Get 20M+ Full-Text Papers For Less Than $1.50/day. Start a 14-Day Trial for You or Your Team.

Learn More →

Case report on surgical repair of unusual dissection of arterial duct involving main pulmonary artery in a child with vein of Galen malformation

Case report on surgical repair of unusual dissection of arterial duct involving main pulmonary... Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 CASE REPORT European Heart Journal - Case Reports (2018) 2, 1–4 doi:10.1093/ehjcr/yty140 Congenital heart disease Case report on surgical repair of unusual dissection of arterial duct involving main pulmonary artery in a child with vein of Galen malformation 1 1 2 Nagarajan Muthialu *, Alex Robertson , Kristian Mortensen , and Sachin Khambadkone 1 2 Department of Cardiothoracic Surgery, Great Ormond Street Hospital, Great Ormond Street, London WC1N 3JH, UK; and Department of Cardiology, Great Ormond Street Hospital, Great Ormond Street, London WC1N 3JH, UK Received 20 April 2018; accepted 26 October 2018; online publish-ahead-of-print 24 November 2018 Background Dissection involving patent arterial duct (PDA) extending to main pulmonary artery (MPA) is an extremely rare condition. ................................................................................................................................................................................................... Case summary We report a case of complex dissection involving PDA extending to MPA, in a child with vein of Galen malforma- tion. This was surgically corrected under deep hypothermic circulatory arrest, and we describe the pathophysiology and surgical rationale in this report. ................................................................................................................................................................................................... Discussion Management of intracardiac lesion in the presence of intracranial arterio-venous malformations such as vein of Galen is very difficult, and employing deep hypothermic circulatory arrest allows one to manage cerebral protec- tion while offering the visualization of intracardiac anatomy more effectively. Keywords Case report Patent arterial duct Main pulmonary artery dissection Vein of Galen malformation • • • Learning points • Introduction Spontaneous dissection of patent arterial duct (PDA) is extremely rare,and oftenseen with otherpre-existingconditionsorany . Spontaneous pulmonary artery dissection is an extremely rare condi- traumatic interventions in children. . 1 • . tion. Reports of dissected pulmonary artery secondary to infective Strong suspicion of infective endocarditis is important for clinical management of dissected PDA. . endocarditis are available. Origin of such a dissection from patent ar- Early surgery is important to help with the haemodynamic man- . terial duct (PDA) in a neonate has not been reported previously. We agement of such a condition, and also to avoid catastrophic spon- report such a condition, where there was a dissected PDA with ex- taneous rupture of either the pulmonary artery or the dilated tension into main pulmonary artery (MPA), which was treated surgi- duct. cally. Informed consent has been obtained from the parents to Deep hypothermic circulatory arrest is of vital use in such a situ- . report this exceptional case report as part of publication. ation, in order to allow for a bloodless field and for surgical expos- ure, thereby optimal surgical procedure is carried out. * Corresponding author. Tel: þ44 2074059200, Fax: þ44 2078138262, Email: Nagarajan.Muthialu@gosh.nhs.uk Handling Editor: Julia Grapsa Peer-reviewers: Cemil Izgi, Julia Grapsa Compliance Editor: Amir Aziz Supplementary Material Editor: Peregrine Green V The Author(s) 2018. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 2 N. Muthialu et al. felt that any interventional approach would risk placing a larger de- Timeline vice within the duct and potentially compromise pulmonary valve be- . cause of its proximity. . Surgery was carried under cardiopulmonary bypass, with deep . hypothermic circulatory arrest at 18 . Neurological monitoring was On admission Initial diagnosis of vein of Galen . with near-infrared spectroscopy and using pH strategy for better malformation . cerebrovascular control. External appearance showed bosselated ap- In the first 2 weeks of Neuro-radiological intervention of arte- . pearance of MPA and a large PDA. Once circulation was stopped, hospital admission rio-venous malformations to vein of division of PDA in its midpoint confirmed two lumens—one small Galen malformation true and another false lumen. The MPA was opened distal to pulmon- On 15th day in hospital Echocardiographic diagnosis of patent ary valve, and false lumen was additionally confirmed. The extent of arterial duct (PDA) diagnosis false lumen towards the pulmonary valve, thereby showing the exit On 18th day in hospital Further evaluation with computerized point near the valve is shown in Figure 2. tomographic scan of chest with con- The exit point was closed using 7/0 polypropylene suture. The trast and delineation of anatomy with false lumen on either side of transected pulmonary artery was further echocardiography sutured using similar 7/0 polypropylene sutures. The divided PDA On 20th day Joint cardiology and cardiothoracic sur- was secured, and MPA was closed. Weaning from bypass was gery conference discussion to make smooth. The child was extubated in 3 days with a normal neurological management decision assessment and post-operative echocardiography confirms normal On 24th day Surgical procedure to correct PDA . flow across pulmonary artery with no evidence of any false lumen. dissection . On 27th day Extubation in cardiac intensive care unit, . and assessment proving normal . Discussion neurological assessment . On Days 26 and 28 (for Post-operative echocardiography con- . . Spontaneous dissection of pulmonary artery or PDA is an extremely maintenance