate of the UT training costs. Although we methodologically limited our analysis to the costs derived from our primary data set, which accrued solely by emergency transport, we assessed the effect of 1 or 2 follow-up fetal echocardiograms for patients with a prenatal diagnosis. The reported prenatal detection rates have varied widely.1 To account for the complete spectrum of possible prenatal detection rates, the sensitivity analysis included primary and secondary detection rates between 0 and 1 in intervals of 0.1. Statistical analysis used SAS, version 9.1 (SAS Institute, Cary, North Carolina), and p ⬍0.05 was considered significant. The cost/benefit analysis was conducted from a payer’s perspective to study the implications of UT training and improved detection rates on future incremental costs of
Mean Incremental Difference
p Value
postnatal CHD diagnosis. A 1- and 5-year prospective period was evaluated assuming the technology would be mostly unchanged throughout these periods. The total savings in the future was not discounted but calculated to today’s present value assuming that the time–value of money would offset the discount factor. The calculations were based on 2009 costs without adjustment for future inflation. The cost/benefit analysis was adjusted for the average population growth and birth rate in California (population growth 0.008%/year, extrapolating 15.1 births/ 1,000) and Nevada (population growth 0.024%/year, extrapolating 15.2 births/1,000) from 2006 to 2008.7,8 A full day UT program at the 2010 American Institute of Ultrasound in Medicine convention in San Diego cost $300.00 – $600.00 per person.9 A similar course at New York University costs $150 for pediatric cardiologists and $75 for UTs. As such, we adopted average 1-day training costs of $500 per person in our analysis and $250 and $750 for the sensitivity analysis. In 2006, there were 1,650 UTs employed in Northern California and North Western Nevada.10 Our analysis considered the cost of training of 1,815 UTs, an additional 10% of the 2006 number assuming 5% population growth and a 5% cushion to mitigate against a conservative estimate. Finally, we determined the mini-
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Figure 3. Total costs of CHD associated with transportation. Right vertical line indicates detection rate of our study. Total costs of CHD associated with transportation in Northern California and North Western Nevada presented. With increasing prenatal detection rate, total costs and those associated with postnatal diagnosis decreased. Expectedly, the costs of prenatal diagnosis increased with increased detection rate. Using estimates of our study, the total costs saved for different detection rates can be extrapolated using these data.
mally required detection rate improvement for a positive net benefit after 1 year and calculated the net benefit at 5 years for a broad range (10% to 100% in steps of 10%) of new detection rates after training. We considered improvements of 19.4% to 50% in the detection rates.1,11 Results The study population is shown in Figure 1. The CHD diagnostic categories for the prenatally and postnatally diagnosed groups and their requirement for transport are shown in Figure 2. A total of 45 infants (30.6%) with a prenatal diagnosis and 102 (69.4%) with a postnatal diagnosis were studied. In aggregate, infants with a prenatal diagnosis were transported 243 miles (mean 5.4) at a cost of $17,505.00 (mean US$389/infant). Infants diagnosed postnatally were transported a total of 7,181 miles (mean 70.4) at a cost of $524,638.00 (mean US$5,143.51/infant). Infants with a postnatal diagnosis were more likely to require emergency transport (relative risk 16.5, 95% confidence interval 4.25 to 64.44; p ⬍0.001). The mean distances and costs, including a ⫾20% sensitivity analysis for costs and follow-up fetal echocardiograms, are listed in Table 2. The mean emergency transport costs and distances traveled were significantly different between the groups ($389 vs $5,143.51; p ⬍0.0001, t test, and p ⬍0.0001, Wilcoxon test). The emergency transportation costs for postnatally diagnosed infants were 13.2 times greater than the costs for
