Endovascular Therapy for Patients with Renal Angiomyolipoma Presenting with Retroperitoneal Haemorrhage

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1 Eur J Vasc Endovasc Surg (2010) 39, 739e744 SHORT REPORT Endovascular Therapy for Patients with Renal Angiomyolipoma Presenting with Retroperitoneal Haemorrhage M. Incedayi, U.C. Turba*, B. Arslan, S.S. Sabri, W.E.A. Saad, A.H. Matsumoto, J.F. Angle Department of Radiology, University of Virginia Health System, Box , Lee Street, Charlottesville, VA 22908, USA Submitted 11 September 2009; accepted 14 December 2009 Available online 21 January 2010 KEYWORDS Angiomyolipoma; Retroperitoneal haemorrhage; Catheter-directed embolisation Abstract We report our experience treating four patients with acutely bleeding angiomyolipoma (AML) of sizes between 4 and 12 cm who were managed with endovascular embolisation with a mean follow-up of 10 months. In our case series, we demonstrate that endovascular embolisation in the acute setting for bleeding AMLs is a viable treatment option. AML should be in the differential diagnosis of acutely bleeding renal masses, even when there is no fat assessed by computed tomography (CT) imaging in the renal mass. ª 2009 European Society for Vascular Surgery. Published by Elsevier Ltd. All rights reserved. Angiomyolipoma (AML) can be treated with conservative management, catheter-directed embolisation of the feeding renal artery or surgery. 1e4 Currently, prophylactic endovascular treatment of AML is widely accepted because of the risk of AML bleeding. 4e6 However, there are different methods of treatment and few reports regarding the management of patients presenting with acutely bleeding renal AML. 1,3,7,8 Our purpose is to review literature and report our experience with four cases of acute renal AML haemorrhage treated with catheter-directed embolisation. * Corresponding author. Tel.: þ ; fax: þ address: uct5d@virginia.edu (U.C. Turba). Materials and Methods After institutional review board approval was received, a retrospective review of our Hi-IQ database (Conexs, Providence, RI, USA) was undertaken between 1998 and We identified four patients with angiomyolipoma who presented with retroperitoneal haemorrhage. Three patients were treated with an ethanol:ethiodol (American Recent, Shirley, NY, USA; Savage Laboratories, Melville, NY, USA) mixture and one patient was treated with polyvinyl alcohol (PVA) particles and coils (Cook, Bloomington, IN, USA) because at presentation, the mass was indeterminate which was subsequently identified as an AML /$36 ª 2009 European Society for Vascular Surgery. Published by Elsevier Ltd. All rights reserved. doi: /j.ejvs

2 740 M. Incedayi et al. Table 1 Summary of cases. Repeat Intervention Follow-up (months) Length of hospital stay (days) Hemorrhage to intervention interval materials Imaging Findings Co-morbidities Kidney-number of lesions Cases # Gender/ Age 5 1 Left side embolized 5 months later with 3:1 Ethanol:Ethiodol Transferred from ER Ratio of 3:2 Ethanol; Ethiodol 11 cm diameter mass on right with perirenal 1 M/26 Tuberous sclerosis Bilateralnumerous 2 None 19 Transferred from ER Ratio of 3:2 Ethanol:lipiodol and PVA particles 2 F/51 None Left - two 6 cm on left mass with perirenal 12 1 Right side embolized three months later 3:1 Ethanol:Ethiodol 6 months post event Ratio of 3:1 Ethanol;Ethiodol Bilateral-four 5 cm left mass with extravasation and perinephritic 3 F/33 After twin pregnancy, bilateral-multiple 5 None 5 Transferred from ER Coils and 500e700 mm embospheres, Left-solitary 12 cm mass with perinephritic 4 M/43 Congenital deafness Results Four patients (two men and two women) were identified with a mean age of 38 years (range, 26e51 years). All had acute haemorrhage identified on computed tomography (CT). Three patients were embolised emergently, and one pregnant patient was embolised electively post-partum (Table 1). The treated tumours were all 4 cm in diameter or larger. Two patients had elective additional AML embolisations 3 and 5 months after primary procedure. In three cases, an ethanol and ethiodol mixture was used, followed by PVA and/or embospheres in the setting of acute haemorrhage. In case 1, renal angiogram demonstrated 11 cm angiomyolipoma with active haemorrhage. An intervention was performed with a renegade Hi-Flo microcatheter (Boston Scientific, Natick, MA, USA). Approximately none 18 ml of 3:2 ethanol:ethiodol mixture was slowly injected, then approximately 1.25 vials of PVA particles (250e355 mm) were used to embolize the larger AML. In addition, there was a capsular arterial branch feeding the tumour, which required 2 ml of additional mixture of 3:2 ethanol:ethiodol and PVA particles. The smaller AML (4 cm) was also treated using approximately 13 ml of 3:2 ethanol:ethidol mixture and 2 ml of PVA particles. Follow-up angiogram demonstrated no arterial flow to either of the AMLs. Similar approaches were used in cases 2 (Figs. 1e6) and 3. Case 4 was unique, in that the presenting CT demonstrated a large (12 cm) renal bleeding mass, but the CT findings were not typical findings of AML (fat was not well distinguished from blood products). Emergent left renal angiogram was performed using a 5 French C2 Cobra catheter (Cook, Bloomington, IN, USA) and selective renal arteriogram demonstrated active extravasation of contrast within two separate areas consistent with ongoing haemorrhage. Embolisation was performed with 500e700 Figure 1 The left kidney demonstrates 6 cm fat containing mass with surrounding. Blood layers in the left pararenal space and anterior pararenal space.

