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

Learn More →

Ductus arteriosus aneurysm presenting as hoarseness: successful repair with an endovascular approach

Ductus arteriosus aneurysm presenting as hoarseness: successful repair with an endovascular approach An aneurysm of the ductus arteriosus is a rare finding, particularly in the adult population. These saccular aneurysms arise at the site of an incompletely obliterated ductus arteriosus along the lesser curvature of the aortic arch. Left untreated, it is associated with a high risk of potentially life-threatening complications including rupture, infection and thromboembolism. As a result, surgical correction is recommended. Previously, options were limited to open repair but as endovascular experi- ence grows, novel techniques afford safer and less invasive alternatives. In contrast, neonatal ductus arteriosus aneurysms may regress spontaneously and expectant treatment can be justified. We present the case of a 74-year-old woman who pre- sented with hoarseness secondary to a ductus arteriosus aneurysm; a diagnosis consistent with Ortner’s syndrome. The patient underwent an uncomplicated endovascular repair using the chimney-graft technique. secondary to aneurysmal dilatation of the diverticulum of the INTRODUCTION ductus arteriosus. We report a successful endovascular repair Aneurysms of the ductus arteriosus in adults are rare findings with good recovery of phonation on follow-up. and the true incidence is unknown. Previously, most cases were discovered at autopsy or in those undergoing cardiac surgery for another indication [1]. Widespread use of cross-sectional ima- CASE REPORT ging in the adult population has led to increased diagnosis of this often asymptomatic abnormality. Correction is warranted A 74-year-old woman was referred to our vascular service with due to the high risk of potentially life-threatening complications a 6-month history of hoarseness of voice and intermittent dry including rupture, thromboembolism and acute fistula forma- cough. The patient’s medical history included hypertension tion [2]. In contrast, ductal aneurysms occurring in neonates and gastroesophageal reflux disease. Flexible laryngoscopy commonly communicate with the pulmonary artery and thereby demonstrated a foreshortened, lateralised and flaccid vocal exist within a patent ductus arteriosus [3]. Operative mortality fold consistent with unilateral vocal cord paralysis. in this group is higher and the disease follows a relatively benign Subsequent computed tomography (CT) imaging of the neck course such that serial echocardiography monitoring is the initi- and thorax revealed a saccular aneurysm in the aortopulmon- al approach. In this case report, an elderly woman presented ary window measuring 39 mm (Fig. 1). Digital subtraction with clinical features of recurrent laryngeal nerve palsy angiography confirmed the presence of a ductal aneurysm Received: February 11, 2016. Accepted: March 16, 2016 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2016. 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 1 2 S. De Freitas et al. Figure 1: Sagittal CT with 3D reconstruction demonstrated a 39-mm saccular aneurysm along the lesser curvature of the aortic arch, expanding in the direction of the pulmonary artery. DISCUSSION During foetal life, the ductus arteriosus permits ejection of oxygen-rich right ventricular blood into the distal aortic arch, bypassing the high-resistance pulmonary circulation. Following the first breath, the smooth muscle in the ductal walls responds to a variety of biochemical stimuli that promote functional clo- sure [4]. Anatomical closure follows as fibrous tissues proliferate from the pulmonic end of the ductus towards the aorta. Incomplete ductal obliteration at the aortic end gives rise to a diverticular remnant at the posteroinferior aspect of the aortic arch. Subsequently, there is potential for aneurysmal dilatation of the diverticulum with exposure to systemic arterial pressures [5]. Apart from previous repair of a patent ductus arteriosus, risk factors for ductus arteriosus aneurysms have been poorly delineated in the literature. Other associations, but not causal relationships, include connective tissue disorders, chromoso- mal anomalies and SMAD3 mutations [3, 6, 7]. Of note, our patient sustained blunt chest wall trauma in a road traffic acci- dent approximately 12 years prior to presentation. Sudden deceleration injuries of the thorax commonly cause aortic transection but an association with aneurysmal dilation of the ductus diverticulum has not previously been described. We postulate that remote shearing forces compromised the tunica Figure 2: Digital subtraction angiogram demonstrating the chimney-graft media at the site of the ductus diverticulum and contributed to approach. Image acquired shortly before simultaneous subclavian and thoracic stent-graft deployment. aneurysmal dilation. Our patient presented with a hoarse, breathy voice and with a well-defined neck and no intraluminal thrombus intermittent bouts of coughing over 6 months. Laryngoscopic (Fig. 2). There was no contrast extravasation into the mediasti- examination demonstrated unilateral vocal cord paralysis con- num