of chron- firming corrected lesion, with normal . rare condition. While there are individual reports of dissected pul- ology, is mentioned flow across pulmonary artery . monary artery, reports on dissected PDA are scanty. These dissec- here as combined . tions are thought to be secondary to primary pulmonary event) . hypertension or in association with conditions such as vascular in- . flammatory disease, aorto-pulmonary fistulas, connective tissue dis- ease, or catheter-induced vessel injury. Some of these conditions predispose to a weak arterial wall. Associated abnormal hydrodynamics, in the presence of extensive AVMs, as seen in our child, with vein of Galen malformation may in- Case presentation . crease the risk of intimal damage, thereby inducing intimal rupture A 20-day-old neonate was evaluated by echocardiography following leading to dissection. a neuro-radiological intervention to vein of Galen malformation, Traumatic dissections can occur in arterial tree in small children in 3,4 whereby coil occlusion of arterio-venous malformations (AVMs) was the presence of abnormal haemodynamics. The traumatic event is done. Clinical examination showed mild pectus deformity of chest. related to any instrumentation, including catheterization, as in our There were occasional tachypnoeic features, but no obvious signs of case. But the timing of origin and site of dissection away from the end cardiac failure. The heart sounds were normal, with no audible mur- of PDA make this unlikely to be the cause in our case. The entry point murs at this stage. Echocardiography demonstrated PDA and con- of dissection was in the PDA itself away from aortic wall. firmed an additional lumen inside PDA, shunting from aorta to MPA, Infective endocarditis can potentially weaken specific area of arter- just proximal to the pulmonary valve. Clinically the child was asymp- ial tree. Bacterial cultures in our child were negative to explain such tomatic, but had features of hyperdynamic circulation with palpable an origin. Despite this, the possibility remained high in our consider- pericardial thrill. Further evaluation was done with a contrast- ation, thereby warranting repeated evaluation and antibiotic cover. enhanced computerized tomographic (CT) scan. This confirmed a . Shi et al. further reports that while infective endocarditis can help dissected PDA with evidence of both true and false lumen (Figure 1) . begin the process of dissection, PDA might also contribute to the with dilated MPA. The exit point of this dissection into MPA was not . progression even in the absence of pulmonary hypertension. clear on the scan. Bacterial cultures were repeatedly negative to ex- . Considering the unquantified risk of rupture in a small child, surgery plain any possibility of infective source for this. Despite this, we kept . was offered. Catheter-based interventions would potentially be difficult the possibility of infection higher in our consideration, thereby war- . in this situation. Surgical strategy depends on the comorbidities and ranting repeated evaluation to seek infective markers and kept the deep hypothermic arrest would allow us to inspect the anatomy clearly. child under antibiotic cover. With a paucity of report, management depends entirely on the Based on an unquantified risk of rupture from any literature evi- haemodynamic status on the child. With a successful early result, we dence, surgery was decided with aim to ligate the PDA, obliterate the suggest surgical intervention on dissected PDA with dissection into false lumen, and repair the MPA. Furthermore, on discussion, it was MPA. Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 Complex PDA dissection 3 Figure 1 Preoperative computerized tomographic scan showing (A): contrast filled false lumen from the aortic end (B): cross-sectional view of main pulmonary artery with true and false lumens (C): reconstructed image showing similar appearance. Figure 2 (A) Probe inside false lumen of distal main pulmonary artery; (B) divided arterial duct with probes in true and false lumens; (C) proximal extent of dissection with probe at exit point near pulmonary valve. Figure 3 Echocardiographic images showing (A) pre-neurointervention with a small ductal ampulla (dotted line) at aortic end; (B) preoperative image showing false lumen of the duct with flow into main pulmonary artery (solid line) via exit point. Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 4 N. Muthialu et al. . References Supplementary material 1. Malm CJ, Ternstrom L, Jorgensen K, Dellgren G. Pulmonary artery dissection in a . patient with undiagnosed pulmonary hypertension—a case report and review of Supplementary material is available at European Heart Journal - Case literature. Heart Lung 2015;44:453–457. Reports online. 2. McElhinney DB, Halbach VV, Silverman NH, Dowd CF, Hanley FL. Congenital cardiac anomalies with vein of Galen malformations in infants. Arch Dis Child 1998; Slide sets: A fully edited slide set detailing this case and suitable for . 78:548–551. local presentation is available online as Supplementary data. 3. Zamorano MMB. Dissection and ruptureof the pulmonaryarteryassociated with patent ductus arteriosus: report of a case. Rev Bras Cir Cardiovasc 1987;2: Consent: The author/s confirm that written consent for submission . 139–144. 4. Cui Y, Yu J, Nie Z, Shi H. A rare case of aortic dissection with concomitant pul- and publication of this case report including image(s) and associated . monary artery dissection extending through a patent ductus arteriosus. J Vasc text has been obtained from the patient in line with COPE guidance. . Interv Radiol 2015;26:1084–1086. . 5. Shi X, Wang X, Wang C, Zhou K, Li Y, Hua Y. A rare case of pulmonary Conflict of interest: none declared. artery dissection associated with infective endocarditis. Medicine 2016;95:e3358. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png European Heart Journal - Case Reports Oxford University Press