3 Renal Angiomyolipoma Presenting with Retroperitoneal Haemorrhage 741 Figure 2 There is a large vascular mass in the inferior pole of the left kidney with extensive neovascularity and puddling of contrast consistent with renal angiomyolipoma. micron embospheres (Biosphere Medical, Rockland, IN, USA). Post-embolisation angiograms confirmed successful occlusion of the feeding artery of the bleeding mass. Then, tornado coils (Cook Inc., Bloomington, IN, USA) were deployed at the origin of the main feeding vessel to completely seal the origin of the feeding artery. In the emergent setting, the operators chose to use embospheres and coils to stabilise the active haemorrhage. At that time, AML diagnosis could not be established, which prompted further evaluation with open exploration by a urology Figure 4 Post arteriogram demonstrates complete obliteration of the AML and stasis of flow to the inferior pole. Successful alcohol and PVA particle embolization of the left inferior pole of renal angiomyolipoma was achieved. team to evaluate the nature of the mass lesion and for possible partial nephrectomy. However, open biopsy later confirmed AML. Discussion Radiological diagnosis of AML is confirmed by demonstration of fatty tissue within the renal mass on CT or magnetic resonance imaging (MRI). However, in 5e15% of cases the fat component may not be visible. 1,3 In case 4, fatty component of the renal mass was masked by haemorrhage, which was later diagnosed by open biopsy. Figure 3 An occlusion balloon catheter was positioned within the feeding lower pole of renal artery and 4 ml of dehydrated alcohol mixed with Lipiodol was infused (3:2 dilution). Figure 5 Follow-up CT demonstrates decreased in size of the left lower pole angiomyolipoma (19 months post embolization) and resolution of the.

4 742 M. Incedayi et al. Figure 6 A) A sketch of renal angiomyolipoma showing a single vessel feeder of the mass. B) A sketch of an occlusion balloon that was placed at the origin of the single feeder artery and embolization was performed through the same occlusion balloon. C) Postembolization sketch showing absence of tumor vascularity. Possibly fatal haemorrhage is a well-described complication of AML. During the past two decades, superselective renal artery embolotheraphy, sparing the normal renal parenchyma, has become increasingly widespread in the management of renal AMLs using alcohol:ethidol liquid embolisation, particles or coils. 2,3,6,9 In 1998, Lee et al. 4 reported 87% success using iodised oil and absolute ethanol mixture to prevent haemorrhagic complications. Although many authors accepted this approach, Rimon et al. 7 increased his clinical success (94%) by adding PVA at completion (Table 2). The natural history of AML is not well recognised. However, it has been reported that AMLs increase in size with time, but growth speed is not predictable. 1 The major risk of tumour progression is haemorrhage, which can be life threatening. The haemorrhagic risk depends on not only its size, but also presence of multiple foci and intralesional aneurysms. 1,5 The incidence of haemorrhage remains unknown. Most series demonstrate that patients presenting with haemorrhage bleed from AMLs at least 4 cm in diameter. 1e3,7 Although there are very limited evidence-based size criteria for prophylactic therapeutic management of AMLs, empirically 4 cm is a widely accepted treatment criteria. 1,2,6,10 The appropriateness of embolisation during pregnancy also remains uncertain. Dabbeche et al. 1 reported two cases of endovascular intervention during pregnancy, using proper radiation protection measures. Morales et al. 11 reported one case during 10 weeks of pregnancy with normal delivery of infant 28 weeks later. Evidence-based reports are very limited with pregnancy and AML haemorrhage, but, intuitively, haemorrhagic risk increases with

