Aberrant left coronary artery from the pulmonary artery with patent ductus arteriosus - a case report and review of the literature.

BACKGROUND: Aberrant left coronary artery from pulmonary artery (ALCAPA) is a very rare congenital heart defect. Its coexistence with patent ductus arteriosus (PDA) is extremely rare. The high pressures created by the left-to-right shunt in the pulmonary arteries can delay symptoms and create a real challenge in diagnosing ALCAPA. Missing this diagnosis can have severe results, including extensive ischemia and sudden death. CASE PRESENTATION: We present a case of an infant born with a large PDA. Initially treated conservatively, however, due to congestive heart failure and lack of weight gain, she underwent surgical ligation of the PDA at the age of four and a half months. Following surgery, she developed pulmonary edema. Echocardiography revealed decreased ventricular function. ECG revealed ST elevations on lateral leads, and serum troponin was significantly increased. The patient underwent cardiac magnetic resonance imaging (MRI), which revealed signs of wall ischemia and decreased function of the left ventricle (LV) with unclear coronary anatomy. Diagnostic catheterization revealed an ALCAPA. She underwent surgical intervention, and the left coronary artery was re-implanted in the aortic sinus. Follow-up revealed slow improvement of cardiac function. DISCUSSION AND LITERATURE REVIEW: The coexistence of PDA and ALCAPA is a very rare occurrence. We found at least 10 reported cases in the literature. Delayed diagnosis might be detrimental. The prognosis of these patients is variable. CONCLUSION: An unusual post-surgical course following PDA repair requires a high index of suspicion and appropriate evaluation for ALCAPA, preferably with angiography.

[Diagnosis and treatment of feline persistent ductus arteriosus: a retrospective evaluation of 17 cats]. / Diagnose und Therapie eines persistierenden Ductus arteriosus: Eine retrospektive Evaluation bei 17 Katzen.

OBJECTIVE: The objective of this study was to evaluate the signalement, clinical features, and echocardiographic findings of cats diagnosed with patent ductus arteriosus (PDA) as well as short- and medium-term outcome after successful ligation of the PDA. MATERIAL AND METHODS: Over a 10-year period 17 cats were diagnosed with PDA by transthoracic echocardiography. Thirteen cats were surgically treated by thoracotomy and ligation of the PDA. RESULTS: In all cats, a heart murmur was detected. In 88.2% of the cases, this presented as grade 4 out of 6 murmur (15/17 cats). A continuous murmur was more common (10/17 cats; 58.9%) than a systolic murmur (7/17 cats; 41.1%). Echocardiography showed that left ventricular internal diameter end diastole (LVIDd) and left ventricular internal diameter end systole (LVIDs) were significantly above reference values in the majority of cats. Mean diameter of the PDA measured at the widest point of the vessel was 3.4 mm (± 1.08 mm) and mean maximum flow velocity amounted to 5,06 m/sec (2,6m/sec-6,4m/sec). Surgery was successfully performed in all cats treated by surgical ligation and all of these patients were discharged after postoperative inpatient therapy. One cat experienced perioperative bleeding from the PDA, which was stopped efficaciously. This cat exhibited a residual shunt directly postoperatively; this could no longer be visualized in a re-check echocardiography 3 months later. Six cats were followed over a longer period of time. CONCLUSIONS: The surgical prognosis in this case study is very good with a postoperative survival rate of 100%. CLINICAL RELEVANCE: Surgical treatment of PDA is curative in animals not displaying advanced cardiac lesions. The auscultation of a heart murmur can provide initial findings indicative of PDA. Therefore, cardiac auscultation is warranted at every first presentation of a kitten. It must however be taken into consideration that not every cat with PDA necessarily has a continuous murmur but may display a systolic heart murmur. Therefore, it is important give utmost attention to the patients' clinical signs.

Massive left pulmonary artery aneurysm with a co-existing patent ductus arteriosus in a five-year-old female child: A case report.

Pulmonary Artery Aneur ysm (PAA), whether congenital or acquired, is a rare diagnostic find ing com pare d to aor tic aneur ysms. There have been fe w cases where PA As were documented as a complication of untreated Patent Ductus Ar teriosus (PDA) due to long-standing Pulmonary Arterial H ypertension (PAH). However, it is quite rare for a case of PAA to be reported with co-existing PDA without PAH. This report highlights a case of a five -year-old girl who was presented with palpitations, easy fatigability, fever, c yanos is, and vomiting. A Chest X-ray s howed mo derate cardiomega ly. A PDA of 6 mm was diagnosed on Transthoracic E chocardiog rap hy ( TTE ) and a large cavity con necte d with LPA raised suspicion of a possible LPA aneur ysm. A Chest CT scan confirm ed the diagnosis of a saccular aneurysm, originating from the distal part of the main Left Pulmonary Artery (LPA) just proximal to the point of bifurcation into lobar branches, measuring 7.5x6.5 cm. During surgery, the aneurysm was opened, emptied with suction and closed without resecting the aneur ysmal walls. The patient had an uneventful post-op course and is doing well during regular interval follow up visits.

