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Multidisciplinary management of complicated bilateral renal artery aneurysm in a woman of childbearing age

Multidisciplinary management of complicated bilateral renal artery aneurysm in a woman of... Ruptured renal artery aneurysm (RAA) during pregnancy is a rare condition associated with high mortality rates to both the mother and the foetus. We report on a 41-year-old woman at her second trimester who presented with shock to the emergency department as a result of a ruptured left RAA. While the bleeding was successfully treated with angio- graphic embolization, a contralateral RAA, also at risk of rupture, was discovered. Due to its position on the artery bifurcation, this lesion was considered not suitable for interventional radiology and was therefore managed by hand- assisted retroperitoneoscopic nephrectomy, ex-vivo repair and autotransplantation. This was done in order to preserve renal mass and give our patient a chance of having future pregnancies without risk of rupture. Three years later, her renal function is normal, there is no evidence of recurrence, and more importantly she had two successful and uncom- plicated pregnancies. INTRODUCTION CASE REPORT Renal artery aneurysm (RAA) accounts for 1% of all aneur- A 41-year-old woman, 21-week pregnant, presented to the ysms and 22% of visceral aneurysms [1, 2]. Rupture during Emergency Department with left-flank pain, hypotension (100/ pregnancy represents a life-threatening condition [2]. If the 60 mmHg) and tachycardia (95 bpm). Blood tests were: haemo- mother can be stabilized and the chances of survival of the globin 7.3 g/dL, leukocytosis 23.4 cell × 10 /L, lactate 3.6 mmol/L, foetus are good, caesarean section precedes any attempts to base excess −8.4 mol/L and serum creatinine 82 mmol/L. Abdominal stop the bleeding. In case of foetus loss or massive maternal ultrasound was normal but foetal monitoring demonstrated a haemorrhage, angiographic embolization or nephrectomy drop in heart rate suggestive for impending demise. Placental remain the safest options. Endovascular techniques have abruption was suspected and following resuscitation she was revolutionized elective management of RAA. However, surgi- brought to theatre. We found a large left-sided retroperitoneal cal treatment is still indicated for selected cases. haematoma and a non-viable foetus. Exploration also revealed a Received: April 3, 2018. Accepted: July 12, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/7/rjy147/5048038 by Ed 'DeepDyve' Gillespie user on 03 July 2018 2 E. Favi et al. Figure 1: Abdomen contrast-enhanced computed tomography scan: massive Figure 3: Intra-operative finding: right saccular renal artery aneurysm (white retroperitoneal haematoma (white arrow) with active bleeding from a ruptured arrow) involving the main arterial branch and extending to the level of its distal 2-cm left renal artery aneurysm (black arrow). bifurcation. remaining two arteries were prepared for implantation (Fig 3). The patient was placed supine and the hand-port access was used for the autotransplant. The renal vein was anastomosed to the external iliac vein whereas the renal arteries were anastomosed to the external iliac artery. The ureteral-vesical anastomosis was performed according to Lich-Gregoire. The procedure took 481 min. Extraction, cold ischaemia and anastomosis times were 2, 48 and 52 min, respectively. Intra-operative blood loss was 280 mL. The postoperative course was uneventful. Histology showed myxoid medial degeneration of the renal artery. Three years later, her ser- um creatinine is 79 mmol/L with no RAA recurrence. She also com- pleted two uncomplicated pregnancies. DISCUSSION Ruptured RAA during pregnancy has been previously described [1]. Nakamura et al. [2] showed that over 50% of arterial aneur- Figure 2: Abdomen contrast-enhanced computed tomography scan showing an ysm ruptures in women under 40 years are pregnancy-related. intact 2.2-cm right renal artery aneurysm (white arrow). RAA is classified as saccular, fusiform, dissecting or mixed. Saccular type is the most common (70%). Aetiology includes ruptured left RAA and a contralateral RAA. At this point, it was atherosclerosis, hypertension, fibromuscular dysplasia, myxoid felt a percutaneous approach would have been more appropriate. medial degeneration, neurofibromatosis, tuberous sclerosis, Contrast-enhanced computed tomography showed a massive Ehlers–Danlos syndrome, infection, neoplasm, radiotherapy, retroperitoneal haematoma, a ruptured left RAA (Fig. 1), and an surgery, trauma and chemotherapy [1]. Most patients with a intact right-sided