Received : 22 October 2014 Accepted : 28 April 2015 Available online 2 July 2015
Available online at
ScienceDirect www.sciencedirect.com
Clinical case Fermeture du canal arte´riel et e´valuation de l’oxyge´nation re´gionale par spectroscopie dans le proche infra-rouge dans les cardiopathies conge´nitales ducto-de´pendantes P.-E. Se´gue´la*, E. Guillet, J.-B. Thambo, P. Mauriat Service de cardiologie pe´diatrique et conge´nitale, hoˆpital Haut-Le´ve`que, CHU de Bordeaux,
Ductal closure and near-infrared spectroscopy for regional oxygenation monitoring in ductus-dependent congenital heart disease
avenue de Magellan, 33604 Pessac cedex, France
Summary
Re´sume´
In ductus-dependent congenital heart disease, preserving the blood flow through the ductus arteriosus (DA) is vital before surgery. We present the cases of three full-term neonates with ductus-dependent congenital heart disease for whom near-infrared spectroscopy (NIRS) monitoring was performed. We recorded cyclical drops in regional oxygen saturation, both cerebral and renal, that corresponded to constrictions of the DA. These findings appeared either simultaneously or previous to SpO2 drops and were corrected by prostaglandin infusion. Through these cases, we assume that cyclical constrictions of ductal cells participate in the DA closure process in its early phase. ß 2015 Elsevier Masson SAS. All rights reserved.
En cas de cardiopathie conge´nitale ducto-de´pendante, il faut maintenir perme´able le canal arte´riel (CA) en attendant la chirurgie. Nous pre´sentons les cas de trois nouveau-ne´s a` terme ayant une cardiopathie ducto-de´pendante pour lesquels l’utilisation de la spectroscopie en proches infrarouges (NIRS) a permis de mettre en e´vidence des diminutions cycliques de l’oxyge´nation re´gionale, aussi bien ce´re´brale que re´nale. Ces chutes de valeur de la NIRS correspondaient a` des constrictions du CA et e´taient observe´es en meˆme temps, ou un peu avant, la diminution des valeurs de saturation pe´riphe´rique. Ces alte´rations ont e´te´ corrige´es par l’administration de prostaglandine intraveineuse. A` travers ces cas, nous e´mettons l’hypothe`se que des constrictions cycliques des cellules ductales participent au processus initial de fermeture du CA. ß 2015 Elsevier Masson SAS. Tous droits re´serve´s.
1. Introduction
ductus-dependent congenital heart disease, preserving the blood flow through the DA is essential for the infant’s survival, before surgery. To maintain it open, prostaglandins are very effective, particularly since they are administered shortly after birth [3]. However, prostaglandin treatment entails certain difficulties. Fever, apnea, hypotension, vasodilatation, and diarrhea have been reported among the frequent short-term side effects using the standard dose [4]. Therefore, this treatment is initiated only when necessary. Preoperative evaluation of neonatal ductus-dependent congenital heart disease is classically based on clinical, biological, and echocardiographic findings. While conventional cardiovascular
By connecting the proximal descending aorta to the roof of the main pulmonary artery, the ductus arteriosus (DA) is an essential fetal structure that normally closes spontaneously after birth [1]. Its closure is described as occurring in two consecutive phases: a functional closure in the first days of life and an anatomical occlusion over the next few days [2]. In
* Corresponding author. e-mail: [email protected] (P.-E. Se´gue´la). http://dx.doi.org/10.1016/j.arcped.2015.04.020 Archives de Pe´diatrie 2015;22:857-860 0929-693X/ß 2015 Elsevier Masson SAS. All rights reserved.
857
Archives de Pe´diatrie 2015;22:857-860
P.-E. Se´gue´la et al.
monitoring does not detect tissue hypoxia, near-infrared spectroscopy (NIRS) (fig. 1) provides non-invasive monitoring of regional oxygen saturation (rSO2) within a wide range of emergency scenarios. NIRS correlates well with venous blood O2 saturation and is an increasingly used method for the measurement of tissue oxygenation. Through the cases of three newborns, we report the particular advantages of rSO2 that we observed during spontaneous ductal closure in cyanotic ductus-dependent congenital heart disease.
