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Figure 1. Flow diagram describing the protocol of the Pulsimat study for the screening of critical congenital heart disease. PO: pulse oximetry. Included patients were screened between 24-48 h after birth, following the protocol shown in Figure 1. Newborn infants with a positive or doubtful screening were admitted to the Neonatal Intensive Care Unit (NICU) where the clinical, analytical, and echocardiographic study was completed by neonatologists and pediatric cardiologists. After reaching a diagnosis, babies remained in the NICU for treatment or were discharged home.

Figure 1 Flow diagram describing the protocol of the Pulsimat study for the screening of critical congenital heart disease. PO: pulse oximetry. Included patients were screened between 24-48 h after birth, following the protocol shown in Figure 1. Newborn infants with a positive or doubtful screening were admitted to the Neonatal Intensive Care Unit (NICU) where the clinical, analytical, and echocardiographic study was completed by neonatologists and pediatric cardiologists. After reaching a diagnosis, babies remained in the NICU for treatment or were discharged home.

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Table 1. Types of Critical Congenital Heart Disease and Associated International Classification of Disease, 10th Version (ICD-10) Diagnosis Codes. The present review summarizes and critiques previously published estimates of the costs or cost-effectiveness of CCHD screening—two UK studies in four publications [12-14,17], three US studies in four publications [10,21—23], and one study from China [20]. Updated estimates of costs, outcomes, and summary measures of the economic value of CCHD screening in the United States are also presented. The new estimates address future disease-related treatment costs, shortened life expectancy among CCHD survivors, and a higher estimated number of avoided infant deaths due to screening.
Table 1. Types of Critical Congenital Heart Disease and Associated International Classification of Disease, 10th Version (ICD-10) Diagnosis Codes. The present review summarizes and critiques previously published estimates of the costs or cost-effectiveness of CCHD screening—two UK studies in four publications [12-14,17], three US studies in four publications [10,21—23], and one study from China [20]. Updated estimates of costs, outcomes, and summary measures of the economic value of CCHD screening in the United States are also presented. The new estimates address future disease-related treatment costs, shortened life expectancy among CCHD survivors, and a higher estimated number of avoided infant deaths due to screening.
Note: all currency conversions were calculated using the purchasing power parity exchange rate for the year of the original cost estimate. Source: https: / /data.oecd.org /conversion/purchasing-power-parities-ppp.htm. Table 2. Summary of data assumptions of micro-costing studies of critical congenital heart disease screening.
Note: all currency conversions were calculated using the purchasing power parity exchange rate for the year of the original cost estimate. Source: https: / /data.oecd.org /conversion/purchasing-power-parities-ppp.htm. Table 2. Summary of data assumptions of micro-costing studies of critical congenital heart disease screening.
CHD: congenital heart disease; CCHD: critical congenital heart disease. Table 1. Pros and cons of screening before and after 24 h of age.
CHD: congenital heart disease; CCHD: critical congenital heart disease. Table 1. Pros and cons of screening before and after 24 h of age.
Table 2. Formalised screening program versus vital sign documentation. Importantly, the detailed requirements needed to satisfy inclusion as a formal country-wide screening test are not needed: these requirements can result in significant delays in the introduction of a screening program. The downside of this approach is that there may not be true nationwide screening, particularly at smaller, under-resourced hospitals. The approach may result in a less-uniform approach and lack of a formalized collection of results to understand the impact of screening. In contrast, a formal application to include pulse oximetry screening for CCHD as a part of a country screening program results in proper resourcing, oversight, and governance. It is more likely that all babies at all levels will be screened. However, the process takes time (5 years and still proceeding in the case of the United Kingdom) and will still suffer from all of the issues discussed above when pulse oximetry measures are linked to screening for CCHD. The Nordic countries have been successful in the approach Figure 1. Pulse oximetry screening: A new paradigm.
Table 2. Formalised screening program versus vital sign documentation. Importantly, the detailed requirements needed to satisfy inclusion as a formal country-wide screening test are not needed: these requirements can result in significant delays in the introduction of a screening program. The downside of this approach is that there may not be true nationwide screening, particularly at smaller, under-resourced hospitals. The approach may result in a less-uniform approach and lack of a formalized collection of results to understand the impact of screening. In contrast, a formal application to include pulse oximetry screening for CCHD as a part of a country screening program results in proper resourcing, oversight, and governance. It is more likely that all babies at all levels will be screened. However, the process takes time (5 years and still proceeding in the case of the United Kingdom) and will still suffer from all of the issues discussed above when pulse oximetry measures are linked to screening for CCHD. The Nordic countries have been successful in the approach Figure 1. Pulse oximetry screening: A new paradigm.
