Echocardiographic Evidence for a Ductal Tissue Sling Causing Discrete Coarctation of the Aorta in the Neonate: Case Report

Callahan, Patrick F.; Quivers, Eric S.; Bradley, Linda M.; Sell, Jeffrey E.; Martin, Gerard R.

doi:10.1007/s002469900276

Cited by 10 publications

(5 citation statements)

References 9 publications

(7 reference statements)

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“…10 The exact pathophysiological mechanisms that lead to the development of CoA are still poorly understood, despite the first such theories being proposed over 180 years ago. [11][12][13][14][15][16][17] The so-called branching theory, proposed by Hutchins 14 in 1971, postulates that reduced or reversed flow at the isthmus leads to the distal arch becoming a functional branch of the DA (Figure 1). This theory is based on the observation, from surgically resected specimens, that the posterior shelf-the ridge of infolded tissue associated with isthmal obstruction following DA closure-is histologically identical to the arterial infolding seen in other major aortic branches.…”

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confidence: 99%

Analysis of 3-Dimensional Arch Anatomy, Vascular Flow, and Postnatal Outcome in Cases of Suspected Coarctation of the Aorta Using Fetal Cardiac Magnetic Resonance Imaging

Lloyd

Poppel

Pushparajah

et al. 2021

Circ: Cardiovascular Imaging

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Background: Identifying fetuses at risk of severe neonatal coarctation of the aorta (CoA) can be lifesaving but is notoriously challenging in clinical practice with a high rate of false positives. Novel fetal 3-dimensional and phase-contrast magnetic resonance imaging (MRI) offers an unprecedented means of assessing the human fetal cardiovascular system before birth. We performed detailed MRI assessment of fetal vascular morphology and flows in a cohort of fetuses with suspected CoA, correlated with the need for postnatal intervention. Methods: Women carrying a fetus with suspected CoA on echocardiography were referred for MRI assessment between 26 and 36 weeks of gestation, including high-resolution motion-corrected 3-dimensional volumes of the fetal heart and phase-contrast flow sequences gated with metric optimized gating. The relationship between aortic geometry and vascular flows was then analyzed and compared with postnatal outcome. Results: Seventy-two patients (51 with suspected fetal CoA and 21 healthy controls) underwent fetal MRI with motion-corrected 3-dimensional vascular reconstructions. Vascular flow measurements from phase-contrast sequences were available in 53 patients. In the CoA group, 25 of 51 (49%) required surgical repair of coarctation after birth; the remaining 26 of 51 (51%) were discharged without neonatal intervention. Reduced blood flow in the fetal ascending aorta and at the aortic isthmus was associated with increasing angulation ( P =0.005) and proximal displacement ( P =0.006) of the isthmus and was seen in both true positive and false positive cases. A multivariate logistic regression model including aortic flow and isthmal displacement explained 78% of the variation in outcome and correctly predicted the need for intervention in 93% of cases. Conclusions: Reduced blood flow though the left heart is associated with important configurational changes at the aortic isthmus in fetal life, predisposing to CoA when the arterial duct closes after birth. Novel fetal MRI techniques may have a role in both understanding and accurately predicting severe neonatal CoA.

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confidence: 99%

Analysis of 3-Dimensional Arch Anatomy, Vascular Flow, and Postnatal Outcome in Cases of Suspected Coarctation of the Aorta Using Fetal Cardiac Magnetic Resonance Imaging

Lloyd

Poppel

Pushparajah

et al. 2021

Circ: Cardiovascular Imaging

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“…The patient was surgically repaired via subclavian flap aortoplasty. 9 In another unique case of coarctation in a patient with a persistent fifth aortic arch, prostaglandin infusion relieved the obstruction, with an improvement in femoral pulses, and echocardiographic narrowing without opening the ductus. 10 It has also been reported that in a premature baby with coarctation, whose ductus had been previously ligated, prostaglandin infusion successfully ameliorated symptoms of heart failure.…”

Section: Discussionmentioning

confidence: 98%

“…Callahan et al reported a nine-day-old neonate with significant coarctation in the juxtaductal region without a patent ductus. 9 Prostaglandin infusion resulted in clinical and echocardiographic resolution and its discontinuation resulted in recurrence of signs of coarctation. The patient was surgically repaired via subclavian flap aortoplasty.…”