2. Case reports 2.1. Patient #1 A full-term male newborn weighing 3.245 kg was referred to our department due to isolated cyanosis observed at 4 h of life. Peripheral oxygen saturation of hemoglobin, measured by pulse oximetry (SpO2), was 88%. Transthoracic echocardiography revealed severe pulmonary valve stenosis and a hypertrophic right ventricle with moderate tricuspid regurgitation. The atrial septal defect was shunting right-to-left and a large DA (2.5 mm) was initially shunting left-to-right. Cerebral and renal rSO2 were monitored using NIRS technology (INVOSW system, Somanetics Corporation, Troy, MI, USA). Prostaglandin E1 (PGE1) infusion was intentionally not started immediately in order to evaluate the ductus-dependency of this cardiac disease. At 6 h of life, cyclical losses of the SpO2 signal and drops in rSO2 (from 80 to 20%) were simultaneously observed. These findings were associated on echocardiography with constriction of the DA (2.2 mm in diameter when rSO2 was 80% versus 0.5 mm in diameter when rSO2 was 20%). Thus, the patient received an intravenous PGE1 infusion (25 ng/kg/ min) to keep the DA open. PGE1 maintained SpO2 above 92% and adequate rSO2 values (80% for renal oxygenation and
70% for cerebral oxygenation). The pulmonary valve was successfully dilated by cardiac catheterization on day 2.
2.2. Patient #2 A full-term female newborn weighing 3.320 kg, who had been prenatally diagnosed with pulmonary atresia associated with a ventricular septal defect, was referred for postnatal management. PGE1 was started just after delivery, through an umbilical venous catheter and SpO2 was 95% on admission. Postnatal echocardiography confirmed the diagnosis of pulmonary atresia with a type 1 ventricular septal defect (native pulmonary arteries present). The DA was large (3 mm) and tortuous, with no aortopulmonary collateral artery. Because this type of extremely malformative DA may sometimes never close spontaneously, PGE1 infusion was interrupted under careful monitoring. On day 7, reintroduction of PGE1 (50 ng/ kg/min) for severe hypoxemia (SpO2 55%) was instantaneously effective, as shown by a SpO2 of 85%. A careful analysis of NIRS curves showed that rSO2 started to decrease significantly (down to 20%), with a cyclical pattern, several minutes before SpO2 dropped (fig. 2). A modified BlalockTaussig shunt was then performed, allowing an early discharge from the hospital.
2.3. Patient #3 A full-term male newborn (3.720 kg) with a prenatal diagnosis of transposition of the great arteries was admitted immediately after delivery. SpO2 was 57%. Given that he had no breathing difficulty, he did not require ventilatory support. The atrial septal defect was restrictive (2 mm in diameter), so a percutaneous atrioseptostomy was performed 2 h after birth. Post-procedure SpO2 was 90%. Because the DA was initially large (3 mm) and atrial mixing seemed to be efficient,
Figure 1. Operating principle of near-infrared spectroscopy (NIRS). Near-infrared light photons are produced by a light-emitting source placed on the patient’s skin. The photons penetrate the body tissue of interest (cerebral tissue in our example) and some fraction of them is reflected toward the detectors. By measuring the quantity of returning photons as a function of wavelength, the spectral absorption of the underlying tissue is measured and the relative amounts of hemoglobin and oxygenated hemoglobin are calculated, giving the regional oxygen saturation.