Case report on surgical repair of unusual dissection of arterial duct involving main pulmonary artery in a child with vein of Galen malformation

Download PDF
4 pages

Loading next page...
 
/lp/oxford-university-press/case-report-on-surgical-repair-of-unusual-dissection-of-arterial-duct-BmZW10i4qE

References (6)

Publisher
Oxford University Press
Copyright
© The Author(s) 2018. Published by Oxford University Press on behalf of the European Society of Cardiology.
eISSN
2514-2119
DOI
10.1093/ehjcr/yty140
Publisher site
See Article on Publisher Site

Abstract

Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 CASE REPORT European Heart Journal - Case Reports (2018) 2, 1–4 doi:10.1093/ehjcr/yty140 Congenital heart disease Case report on surgical repair of unusual dissection of arterial duct involving main pulmonary artery in a child with vein of Galen malformation 1 1 2 Nagarajan Muthialu *, Alex Robertson , Kristian Mortensen , and Sachin Khambadkone 1 2 Department of Cardiothoracic Surgery, Great Ormond Street Hospital, Great Ormond Street, London WC1N 3JH, UK; and Department of Cardiology, Great Ormond Street Hospital, Great Ormond Street, London WC1N 3JH, UK Received 20 April 2018; accepted 26 October 2018; online publish-ahead-of-print 24 November 2018 Background Dissection involving patent arterial duct (PDA) extending to main pulmonary artery (MPA) is an extremely rare condition. ................................................................................................................................................................................................... Case summary We report a case of complex dissection involving PDA extending to MPA, in a child with vein of Galen malforma- tion. This was surgically corrected under deep hypothermic circulatory arrest, and we describe the pathophysiology and surgical rationale in this report. ................................................................................................................................................................................................... Discussion Management of intracardiac lesion in the presence of intracranial arterio-venous malformations such as vein of Galen is very difficult, and employing deep hypothermic circulatory arrest allows one to manage cerebral protec- tion while offering the visualization of intracardiac anatomy more effectively. Keywords Case report Patent arterial duct Main pulmonary artery dissection Vein of Galen malformation • • • Learning points • Introduction Spontaneous dissection of patent arterial duct (PDA) is extremely rare,and oftenseen with otherpre-existingconditionsorany . Spontaneous pulmonary artery dissection is an extremely rare condi- traumatic interventions in children. . 1 • . tion. Reports of dissected pulmonary artery secondary to infective Strong suspicion of infective endocarditis is important for clinical management of dissected PDA. . endocarditis are available. Origin of such a dissection from patent ar- Early surgery is important to help with the haemodynamic man- . terial duct (PDA) in a neonate has not been reported previously. We agement of such a condition, and also to avoid catastrophic spon- report such a condition, where there was a dissected PDA with ex- taneous rupture of either the pulmonary artery or the dilated tension into main pulmonary artery (MPA), which was treated surgi- duct. cally. Informed consent has been obtained from the parents to Deep hypothermic circulatory arrest is of vital use in such a situ- . report this exceptional case report as part of publication. ation, in order to allow for a bloodless field and for surgical expos- ure, thereby optimal surgical procedure is carried out. * Corresponding author. Tel: þ44 2074059200, Fax: þ44 2078138262, Email: Nagarajan.Muthialu@gosh.nhs.uk Handling Editor: Julia Grapsa Peer-reviewers: Cemil Izgi, Julia Grapsa Compliance Editor: Amir Aziz Supplementary Material Editor: Peregrine Green V The Author(s) 2018. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 2 N. Muthialu et al. felt that any interventional approach would risk placing a larger de- Timeline vice within the duct and potentially compromise pulmonary valve be- . cause of its proximity. . Surgery was carried under cardiopulmonary bypass, with deep . hypothermic circulatory arrest at 18 . Neurological monitoring was On admission Initial diagnosis of vein of Galen . with near-infrared spectroscopy and using pH strategy for better malformation . cerebrovascular control. External appearance showed bosselated ap- In the first 2 weeks of Neuro-radiological intervention of arte- . pearance of MPA and a large PDA. Once circulation was stopped, hospital admission rio-venous malformations to vein of division of PDA in its midpoint confirmed two lumens—one small Galen malformation true and another false lumen. The MPA was opened distal to pulmon- On 15th day in hospital Echocardiographic diagnosis of patent ary valve, and false lumen was additionally confirmed. The extent of arterial duct (PDA) diagnosis false lumen towards the pulmonary valve, thereby showing the exit On 18th day in hospital Further evaluation with computerized point near the valve is shown in Figure 2. tomographic scan of chest with con- The exit point was closed using 7/0 polypropylene suture. The trast and delineation of anatomy with false lumen on either side of transected pulmonary artery was further echocardiography sutured using similar 7/0 polypropylene sutures. The divided PDA On 20th day Joint cardiology and cardiothoracic sur- was secured, and MPA was closed. Weaning from bypass was gery conference discussion to make smooth. The child was extubated in 3 days with a normal neurological management decision assessment and post-operative echocardiography confirms normal On 24th day Surgical procedure to correct PDA . flow across pulmonary artery with no evidence of any false lumen. dissection . On 27th day Extubation in cardiac intensive care unit, . and assessment proving normal . Discussion neurological assessment . On Days 26 and 28 (for Post-operative echocardiography con- . . Spontaneous dissection of pulmonary artery or PDA is an extremely maintenance of chron- firming corrected lesion, with normal . rare condition. While there are individual reports of dissected pul- ology, is mentioned flow across pulmonary artery . monary artery, reports on dissected PDA are scanty. These dissec- here as combined . tions are thought to be secondary to primary pulmonary event) . hypertension or in association with conditions such as vascular in- . flammatory disease, aorto-pulmonary fistulas, connective tissue dis- ease, or catheter-induced vessel injury. Some of these conditions predispose to a weak arterial wall. Associated abnormal hydrodynamics, in the presence of extensive AVMs, as seen in our child, with vein of Galen malformation may in- Case presentation . crease the risk of intimal damage, thereby inducing intimal rupture A 20-day-old neonate was evaluated by echocardiography following leading to dissection. a neuro-radiological intervention to vein of Galen malformation, Traumatic dissections can occur in arterial tree in small children in 3,4 whereby coil occlusion of arterio-venous malformations (AVMs) was the presence of abnormal haemodynamics. The traumatic event is done. Clinical examination showed mild pectus deformity of chest. related to any instrumentation, including catheterization, as in our There were occasional tachypnoeic features, but no obvious signs of case. But the timing of origin and site of dissection away from the end cardiac failure. The heart sounds were normal, with no audible mur- of PDA make this unlikely to be the cause in our case. The entry point murs at this stage. Echocardiography demonstrated PDA and con- of dissection was in the PDA itself away from aortic wall. firmed an additional lumen inside PDA, shunting from aorta to MPA, Infective endocarditis can potentially weaken specific area of arter- just proximal to the pulmonary valve. Clinically the child was asymp- ial tree. Bacterial cultures in our child were negative to explain such tomatic, but had features of