RAA measuring 2.2 cm (Fig. 2). Under selective RAA are asymptomatic or complain of non-specific symptoms angiography, the aneurysm was embolized and the bleeding con- (haematuria, hypertension, abdominal pain). Albeit rare (3%), trolled [3]. Recovery was rapid but a DMSA scan performed 2 rupture represents a catastrophic event [4]. Indications for weeks later, demonstrated reduced function in the treated kidney treatment are: haemorrhage, refractory hypertension, high- (37%). The risk of rupture of the right RAA was deemed significant flow arteriovenous fistula, embolization and high risk of rup- and a plan for repair was made. The lesion was saccular, wide- ture (enlargement, distal location, calcification, hypertension necked and located at the artery bifurcation thus preventing endo- and pregnancy) [2]. Intervention is also recommended for vascular treatment. We opted for hand-assisted retroperitoneo- asymptomatic lesions larger than 2 or 1 cm in women of child- scopic nephrectomy, ex-vivo repair and autotransplant. bearing age [5]. In case of rupture, open nephrectomy or embol- The patient was placed in left lateral decubitus. An 8-cm- ization are the safest options. Successful repair has been long suprapubic incision extended to the right iliac fossa was reported but it remains anecdotal [6]. For many years, elective performed and the retroperitoneum was entered. A hand-port treatment of RAA has been mainly surgical but recent out- (GelPort Laparoscopic System, Applied Medical, USA) and comes of endovascular procedures have increased the number three 12-mm ports, one for the 30° camera and the others for of patients referred to interventional radiology. Major advan- the instruments were inserted. Ureter, renal artery and renal tages are lower invasiveness and reduced morbidity [7]. vein were divided. The kidney was extracted through the inci- However, severe complications have been described and no sion, flushed and immerged into a cold solution (Soltran, long-term follow-up is available. Multiple, wide-necked or dis- Baxter Healthcare, USA). The aneurysm was resected and the tal aneurysms involving small branches of the renal artery are Downloaded from https://academic.oup.com/jscr/article-abstract/2018/7/rjy147/5048038 by Ed 'DeepDyve' Gillespie user on 03 July 2018 Treatment of bilateral renal artery aneurism 3 considered not suitable for endovascular treatment. Surgery CONFLICT OF INTEREST STATEMENT includes nephrectomy, aorto-renal by-pass and vascular repair. The authors have no financial conflicts of interest. The subject Repair involves excision of the lesion and reconstruction of the of the present case report gave informed consent for publication artery. The procedure requires prolonged interruption of the of the article. The procedures followed were in accordance with renal blood flow with possible development of acute tubular the ethical standards of the Helsinki Declaration (1964, amended necrosis. Cold perfusion of the kidney reduces ischaemia- most recently in 2008) of the World Medical Association. reperfusion injury thus allowing time-consuming manoeuvres [8]. Back-table-surgery also gives the chance of operating with- out anatomical restrains. For all these reasons, nephrectomy REFERENCES with extracorporeal repair under cold perfusion followed by 1. Rijbroek A, van Dijk HA, Roex AJ. Rupture of renal artery reimplantation represents the best option [9]. Open nephrectomy aneurysm during pregnancy. Eur J Vasc Surg 1994;8:375–6. is considered the golden standard, but latest results support mini- 2. Nakamura R, Koyama S, Maeda M, Kobayashi M, Tanaka Y, invasive surgery even in complex cases. The experience gained Kubota S, et al. Rupture of renal artery aneurysm during the with living donation demonstrates that laparoscopic nephrectomy early post-partum period. JObstetGynaecolRes 2013;39:1476–9. is safe and offers better results than open procedures in terms of 3. Maughan E, Webster C, Konig T, Renfrew I. Endovascular postoperative complications, pain and length of hospitalization. management of renal artery aneurysm rupture in preg- Renal function and long-term transplant outcomes are also nancy—a case report. Int J Surg Case Rep 2015;12:41–3. equivalent [10]. 