or pulmonary circulation. Following a cardiovascular sistent with a left recurrent laryngeal nerve palsy. The left multidisciplinary meeting, treatment options were discussed recurrent laryngeal nerve passes inferior to the arch of the aor- with the patient and consensus favoured endovascular repair. ta before ascending in the tracheo-oesophageal groove to sup- The distance from the proximal margin of the aneurysmal ply all the intrinsic muscles of the larynx, apart from the neck to the origin of the left subclavian artery measured just cricothyroid. Therefore, the clinical presentation in this case is under 2 cm and therefore did not meet the manufacturer’s consistent with Ortner’s cardiovocal syndrome, in which left minimal landing zone requirements. To circumnavigate this recurrent laryngeal nerve palsy arises from cardiovascular dis- dilemma, a left subclavian stent graft was inserted with its ease. In 1897 Norbert Ortner, an Austrian physician, first proximal end parallel to the main thoracic endoprosthesis. described three patients with hoarseness of voice secondary to The procedure was uncomplicated and repeat imaging a dilated left atrium in the setting of mitral stenosis [8]. Less revealed widely patent stent grafts with satisfactory position- commonly reported causes of Ortner’s syndrome include ing and absence of endoleak (Fig. 3). Our patient was dis- degenerative aneurysms of the aortic arch, dilation of the pul- charged home on the second postoperative day. She monary artery, masses in the aortopulmonary window, aortic continued speech and language therapy postoperatively and dissection and iatrogenic injury. Hoarseness in the setting of has made remarkable recovery of phonation, declining any recurrent laryngeal nerve palsy may be permanent but typically further treatment options for same. there is some functional recovery as the uninvolved cord Ductus arteriosus aneurysm presenting as hoarseness 3 Figure 3: Sagittal CT with 3D reconstruction at 1-year post-intervention, showing satisfactory placement of stent grafts. develops the ability to hyperadduct and appose the paralysed 2. Mitchell RS, Seifert FC, Miller DC, Jamieson SW, Shumway cord during phonation. NE. Aneurysm of the diverticulum of the ductus arteriosus Correction is recommended when aneurysms of the ductus in the adult. Successful surgical treatment in five patients arteriosus are >3 cm, enlarge significantly over time or cause and review of the literature. J Thorac Cardiovasc Surg symptoms [2]. In this case, there were two indications for inter- 1983;86:400–8. vention, albeit the most appropriate method of repair was not 3. Dyamenahalli U, Smallhorn JF, Geva T, Fouron JC, Cairns P, clearly defined. Given the substantial morbidity associated with Jutras L, et al. Isolated ductus arteriosus aneurysm in the open repair, endovascular techniques provide for an attractive, fetus and infant: a multi-institutional experience. J Am Coll less invasive alternative. However, there are only a small num- Cardiol 2000;36:262–9. ber of reports of ductus arteriosus aneurysms corrected with an 4. Schneider DJ, Moore JW. Patent ductus arteriosus. endovascular approach [9, 10]. Standard thoracic stent grafts Circulation 2006;114:1873–82. may be used when there is an appropriate landing zone [10]. 5. Pastuszko P, Eisenberg JA, Diehl JT. Ductus arteriosus Otherwise, as illustrated in this unique case, preservation of aneurysm in an adult patient presenting with hoarseness. J the left subclavian artery with the chimney-graft technique is Card Surg 2005;20:386–8. possible. Another option would be to perform a hybrid proce- 6. Chang JP, Chang CH, Sheih MJ. Aneurysmal dilatation of dure in which a surgical bypass is created before the stent is patent ductus arteriosus in a case of Ehlers-Danlos syn- placed over the left subclavian orifice. drome. Ann Thorac Surg 1987;44:656–7. This case highlights the feasibility of a total endovascular 7. van der Lind D, Witsenburg M, van de Laar I, Moelker A, approach to complex thoracic aneurysms as an alternative to Roos-Hesselink J. Saccular aneurysm within a patent ductus open surgery. Ultimately, the most appropriate decision should arteriosus. Lancet 2012;379:e33. follow multidisciplinary discussion, taking into consideration 8. Ortner N. Recurrent laryngeal nerve paralysis due to mitral the expertise of the available endovascular services. value stenosis. Wien Klin Wochenschr 1897;10:753–5. 9. Saito N, Kimura T, Toma M, Teragaki M, Minaminimura H, Kita T, et al. Successful endovascular repair of an aneur- CONFLICT OF INTEREST STATEMENT ysm of the ductus diverticulum with a branched stent None declared. graft: case report and review of literature. J Vasc Surg 2004;40:1228–33. REFERENCES 10. Runge V, Pieper CC, Schiller W, Praeger AJ, Probst C, 1. Lund JT, Jensen MB, Hjelms E. Aneurysm of the ductus Wilhelm KE. Endovascular repair of an ductus arteriosus aneurysm causing Ortner syndrome. Vasc Endovasc Surg arteriosus. A review of the literature and the surgical impli- cations. Eur J Cardiothorac Surg 1991;5:566–70. 2014;48:271–4. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Ductus arteriosus aneurysm presenting as hoarseness: successful repair with an endovascular approach