the prenatally diagnosed group. The total emergency transportation costs for all study subjects, in the 1-year study period, were US$542,143 (mean US$3,688.05/infant). The effect of the varying detection rates along a continuum from 10% to 100% on the predicted total 1-year costs in Northern California and North Western Nevada are shown in Figure 3. The costs of prenatal detection increased by a relatively small degree with an increasing detection rate but with a greater decrease in the transport costs with prenatal detection. In 2004 to 2005, when the study cohort was born, there were an estimated 220,000 births in Northern California and North Western Nevada. Using our cohort of 147 infants, we obtained a 0.68/1000 birth rate with major CHD (likely underestimated secondary to excluding common CHD types, estimates of birth rates, and possible referral of infants to other centers). In 2009, the population of Northern California and North Western Nevada was estimated at 14.9 million. The 5-year prospective cost/benefit analysis accounted for population growth and birthrate (although not migration such as might occur in agricultural areas) and the incidence rate of CHD requiring intervention from our study sample (0.68/1,000 births). The cost/benefit analysis results are shown in Figure 4. In the worst-case scenario (with no follow-up echocardiograms), assuming an improvement in prenatal detection from 30.6% to 40% and $750.00 training costs for 1,815 UTs ($750.00 ⫻ 1,815 UT ⫽ $1,361,250.00),
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Figure 4. Cost/benefit analysis. Right vertical line indicates detection rate of our study. Graph shows cost benefit for UTs training at a cost of $250, $500, and $750 and 0, 1, and 2 follow-up fetal echocardiograms for continuous numbers of improved detection rates at a 1-year perspective. Although the number of fetal echocardiograms clearly affects the net cost benefit, the cost of UT training is of secondary importance. Table 3 Detection rates with positive net benefit Training Costs
$250 $500 $750
Follow-Up Echocardiograms for Prenatally Diagnosed Patients None
One
Two
0.32 0.32 0.33
0.35 0.39 0.43
0.71 0.73 0.75
would result in a cost benefit within 1 year and would save US$21.3 million within 5 years. Assuming a best-case scenario with high detection rates of 70%, as reported previously,1 and using $250 training costs, the cost benefit would be positive within 1 year and would be US$94.5 million within 5 years. Because prenatal detection usually leads to a fetal echocardiogram, as well as a variable number of follow-up fetal echocardiograms, we assessed their effect on the cost/benefit analysis (Table 3). The results are also given for the entire spectrum of prenatal detection from 10% to 100%, stratified by the number of fetal echocardiograms and training costs (Table 4). According to our study institutions’ practice, prenatal detection led to a fetal echocardiogram with 1 follow-up echocardiogram. This would affect the net benefit after 1 and 5 years as follows: with training costs of $500, the net benefit would be positive after 1 year, ranging from US$4.4 and 38.2 within 5 years. For training costs of $250 and $750, the net benefit would be positive after 1 year, with savings at 5 years of ⱕUS$37.8 million. For 2
follow-up fetal echocardiograms, the net benefit becomes positive only after 1 year at an improved detection rate of 71% for training costs of $250 and 75% for training costs of $750 (Table 3). In this optimistic scenario of high prenatal detection rates, after 5 years, the investment of UT training at $250 would be cost beneficial at an improved detection rate of 70%, yielding savings of US$0.3 million, and at an improved detection rate of 80%, savings of US$12.22 million (Table 4). Discussion Although previous studies have investigated the effect of a prenatal diagnosis of CHD on the postnatal course and outcomes, including the cost related to the initial hospitalization,11–19 our study is the first to estimate the costs associated with postnatal transportation of infants born with CHD. Our results showed that a missed prenatal diagnosis of infants with CHD results in greater costs related to emergency transportation and that improving the prenatal detection rates through improved UT training could reduce these costs.1,11,20 Our analysis provides a generalizable picture of the incremental costs associated with a prenatal versus a postnatal diagnosis of CHD for tertiary care centers, using patientlevel data that included the need for emergency transport and related ventilation. As such, we used cost values and adopted a payer’s perspective. However, a cost-effectiveness analysis could not be performed, because data on
