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6 Cotran RS, Kumar V, Abbas AK, Collins T, Robbins SL, Schmitt B. Pathologic Basis of the Disease, 6th ed. Philadelphia, PA: Saunders. 1999; 498–510. 7 Son CN, Jung KH, Song SY, Jun JB. Monckeberg’s sclerosis in a patient with systemic sclerosis. Rheumatol Int. 2009; 30:105–107. 8 Tsai CW, Kuo CC, Hwang JJ. Monckeberg’s sclerosis. Acta Clin Belg. 2010; 65:361. 9 McCullough PA, Chinnaiyan KM, Agrawal V, Danielewicz E, Abela GS. Amplification of atherosclerotic calcification and Monckeberg’s sclerosis: A spectrum of the same disease process. Adv Chronic Kidney Dis. 2008; 15:396–412. 10 Sorenson MD, Long LO, Wessells H, Kuan J. Monckeberg’s calciphylaxis with necrosis of the glans penis: A case presentation. Hemodial Int. 2007; 11:300–302. 11 Moe SM, O’Neill KD, Duan D, et al. Medial artery calcification in ESRD patients is associated with deposition of bone matrix proteins. Kidney Int. 2002; 61:638–647. 12 Demer LL, Tintut Y, Parhami F. Novel mechanisms in accelerated vascular calcification in renal disease patients. Curr Opin Nephrol Hypertens. 2002; 11:437–443.

13 Yilmaz MI, Sonmez A, Saglam M, et al. FGF-23 and vascular dysfunction in patients with stage 3 and 4 chronic kidney disease. Kidney Int. 2010; 78:679–685. 14 Nasrallah MM, El-Shehaby AR, Salem MM, Osman NA, El Sheikh E, Sharaf El Din UA. Fibroblast growth factor-23 (FGF-23) is independently correlated to aortic calcification in haemodialysis patients. Nephrol Dial Transplant. 2010; 25:2679–2685. 15 Yatzidis H. Successful sodium thiosulfate treatment for recurrent calcium urolithiasis. Clin Nephrol. 1985; 23:63– 67. 16 Agroyannis BJ, Koutsikos DK, Tzanatos HA, Konstadinidou IK. Sodium thiosulfate in the treatment of renal tubular acidosis I with nephrocalcinosis. Scand J Urol Nephrol. 1994; 28:107–108. 17 Kyriakopoulos G, Kontogianni K. Sodium thiosulfate treatment of tumoral calcinosis in patients with end-stage renal disease. Ren Fail. 1990; 12:213–219. 18 Papadakis JT, Patrikarea A, Digenis GE, et al. Sodium thiosulfate in the treatment of tumoral calcifications in a hemodialysis patient without hyperparathyroidism. Nephron. 1996; 72:308–312.

Thrombosed hemodialysis access as an unusual source of emboli in the upper extremity of a kidney transplant recipient Mi-Hyeong KIM,1 Jeong Kye HWANG,3 Ho Jong CHUN,2 In Sung MOON,1 Ji Il KIM1 1

Department of Surgery, 2Department of Interventional Radiology, Seoul St. Mary’s Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea; 3Department of Surgery, Daejeon St. Mary’s Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea

Abstract Arteriovenous fistula (AVF) is no longer used in kidney transplant recipients. However, there is no consensus regarding whether or not to ligate a well-functioning AVF after successful kidney transplantation, particularly in patients with well and stably functioning kidney transplants. Most AVFs without complications are left in situ and more than one-third of native AVFs close spontaneously. The currently accepted policy toward thrombosed AVFs is retention within the patient’s extremity without treatment. These thrombosed AVFs seldom cause serious problems. However, when combined with aneurysmal dilatation of the proximal vein adjacent to the arterial anastomotic area, the AVF could act as the source of distal arterial emboli. This is very similar clinical scenario

Correspondence to; J. I. Kim, MD, PhD, Seoul St. Mary’s Hospital, 505 Banpo-Dong, Secho-Gu, Seoul 137-040, Korea. E-mail: [email protected] Conflict of interest and financial disclosure: None to declare.