858
NIRS in ductus-dependent congenital heart disease
Figure 2. Regional oxygen saturation (rSO2) monitoring of patients with ductus-dependent congenital heart disease. rSO2 monitoring showed regular and cyclical drops in tissue oxygenation (every 6–9 min, depending on the patients). Patient #1 had a severe pulmonary valve stenosis. When the rSO2 values were normal, the ductus arteriosus was wide open on echocardiography, whereas drops in rSo2 were contemporaneous with ductus arteriosus constriction. Patient #2 had pulmonary atresia with a ventricular septal defect. Patient #3 had a transposition of the great arteries. PGE1 infusion allowed restoring normal rSO2 values. PGE1: prostaglandin E1.
no first-intention PGE1 infusion was started. Twelve hours after birth, NIRS showed deep and cyclical decreases in saturation of rSO2 in both cerebral and renal tissues, whereas a stable SpO2 value (80%) was recorded (fig. 2). Echocardiography showed nearly full closure of the DA and the serum lactate level began to increase (3 mmol/L). PGE1 was started and normal rSO2 values were recovered, as was the serum lactate level. An arterial switch operation was performed on day 3 with a good outcome.
3. Discussion Previous studies have shown that, after birth, closure of the DA occurs due to several factors. The initial functional closure is responsible for local hypoxia that induces cell apoptosis, endothelial proliferation, extracellular matrix production, and fibrosis. Finally, these phenomena result in a definitive anatomical occlusion. Our observations suggest that the functional constriction of the DA probably follows a cyclical pattern, consisting in alternate phases of closure and opening. This
hypothesis is supported by the fact that, when performed in patient #1, echocardiography showed a reduced ductal flow when NIRS values were the lowest and a higher flow when rSO2 values were normal. In ductus-dependent congenital heart disease, the patency of the DA is necessary to maintain a sufficient pulmonary or systemic blood flow. In patients #2 and 3, the monitoring of tissue oxygenation using NIRS allowed the early detection of ductal closure. While the periodic cyanosis was difficult to demonstrate using conventional monitoring because of a complete loss of the signal, rSO2 curves showed it perfectly. Moreover, rSO2 monitoring has been previously proved to be more effective for the detection of both hypoxia and tissue oxygenation modifications than SpO2 [5,6]. Finally, NIRS is useful to evaluate the effectiveness of therapeutics. Adequate tissue oxygenation thus correlated with the use of prostaglandins. These cases also illustrate the difficulty of managing ductus-dependent congenital cardiac disease in the neonatal period and show the crucial importance of reliable cardiovascular monitoring in this context. We decided to assess the ductus-dependency of our patients (by either suspending or
859
Archives de Pe´diatrie 2015;22:857-860
P.-E. Se´gue´la et al.
waiting before starting PGE1 infusion), and NIRS seemed to be highly valuable for monitoring this situation.
References [1]
4. Conclusion Postnatal closure of the DA is governed by various mechanisms that today remain incompletely known. In patients with ductus-dependent congenital heart disease, the monitoring of rSO2 using NIRS may be complementary to the measurement of SpO2. Indeed, this technique may help detect ductal closure early, thus helping make therapeutic decisions.
[4]
Disclosure of interest
[5]
The authors declare that they have no conflicts of interest concerning this article. Financial disclosure: none.
860
[2] [3]
[6]
Desligneres S, Larroche JC. Ductus arteriosus. I. Anatomical and histological study of its development during the second half of gestation and its closure after birth. II. Histological study of a few cases of patent ductus arteriosus in infancy. Biol Neonat 1970;16:278–96. Coceani F, Baragatti B. Mechanisms for ductus arteriosus closure. Semin Perinatol 2012;36:92–7. Kramer HH, Sommer M, Rammos S, et al. Evaluation of low dose prostaglandin E1 treatment for ductus dependent congenital heart disease. Eur J Pediatr 1995;154:700–7. Lewis AB, Freed MD, Heymann MA, et al. Side effects of therapy with prostaglandin E1 in infants with critical congenital heart disease. Circulation 1981;64:893–8. Ricci Z, Garisto C, Favia I, et al. Cerebral NIRS as a marker of superior vena cava oxygen saturation in neonates with congenital heart disease. Paediatr Anaesth 2010;20:1040–5. Giliberti P, Mondı` V, Conforti A, et al. Near infrared spectroscopy in newborns with surgical disease. J Matern Fetal Neonatal Med 2011;24(Suppl. 1):56–8.
