Long-term complications of cuffed tunneled central

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1 IMAGING TEACHING CASE Epistaxis as a Rare Complication of Catheter-Related Central Venous Stenosis Jin-Ju Tsai, MD, 1 Ching-Chih Hsia, MD, 1,2 Dong-Ming Tsai, MD, 1 Wei-Tsung Chen, MD, 3 and Yung-Hsuen Hsu, MD 1,2 INDEX WORDS: Epistaxis; hemodialysis; tunneled cuffed catheter; subclavian vein obstruction; central venous stenosis. Long-term complications of cuffed tunneled central venous catheters include exit-site and tunnel infections, catheter-related bacteremia, and extrinsic and intrinsic thrombosis or stenosis. 1 Catheterization of the subclavian vein should be avoided because of high rates of vessel stenosis. 2,3 Events leading to the clinical detection of subclavian vein thrombosis include arm edema and increased venous pressure during hemodialysis. 2 The clinical presentations of central venous stenosis may be obscure, and imaging studies may be needed in patients with a high suspicion of central venous stenosis. Imaging studies are important for showing the site of obstruction, collateral vessel architecture, and blood flow. We report a patient with end-stage renal disease who presented with facial edema and repeated episodes of epistaxis as a result of subclavian vein obstruction caused by repeated placements of cuffed tunneled dialysis catheters. CASE REPORT Clinical History A 48-year-old man with end-stage renal disease caused by hypertension began hemodialysis therapy in February The patient s blood pressure was well controlled after initiation of dialysis therapy. In March 2006, he reported repeated episodes of epistaxis. He had no history of nasal disease, coagulation disorders, or trauma to the nose. When the patient started hemodialysis therapy, a silicone cuffed tunneled catheter (Hickman; Bard Access System, Salt Lake City, UT) was inserted into the right subclavian vein for vascular access. A left forearm arteriovenous (AV) fistula was constructed in April 2003, but became nonfunctional in May 2003, and a right forearm AV fistula was then created. The right AV fistula worked well until April 2004, when it thrombosed. Another cuffed tunneled catheter was placed in the right subclavian vein, but was removed after 1 month because of poor performance. An AV graft was next placed in the left forearm, but failed after 2 months. Additional cuffed tunneled catheters were inserted, 1 in the right internal jugular vein that remained in place for 3 weeks, and then in the right subclavian vein, which remained functional for 2 weeks. This was the third catheter placed in the right subclavian vein. In August 2004, a right forearm AV graft was constructed, finally providing adequate blood flow. The patient s right arm developed edema shortly after AV graft creation; this initially was considered to be the result of increased venous pressure from shunting of arterial blood and inadequate vascular anatomy in the arm. In January 2006, prominent facial edema developed. The facial edema did not resolve after reducing the patient s dry weight. The exact cause of his arm and facial edema was not evaluated until the patient reported repeated episodes of epistaxis in March Epistaxis occurred spontaneously once every 1 to 2 weeks. It was not accompanied by sneezing, coughing, or straining. There was no relationship between epistaxis episodes and dialysis sessions. Epistaxis persisted even when heparin was withheld during hemodialysis. The patient reported that the bleeding was mainly from the right nostril, lasted for a few minutes, and could be stopped by packing the nostril with cotton. The patient denied symptoms of headache, hoarseness, dyspnea, hemoptysis, cyanosis, dysphagia, or chest pain. Physical examination showed a well-developed man with mild facial edema, an engorged right external jugular vein, and mild edema of the right arm. No collateral veins were noted in the upper torso. Results from liver function and coagulation tests were within normal limits. Dynamic venous pressure during hemodialysis at a blood flow of 200 ml/min was less than 150 mm Hg at the start of the dialysis treatment at both initial use of the right forearm AV graft and the time of epistaxis. Based on the facial and arm edema and unexplained recurrent epistaxis, a preliminary diagnosis of central venous obstruction was made. Imaging Studies Antegrade venography through the right arm showed obstruction of the right subclavian vein, with blood shunted into the right external jugular vein (Fig 1; Movie S1, available online at This was confirmed by using contrast-enhanced magnetic resonance venography. From the 1 Department of Nephrology, Taipei City Hospital, Jen-Ai Branch; 2 National Yang-Ming University; and 3 Department of Radiology, Taipei City Hospital, Jen-Ai Branch, Taipei, Taiwan, ROC. Received March 13, Accepted in revised form August 14, Originally published online as doi: /j.ajkd on December 15, Address correspondence to Yung-Hsuen Hsu, MD, No 10, Sec 4, Jen Ai Rd, Taipei, Taiwan, ROC. hsueen@ yahoo.com.tw 2009 by the National Kidney Foundation, Inc /09/ $36.00/0 doi: /j.ajkd American Journal of Kidney Diseases, Vol 53, No 3 (March), 2009: pp