4. Tham G, Ekelund L, Herrlin K, Lindstedt EL, Olin T, Bergentz Our patient was pregnant and presented with massive SE. Renal artery aneurysms. Natural history and prognosis. bleeding. The haemorrhage was managed by embolization but Ann Surg 1983;197:348–52. loss of renal function developed. Considering her strong will to 5. Klausner JQ, Harlander-Locke MP, Plotnik AN, Lehrman E, have a child and the risk of rupture of the remaining aneurysm, DeRubertis BG, Lawrence PF. Current treatment of renal a plan for repair was made. The lesion was unsuitable for endo- artery aneurysms may be too aggressive. J Vasc Surg 2014; vascular treatment and we opted for ex-vivo repair and auto- 59:1356–61. transplant. We preferred the retroperitoneoscopic technique 6. Lacroix H, Bernaerts P, Nevelsteen A, Hanssens M. Ruptured because adhesions from previous surgery and anatomy altered renal artery aneurysm during pregnancy: successful ex situ by the organized haematoma, would have been difficult to repair and autotransplantation. J Vasc Surg 2001;33:188–90. manage using a transperitoneal approach. Moreover, the same 7. Xiong J, Guo W, Liu X, Yin T, Jia X, Zhang M. Renal artery incision for nephrectomy could be used for autotransplant aneurysm treatment with stent plus coil embolization. Ann without extra dissection. Hypothermic perfusion reduced pos- Vasc Surg 2010;24:e1–3. sible parenchymal damage. 8. Thompson RH, Frank I, Lohse CM, Saad IR, Fergany A, RAA rupture is a life-threatening condition that needs to be Zincke H, et al. The impact of ischemia time during open considered in pregnant patients with abdominal pain and hemo- nephron sparing surgery on solitary kidneys: a multi- dynamic instability. In selected cases not suitable for endovascular institutional study. J Urol 2007;177:471–6. treatment and for functionally uninephric patients, prophylactic 9. Morin J, Chavent B, Duprey A, Albertini JN, Favre JP, Barral X. repair should be attempted. Hand-assisted retroperitoneoscopic Early and late results of ex vivo repair and autotransplantation nephrectomy, ex-vivo repair and autotransplant represents a safe in solitary kidneys. Eur J Vasc Endovasc Surg 2012;43:716–20. and feasible option. 10. Brook NR, Gibbons N, Nicol DL, McDonald SP. Open and lap- aroscopic donor nephrectomy: activity and outcomes from ACKNOWLEDGEMENTS all Australasian transplant centers. Transplantation 2010;89: None. 1482–8. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/7/rjy147/5048038 by Ed 'DeepDyve' Gillespie user on 03 July 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Multidisciplinary management of complicated bilateral renal artery aneurysm in a woman of childbearing age

Journal of Surgical Case Reports , Volume Advance Article (7) – Jul 3, 2018

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Oxford University Press
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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018.
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2042-8812
DOI
10.1093/jscr/rjy147
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Abstract

Ruptured renal artery aneurysm (RAA) during pregnancy is a rare condition associated with high mortality rates to both the mother and the foetus. We report on a 41-year-old woman at her second trimester who presented with shock to the emergency department as a result of a ruptured left RAA. While the bleeding was successfully treated with angio- graphic embolization, a contralateral RAA, also at risk of rupture, was discovered. Due to its position on the artery bifurcation, this lesion was considered not suitable for interventional radiology and was therefore managed by hand- assisted retroperitoneoscopic nephrectomy, ex-vivo repair and autotransplantation. This was done in order to preserve renal mass and give our patient a chance of having future pregnancies without risk of rupture. Three years later, her renal function is normal, there is no evidence of recurrence, and more importantly she had two successful and uncom- plicated pregnancies. INTRODUCTION CASE REPORT Renal artery aneurysm (RAA) accounts for 1% of all aneur- A 41-year-old woman, 21-week pregnant, presented to the ysms and 22% of visceral aneurysms [1, 2]. Rupture during Emergency Department with left-flank pain, hypotension (100/ pregnancy represents a life-threatening condition [2]. If the 60 mmHg) and tachycardia (95 bpm). Blood tests were: haemo- mother can be stabilized and the chances of survival of the globin 7.3 g/dL, leukocytosis 23.4 cell × 10 /L, lactate 3.6 mmol/L, foetus are good, caesarean section precedes any attempts to base excess −8.4 mol/L and serum creatinine 82 mmol/L. Abdominal stop the bleeding. In case of foetus loss or massive maternal ultrasound was normal but foetal monitoring demonstrated a haemorrhage, angiographic embolization or nephrectomy drop in heart rate suggestive for impending demise. Placental remain the safest options. Endovascular techniques have abruption was suspected and following resuscitation she was revolutionized elective management of RAA. However, surgi- brought