Available online at
ScienceDirect www.sciencedirect.com
Clinical case Fermeture du canal arte´riel et e´valuation de l’oxyge´nation re´gionale par spectroscopie dans le proche infra-rouge dans les cardiopathies conge´nitales ducto-de´pendantes P.-E. Se´gue´la*, E. Guillet, J.-B. Thambo, P. Mauriat Service de cardiologie pe´diatrique et conge´nitale, hoˆpital Haut-Le´ve`que, CHU de Bordeaux,
Ductal closure and near-infrared spectroscopy for regional oxygenation monitoring in ductus-dependent congenital heart disease
avenue de Magellan, 33604 Pessac cedex, France
Summary
Re´sume´
In ductus-dependent congenital heart disease, preserving the blood flow through the ductus arteriosus (DA) is vital before surgery. We present the cases of three full-term neonates with ductus-dependent congenital heart disease for whom near-infrared spectroscopy (NIRS) monitoring was performed. We recorded cyclical drops in regional oxygen saturation, both cerebral and renal, that corresponded to constrictions of the DA. These findings appeared either simultaneously or previous to SpO2 drops and were corrected by prostaglandin infusion. Through these cases, we assume that cyclical constrictions of ductal cells participate in the DA closure process in its early phase. ß 2015 Elsevier Masson SAS. All rights reserved.
En cas de cardiopathie conge´nitale ducto-de´pendante, il faut maintenir perme´able le canal arte´riel (CA) en attendant la chirurgie. Nous pre´sentons les cas de trois nouveau-ne´s a` terme ayant une cardiopathie ducto-de´pendante pour lesquels l’utilisation de la spectroscopie en proches infrarouges (NIRS) a permis de mettre en e´vidence des diminutions cycliques de l’oxyge´nation re´gionale, aussi bien ce´re´brale que re´nale. Ces chutes de valeur de la NIRS correspondaient a` des constrictions du CA et e´taient observe´es en meˆme temps, ou un peu avant, la diminution des valeurs de saturation pe´riphe´rique. Ces alte´rations ont e´te´ corrige´es par l’administration de prostaglandine intraveineuse. A` travers ces cas, nous e´mettons l’hypothe`se que des constrictions cycliques des cellules ductales participent au processus initial de fermeture du CA. ß 2015 Elsevier Masson SAS. Tous droits re´serve´s.
1. Introduction
ductus-dependent congenital heart disease, preserving the blood flow through the DA is essential for the infant’s survival, before surgery. To maintain it open, prostaglandins are very effective, particularly since they are administered shortly after birth [3]. However, prostaglandin treatment entails certain difficulties. Fever, apnea, hypotension, vasodilatation, and diarrhea have been reported among the frequent short-term side effects using the standard dose [4]. Therefore, this treatment is initiated only when necessary. Preoperative evaluation of neonatal ductus-dependent congenital heart disease is classically based on clinical, biological, and echocardiographic findings. While conventional cardiovascular
By connecting the proximal descending aorta to the roof of the main pulmonary artery, the ductus arteriosus (DA) is an essential fetal structure that normally closes spontaneously after birth [1]. Its closure is described as occurring in two consecutive phases: a functional closure in the first days of life and an anatomical occlusion over the next few days [2]. In
* Corresponding author. e-mail: [email protected] (P.-E. Se´gue´la). http://dx.doi.org/10.1016/j.arcped.2015.04.020 Archives de Pe´diatrie 2015;22:857-860 0929-693X/ß 2015 Elsevier Masson SAS. All rights reserved.
857
Archives de Pe´diatrie 2015;22:857-860
P.-E. Se´gue´la et al.
monitoring does not detect tissue hypoxia, near-infrared spectroscopy (NIRS) (fig. 1) provides non-invasive monitoring of regional oxygen saturation (rSO2) within a wide range of emergency scenarios. NIRS correlates well with venous blood O2 saturation and is an increasingly used method for the measurement of tissue oxygenation. Through the cases of three newborns, we report the particular advantages of rSO2 that we observed during spontaneous ductal closure in cyanotic ductus-dependent congenital heart disease.