Percutaneous closure of severe calcified patent ductus arteriosus with two amplatzer devices.

An 89-year-old woman was referred for closure of a patent ductus arteriosus (PDA). Contrast-computed tomography showed Krichenko type C PDA with severe calcification (Figure 1). Initial angiography revealed severe calcification of the PDA (Figure 2, Video 1), and the mid-ductus diameter was 6 mm and the ductus length was 14 mm..

Patent Ductus Arteriosus Exclusion Technique Using Thoracic Endovascular Aortic Repair.

A large patent ductus arteriosus is an uncommon discovery in an adult. A 2.7-cm patent ductus arteriosus was found in a 31-year-old man with heart failure symptoms. Owing to the size, an occluder device failed to prevent left-to-right shunting, and consideration was given for alternatives to percutaneous closure, including traditional open repair vs thoracic endovascular aortic repair (TEVAR). After a left carotid-subclavian artery bypass was performed, the patient underwent a zone 2 deployment of TEVAR graft. TEVAR exclusion is a useful technique in adults, particularly in the setting of a large or calcified ductus.

Treatment-refractory heart failure as a manifestation of aortic arch atresia.

BACKGROUND: Distal segment atresia (isthmus) is an extremely rare anatomical variant of obstructive aortic arch anomalies. CASE REPORT: We present the case of a newborn who, at 48 hours of life, presented a clinical picture of heart failure. The initial echocardiogram showed a congenital interrupted aortic arch type A, patent ductus arteriosus, and ventricular septal defect. Prostaglandins were initially indicated. Subsequently, a second echocardiogram showed the absence of ductus arteriosus; the CT angiography study confirmed this finding and revealed blood flow to the descending aorta through small intercostal blood vessels. The possibility of atresia of the distal segment (isthmus) of the aortic arch was considered and confirmed at the time of surgery. CONCLUSIONS: Aortic atresia should be considered a diagnostic possibility in the presence of type A interrupted aortic arch since the hemodynamic behavior between them is similar. Surgical medical treatment should be individualized since this condition is frequently an emergency in the neonatal period. However, this is not always the case, as other cases have been reported in schoolchildren and adults.

INTRODUCCIÓN: La atresia de segmento distal (istmo) de arco aórtico es una variante anatómica extremadamente rara de las anomalías obstructivas del arco aórtico. CASO CLÍNICO: Se presenta el caso de un recién nacido que a las 48 horas de vida presentó un cuadro clínico de insuficiencia cardiaca. El estudio de ecocardiograma inicial mostró una anomalía congénita de interrupción de arco aórtico tipo A, conducto arterioso y comunicación interventricular. De inicio se indicaron prostaglandinas. Posteriormente, el segundo ecocardiograma mostró la ausencia del conducto arterioso; el estudio de angiotomografía confirmó este hallazgo y también reveló flujo sanguíneo hacia aorta descendente a través de pequeños vasos sanguíneos intercostales. Se consideró la posibilidad de atresia del segmento distal (istmo) de arco aórtico y se confirmó al momento del acto quirúrgico. CONCLUSIONES: La atresia aórtica debe ser considerada como posibilidad diagnóstica en presencia de interrupción de arco aórtico tipo A, ya que el comportamiento hemodinámico entre ellos es similar. El tratamiento médico quirúrgico debe individualizarse, ya que es frecuente que sea una urgencia en el periodo neonatal. Sin embargo, no sucede así siempre, ya que se han reportado casos en escolares y adultos.

Patent ductus arteriosus in a late preterm neonate: think congenital hypopituitarism.

A late preterm female neonate presented with initial respiratory distress and heart murmur attributed to a haemodynamically significant patent ductus arteriosus (hsPDA) not responding to two courses of ibuprofen. Thyroid function performed for prolonged neonatal jaundice at 3 weeks of life suggested central hypothyroidism. Subsequent adrenocorticotropic hormone stimulation test showing hypocortisolism and MRI revealing adenohypophysis hypoplasia confirmed the diagnosis of congenital hypopituitarism (CH). Commencement of hydrocortisone followed by thyroxine replacement coincided with clinical closure of the hsPDA within 72 hours of treatment. Hypothyroidism and hypocortisolism may have contributed to persistent hsPDA. Thyroid hormone increases cytochrome P450 activity, endothelin-1 and fibronectin expression. Hydrocortisone decreases sensitivity of ductus arteriosus to PGE2 These mechanisms have been postulated to cause ductal constriction and closure. Our case supports this association. hsPDA in a term and near-term neonate with a protracted disease course or associated midline defects should prompt the clinician to suspect CH (hypothyroidism and/or hypocortisolism).