to theatre. We found a large left-sided retroperitoneal cal treatment is still indicated for selected cases. haematoma and a non-viable foetus. Exploration also revealed a Received: April 3, 2018. Accepted: July 12, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/7/rjy147/5048038 by Ed 'DeepDyve' Gillespie user on 03 July 2018 2 E. Favi et al. Figure 1: Abdomen contrast-enhanced computed tomography scan: massive Figure 3: Intra-operative finding: right saccular renal artery aneurysm (white retroperitoneal haematoma (white arrow) with active bleeding from a ruptured arrow) involving the main arterial branch and extending to the level of its distal 2-cm left renal artery aneurysm (black arrow). bifurcation. remaining two arteries were prepared for implantation (Fig 3). The patient was placed supine and the hand-port access was used for the autotransplant. The renal vein was anastomosed to the external iliac vein whereas the renal arteries were anastomosed to the external iliac artery. The ureteral-vesical anastomosis was performed according to Lich-Gregoire. The procedure took 481 min. Extraction, cold ischaemia and anastomosis times were 2, 48 and 52 min, respectively. Intra-operative blood loss was 280 mL. The postoperative course was uneventful. Histology showed myxoid medial degeneration of the renal artery. Three years later, her ser- um creatinine is 79 mmol/L with no RAA recurrence. She also com- pleted two uncomplicated pregnancies. DISCUSSION Ruptured RAA during pregnancy has been previously described [1]. Nakamura et al. [2] showed that over 50% of arterial aneur- Figure 2: Abdomen contrast-enhanced computed tomography scan showing an ysm ruptures in women under 40 years are pregnancy-related. intact 2.2-cm right renal artery aneurysm (white arrow). RAA is classified as saccular, fusiform, dissecting or mixed. Saccular type is the most common (70%). Aetiology includes ruptured left RAA and a contralateral RAA. At this point, it was atherosclerosis, hypertension, fibromuscular dysplasia, myxoid felt a percutaneous approach would have been more appropriate. medial degeneration, neurofibromatosis, tuberous sclerosis, Contrast-enhanced computed tomography showed a massive Ehlers–Danlos syndrome, infection, neoplasm, radiotherapy, retroperitoneal haematoma, a ruptured left RAA (Fig. 1), and an surgery, trauma and chemotherapy [1]. Most patients with a intact right-sided RAA measuring 2.2 cm (Fig. 2). Under selective RAA are asymptomatic or complain of non-specific symptoms angiography, the aneurysm was embolized and the bleeding con- (haematuria, hypertension, abdominal pain). Albeit rare (3%), trolled [3]. Recovery was rapid but a DMSA scan performed 2 rupture represents a catastrophic event [4]. Indications for weeks later, demonstrated reduced function in the treated kidney treatment are: haemorrhage, refractory hypertension, high- (37%). The risk of rupture of the right RAA was deemed significant flow arteriovenous fistula, embolization and high risk of rup- and a plan for repair was made. The lesion was saccular, wide- ture (enlargement, distal location, calcification, hypertension necked and located at the artery bifurcation thus preventing endo- and pregnancy) [2]. Intervention is also recommended for vascular treatment. We opted for hand-assisted retroperitoneo- asymptomatic lesions larger than 2 or 1 cm in women of child- scopic nephrectomy, ex-vivo repair and autotransplant. bearing age [5]. In case of rupture, open nephrectomy or embol- The patient was placed in left lateral decubitus. An 8-cm- ization are the safest options. Successful repair has been long suprapubic incision extended to the right iliac fossa was reported but it remains anecdotal [6]. For many years, elective performed and the retroperitoneum was entered. A hand-port treatment of RAA has been mainly surgical but recent out- (GelPort Laparoscopic System, Applied Medical, USA) and comes of endovascular procedures have increased the number three 12-mm ports, one for the 30° camera and the others for of patients referred to interventional radiology. Major advan- the instruments were inserted. Ureter, renal artery and renal tages are lower invasiveness and reduced morbidity [7]. vein were divided. The kidney was extracted through the inci- However, severe complications have been described and no sion, flushed and immerged into a cold solution (Soltran, long-term follow-up is available. Multiple, wide-necked or dis- Baxter Healthcare, USA). The aneurysm was resected and the tal aneurysms involving small branches of the renal artery are Downloaded from https://academic.oup.com/jscr/article-abstract/2018/7/rjy147/5048038 