© 2013 International Society for Hemodialysis DOI:10.1111/hdi.12118

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to that observed in embolization from a peripheral arterial aneurysm. Here we describe a case report of upper extremity ischemia following massage of a thrombosed aneurysmal AVF. The patient was successfully treated with a combination of catheter-directed thromboaspiration, thrombolysis, and surgical repair of the thrombosed AVF. To the best of our knowledge, this is the first report of upper extremity embolism after massage of a thrombosed aneurysmal AVF involving this combined treatment. Key words: Embolism, thrombosed arteriovenous fistula, aneurysm, kidney transplant recipient

INTRODUCTION Native arteriovenous fistula (AVF) is the procedure of choice for patients with end-stage renal disease requiring long-term hemodialysis. However, access for hemodialysis is no longer required in kidney transplant recipients (KTRs). There is a lack of published data and consensus in the literature regarding the management of vascular access after kidney transplantation (KT). Generally accepted indications for AVF closure in patients with wellfunctioning renal allografts are high-output cardiac failure, arterial steal syndrome, venous hypertension, infection, and traumatic bleeding.1,2 AVFs without these complications are commonly kept in situ. More than onethird of these retained AVFs closed spontaneously over time.2,3 These thrombosed AVFs have a benign nature and remain clinically well tolerated. Most physicians do not consider them as a possible source of embolism. However, an AVF with aneurysmal dilatation of proximal venous outflow and distal outflow obstruction could be a source of distal arterial ischemia. Arterial emboli in an upper extremity (UE) can originate from a cardiac source, iatrogenic catheter-induced thrombus, or proximal vascular disease.4–8 Here we report an unusual cause of UE emboli after massage of AVF in a KTR who had a recently thrombosed AVF with aneurysmal degeneration of the proximal venous segment.

CASE A 64-year-old female was admitted with a 2-day history of acute onset of pain, numbness, coldness, and weakness of the left forearm, especially the third, fourth, and fifth fingers. She had a history of diabetes mellitus and hypertension as a risk factor for arteriosclerosis. Two years earlier, she had a successful KT, which was still functioning well. She has also undergone two hemodialysis access procedures before KT. Ten years earlier, she underwent placement of a left forearm brachial-antecubital loop expanded

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polytetrafluoroethylene graft, which was thrombosed 6 years after its construction. This was followed by an upper arm brachiocephalic fistula, which was well functioning until just before symptoms appeared. The thrill and bruit had disappeared on the distal segment of this AVF, and the nature of the aneurysmal segment on the juxta-anastomotic area changed from soft to hard and pulsating. The patient performed massage and applied a hot bag on an aneurysmal segment to resolve hardness. Her presenting symptoms had developed just after massage of AVF and were exacerbated by physical activity. The patient’s left distal forearm and hand were cyanotic and cooler than the right side. She had a palpable brachial pulse at the elbow crease, but no ulnar and radial pulse. There was a 3 × 4-cm-sized pulsating mass on the juxtaanastomotic area of AVF. The distal outflow vein of this AVF had no thrill or bruit. There were no abnormal laboratory data and the electrocardiogram showed a regular sinus rhythm. Color duplex ultrasound imaging showed a 2.6 × 2.5-cm-sized, partially thrombosed venous aneurysm just distal to the anastomosis. The distal outflow vein, radial and ulnar arteries were occluded. We performed echocardiography and UE computed tomographic angiography to identify other embolic sources but could not find any other embolic source. With the presumptive diagnosis of distal embolism originating from the thrombosed aneurysm, anticoagulation was initiated and the patient was subsequently taken to the angiographic suite. Angiography revealed normal subclavian, axillary, and proximal brachial arteries, but thrombosed aneurysmal dilatation of the proximal venous segment and multisegmental acute thromboembolism at the ulnar and radial arteries (Figure 1A,B). The occluded segments along the left radial and ulnar arteries were dilated and macerated with a Nanocross balloon catheter (ev3 Corp., Plymouth, MN, USA). The thrombi were aspirated via a 5-Fr Angel sheath (Cook Medical Inc., Bloomington, IN, USA). Follow-up arteriography was performed and some

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Figure 1 Patent brachial artery and aneurysmal formation of the thrombosed arteriovenous fistula (A). Multisegmental emboli at the ulnar and radial arteries (B). Residual thrombi in the distal ulnar artery after finishing catheter-directed thromboaspiration (C). Complete recanalization of the radial artery and improvement of the proximal ulnar artery and carpal arch after finishing thrombolysis (D).