Loading next page...
 
/lp/oxford-university-press/ductus-arteriosus-aneurysm-presenting-as-hoarseness-successful-repair-UnP0wO79R3

References (13)

Publisher
Oxford University Press
Copyright
Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2016.
eISSN
2042-8812
DOI
10.1093/jscr/rjw060
pmid
27141045
Publisher site
See Article on Publisher Site

Abstract

An aneurysm of the ductus arteriosus is a rare finding, particularly in the adult population. These saccular aneurysms arise at the site of an incompletely obliterated ductus arteriosus along the lesser curvature of the aortic arch. Left untreated, it is associated with a high risk of potentially life-threatening complications including rupture, infection and thromboembolism. As a result, surgical correction is recommended. Previously, options were limited to open repair but as endovascular experi- ence grows, novel techniques afford safer and less invasive alternatives. In contrast, neonatal ductus arteriosus aneurysms may regress spontaneously and expectant treatment can be justified. We present the case of a 74-year-old woman who pre- sented with hoarseness secondary to a ductus arteriosus aneurysm; a diagnosis consistent with Ortner’s syndrome. The patient underwent an uncomplicated endovascular repair using the chimney-graft technique. secondary to aneurysmal dilatation of the diverticulum of the INTRODUCTION ductus arteriosus. We report a successful endovascular repair Aneurysms of the ductus arteriosus in adults are rare findings with good recovery of phonation on follow-up. and the true incidence is unknown. Previously, most cases were discovered at autopsy or in those undergoing cardiac surgery for another indication [1]. Widespread use of cross-sectional ima- CASE REPORT ging in the adult population has led to increased diagnosis of this often asymptomatic abnormality. Correction is warranted A 74-year-old woman was referred to our vascular service with due to the high risk of potentially life-threatening complications a 6-month history of hoarseness of voice and intermittent dry including rupture, thromboembolism and acute fistula forma- cough. The patient’s medical history included hypertension tion [2]. In contrast, ductal aneurysms occurring in neonates and gastroesophageal reflux disease. Flexible laryngoscopy commonly communicate with the pulmonary artery and thereby demonstrated a foreshortened, lateralised and flaccid vocal exist within a patent ductus arteriosus [3]. Operative mortality fold consistent with unilateral vocal cord paralysis. in this group is higher and the disease follows a relatively benign Subsequent computed tomography (CT) imaging of the neck course such that serial echocardiography monitoring is the initi- and thorax revealed a saccular aneurysm in the aortopulmon- al approach. In this case report, an elderly woman presented ary window measuring 39 mm (Fig. 1). Digital subtraction with clinical features of recurrent laryngeal nerve palsy angiography confirmed the presence of a ductal aneurysm Received: February 11, 2016. Accepted: March 16, 2016 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2016. 