5 Table 2 Literature review. Study year Number of patients/ Number of AMLs embolized technique Lesion size (cm) Emergent/ Prophylacticelective Technique success Clinical success Earthman et al /3 Absolute alcohol 4 < 2/0 100% 100% 24 Soulen et al /5 Absolute alcohol 4 < 0/5 100% 90% 21 Absolute alcohol/ Ethiodol Mourikis et al /8 Polyvinyl alcohol, 5e12 5/0 100% 80% 5 Coils Chiarugi et al /Numerous Coils 4 1/0 100% Unknown/ 36 mild renal insufficiency Kothary et al /30 Ethanol/Ethiodol 4 < 7/12 100% 70% 51 Coils þ Ethanol/ Ethiodol Rimon et al /18 Ethanol and 5.5e20 3/14 94% 94% 14 Polyvinyl alcohol particles Dabbech et al /35 Ethanol, Particles 4e11 16/19 80% 50% 18 Coils Seyam et al /6 Ethanol 2e21 0/6 100% 83.3% 39 (additionally 12 nephrectomy 11 partial nephrectomy) Current study /6 Ethanol/Ethiodol mixture Embospheres, Polyvinyl alcohol particles and Coils 4e12 3/3 100% 100% 10 Mean follow-up (months) Renal Angiomyolipoma Presenting with Retroperitoneal Haemorrhage 743

6 744 M. Incedayi et al. altered haemodymamics and external pressure similarly in our case 3. In this case series, the clinical decision to defer treatment until after deliver was made, but further evidence is needed to dictate if this is safe practice. In conclusion, embolisation in the acute setting for bleeding AMLs appears effective and safe. The role of emergent embolisation in pregnant patients with contained acute haemorrhage AML remains unclear. The previously reported ethiodol and alcohol mixture is well tolerated by patients in combination with particle embolisation. Lastly, AML should be in the differential diagnosis of acutely bleeding renal masses, even when there is no fat in the renal mass. Conflict of Interest/Funding None Acknowledgements We would like to thank Leanne Dore Lessley RT(R) VI for her artistic contribution of sketches 6A, 6B and 6C and Lauren J. Germain for her help in preparing this article. References 1 Dabbeche C, Chaker M, Chemali R, Perot V, El Hajj L, Ferriere JM, et al. [Role of embolisation in renal angiomyolipomas] Pt. J Radiol 2006 Dec;1:1859e67. 2 Seyam RM, Bissada NK, Kattan SA, Mokhtar AA, Aslam M, Fahmy WE, et al. Changing trends in presentation, diagnosis and management of renal angiomyolipoma: comparison of sporadic and tuberous sclerosis complex-associated forms. Urology 2008 Nov;72(5):1077e82. 3 Mourikis D, Chatziioannou A, Antoniou A, Kehagias D, Gikas D, Vlahos L. Selective arterial embolisation in the management of symptomatic renal angiomyolipomas. Eur J Radiol 1999 Dec; 32(3):153e9. 4 Lee W, Kim TS, Chung JW, Han JK, Kim SH, Park JH. Renal angiomyolipoma: embolotherapy with a mixture of alcohol and iodized oil. J Vasc Interv Radiol 1998 MareApr;9(2):255e61. 5 Lenton J, Kessel D, Watkinson AF. Embolisation of renal angiomyolipoma: immediate complications and long-term outcomes. Clin Radiol 2008 Aug;63(8):864e70. 6 Kothary N, Soulen MC, Clark TW, Wein AJ, Shlansky- Goldberg RD, Crino PB, et al. Renal angiomyolipoma: long-term results after arterial embolisation. J Vasc Interv Radiol 2005 Jan;16(1):45e50. 7 Rimon U, Duvdevani M, Garniek A, Golan G, Bensaid P, Ramon J, et al. Ethanol and polyvinyl alcohol mixture for transcatheter embolisation of renal angiomyolipoma. AJR Am J Roentgenol 2006 Sep;187(3):762e8. 8 Chiarugi M, Martino MC, Pucciarelli M, Decanini L, Vignali C. Recurrent retroperitoneal haemorrhage in a patient with tuberous sclerosis complex: a case report. Cases J 2008;1(1): Earthman WJ, Mazer MJ, Winfield AC. Angiomyolipomas in tuberous sclerosis: subselective embolotherapy with alcohol, with long-term follow-up study. Radiology 1986 Aug;160(2): 437e Soulen MC, Faykus Jr MH, Shlansky-Goldberg RD, Wein AJ, Cope C. Elective embolisation for prevention of hemorrhage from renal angiomyolipomas. J Vasc Interv Radiol 1994 JuleAug;5(4):587e Morales JP, Georganas M, Khan MS, Dasgupta P, Reidy JF. of a bleeding renal angiomyolipoma in pregnancy: case report and review. Cardiovasc Intervent Radiol 2005 MareApr;28(2):265e8.