Abstract
Pseudoaneurysms (PAs) of arteries in the lower extremities are uncommon. In most cases, a PA of the common femoral artery develops following percutaneous access and treatment with ultrasound guided thrombin injection achieves success rates approaching 98%. In contrast, the management of a PA of the distal leg vessels is more complex and may require additional endovascular and/or surgical treatments. We present a case of a recalcitrant PA involving the distal peroneal artery that developed following blunt trauma in a patient with Hemophilia B who failed ultrasound guided thrombin injection, para-aneurysmal saline injection and required two coil embolization procedures. Our observations suggest that Factor IX supplementation combined with aggressive coil embolization is the most effective treatment approach.
1
Introduction
Aneurysms of the peroneal artery are uncommon with the majority being pseudoaneurysms (PAs) . In most cases, ultrasound guided thrombin injection is effective . In contrast, management of PA in patients with a coagulopathy is more complex and may require additional endovascular and/or surgical treatments. We present a case of a recalcitrant PA of the distal peroneal artery that developed following blunt trauma in a patient with Hemophilia B. Hemophilia B (Christmas disease or Factor IX hemophilia) is a hereditary bleeding disorder caused by deficiency of blood clotting factor IX. Persons with this disorder cannot achieve adequate hemostasis and thus are prone to excessive hemorrhage following injury or trauma. A PA in the presence of Hemophilia B is a challenging combination where control of bleeding and effective occlusion of the PA can be difficult. There are few published studies describing the appropriate management of patients with a PA and Hemophilia B .
2
Case presentation
A 40-year Latino male with a diagnosis of Hemophilia B presented for evaluation of a large 7 x 7 cm mass on the lateral aspect of his right lower leg. Three months prior, the patient twisted his right ankle. At the time of injury, the entire right lower extremity was edematous which subsequently evolved over three weeks to a 7 x 7 centimeter mass on his right lower leg, just superior to the lateral malleolus. The patient initially presented to an outside emergency room, where a needle was inserted into the mass, with return of pulsatile blood flow ( Fig. 1 ). Computed tomographic angiography showed a PA measuring approximately 6 cm, communicating with the distal right peroneal artery ( Fig. 2 ). The patient was transferred to our tertiary care facility for further management. Duplex ultrasonography (D-US; 7.0 to 11.0 MHz; Sonos 5500; Hewlett-Packard Corporation, Palo Alto, California) confirmed the diagnosis of the PA ( Fig. 2 C). Ultrasound guided thrombin injection of the PA at the patient’s bedside was performed . Thrombin (Bovine thrombin; Vascular Solutions Inc., Minneapolis, USA) reconstituted in normal saline as a 100 U/mL solution was sequentially injected in 100 U increments directly into the PA under D-US guidance. A total of 300 U was injected with continuous D-US imaging with subsequent filling of the PA with thrombus and no flow from the adjacent peroneal artery into the aneurysm sac. D-US imaging the following day showed a small amount of flow within the PA. Expecting that the PA would spontaneously occlude over time given the minimal amount of flow, repeat D-US imaging was planned after an additional week.
Over the next week, the patient experienced increasing pain and difficulty ambulating. Repeat D-US showed the PA had returned and was similar in size to previous. After the administration of 1% lidocaine to the subcutaneous tissue, an 18-gauge needle mounted on a plastic syringe filled with 0.9% saline was advanced and positioned within 2 to 5 mm along the neck communicating the peroneal artery and the PA. After confirmation of extravascular and extra-aneurysmal needle position, saline was slowly injected until the resultant tissue swelling completely obliterated the PA neck (total volume of saline 4 cc) . Thrombin (Bovine thrombin; Vascular Solutions Inc., Minneapolis, USA) reconstituted in normal saline as a 100 U/mL solution was also injected in 100 U increments to a total dose of 500 U under D-US guidance directly into the PA. The immediate post-injection D-US showed complete thrombosis of the PA with no flow from the adjacent peroneal artery. However, D-US the following day showed the PA had once again returned.
The patient was then brought to the cardiac catheterization laboratory for coil embolization. Recombinant Factor IX (BeneFIX, Pfizer Inc., New York, NY) 7000 U was given intravenously immediately prior to the procedure. Common femoral access was obtained through an antegrade puncture. A 6 French Pinnacle sheath, 10 cm in length, (Terumo Interventional Systems, Somerset, New Jersey) was placed and the peroneal artery was selectively cannulated using a .014-inch PT2 wire (Boston Scientific, Natick, Massachusetts) and a .014-inch support catheter (Cross-it, Spectranetics Corporation, Colorado Springs, Colorado). A 3 mm × 2 mm Tornado embolization coil (Cook Medical Inc., Bloomington, Indiana) was placed into the distal peroneal artery, proximal to the origin of the PA ( Fig. 3 ). Completion angiography showed no flow within the PA.
D-US performed one week later showed the PA to have once again returned. A final attempt at endovascular intervention was planned. Access was obtained through the contralateral common femoral artery. A 55 cm 6 French Raabe sheath (Cook Medical Inc., Bloomington, Indiana) was placed and the peroneal artery was selectively cannulated using the same technique as above. Recombinant Factor IX 7000 U was given intravenously immediately prior to the procedure. Three Helix coils (3 mm × 8 cm, 4 mm × 8 cm and 4 mm × 12 cm, ev3 Endovascular Inc., Plymouth, Minnesota) were placed and packed tightly within the distal peroneal artery ( Fig. 4 ). The following day D-US demonstrated obliteration of the PA with no flow. Repeat D-US one week later demonstrated continued obliteration of the PA. The patient noted improvement in pain with a reduction in size of the mass.
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