⫺77,478,764 ⫺64,664,368 ⫺51,849,972 ⫺39,035,575 ⫺26,221,179 ⫺13,406,783 ⫺592,386 12,222,010 25,036,407 37,850,803 ⫺77,025,014 ⫺64,210,618 ⫺51,396,222 ⫺38,581,825 ⫺25,767,429 ⫺12,953,033 ⫺138,636 12,675,760 25,490,157 38,304,553 ⫺76,571,264 ⫺63,756,868 ⫺50,942,472 ⫺38,128,075 ⫺25,313,679 ⫺12,499,283 315,113 13,129,510 25,943,906 38,758,303 ⫺12,971,360 ⫺7,335,384 ⫺1,699,409 3,936,567 9,572,543 15,208,518 20,844,494 26,480,470 32,116,445 37,752,421 ⫺12,517,610 ⫺6,881,634 ⫺1,245,659 4,390,317 10,026,293 15,662,268 21,298,244 26,934,220 32,570,195 38,206,171 ⫺12,063,860 ⫺6,427,884 ⫺791,909 4,844,067 10,480,043 16,116,018 21,751,994 27,387,970 33,023,945 38,659,921 ⫺50,525,376 ⫺26,439,029 ⫺2,352,681 21,733,667 45,820,015 69,906,362 93,992,710 118,079,057 142,165,405 166,251,753 10% 20% 30% 40% 50% 60% 70% 80% 90% 100%
⫺50,071,626 ⫺25,985,279 ⫺1,898,931 22,187,417 46,273,765 70,360,112 94,446,460 118,532,807 142,619,155 166,705,503
⫺50,979,126 ⫺26,892,779 ⫺2,806,431 21,279,917 45,366,264 69,452,612 93,538,960 117,625,307 141,711,655 165,798,003
$750 Training Costs $500 Training Costs $250 Training Costs $750 Training Costs $500 Training Costs $250 Training Costs $750 Training Costs $500 Training Costs $250 Training Costs
No Follow-Up Echocardiograms for Prenatally Diagnosed Patients
Prenatal Detection Rate
Table 4 Cost/benefit analysis for study region: savings at 5 years
One Follow-Up Echocardiogram for Prenatally Diagnosed Patients
Two Follow-Up Echocardiograms for Prenatally Diagnosed Patients
Congenital Heart Disease/Cost/Benefit Ratio of Prenatal Detection
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outcomes such as mortality, number of interventions, or days of hospital stay, were unavailable. Although older studies have suggested that a prenatal diagnosis improves morbidity, mortality, and length of stay;21–23 recent studies have found that only the preoperative status is affected.24,25 Despite a poorer preoperative status;25,26 the interval to surgery, postoperative ventilator days,27 and length of hospital stay26,27 could be similar between prenatally and postnatally diagnosed groups. This would suggest that the costs related to initial hospitalization are similar as intensive care and surgical techniques have improved. It is as yet unknown whether the observation that preoperative status is better in prenatally diagnosed infants with CHD will translate to better long-term neurodevelopmental and functional outcomes.28,29 An increased detection rate could also lead to a greater false-positive rate with associated costs and termination rates. Because no important data on false-positive rates have been published, we did not include this aspect in our analysis. We only included infants transported ⬍230 miles. Although in reality, 13% were transported longer distances, we chose a conservative approach in deeming that distant transfers might not be generalizable to other regions. Accounting for these infants would increase the costs of postnatal diagnosis. We increased the robustness of our study by accounting for the variability in CHD transport costs and detection rates in a sensitivity analysis. The effectiveness of UT training has been previously demonstrated.1,30 Training UTs to obtain outlet views in addition to the 4-chamber view1 increases the prenatal diagnosis of lesions such as transposition of the great arteries, which, in our cohort, was associated with low detection rates.3 The reported increases in detection rates through training have varied from 8% to 19%.1,30 To capture all possible detection improvements, we plotted the cost benefit against a continuous range of improved detection rates, although we realize that high detection rates might not be realistic after a 1-day course. Because we expect training to be effective for years, we calculated the cost benefit for a 5-year period. We did not adjust for potentially increased costs resulting from longer ultrasound examinations per patient (or a reduced number of ultrasound scans per day) or the potential need for refreshment training. UTs spend 2 to 6 minutes visualizing the heart.1 Because of this variability, we assumed that a more detailed