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 1 2 S. De Freitas et al. Figure 1: Sagittal CT with 3D reconstruction demonstrated a 39-mm saccular aneurysm along the lesser curvature of the aortic arch, expanding in the direction of the pulmonary artery. DISCUSSION During foetal life, the ductus arteriosus permits ejection of oxygen-rich right ventricular blood into the distal aortic arch, bypassing the high-resistance pulmonary circulation. Following the first breath, the smooth muscle in the ductal walls responds to a variety of biochemical stimuli that promote functional clo- sure [4]. Anatomical closure follows as fibrous tissues proliferate from the pulmonic end of the ductus towards the aorta. Incomplete ductal obliteration at the aortic end gives rise to a diverticular remnant at the posteroinferior aspect of the aortic arch. Subsequently, there is potential for aneurysmal dilatation of the diverticulum with exposure to systemic arterial pressures [5]. Apart from previous repair of a patent ductus arteriosus, risk factors for ductus arteriosus aneurysms have been poorly delineated in the literature. Other associations, but not causal relationships, include connective tissue disorders, chromoso- mal anomalies and SMAD3 mutations [3, 6, 7]. Of note, our patient sustained blunt chest wall trauma in a road traffic acci- dent approximately 12 years prior to presentation. Sudden deceleration injuries of the thorax commonly cause aortic transection but an association with aneurysmal dilation of the ductus diverticulum has not previously been described. We postulate that remote shearing forces compromised the tunica Figure 2: Digital subtraction angiogram demonstrating the chimney-graft media at the site of the ductus diverticulum and contributed to approach. Image acquired shortly before simultaneous subclavian and thoracic stent-graft deployment. aneurysmal dilation. Our patient presented with a hoarse, breathy voice and with a well-defined neck and no intraluminal thrombus intermittent bouts of coughing over 6 months. Laryngoscopic (Fig. 2). There was no contrast extravasation into the mediasti- examination demonstrated unilateral vocal cord paralysis con- num or pulmonary circulation. Following a cardiovascular sistent with a left recurrent laryngeal nerve palsy. The left multidisciplinary meeting, treatment options were discussed recurrent laryngeal nerve passes inferior to the arch of the aor- with the patient and consensus favoured endovascular repair. ta before ascending in the tracheo-oesophageal groove to sup- The distance from the proximal margin of the aneurysmal ply all the intrinsic muscles of the larynx, apart from the neck to the origin of the left subclavian artery measured just cricothyroid. Therefore, the clinical presentation in this case is under 2 cm and therefore did not meet the manufacturer’s consistent with Ortner’s cardiovocal syndrome, in which left minimal landing zone requirements. To circumnavigate this recurrent laryngeal nerve palsy arises from cardiovascular dis- dilemma, a left subclavian stent graft was inserted with its ease. In 1897 Norbert Ortner, an Austrian physician, first proximal end parallel to the main thoracic endoprosthesis. described three patients with hoarseness of voice secondary to The procedure was uncomplicated and repeat imaging a dilated left atrium in the setting of mitral stenosis [8]. Less revealed widely patent stent grafts with satisfactory position- commonly reported causes of Ortner’s syndrome include ing and absence of endoleak (Fig. 3). Our patient was dis- degenerative aneurysms of the aortic arch, dilation of the pul- charged home on the second postoperative day. She monary artery, masses in the aortopulmonary window, aortic continued speech and language therapy postoperatively and dissection and iatrogenic injury. Hoarseness in the setting of has made remarkable recovery of