Table 1. Overview of screening parameters for pilot and accuracy study 6. Costs Before screening programs can be recommended for universal implementation, cost effectiveness should be considered. Cost analyses have shown that the PO screening is likely to be cost effective, but only screening in hospitals was taken into account [3,4,16]. The outcome of children after pediatric cardiac surgery has considerably improved in the last decades, but recent data on gained quality-adjusted life years (QALYs) are lacking. However, it is known that a timely diagnosis of CCHD decreases the risk of mortality and morbidity, and also the length of hospital stay [17,18]. ee | ee? ee? ee ee 1]... ...5... 2 4 ee, ee” ae In order to assess the accuracy of the adapted PO screening, we performed an implementatior study in a larger cohort in a much larger region (Leiden—Amsterdam Region (POLAR) study) [15] This study was carried out in three academic hospitals, 11 regional hospitals and 75 midwifery practices and included 23,959 newborns. The prenatal detection rate was 73% and the sensitivity was 50%, with a specificity of 99%. Four out of five detected CCHD were identified in the first screening test, in the first hours after birth. The fifth CCHD was diagnosed at 12 h after birth, on the day after the date of birth (day 2 of life). Serious illnesses such as infections and respiratory pathology were detected in 61% of all newborns with false positive screening results (Table 1). This study demonstrated thai PO screening adapted to home births and early post-delivery hospital discharge contributes to the detection of CCHD in an early, asymptomatic stage. The early detection of CCHD, but also othe: significant pathologies, such as infections and respiratory morbidity, could be considered as a safety net when newborns are born at home or early discharged after delivery in hospital. In that view, the PC screening has the potential to decrease morbidity and mortality of newborns in The Netherlands.
Table 1. Overview of screening parameters for pilot and accuracy study 6. Costs Before screening programs can be recommended for universal implementation, cost effectiveness should be considered. Cost analyses have shown that the PO screening is likely to be cost effective, but only screening in hospitals was taken into account [3,4,16]. The outcome of children after pediatric cardiac surgery has considerably improved in the last decades, but recent data on gained quality-adjusted life years (QALYs) are lacking. However, it is known that a timely diagnosis of CCHD decreases the risk of mortality and morbidity, and also the length of hospital stay [17,18]. ee | ee? ee? ee ee 1]... ...5... 2 4 ee, ee” ae In order to assess the accuracy of the adapted PO screening, we performed an implementatior study in a larger cohort in a much larger region (Leiden—Amsterdam Region (POLAR) study) [15] This study was carried out in three academic hospitals, 11 regional hospitals and 75 midwifery practices and included 23,959 newborns. The prenatal detection rate was 73% and the sensitivity was 50%, with a specificity of 99%. Four out of five detected CCHD were identified in the first screening test, in the first hours after birth. The fifth CCHD was diagnosed at 12 h after birth, on the day after the date of birth (day 2 of life). Serious illnesses such as infections and respiratory pathology were detected in 61% of all newborns with false positive screening results (Table 1). This study demonstrated thai PO screening adapted to home births and early post-delivery hospital discharge contributes to the detection of CCHD in an early, asymptomatic stage. The early detection of CCHD, but also othe: significant pathologies, such as infections and respiratory morbidity, could be considered as a safety net when newborns are born at home or early discharged after delivery in hospital. In that view, the PC screening has the potential to decrease morbidity and mortality of newborns in The Netherlands.
Figure 1. Pulse oximetry screening in Germany in December 2009. Green bars indicate the percentage of perinatal and neonatal units performing pulse oximetry screening as per December 2009 in five regions in Germany. The black lines indicate the level of significance (dotted line: p < 0.05, dashed line: p < 0.01, solid line, p < 0.001). The asterisk indicates the federal state of Saxony. Source of the map: adaptation from svg/2000px-Germany_location_map.svg.png; author: NordNordWest; http:/ /creativecommons.org /licenses/by-sa/3.0/de/legalcode. In December 2009, a nationwide survey on pulse oximetry screening in Germany was conducted. A questionnaire was sent to all 890 perinatal and neonatal units; the response rate was 29% (n = 255). In 46% of the responding units, pulse oximetry screening had already been established. Although, in the majority of cases, it had only been established in the last three years. However, the percentage of units performing pulse oximetry screening showed remarkable regional differences, possibly at least in part related to the regional effects of the activities in Saxony (Figure 1).