2. Case reports 2.1. Patient #1 A full-term male newborn weighing 3.245 kg was referred to our department due to isolated cyanosis observed at 4 h of life. Peripheral oxygen saturation of hemoglobin, measured by pulse oximetry (SpO2), was 88%. Transthoracic echocardiography revealed severe pulmonary valve stenosis and a hypertrophic right ventricle with moderate tricuspid regurgitation. The atrial septal defect was shunting right-to-left and a large DA (2.5 mm) was initially shunting left-to-right. Cerebral and renal rSO2 were monitored using NIRS technology (INVOSW system, Somanetics Corporation, Troy, MI, USA). Prostaglandin E1 (PGE1) infusion was intentionally not started immediately in order to evaluate the ductus-dependency of this cardiac disease. At 6 h of life, cyclical losses of the SpO2 signal and drops in rSO2 (from 80 to 20%) were simultaneously observed. These findings were associated on echocardiography with constriction of the DA (2.2 mm in diameter when rSO2 was 80% versus 0.5 mm in diameter when rSO2 was 20%). Thus, the patient received an intravenous PGE1 infusion (25 ng/kg/ min) to keep the DA open. PGE1 maintained SpO2 above 92% and adequate rSO2 values (80% for renal oxygenation and
70% for cerebral oxygenation). The pulmonary valve was successfully dilated by cardiac catheterization on day 2.
2.2. Patient #2 A full-term female newborn weighing 3.320 kg, who had been prenatally diagnosed with pulmonary atresia associated with a ventricular septal defect, was referred for postnatal management. PGE1 was started just after delivery, through an umbilical venous catheter and SpO2 was 95% on admission. Postnatal echocardiography confirmed the diagnosis of pulmonary atresia with a type 1 ventricular septal defect (native pulmonary arteries present). The DA was large (3 mm) and tortuous, with no aortopulmonary collateral artery. Because this type of extremely malformative DA may sometimes never close spontaneously, PGE1 infusion was interrupted under careful monitoring. On day 7, reintroduction of PGE1 (50 ng/ kg/min) for severe hypoxemia (SpO2 55%) was instantaneously effective, as shown by a SpO2 of 85%. A careful analysis of NIRS curves showed that rSO2 started to decrease significantly (down to 20%), with a cyclical pattern, several minutes before SpO2 dropped (fig. 2). A modified BlalockTaussig shunt was then performed, allowing an early discharge from the hospital.
2.3. Patient #3 A full-term male newborn (3.720 kg) with a prenatal diagnosis of transposition of the great arteries was admitted immediately after delivery. SpO2 was 57%. Given that he had no breathing difficulty, he did not require ventilatory support. The atrial septal defect was restrictive (2 mm in diameter), so a percutaneous atrioseptostomy was performed 2 h after birth. Post-procedure SpO2 was 90%. Because the DA was initially large (3 mm) and atrial mixing seemed to be efficient,
Figure 1. Operating principle of near-infrared spectroscopy (NIRS). Near-infrared light photons are produced by a light-emitting source placed on the patient’s skin. The photons penetrate the body tissue of interest (cerebral tissue in our example) and some fraction of them is reflected toward the detectors. By measuring the quantity of returning photons as a function of wavelength, the spectral absorption of the underlying tissue is measured and the relative amounts of hemoglobin and oxygenated hemoglobin are calculated, giving the regional oxygen saturation.