cardiac examination by an experienced UT could be completed within a similar period, although we recognize that scanning is longer when abnormalities are found. In the best-case scenario, training would be cost beneficial for an increased detection rate of just 1% assuming training costs of $250 to $500 and no follow-up fetal echocardiograms. In the “worst” case of $750 training costs and 2 follow-up echocardiograms, a 44% improvement would be necessary for a positive net cost benefit. These findings are related to the crude number of UTs in the study region. However, not all UTs perform prenatal screening, reducing the training costs. Furthermore, training could be organized locally to reduce costs and tele-echocardiography might be an option in some areas. We did not account for the indirect costs associated with training such as transportation, lodging, and loss of revenue from the UTs attending training programs. These costs vary, depending on the location of
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the training, and are often borne by the UT, making it difficult to accurately account for. In general, these would increase the costs of improving prenatal detection. Our study was susceptible to selection bias. However, because a randomized design is not applicable, our analysis was appropriately based on a prospective cohort study. Our data might be applicable to other centers with similar referral patterns. However, because healthcare systems and costs vary, the generalization of our data must be done prudently. Including comprehensive outcome data would allow a costeffectiveness analysis and would decrease the number of assumptions needed. This requires additional study. A cost analysis is based on several assumptions from published data and billing agencies. Although we accounted for uncertainty by using sensitivity analyses, the results are macroeconomic estimates and not absolute true values. A microeconomic costing approach might allow a more exact calculation of the costs of a postnatal diagnosis of CHD. We were unable to account for the possibility of increased clinical visits, testing, and caesarean sections secondary to prenatal diagnosis, because these data were not available. Likewise, pregnancy termination rates would affect the costs because postnatal transportation would become irrelevant. 1. Carvalho JS, Mavrides E, Shinebourne EA, Campbell S, Thilaganathan B. Improving the effectiveness of routine prenatal screening for major congenital heart defects. Heart 2002;88:387–391. 2. Sharland GK, Allan LD. Screening for congenital heart disease prenatally: results of a 2 1/2-year study in the South East Thames Region. Br J Obstet Gynaecol 1992;99:220 –225. 3. Friedberg MK, Silverman NH, Moon-Grady AJ, Tong E, Nourse J, Sorenson B, Lee J, Hornberger LK. Prenatal detection of congenital heart disease. J Pediatr 2009;155:26 –31. 4. UCSF Children’s Hospital – Transport Services. Available at: http:// www.ucsfchildrenshospital.org/services/transport_service/index.html. Accessed on November 30, 2009. 5. Briggs AH, O’Brien BJ. The death of cost-minimization analysis? Health Econ 2001;10:179 –184. 6. Briggs A, Nixon R, Dixon S, Thompson S. Parametric modelling of cost data: some simulation evidence. Health Econ 2005;14:421– 428. 7. U.S. Census Bureau – Annual Estimates of the Resident Population for the United States, Regions, States, and Puerto Rico. Available at: http://www.census.gov/popest/states/tables/NST-EST2008-01.xls. Accessed on November 30, 2009. 8. Births, Birth Rates, and Fertility Rates. Available at: http://www.infoplease.com/ipa/A0763849.html. Accessed November 30, 2009. 9. 2010. AIUM Annual Convention and Preconvention Program—Preliminary Program. Available at: http://www.aium.org/cme/events/ ann2010/programFull.pdf. Accessed on November 30, 2009. 10. Job Bank USA – Ultrasound Technicians Salaries, Locations, and Schools. Available at: http://www.jobbankusa.com/career-profiles/ ultrasound-technicians.shtml. Accessed on November 30, 2009. 11. Bahtiyar MO, Copel JA. Improving detection of fetal cardiac anomalies: a fetal echocardiogram for every fetus? J Ultrasound Med 2007; 26:1639 –1641. 12. Ewigman BG, Crane JP, Frigoletto FD, LeFevre ML, Bain RP, McNellis D. Effect of prenatal ultrasound screening on perinatal outcome. RADIUS Study Group. N Engl J Med 1993;329:821– 827.