Aberrant Right Subclavian Artery with Kommerell's Diverticulum and Patent Ductus Arteriosus: Unusual Combination of Congenital Heart Anomalies and Clinical Manifestations in A Neonate.

BACKGROUND: A one-month-old neonate presented to the hospital with dyspnea and bloody stool, which happened suddenly and progressed over two days. CASE PRESENTATION: Computed tomography and three-dimensional reconstruction of the trachea and heart was done, demonstrating significant trachea stenosis, aberrant right subclavian artery arising from Kommerell's diverticulum, and patent ductus arteriosus. Reconstruction of the aberrant subclavian artery, resection of the diverticulum, and ligation of ductus arteriosus was performed. CONCLUSION: The case reported a rare combination of congenital anomalies and rare clinical manifestations at the same time. We thought the anatomical anomalies caused necrotizing enterocolitis (NEC), which lead to bloody stool.

Type A Acute Aortic Dissection in a Patient with Patent Ductus Arteriosus.

Cardiac or aortic surgeries requiring cardiopulmonary bypass in combination with patent ductus arteriosus in adults present a special problem that requires a tailored approach. We report a case of ruptured type A acute aortic dissection in a 73-year-old woman with patent ductus arteriosus. In this case, we highlight that transpulmonary direct ductus arteriosus closure and subsequent frozen elephant deployment with total arch replacement are reasonable and reliable treatments for type A acute aortic dissection in a patient with patent ductus arteriosus.

Stent implantation of an unusual morphology patent ductus arteriosus via Glidesheath slender.

The procedure of stenting the patent ductus arteriosus (PDA) is a palliative procedure applied as an alternative to surgery in newborns with ductus-dependent pulmonary circulation. However, it is still a very challenging method in patients with aortic arch anomalies. We describe our experience with a newborn with right atrial isomerism and dextrocardia, complete atrioventricular septal defect, aortic outlet right ventricle with pulmonary atresia, right aortic arch, and a PDA from the left innominate artery. Because the PDA was long and tortuous, we preferred placing three short stents instead of a single long stent. The procedure applied the femoral artery approach with a Glidesheath Slender to decrease arterial injuries. PDA stenting in challenging morphologies can be performed successfully using multiple short stents and via Glidesheath Slenders.

Ductus arteriosus aneurysm with left pulmonary artery obstruction.

The echogenic mass in the pulmonary artery is a rare finding in newborns. Differential diagnoses include pulmonary artery thrombosis, ductal aneurysm, and malignant tumor. We report a newborn case who presented with mild desaturation and was found to have a large echogenic mass at the pulmonary bifurcation on echocardiography, which caused partial flow obstruction in the proximal left pulmonary artery. Along with the findings of cardiac computed tomography and magnetic resonance imaging, the diagnosis of ductus arteriosus aneurysm (DAA) was made. This DAA spontaneously regressed without any intervention at 4 months follow-up.

Patent ductus arteriosus treatment trends and associated morbidities in neonates.

To evaluate national epidemiologic data on infants treated for patent ductus arteriosus (PDA) in Korea and analyze outcomes associated with different PDA treatments. We retrospectively evaluated data on 12,336 patients diagnosed with PDA (International Classification of Diseases-10 code: Q250) between 2015 and 2018 from the Health Insurance Review and Assessment database. Among them, 1623 patients underwent surgical ligation (code: O1671). We used birth certificate data from Statistics Korea to estimate the prevalence, diagnosis, and treatment of PDA. The prevalence of infants with PDA was 81 infants per 10,000 live births and 45.2% in very low birth weight (VLBW) infants, which increased from 2015 to 2018. PDA ligation was performed in 2571 infants and 22% VLBW infants. Medical treatment was administered to 4202 infants, which decreased significantly, especially in VLBW infants (62% to 53%). The proportion of treatment was as follows: conservative treatment (53.1%), intravenous ibuprofen (24.4%), surgery (20.4%), and oral ibuprofen (10.7%); that among 4854 VLBW infants was as follows: intravenous ibuprofen (46.3%), conservative treatment (33.2%), surgery (22.2%), and oral ibuprofen (14.2%). Surgical treatment had a significantly higher risk (odds ratio 1.36) of mortality than conservative treatment. Surgical and/or medical treatments were associated with a higher risk of morbidity. Recently, increased use of conservative management of PDA has contributed to improved neonatal outcomes in VLBW infants. Select patients may still benefit from surgical ligation following careful consideration.

Hypertrophic cardiomyopathy in an extremely preterm infant.