residual thromboemboli were noted in the distal ulnar artery (Figure 1C). Catheter-directed thrombolysis with urokinase (Green Cross Corp., Seoul, Korea): (300,000 unit bolus followed by 50,000 units/hour) was performed. Simultaneously, a low dose of heparin infusion (500 units/ hour) was initiated through the sheath port. Her left hand became warm and the pulse of the radial and ulnar arteries was palpated. After 12 hours, follow-up angiography was performed, and this showed complete recanalization of the left radial artery and proximal ulnar artery. The carpal arch was partly recanalized with patent first, second, and fourth metacarpal arteries. The first, fourth, and fifth digital arteries were also patent (Figure 1D). She was subsequently anticoagulated with warfarin. After 2 days, the patient underwent an operation to remove the embolic source. We performed a longitudinal incision over the pulsating mass in the elbow region. The anastomosis site of AVF was isolated along the brachial artery and encircled with a vessel loop (Figure 2A). After administering heparin, we clamped the proximal and distal brachial artery of involved segment and resected the anastomotic segment of brachial artery with the thrombosed aneurysmal segment of the AVF (Figure 2B,C). The brachial artery was repaired with an end-to-end anastomosis (Figure 2D). The resected aneurysmal sac was filled with soft red and multi-lamellated thrombus (Figure 2B). Three days later, the patient was discharged in good general condition.

DISCUSSION This case illustrated an unusual complication of UE embolism from a thrombosed AVF with aneurysmal degeneration in a KTR. Upper extremity emboli comprise 15% to 20% of all emboli to the extremities.4 The most common

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cause of embolism to an UE is cardiac,4,5 but it can also be caused by proximal arterial lesions.6–8 Kidney transplant recipients have another potential source of embolism. Although it does not attract the attention of vascular or transplant surgeons or nephrologists, retained thrombosed aneurysmal AVF can cause UE embolism, as described in this case. This is the first report of the development of distal arterial embolism after massage of a partially thrombosed aneurysmal AVF. In the medical literature, there are 6 cases of UE embolism, mainly in the artery of the forearm and/or hand, which were associated with thrombosed AVF.9–11 There are several common clinical features of these cases. First, all patients had a dilated aneurysmal venous segment filled with thrombus on a juxtaanastomotic area of AVF. Second, the distal drainage vein of the AVF was already thrombosed before embolic attacks (15 days to 10 years). Lastly, all events occurred spontaneously. An aneurysmal segment of thrombosed AVF acts as an aneurysm of the artery under these circumstances. When outflow is occluded, high turbulence flow and transmission of systemic blood pressure can erode into the thrombus at the aneurysmal venous segment generating emboli which can lead to ischemia of the distal arterial circulation. Contrary to other reports, in this case ischemic symptoms developed after massage of the thrombosed aneurysmal segment of the AVF. The aneurysmal segment was filled with a mixture of fresh and old thrombus (Figure 2B). This newly formed thrombus was poorly organized and friable and disintegrated easily from the firm, laminated organized thrombus with minimal manipulation. Emboli detached from fresh thrombus by massage were thus likely the cause of UE ischemia in our patient.

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Figure 2 Anastomotic site between the brachial artery and cephalic vein and the aneurysmal segment of arteriovenous fistula (A). Aneurysm filled with thrombus (B). Segmental resection of the involved brachial artery and aneurysmal segment (C). Repair of the brachial artery by end-to-end anastomosis (D).

The objectives of treatment were to restore arterial blood flow and prevent recurrent embolic attack. To restore arterial circulation, surgical thrombectomy, catheter-directed thrombolysis, or catheter-directed thromboaspiration can be used alone or in combination, but no clear consensus exists as to the optimum technique.12–14 We used catheter-directed thromboaspiration followed by thrombolysis to restore blood flow. To the best of our knowledge, this is the first report of treatment of this entity with this combined modality. The advantages of this combined approach are: reduction of the dose of thrombolytic agent by reducing the burden of thrombus; reduction of complications related to the dose of thrombolytic agent; and reduction of the timerequired thrombolysis. Next, the thrombosed aneurysmal AVF was removed to prevent recurrent embolic attack. This can be performed with open ligation, excision, and/or arterial reconstruction. Some authors performed ligation only.9,11