Figure 1. Pulse oximetry screening in Germany in December 2009. Green bars indicate the percentage of perinatal and neonatal units performing pulse oximetry screening as per December 2009 in five regions in Germany. The black lines indicate the level of significance (dotted line: p < 0.05, dashed line: p < 0.01, solid line, p < 0.001). The asterisk indicates the federal state of Saxony. Source of the map: adaptation from svg/2000px-Germany_location_map.svg.png; author: NordNordWest; http:/ /creativecommons.org /licenses/by-sa/3.0/de/legalcode. In December 2009, a nationwide survey on pulse oximetry screening in Germany was conducted. A questionnaire was sent to all 890 perinatal and neonatal units; the response rate was 29% (n = 255). In 46% of the responding units, pulse oximetry screening had already been established. Although, in the majority of cases, it had only been established in the last three years. However, the percentage of units performing pulse oximetry screening showed remarkable regional differences, possibly at least in part related to the regional effects of the activities in Saxony (Figure 1).
Figure 2. Pulse oximetry screening and level of neonatal care. Green bars indicate percentage of units using pulse oximetry screening as per December 2009 in Germany. Black lines indicate level of significance (dashed line: p < 0.01, solid line, p < 0.001). In light of the increasing body of evidence on the benefits of pulse oximetry screening [11,12], the German Society for Paediatric Cardiology supported the use of pulse oximetry screening in 2011 and formulated a statement with recommendations for practical aspects of its implementation in 2013 [13,14]. Likewise, the German Society for Neonatology and Paediatric intensive care (GNPI) recommended the use of pulse oximetry screening in its guidelines for neonatal care in 2012 [15]. However, the effects of these nonbinding recommendations remained unknown.
Figure 2. Pulse oximetry screening and level of neonatal care. Green bars indicate percentage of units using pulse oximetry screening as per December 2009 in Germany. Black lines indicate level of significance (dashed line: p < 0.01, solid line, p < 0.001). In light of the increasing body of evidence on the benefits of pulse oximetry screening [11,12], the German Society for Paediatric Cardiology supported the use of pulse oximetry screening in 2011 and formulated a statement with recommendations for practical aspects of its implementation in 2013 [13,14]. Likewise, the German Society for Neonatology and Paediatric intensive care (GNPI) recommended the use of pulse oximetry screening in its guidelines for neonatal care in 2012 [15]. However, the effects of these nonbinding recommendations remained unknown.
Figure 3. Algorithm for pulse oximetry screening in Germany as recommended by the Common Federal Committee (Gemeinsamer Bundesausschuss, G-BA), modified after [19] by a specialist in paediatrics, ideally with subspecialty training in neonatology /paediatric cardiology.
Figure 3. Algorithm for pulse oximetry screening in Germany as recommended by the Common Federal Committee (Gemeinsamer Bundesausschuss, G-BA), modified after [19] by a specialist in paediatrics, ideally with subspecialty training in neonatology /paediatric cardiology.
Timeline of U.S. Implementation
Timeline of U.S. Implementation
Table A1. Showing Components for Successful U.S. Implementation & Barriers Addressed.
Table A1. Showing Components for Successful U.S. Implementation & Barriers Addressed.