We present a case of an extreme preterm infant (Baby X) born at 24-week gestation. The echocardiogram showed evidence of hypertrophic cardiomyopathy (HCM) and a patent ductus arteriosus (PDA). There are a number of well-known causes of neonatal HCM including genetic, metabolic and endocrine. PDA is commonly present in preterm infants, and this can contribute to cardiac remodelling and result in cardiac changes mimicking HCM. Furthermore, medications such as steroids can also cause HCM through various mechanisms. A careful consideration of all the different aetiologies for HCM is important for appropriate management of such cases. This report examines the evidence in the literature for the above differential diagnoses and highlights the challenges in diagnosing the underlying cause of HCM in a preterm infant.

Clinical Outcomes of Extrapleural Closure of a Patent Ductus Arteriosus Concomitant with Aortic Coarctation Repair.

BACKGROUND: The aim of this study was to present an extrapleural approach for the closure of patent ductus arteriosus (PDA), with the repair of aortic coarctation (CoA) in the same session, in critically ill newborns and infants as an alternative to the transpleural surgical technique. METHODS: Between December 2007 and November 2010, 44 critically ill patients with PDA and coarctation of the aorta were operated on during the same session with the extrapleural approach. The diagnoses of the patients were made by transthoracic echocardiography (TTE). We investigated the aortic arch, the length of the coarctation segment, peak-to-peak gradients, the aortic valve, and intracardiac defects prior to the surgery using TTE. Cardiac angiography was performed to determine whether the patients were suitable for an interventional approach in hemodynamically stable patients. Twenty-eight patients had congestive heart failure with mild to moderate pulmonary and systemic hypertension. The median gestational age and weight of neonates were 2.1 kg (range: 1.4 to 2.9 kg), and 31.4 weeks (range, 28.6 to 37 weeks), respectively. During the operations, PDA was closed using double clips. Resection of coarctation with an extended end-to-end anastomosis was performed in 27 patients. Subclavian flap angioplasty was performed in four patients, and an aortic patch repair was performed in two infants. Postoperative PDA flow and residual aortic gradient were evaluated using echocardiography prior to discharge from the hospital and during the follow-up period. RESULTS: There were three in-hospital deaths (6.8%). During the follow-up period, two patients died (4.8%). The mean follow-up period was 48.3±21.5 months (range: 29-56 months). Patent foramen ovale, atrial septal defect, and ventricular septal defect were the additional cardiac pathologies. These were hemodynamically insignificant. We detected that the intracardiac defects closed spontaneously. During the follow-up period, recoarctation developed in six patients (20%). We found that the risk factors for recoarctation in patients were to have a gradient from coarctation area, which was higher than ≥ 50 mmHg, and the length of coarctation segment that was longer than 1 cm in their first operation (P = 0.033). The median time from the first surgery to recoarctation was 25.4±13.2 months (range: 16-36 months). Balloon dilatation was performed in four patients. We performed redo-surgery in the remaining two patients with recoarctation. The mean intubation time was 9.1±13.4 hours (range: 5.8-19.8 hours). Transthoracic echocardiography showed normal left ventricular dimensions and systolic function in 34 patients during follow up (87.1%). CONCLUSION: Our experiences show that surgical repair of aortic coarctation and PDA closure at the same session may be performed safely and with acceptable mortality and morbidity via an extrapleural approach. Interventional approach as a less invasive method may be used in patients who have developed recoarctation.

Optimization of surgical timing of congenital diaphragmatic hernia using the quantified flow patterns of patent ductus arteriosus.

PURPOSE: The optimal timing of surgery for congenital diaphragmatic hernia (CDH) is controversial. We aimed to validate our protocol for the timing of CDH repair using the quantified patent ductus arteriosus (PDA) flow pattern. METHODS: This retrospective comparative study analyzed patients with a prenatal diagnosis of isolated CDH between 2007 and 2020. We defined the "LR ratio" as the percentage of velocity-time integral (VTI) of the left-to-right flow of PDA against overall VTI on echocardiography. Since 2010, we followed the decision criterion of performing surgery when LR ratio of > 50% has been achieved in the patients (protocol group). The protocol group (2010-2020) was compared with the historical control group (2007-2009). RESULTS: The average age at surgery was 104.1 ± 175.9 and 37.3 ± 30.6 h in the control and protocol groups, respectively (p = 0.11). Survival rate (88.9% vs. 95.0%, p = 0.53) and the rate of worsening of pulmonary hypertension within 24 h after surgery (22.2% vs. 10.0%, p = 0.57) were not different between the groups. The protocol group had a significantly shorter duration of tracheal intubation (26.9 ± 21.1 vs. 13.3 ± 9.5 days, p = 0.03). CONCLUSION: Our decision criterion might have the advantage of facilitating early and safe surgery for patients with CDH.