13. Germanakis I, Sifakis S. The impact of fetal echocardiography on the prevalence of liveborn congenital heart disease. Pediatr Cardiol 2006; 27:465– 472. 14. Bull C. Current and potential impact of fetal diagnosis on prevalence and spectrum of serious congenital heart disease at term in the UK. British Paediatric Cardiac Association. Lancet 1999;354:1242–1247 ik. 15. Jaeggi ET, Fouron JC, Silverman ED, Ryan G, Smallhorn J, Hornberger LK. Transplacental fetal treatment improves the outcome of prenatally diagnosed complete atrioventricular block without structural heart disease. Circulation 2004;110:1542–1548. 16. Simpson JM, Sharland GK. Fetal tachycardias: management and outcome of 127 consecutive cases. Heart 1998;79:576 –581. 17. Johnson BA, Ades A. Delivery room and early postnatal management of neonates who have prenatally diagnosed congenital heart disease. Clin Perinatol 2005;32:921–946, ix. 18. Verheijen PM, Lisowski LA, Stoutenbeek P, Hitchcock JF, Brenner JI, Copel JA, Kleinman CS, Meijboom EJ, Bennink GB. Prenatal diagnosis of congenital heart disease affects preoperative acidosis in the newborn patient. J Thorac Cardiovasc Surg 2001;121:798 – 803. 19. Hornberger LK, Barrea C. Diagnosis, natural history, and outcome of fetal heart disease. Semin Thorac Cardiovasc Surg Pediatr Card Surg Annu 2001;4:229 –243. 20. Garne E, Stoll C, Clementi M; Euroscan Group. Evaluation of prenatal diagnosis of congenital heart diseases by ultrasound: experience from 20 European registries. Ultrasound Obstet Gynecol 2001;17:386 –391. 21. Copel JA, Tan AS, Kleinman CS. Does a prenatal diagnosis of congenital heart disease alter short-term outcome? Ultrasound Obstet Gynecol 1997;10:237–241. 22. Kumar RK, Newburger JW, Gauvreau K, Kamenir SA, Hornberger LK. Comparison of outcome when hypoplastic left heart syndrome and transposition of the great arteries are diagnosed prenatally versus when diagnosis of these two conditions is made only postnatally. Am J Cardiol 1999;83:1649 –1653. 23. Tworetzky W, McElhinney DB, Reddy VM, Brook MM, Hanley FL, Silverman NH. Improved surgical outcome after fetal diagnosis of hypoplastic left heart syndrome. Circulation 2001;103:1269 –1273. 24. Mahle WT, Clancy RR, McGaurn SP, Goin JE, Clark BJ. Impact of prenatal diagnosis on survival and early neurologic morbidity in neonates with the hypoplastic left heart syndrome. Pediatrics 2001;107: 1277–1282. 25. Sivarajan V, Penny DJ, Filan P, Brizard C, Shekerdemian LS. Impact of antenatal diagnosis of hypoplastic left heart syndrome on the clinical presentation and surgical outcomes: the Australian experience. J Paediatr Child Health 2009;45:112–117. 26. Levey A, Glickstein JS, Kleinman CS, Levasseur SM, Chen J, Gersony WM, Williams IA. The impact of prenatal diagnosis of complex congenital heart disease on neonatal outcomes. Pediatr Cardiol 2010; 31:587–597. 27. Bartlett JM, Wypij D, Bellinger DC, Rappaport LA, Heffner LJ, Jonas RA, Newburger JW. Effect of prenatal diagnosis on outcomes in D-transposition of the great arteries. Pediatrics 2004;113:e335– e340. 28. Mahle WT, Wernovsky G. Long-term developmental outcome of children with complex congenital heart disease. Clin Perinatol 2001;28: 235–247. 29. Tabbutt S, Nord AS, Jarvik GP, Bernbaum J, Wernovsky G, Gerdes M, Zackai E, Clancy RR, Nicolson SC, Spray TL, Gaynor JW. Neurodevelopmental outcomes after staged palliation for hypoplastic left heart syndrome. Pediatrics 2008;121:476 – 483. 30. Tegnander E, Eik-Nes SH. The examiner’s ultrasound experience has a significant impact on the detection rate of congenital heart defects at the second-trimester fetal examination. Ultrasound Obstet Gynecol 2006;28:8 –14.