858
NIRS in ductus-dependent congenital heart disease
Figure 2. Regional oxygen saturation (rSO2) monitoring of patients with ductus-dependent congenital heart disease. rSO2 monitoring showed regular and cyclical drops in tissue oxygenation (every 6–9 min, depending on the patients). Patient #1 had a severe pulmonary valve stenosis. When the rSO2 values were normal, the ductus arteriosus was wide open on echocardiography, whereas drops in rSo2 were contemporaneous with ductus arteriosus constriction. Patient #2 had pulmonary atresia with a ventricular septal defect. Patient #3 had a transposition of the great arteries. PGE1 infusion allowed restoring normal rSO2 values. PGE1: prostaglandin E1.
no first-intention PGE1 infusion was started. Twelve hours after birth, NIRS showed deep and cyclical decreases in saturation of rSO2 in both cerebral and renal tissues, whereas a stable SpO2 value (80%) was recorded (fig. 2). Echocardiography showed nearly full closure of the DA and the serum lactate level began to increase (3 mmol/L). PGE1 was started and normal rSO2 values were recovered, as was the serum lactate level. An arterial switch operation was performed on day 3 with a good outcome.
3. Discussion Previous studies have shown that, after birth, closure of the DA occurs due to several factors. The initial functional closure is responsible for local hypoxia that induces cell apoptosis, endothelial proliferation, extracellular matrix production, and fibrosis. Finally, these phenomena result in a definitive anatomical occlusion. Our observations suggest that the functional constriction of the DA probably follows a cyclical pattern, consisting in alternate phases of closure and opening. This
hypothesis is supported by the fact that, when performed in patient #1, echocardiography showed a reduced ductal flow when NIRS values were the lowest and a higher flow when rSO2 values were normal. In ductus-dependent congenital heart disease, the patency of the DA is necessary to maintain a sufficient pulmonary or systemic blood flow. In patients #2 and 3, the monitoring of tissue oxygenation using NIRS allowed the early detection of ductal closure. While the periodic cyanosis was difficult to demonstrate using conventional monitoring because of a complete loss of the signal, rSO2 curves showed it perfectly. Moreover, rSO2 monitoring has been previously proved to be more effective for the detection of both hypoxia and tissue oxygenation modifications than SpO2 [5,6]. Finally, NIRS is useful to evaluate the effectiveness of therapeutics. Adequate tissue oxygenation thus correlated with the use of prostaglandins. These cases also illustrate the difficulty of managing ductus-dependent congenital cardiac disease in the neonatal period and show the crucial importance of reliable cardiovascular monitoring in this context. We decided to assess the ductus-dependency of our patients (by either suspending or
859
Archives de Pe´diatrie 2015;22:857-860
P.-E. Se´gue´la et al.
waiting before starting PGE1 infusion), and NIRS seemed to be highly valuable for monitoring this situation.
References [1]
4. Conclusion Postnatal closure of the DA is governed by various mechanisms that today remain incompletely known. In patients with ductus-dependent congenital heart disease, the monitoring of rSO2 using NIRS may be complementary to the measurement of SpO2. Indeed, this technique may help detect ductal closure early, thus helping make therapeutic decisions.
[4]
Disclosure of interest
[5]
The authors declare that they have no conflicts of interest concerning this article. Financial disclosure: none.
860
[2] [3]
[6]
Desligneres S, Larroche JC. Ductus arteriosus. I. Anatomical and histological study of its development during the second half of gestation and its closure after birth. II. Histological study of a few cases of patent ductus arteriosus in infancy. Biol Neonat 1970;16:278–96. Coceani F, Baragatti B. Mechanisms for ductus arteriosus closure. Semin Perinatol 2012;36:92–7. Kramer HH, Sommer M, Rammos S, et al. Evaluation of low dose prostaglandin E1 treatment for ductus dependent congenital heart disease. Eur J Pediatr 1995;154:700–7. Lewis AB, Freed MD, Heymann MA, et al. Side effects of therapy with prostaglandin E1 in infants with critical congenital heart disease. Circulation 1981;64:893–8. Ricci Z, Garisto C, Favia I, et al. Cerebral NIRS as a marker of superior vena cava oxygen saturation in neonates with congenital heart disease. Paediatr Anaesth 2010;20:1040–5. Giliberti P, Mondı` V, Conforti A, et al. Near infrared spectroscopy in newborns with surgical disease. J Matern Fetal Neonatal Med 2011;24(Suppl. 1):56–8.