SELU Ie OOO Doha UY Ute be eats 2 | UL OLALE VV IMA OL LET aa L4V]- Based on our experience and the literature, it was estimated that pulse oximetry would detect CCHD in 5 to 7 Tennessee infants who would otherwise be missed. It was also suggested that the upper extremity pulse oximetry reading would be unnecessary if an initial foot pulse oximetry reading was 97% or higher, since it would be impossible to have a difference of greater than 3%. Therefore, the Genetics Advisory Committee of the state of Tennessee presented to the Commissioner of Health a modified Tennessee algorithm with an initial assessment of a single lower extremity reading, which, if 97% or higher a second, upper extremity, test was not required. Figure 1 The two-year results of this screening algorithm are presented in this report. Figure 1. Foot only screen, if the pulse oximetry level is 97% or higher, the test is a pass and no upper extremity result is obtained, if the level is less than 90% the infant fails and is referred for further evaluation. A measurement of 90-96% results in a default to the AHA, CDC algorithm. 2. Materials and Methods
SELU Ie OOO Doha UY Ute be eats 2 | UL OLALE VV IMA OL LET aa L4V]- Based on our experience and the literature, it was estimated that pulse oximetry would detect CCHD in 5 to 7 Tennessee infants who would otherwise be missed. It was also suggested that the upper extremity pulse oximetry reading would be unnecessary if an initial foot pulse oximetry reading was 97% or higher, since it would be impossible to have a difference of greater than 3%. Therefore, the Genetics Advisory Committee of the state of Tennessee presented to the Commissioner of Health a modified Tennessee algorithm with an initial assessment of a single lower extremity reading, which, if 97% or higher a second, upper extremity, test was not required. Figure 1 The two-year results of this screening algorithm are presented in this report. Figure 1. Foot only screen, if the pulse oximetry level is 97% or higher, the test is a pass and no upper extremity result is obtained, if the level is less than 90% the infant fails and is referred for further evaluation. A measurement of 90-96% results in a default to the AHA, CDC algorithm. 2. Materials and Methods
Figure 2. Consort diagram describing eligible patients, disregarded, and finally screened for critical congenital heart disease (CCHD) in the Pulsimat study. Figure 2 shows the flow diagram of the study. From a total of 11,531 newborn infants born in the participating hospitals, 66 babies were diagnosed with CCHD, representing 5.8 per thousand newborn infants. Out of these, 36 patients had CCHD detected in utero and confirmed at birth (3.1 per 1000 births), 27 were diagnosed after birth based on clinical symptoms and echocardiography (2.3 per 1000 births), and three were detected by pulse oximetry screening at discharge (0.26 per thousand live births). A total of 8856 representing 78.6% of all births were screened for critical CCHD. Out of these, five had a positive pulse oximetry screen. However, two cases were caused by respiratory distress secondary to early onset sepsis; thus, only the remaining three cases were due to CCDH, specifically, total anomalous venous return.
Figure 2. Consort diagram describing eligible patients, disregarded, and finally screened for critical congenital heart disease (CCHD) in the Pulsimat study. Figure 2 shows the flow diagram of the study. From a total of 11,531 newborn infants born in the participating hospitals, 66 babies were diagnosed with CCHD, representing 5.8 per thousand newborn infants. Out of these, 36 patients had CCHD detected in utero and confirmed at birth (3.1 per 1000 births), 27 were diagnosed after birth based on clinical symptoms and echocardiography (2.3 per 1000 births), and three were detected by pulse oximetry screening at discharge (0.26 per thousand live births). A total of 8856 representing 78.6% of all births were screened for critical CCHD. Out of these, five had a positive pulse oximetry screen. However, two cases were caused by respiratory distress secondary to early onset sepsis; thus, only the remaining three cases were due to CCDH, specifically, total anomalous venous return.
Table 1. Screening for congenital cardiac heart defects: analysis of results. 4. Discussion It has been estimated that 30% of all CCHD patients have a late diagnosis and could benefit from neonatal screening [16]. As most previous studies demonstrated that non-cardiac conditions could also be detected by pulse oximetry, false positive detection can represent another advantage of the screening [13,17]. In our study, the detection of CCHD in utero (fetal echocardiography) plus ex utero after clinical examination represented 95.5% of the total, while 4.5% of the babies with CCHD were detected by pre-discharge screening. Interestingly, two patients that would have been discharged home with early onset sepsis had a positive screening and were admitted to the NICU. In June 2013, an international group of neonatologists and cardiologists met in Torino (Italy) with the chief investigators from two European screening studies of pulse oximetry to discuss strategies to
Table 1. Screening for congenital cardiac heart defects: analysis of results. 4. Discussion It has been estimated that 30% of all CCHD patients have a late diagnosis and could benefit from neonatal screening [16]. As most previous studies demonstrated that non-cardiac conditions could also be detected by pulse oximetry, false positive detection can represent another advantage of the screening [13,17]. In our study, the detection of CCHD in utero (fetal echocardiography) plus ex utero after clinical examination represented 95.5% of the total, while 4.5% of the babies with CCHD were detected by pre-discharge screening. Interestingly, two patients that would have been discharged home with early onset sepsis had a positive screening and were admitted to the NICU. In June 2013, an international group of neonatologists and cardiologists met in Torino (Italy) with the chief investigators from two European screening studies of pulse oximetry to discuss strategies to
Table 1. Baseline subject characteristics (1 = 214).