phonation, declining any recurrent laryngeal nerve palsy may be permanent but typically further treatment options for same. there is some functional recovery as the uninvolved cord Ductus arteriosus aneurysm presenting as hoarseness 3 Figure 3: Sagittal CT with 3D reconstruction at 1-year post-intervention, showing satisfactory placement of stent grafts. develops the ability to hyperadduct and appose the paralysed 2. Mitchell RS, Seifert FC, Miller DC, Jamieson SW, Shumway cord during phonation. NE. Aneurysm of the diverticulum of the ductus arteriosus Correction is recommended when aneurysms of the ductus in the adult. Successful surgical treatment in five patients arteriosus are >3 cm, enlarge significantly over time or cause and review of the literature. J Thorac Cardiovasc Surg symptoms [2]. In this case, there were two indications for inter- 1983;86:400–8. vention, albeit the most appropriate method of repair was not 3. Dyamenahalli U, Smallhorn JF, Geva T, Fouron JC, Cairns P, clearly defined. Given the substantial morbidity associated with Jutras L, et al. Isolated ductus arteriosus aneurysm in the open repair, endovascular techniques provide for an attractive, fetus and infant: a multi-institutional experience. J Am Coll less invasive alternative. However, there are only a small num- Cardiol 2000;36:262–9. ber of reports of ductus arteriosus aneurysms corrected with an 4. Schneider DJ, Moore JW. Patent ductus arteriosus. endovascular approach [9, 10]. Standard thoracic stent grafts Circulation 2006;114:1873–82. may be used when there is an appropriate landing zone [10]. 5. Pastuszko P, Eisenberg JA, Diehl JT. Ductus arteriosus Otherwise, as illustrated in this unique case, preservation of aneurysm in an adult patient presenting with hoarseness. J the left subclavian artery with the chimney-graft technique is Card Surg 2005;20:386–8. possible. Another option would be to perform a hybrid proce- 6. Chang JP, Chang CH, Sheih MJ. Aneurysmal dilatation of dure in which a surgical bypass is created before the stent is patent ductus arteriosus in a case of Ehlers-Danlos syn- placed over the left subclavian orifice. drome. Ann Thorac Surg 1987;44:656–7. This case highlights the feasibility of a total endovascular 7. van der Lind D, Witsenburg M, van de Laar I, Moelker A, approach to complex thoracic aneurysms as an alternative to Roos-Hesselink J. Saccular aneurysm within a patent ductus open surgery. Ultimately, the most appropriate decision should arteriosus. Lancet 2012;379:e33. follow multidisciplinary discussion, taking into consideration 8. Ortner N. Recurrent laryngeal nerve paralysis due to mitral the expertise of the available endovascular services. value stenosis. Wien Klin Wochenschr 1897;10:753–5. 9. Saito N, Kimura T, Toma M, Teragaki M, Minaminimura H, Kita T, et al. Successful endovascular repair of an aneur- CONFLICT OF INTEREST STATEMENT ysm of the ductus diverticulum with a branched stent None declared. graft: case report and review of literature. J Vasc Surg 2004;40:1228–33. REFERENCES 10. Runge V, Pieper CC, Schiller W, Praeger AJ, Probst C, 1. Lund JT, Jensen MB, Hjelms E. Aneurysm of the ductus Wilhelm KE. Endovascular repair of an ductus arteriosus aneurysm causing Ortner syndrome. Vasc Endovasc Surg arteriosus. A review of the literature and the surgical impli- cations. Eur J Cardiothorac Surg 1991;5:566–70. 2014;48:271–4.

Journal

Journal of Surgical Case ReportsOxford University Press

Published: Apr 1, 2016

There are no references for this article.