Section: Discussionmentioning

confidence: 99%

Prostaglandin Infusion in Neonate With Severe Coarctation of the Aorta With Closed Ductus Arteriosus—A Case Report and Review of the Literature

Bansal

Balakrishnan

Aggarwal

2019

World J Pediatr Congenit Heart Surg

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We report the case of a premature newborn diagnosed with coarctation of the aorta after spontaneous closure of ductus arteriosus who was successfully managed with prostaglandin E1 infusion until surgical repair could be performed. This case, together with a review of the literature, suggests an important role for prostaglandin in the management of coarctation even in the absence of a patent ductus arteriosus. The putative mechanism for the utility of prostaglandin infusion is that it may relieve the obstruction in neonates with severe coarctation by not only opening of the ductus but, in select cases, relaxing the ductal tissue encircling the aortic isthmus region. We also found a possible dose dependence of the efficacy of the prostaglandin infusion when the ductus is closed.

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“…Liberman reported three term neonates with coarctation of the aorta and a closed ductus who were treated with prostaglandin E 1 infusions; prostaglandin therapy was only used for short periods since each infant underwent early corrective surgery 1. Callahan et al also reported the short-term use of prostaglandin E 1 in a near-term baby in whom a trial of cessation of prostaglandin therapy resulted in a worsening of the aortic constriction 4…”

Section: Discussionmentioning

confidence: 99%

Prostaglandin E_₂in a preterm infant with coarctation of the aorta

2019

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Prostaglandins are widely used in aortic coarctation to maintain ductal patency and preserve systemic perfusion until surgical intervention can be performed. Although the short-term use of prostaglandins to ameliorate aortic narrowing in neonates with a closed ductus has been reported, it has not been described as a longer term therapy in extremely preterm neonates. A 27-week gestation baby weighing 560 g presented at 40 days of age with coarctation and a closed ductus arteriosus. He was successfully treated with a 7-week course of prostaglandin E2 therapy because surgical intervention was not deemed feasible in view of his size. Treatment resulted in a relaxation of the aortic constriction and improvement in aortic blood flow velocity profile, highlighting the value of long-term prostaglandin therapy in this population and supporting the hypothesis that the presence of ductal tissue contributes to the development of juxtaductal aortic constriction in some extremely preterm infants.

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Echocardiographic Evidence for a Ductal Tissue Sling Causing Discrete Coarctation of the Aorta in the Neonate: Case Report

Cited by 10 publications

References 9 publications

Analysis of 3-Dimensional Arch Anatomy, Vascular Flow, and Postnatal Outcome in Cases of Suspected Coarctation of the Aorta Using Fetal Cardiac Magnetic Resonance Imaging

Analysis of 3-Dimensional Arch Anatomy, Vascular Flow, and Postnatal Outcome in Cases of Suspected Coarctation of the Aorta Using Fetal Cardiac Magnetic Resonance Imaging

Prostaglandin Infusion in Neonate With Severe Coarctation of the Aorta With Closed Ductus Arteriosus—A Case Report and Review of the Literature

Prostaglandin E_₂in a preterm infant with coarctation of the aorta

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Echocardiographic Evidence for a Ductal Tissue Sling Causing Discrete Coarctation of the Aorta in the Neonate: Case Report

Cited by 10 publications

References 9 publications

Analysis of 3-Dimensional Arch Anatomy, Vascular Flow, and Postnatal Outcome in Cases of Suspected Coarctation of the Aorta Using Fetal Cardiac Magnetic Resonance Imaging

Analysis of 3-Dimensional Arch Anatomy, Vascular Flow, and Postnatal Outcome in Cases of Suspected Coarctation of the Aorta Using Fetal Cardiac Magnetic Resonance Imaging

Prostaglandin Infusion in Neonate With Severe Coarctation of the Aorta With Closed Ductus Arteriosus—A Case Report and Review of the Literature

Prostaglandin E2in a preterm infant with coarctation of the aorta

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Prostaglandin E_₂in a preterm infant with coarctation of the aorta