Table 1. Baseline subject characteristics (1 = 214).
Diagnoses are abbreviated: HLHS, hypoplastic left heart syndrome; TOF, tetralogy of Fallot; TGA, transposition of the great arteries; TA, truncus arteriosus; TAPVR, total anomalous pulmonary venous return; TVA, tricuspid valve atresia; PA/IVS, pulmonary valve atresia with intact ventricular septum; CCHD, critical congenital heart disease. + p< 0.05. Figure 1. Pre- and post-ductal pulse oximetry data at 24, 28, 36, and 48 h of life (n= 158). Mean saturation at each time point indicated by the diamond on each box plot.
Diagnoses are abbreviated: HLHS, hypoplastic left heart syndrome; TOF, tetralogy of Fallot; TGA, transposition of the great arteries; TA, truncus arteriosus; TAPVR, total anomalous pulmonary venous return; TVA, tricuspid valve atresia; PA/IVS, pulmonary valve atresia with intact ventricular septum; CCHD, critical congenital heart disease. + p< 0.05. Figure 1. Pre- and post-ductal pulse oximetry data at 24, 28, 36, and 48 h of life (n= 158). Mean saturation at each time point indicated by the diamond on each box plot.
Table 1. Cont.
Table 1. Cont.
Figure 2. Pulse oximetry by saturation group at 24, 28, 36, and 48 h of life (n = 158).
Figure 2. Pulse oximetry by saturation group at 24, 28, 36, and 48 h of life (n = 158).
Table 2. Pre- and post-ductal pulse oximetry readings, PGE administration, and oxygen supplementation data at 24, 28, 36, and 48 h of life (n = 158). * p < 0.05. 3.2. Secondary Analysis: Hypothetical Screening of Study Cohort
Table 2. Pre- and post-ductal pulse oximetry readings, PGE administration, and oxygen supplementation data at 24, 28, 36, and 48 h of life (n = 158). * p < 0.05. 3.2. Secondary Analysis: Hypothetical Screening of Study Cohort
Figure 3. Application of the American Academy of Pediatrics (AAP) protocol to retrospective data in newborns with known CCHD (n = 149) results in 120 true positive screens and 80.5% sensitivity. % Exclusion criteria: insufficient pulse oximetry data in the electronic health record (EHR), death prior to 24h of life, atrial septostomy or surgery prior to 24h of life. © Inadequate number of pulse oximetry readings to complete the AAP protocol. applied to 149 subjects as 9 of the 158 subjects were excluded due to an inadequate number of pulse oximetry readings to apply the AAP protocol (Figure 3). Twenty-nine subjects who screened negative with passing pulse oximetry readings resulted in a 19.5% hypothetical false negative screening rate in our cohort. The 29 false negative screened subjects represented all diagnosis categories. The false negative screening rate was 18.8% (n = 80) in subjects with a prenatal concern for CCHD and 20.3% (n = 69) in subjects without a prenatal concern for CCHD.
Figure 3. Application of the American Academy of Pediatrics (AAP) protocol to retrospective data in newborns with known CCHD (n = 149) results in 120 true positive screens and 80.5% sensitivity. % Exclusion criteria: insufficient pulse oximetry data in the electronic health record (EHR), death prior to 24h of life, atrial septostomy or surgery prior to 24h of life. © Inadequate number of pulse oximetry readings to complete the AAP protocol. applied to 149 subjects as 9 of the 158 subjects were excluded due to an inadequate number of pulse oximetry readings to apply the AAP protocol (Figure 3). Twenty-nine subjects who screened negative with passing pulse oximetry readings resulted in a 19.5% hypothetical false negative screening rate in our cohort. The 29 false negative screened subjects represented all diagnosis categories. The false negative screening rate was 18.8% (n = 80) in subjects with a prenatal concern for CCHD and 20.3% (n = 69) in subjects without a prenatal concern for CCHD.
Related topics:
Congenital Heart DefectsPulse OximetryHome BirthsCoarctation of Aorta
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