by Ed 'DeepDyve' Gillespie user on 03 July 2018 Treatment of bilateral renal artery aneurism 3 considered not suitable for endovascular treatment. Surgery CONFLICT OF INTEREST STATEMENT includes nephrectomy, aorto-renal by-pass and vascular repair. The authors have no financial conflicts of interest. The subject Repair involves excision of the lesion and reconstruction of the of the present case report gave informed consent for publication artery. The procedure requires prolonged interruption of the of the article. The procedures followed were in accordance with renal blood flow with possible development of acute tubular the ethical standards of the Helsinki Declaration (1964, amended necrosis. Cold perfusion of the kidney reduces ischaemia- most recently in 2008) of the World Medical Association. reperfusion injury thus allowing time-consuming manoeuvres [8]. Back-table-surgery also gives the chance of operating with- out anatomical restrains. For all these reasons, nephrectomy REFERENCES with extracorporeal repair under cold perfusion followed by 1. Rijbroek A, van Dijk HA, Roex AJ. Rupture of renal artery reimplantation represents the best option [9]. Open nephrectomy aneurysm during pregnancy. Eur J Vasc Surg 1994;8:375–6. is considered the golden standard, but latest results support mini- 2. Nakamura R, Koyama S, Maeda M, Kobayashi M, Tanaka Y, invasive surgery even in complex cases. The experience gained Kubota S, et al. Rupture of renal artery aneurysm during the with living donation demonstrates that laparoscopic nephrectomy early post-partum period. JObstetGynaecolRes 2013;39:1476–9. is safe and offers better results than open procedures in terms of 3. Maughan E, Webster C, Konig T, Renfrew I. Endovascular postoperative complications, pain and length of hospitalization. management of renal artery aneurysm rupture in preg- Renal function and long-term transplant outcomes are also nancy—a case report. Int J Surg Case Rep 2015;12:41–3. equivalent [10]. 4. Tham G, Ekelund L, Herrlin K, Lindstedt EL, Olin T, Bergentz Our patient was pregnant and presented with massive SE. Renal artery aneurysms. Natural history and prognosis. bleeding. The haemorrhage was managed by embolization but Ann Surg 1983;197:348–52. loss of renal function developed. Considering her strong will to 5. Klausner JQ, Harlander-Locke MP, Plotnik AN, Lehrman E, have a child and the risk of rupture of the remaining aneurysm, DeRubertis BG, Lawrence PF. Current treatment of renal a plan for repair was made. The lesion was unsuitable for endo- artery aneurysms may be too aggressive. J Vasc Surg 2014; vascular treatment and we opted for ex-vivo repair and auto- 59:1356–61. transplant. We preferred the retroperitoneoscopic technique 6. Lacroix H, Bernaerts P, Nevelsteen A, Hanssens M. Ruptured because adhesions from previous surgery and anatomy altered renal artery aneurysm during pregnancy: successful ex situ by the organized haematoma, would have been difficult to repair and autotransplantation. J Vasc Surg 2001;33:188–90. manage using a transperitoneal approach. Moreover, the same 7. Xiong J, Guo W, Liu X, Yin T, Jia X, Zhang M. Renal artery incision for nephrectomy could be used for autotransplant aneurysm treatment with stent plus coil embolization. Ann without extra dissection. Hypothermic perfusion reduced pos- Vasc Surg 2010;24:e1–3. sible parenchymal damage. 8. Thompson RH, Frank I, Lohse CM, Saad IR, Fergany A, RAA rupture is a life-threatening condition that needs to be Zincke H, et al. The impact of ischemia time during open considered in pregnant patients with abdominal pain and hemo- nephron sparing surgery on solitary kidneys: a multi- dynamic instability. In selected cases not suitable for endovascular institutional study. J Urol 2007;177:471–6. treatment and for functionally uninephric patients, prophylactic 9. Morin J, Chavent B, Duprey A, Albertini JN, Favre JP, Barral X. repair should be attempted. Hand-assisted retroperitoneoscopic Early and late results of ex vivo repair and autotransplantation nephrectomy, ex-vivo repair and autotransplant represents a safe in solitary kidneys. Eur J Vasc Endovasc Surg 2012;43:716–20. and feasible option. 10. Brook NR, Gibbons N, Nicol DL, McDonald SP. Open and lap- aroscopic donor nephrectomy: activity and outcomes from ACKNOWLEDGEMENTS all Australasian transplant centers. Transplantation 2010;89: None. 1482–8. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/7/rjy147/5048038 by Ed 'DeepDyve' Gillespie user on 03 July 2018

Journal

Journal of Surgical Case ReportsOxford University Press

Published: Jul 3, 2018

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