2 556 Tsai et al central venous stenosis complicated by arm and facial edema and repeated epistaxis episodes. Diagnosis Cuffed tunneled dialysis catheter related subclavian vein stenosis. Clinical Follow-up The epistaxis resolved spontaneously about 3 months later. The patient refused to undergo both percutaneous transluminal angioplasty and surgical reconstruction of the subclavian vein obstruction. He also refused AV graft ligation or conversion to peritoneal dialysis therapy. At the 24-month follow-up, the patient remained on hemodialysis therapy through the right forearm AV graft and was still free of symptoms and signs of central venous obstruction with the exception of facial and right arm edema; no further episodes of epistaxis had occurred. Figure 1. Antegrade venography shows obstruction of the right subclavian vein (arrowhead). Shunted blood runs collaterally to the right external jugular vein (black arrow). Other collateral vessels and the right internal jugular vein are also evident (white arrow). Collateral flow returned not only through the right internal jugular vein, but also by crossing through the cavernous sinus to the left external and internal jugular veins and finally to the left brachiocephalic vein (Fig 2; Movie S2). The pterygoid plexus also was markedly congested. We concluded that the patient s congested cranial venous system, including that of his nasal cavity, was a complication of catheter-related central venous stenosis. Repeated central venous dialysis catheter placement contributed to DISCUSSION We report a patient with end-stage renal disease who presented with repeated episodes of epistaxis caused by cuffed tunneled dialysis catheter induced subclavian vein obstruction. A cuffed tunneled catheter is used for dialysis access when all other types of vascular access have failed. Long-term complications of central venous catheters include exit-site and tunnel infection, catheter-related bacteremia, and extrinsic (mural, central vein, and atrial thrombus) and intrinsic (intraluminal and catheter-tip thrombo- Figure 2. Head-and-neck first-pass contrast-enhanced magnetic resonance venography shows occlusion of the right subclavian vein. The venous return of the right arm is diverted to the right external jugular vein (F). The venous return then descends not only through the right internal jugular vein (E), but also through the cavernous sinus to the left external (I) and left internal (J) jugular veins. It finally flows to the left brachiocephalic vein (M). The left pterygoid plexus (H) also is markedly congested. A, right heart; B, pulmonary trunk; C, superior vena cava; D, right brachiocephalic vein; G, right and left cavernous sinuses; K, right and left superior and middle thyroid veins; L, left anterior jugular vein.

3 Hemodialysis Catheter Related Epistaxis 557 sis and fibrin sheath) thrombosis. 1 Catheterization of the subclavian vein should be avoided because of its high rate of stenosis. 2,3 Causes of subclavian vein thrombosis include trauma; strenuous use of the arm; transvenous pacemaker placement; catheterization for parenteral nutrition, chemotherapy, and dialysis; and thrombophilic disorder. 4 Our patient did not have a history of trauma and did not participate in activities that required vigorous exercise of his arms. However, he had had 3 cuffed tunneled catheters placed in his right subclavian vein. Arm edema is the most common clinical presentation of subclavian vein thrombosis. Other signs are increased venous pressure during dialysis and dilated collateral vessels. The development of collateral circulation can sometimes mask the obstruction, 5,6 which may be apparent only after an AV shunt is established in the ipsilateral arm. Thus, imaging studies are needed in patients with a high suspicion of central venous thrombosis. They are also important in showing the level of obstruction, collateral vessel architecture, and blood flow. We believe that our patient developed progressive right subclavian vein thrombosis as a consequence of repeated catheter placement. The patient did not present with signs or symptoms of stenosis until venous return had become overwhelmed by the creation of the right forearm AV graft, thus leading to arm edema. When the subclavian vein finally occluded, shunted blood increasingly flowed through the right external jugular vein to the cranial venous system and nasal venous plexus. The progressive increase in nasal venous pressure eventually caused spontaneous rupture of the vessels and epistaxis. The spontaneous resolution of epistaxis in our patient might have been the result of the efficient collateral venous system that developed, which diverted most of the blood flow away from the congested nasal vessels. Normal antegrade venography through the right arm (Fig 3) showed the contrast medium running from the axillary vein into the right subclavian vein, right brachiocephalic vein, and superior vena cava, then terminating in the right atrium. When the central venous system is obstructed, collateral pathways develop. The groups of collateral vessels resulting from the central venous occlusion included the jugular venous systems, azygos and hemiazygos veins (Fig 4; Movie S3), lateral thoracic and superficial thoracoabdominal veins, internal mammary veins, vertebral venous plexus, and small mediastinal collateral veins. 7,8 Duration of venous obstruction, adequacy of collateralization, and level of obstruction determine the clinical presentation of central venous obstruction. For example, epistaxis, as in our patient, resulted from the Figure 3. Normal antegrade venography through the right arm shows the contrast medium running from the axillary vein (A); joining the cephalic vein (B); draining into the right subclavian vein, right brachiocephalic vein (E), and superior vena cava (F); and finally terminating in the right atrium (G). C, dialysis catheter; D, subclavian vein.