hyperdynamic circulation with palpable an origin. Despite this, the possibility remained high in our consider- pericardial thrill. Further evaluation was done with a contrast- ation, thereby warranting repeated evaluation and antibiotic cover. enhanced computerized tomographic (CT) scan. This confirmed a . Shi et al. further reports that while infective endocarditis can help dissected PDA with evidence of both true and false lumen (Figure 1) . begin the process of dissection, PDA might also contribute to the with dilated MPA. The exit point of this dissection into MPA was not . progression even in the absence of pulmonary hypertension. clear on the scan. Bacterial cultures were repeatedly negative to ex- . Considering the unquantified risk of rupture in a small child, surgery plain any possibility of infective source for this. Despite this, we kept . was offered. Catheter-based interventions would potentially be difficult the possibility of infection higher in our consideration, thereby war- . in this situation. Surgical strategy depends on the comorbidities and ranting repeated evaluation to seek infective markers and kept the deep hypothermic arrest would allow us to inspect the anatomy clearly. child under antibiotic cover. With a paucity of report, management depends entirely on the Based on an unquantified risk of rupture from any literature evi- haemodynamic status on the child. With a successful early result, we dence, surgery was decided with aim to ligate the PDA, obliterate the suggest surgical intervention on dissected PDA with dissection into false lumen, and repair the MPA. Furthermore, on discussion, it was MPA. Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 Complex PDA dissection 3 Figure 1 Preoperative computerized tomographic scan showing (A): contrast filled false lumen from the aortic end (B): cross-sectional view of main pulmonary artery with true and false lumens (C): reconstructed image showing similar appearance. Figure 2 (A) Probe inside false lumen of distal main pulmonary artery; (B) divided arterial duct with probes in true and false lumens; (C) proximal extent of dissection with probe at exit point near pulmonary valve. Figure 3 Echocardiographic images showing (A) pre-neurointervention with a small ductal ampulla (dotted line) at aortic end; (B) preoperative image showing false lumen of the duct with flow into main pulmonary artery (solid line) via exit point. Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/4/1/5204429 by Ed 'DeepDyve' Gillespie user on 15 January 2019 4 N. Muthialu et al. . References Supplementary material 1. Malm CJ, Ternstrom L, Jorgensen K, Dellgren G. Pulmonary artery dissection in a . patient with undiagnosed pulmonary hypertension—a case report and review of Supplementary material is available at European Heart Journal - Case literature. Heart Lung 2015;44:453–457. Reports online. 2. McElhinney DB, Halbach VV, Silverman NH, Dowd CF, Hanley FL. Congenital cardiac anomalies with vein of Galen malformations in infants. Arch Dis Child 1998; Slide sets: A fully edited slide set detailing this case and suitable for . 78:548–551. local presentation is available online as Supplementary data. 3. Zamorano MMB. Dissection and ruptureof the pulmonaryarteryassociated with patent ductus arteriosus: report of a case. Rev Bras Cir Cardiovasc 1987;2: Consent: The author/s confirm that written consent for submission . 139–144. 4. Cui Y, Yu J, Nie Z, Shi H. A rare case of aortic dissection with concomitant pul- and publication of this case report including image(s) and associated . monary artery dissection extending through a patent ductus arteriosus. J Vasc text has been obtained from the patient in line with COPE guidance. . Interv Radiol 2015;26:1084–1086. . 5. Shi X, Wang X, Wang C, Zhou K, Li Y, Hua Y. A rare case of pulmonary Conflict of interest: none declared. artery dissection associated with infective endocarditis. Medicine 2016;95:e3358.

Journal

European Heart Journal - Case Reports – Oxford University Press

Published: Dec 1, 2018

There are no references for this article.