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In conclusion, distal arterial embolism associated with a functioning AVF is extremely rare, as flow preferentially directs any emboli into the venous circulation. However, a patient with a partially thrombosed aneurysmal AVF in the juxta-anastomotic area with distal venous occlusion has a high risk of distal UE embolism caused by dislodgement of thrombus, either spontaneously or inadvertently by external manipulation of the AVF. In addition, majority of such emboli are asymptomatic and the real incidence of embolism could be underestimated.15,16 For these reasons, we suggest two recommendations to reduce or avoid the risk of embolism under these circumstances. We suggest prophylactic ligation of such AVFs in patients with functioning renal allografts, and we recommend that patients with functioning AVFs with aneurysmal changes are educated about the danger of massaging a thrombosed AVF.

Manuscript received August 2013; revised October 2013.

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REFERENCES 1 Manca O, Pisano GL, Carta P, et al. The management of hemodialysis arteriovenous fistulas in well functioning renal transplanted patients: Many doubts, few certainties. J Vasc Access. 2005; 6:182–186. 2 Patard JJ, Bensalah K, Lucas A, et al. Management of vascular access for hemodialysis after successful kidney transplantation. Scand J Urol Nephrol. 2002; 36:373–376. 3 Feldman HI, Kobrin S, Wasserstein A. Hemodialysis vascular access morbidity. J Am Soc Nephrol. 1996; 7:523–535. 4 Muller-Wiefel H, Sello M. Peripheral arterial embolism: Experiences with 174 treated extremities. J Cardiovasc Surg (Torino). 1973; 14:312–316. 5 Connett MC, Murray DH, Jr, Wenneker WW. Peripheral arterial emboli. Am J Surg. 1984; 148:14–19. 6 Moon IS, Hwang JK, Kim JI. Recurrent upper extremity embolism due to a crutch-induced arterial injury: A different cause of upper extremity embolism. Ann Vasc Surg. 2010; 24:554.e7–554.e12. 7 Fields WS, Lemak NA, Ben-Menachem Y. Thoracic outlet syndrome: Review and reference to stroke in a major league pitcher. AJR Am J Roentgenol. 1986; 146:809–814. 8 Lee AW, Hopkins SF, Griffen WO, Jr. Axillary artery aneurysm as an occult source of emboli to the upper extremity. Am Surg. 1987; 53:485–486. 9 Lacombe M. Digital arterial embolism after thrombosis of arteriovenous fistula. Ann Cardiol Angeiol (Paris). 1993; 42:199–202.

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10 Simosa HF, Mudumbi SV, Pomposelli FB, et al. Distal digital embolization from a thrombosed aneurysmal hemodialysis arteriovenous fistula: The benefit of a hybrid approach. Semin Dial. 2009; 22:93–96. 11 Journet J, Bui HT, Capdevila C, et al. [Digital arterial embolization from a previously thrombosed arteriovenous access: A rare and misdiagnosed complication]. Nephrol Ther. 2010; 6:121–124. 12 Valji K, Bookstein JJ, Roberts AC, et al. Pulse-spray pharmacomechanical thrombolysis of thrombosed hemodialysis access grafts: Long-term experience and comparison of original and current techniques. AJR Am J Roentgenol. 1995; 164:1495–1500; discussion 1501– 1493. 13 Patel AA, Tuite CM, Trerotola SO. Mechanical thrombectomy of hemodialysis fistulae and grafts. Cardiovasc Intervent Radiol. 2005; 28:704–713. 14 Cynamon J, Pierpont CE. Thrombolysis for the treatment of thrombosed hemodialysis access grafts. Rev Cardiovasc Med. 2002; 3(Suppl 2):S84–S91. 15 Trerotola SO, Vesely TM, Lund GB, et al. Treatment of thrombosed hemodialysis access grafts: Arrow-Trerotola percutaneous thrombolytic device versus pulse-spray thrombolysis. Arrow-Trerotola Percutaneous Thrombolytic Device Clinical Trial. Radiology. 1998; 206:403– 414. 16 Weng FL, Berns JS. Complications of percutaneous treatment of thrombosed hemodialysis access grafts. Semin Dial. 2003; 16:257–262.

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Thrombosed hemodialysis access as an unusual source of emboli in the upper extremity of a kidney transplant recipient.

Arteriovenous fistula (AVF) is no longer used in kidney transplant recipients. However, there is no consensus regarding whether or not to ligate a wel...
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