4 558 Tsai et al Figure 4. Antegrade venography through the right arm of a patient with dialysis catheter related superior vena cava (SVC) occlusion after removal of the tunneled catheter shows 2 collateral pathways to return blood to the inferior vena cava; 1 through the lateral thoracic vein (A) and the other through the vertebral venous plexus (E) and intercostal veins (D) to the azygos (G) and hemiazygos (H) system, thereby bypassing the obstructed SVC (F). B, obstructed orifice of the azygos vein entering into the SVC; C, axillary vein. collateral jugular venous vessels. Another patient with dialysis catheter related central venous obstruction presented with esophageal varices caused by shunted blood draining into the azygos-hemiazygos system. 9 Management of central venous thrombosis includes thrombolysis, percutaneous transluminal balloon angioplasty, or surgical bypass. 10,11 Our patient refused further invasive management because his epistaxis completely resolved and he tolerated the facial edema. In conclusion, the subclavian vessels should not be chosen for placement of dialysis catheters in patients with inadequate vascular anatomy. 3 Epistaxis occurring in patients with a history of central venous catheter placement into the subclavian vein should raise suspicion of subclavian vein thrombosis. In addition, the correct diagnosis of catheter-related central venous stenosis requires a combination of clinical findings and imaging studies. ACKNOWLEDGEMENTS Support: None. Financial Disclosure: None. SUPPLEMENTARY MATERIALS Movie S1: Antegrade venography through the right arm. Movie S2: Magnetic resonance venography of the head and neck. Movie S3: Antegrade venography through the right arm from another patient. Note: The supplementary material accompanying this article (doi: /j.ajkd ) is available at www. ajkd.org. REFERENCES 1. Schwab SJ, Beathard G: The hemodialysis catheter conundrum: Hate living with them, but can t live without them. Kidney Int 56:1-17, Schwab SJ, Quarles LD, Middleton JP, Cohan RH, Saeed M, Dennis VW: Hemodialysis-associated subclavian vein stenosis. Kidney Int 33: , National Kidney Foundation: KDOQI Clinical Practice Guidelines and Clinical Practice Recommendations for 2006 Updates: Hemodialysis Adequacy, Peritoneal Dialysis Adequacy and Vascular Access. Am J Kidney Dis 48:S1- S322, 2006 (suppl 1) 4. Aburahma AF, Sadler DL, Robinson PA: Axillary subclavian vein thrombosis. Changing patterns of etiology, diagnostic, and therapeutic modalities. Am Surg 57: , Clark DD, Albina JE, Chazan JA: Subclavian vein stenosis and thrombosis: A potential serious complication in chronic hemodialysis patients. Am J Kidney Dis 15: , Glaze RC, MacDougall ML, Wiegmann TB: Thrombotic arm edema as a complication of subclavian vein catheterization and arteriovenous fistula formation for hemodialysis. Am J Kidney Dis 7: , Bashist B, Parisi A, Frager DH, Suster B: Abdominal CT findings when the superior vena cava, brachiocephalic vein, or subclavian vein is obstructed. AJR Am J Roentgenol 167: , 1996

5 Hemodialysis Catheter Related Epistaxis Muramatsu T, Miyamae T, Dohi Y: Collateral pathways observed by radionuclide superior cavography in 70 patients with superior vena caval obstruction. Clin Nucl Med 16: , Hsu YH, Yang MT, Hsia CC, Tsai DM: Esophageal varices as a rare complication of central venous dialysis tunneled cuffed catheter. Am J Kidney Dis 43:e20-e24, Gadallah MF, el-shahawy MA, Campese VM: Unilateral breast enlargement secondary to hemodialysis arteriovenous fistula and subclavian vein occlusion. Nephron 63: , Lee SJ, Neiberger R: Subclavian vein stenosis: Complication of subclavian vein catheterization for hemodialysis. Child Nephrol Urol 11: , 1991