Combined transcatheter managements of a huge spontaneous iliac pseudoaneurysm presenting with fever of unknown origin: a case report
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We present a successful combined endovascular repair of a rare huge spontaneous pseudoaneurysm in a patient troubled solely with fever of unknown origin.
A 79-year-old Chinese man complained of repeated episodes of fever for 10 days. His medical history, physical examination and laboratory tests were not significant. Routine antibiotics were given for suspected sepsis lasting 4 weeks without clinical improvement. Finally, an 81.9×61.6mm iliac pseudoaneurysm was found. The pseudoaneurysm originated from his left iliac arteries and covered the bifurcation of the left common iliac artery and proximal ends of both internal and external iliac arteries. A combination of endovascular repair with coil embolization and stent graft implantation was successfully performed. He underwent an uneventful recovery.
Spontaneous pseudoaneurysm with fever of unknown origin should not be ignored, especially for patients with a high risk for atherosclerosis. Combined transcatheter managements might be an alternative approach to deal with complex pseudoaneurysms, effectively and safely.
KeywordsIliac Artery Stent Graft Endovascular Repair Ceftizoxime Suspected Sepsis
Computed tomographic angiography
Fever of unknown origin
Magnetic resonance imaging
Pseudoaneurysm (PA) or false aneurysm is a relatively rare but threatening clinical disease . PAs are always the results of trauma , inflammation  or iatrogenic procedures [4, 5], presenting with pulsatile masses, compressive feelings or hemorrhage as the most notable manifestations . We report a case of a patient troubled with fever of unknown origin (FUO) that was solely due to the spontaneous formation of a huge PA in the bifurcation of his left common iliac artery. Combined endovascular managements using coils, balloon and stent graft were successfully performed.
Isolated PAs are always secondary to blunt trauma , inflammation , and iatrogenic procedures such as organ transplantation , interventional procedures  or neoplasia  that cause laceration of part of the vessel wall and extravasation of blood into surrounding tissues, followed by tamponade and clot formation. Typically, PAs present with pulsatile masses, compressive symptoms, secondary hemorrhage and neurologic deficit as the most common clinical manifestations. However, even spontaneous PAs or PAs without clear origins are clinically rare; they have been reported to occur in the facial artery , tibioperoneal trunk and anterior tibial artery , lumbar artery  and superficial femoral artery , presenting as pulsatile masses in the former two cases and as ruptured hemorrhage in the latter two respectively.
FUO is defined as a temperature higher than 38.3°C on several occasions and lasting longer than 3 weeks, with a diagnosis that remains uncertain after 1 week of investigation. There are well over 200 different reported causes of FUO and five major categories of conditions: infections, neoplasms, connective tissue diseases, miscellaneous disorders and undiagnosed conditions [10, 11].
To the best of our knowledge, spontaneous formations of PAs originating from iliac arteries with atypical clinical manifestations are extremely rare. The mechanism of this case may be correlated with iliac arteriosclerosis. Underlying brittleness of the arterial wall due to extensive lesions of calcification (Figures 1a and 1b) both in the abdominal aorta and bilateral iliac arteries made them predisposed to rupture. Some cases of PAs were reported to be associated with the process of calcification or atherosclerosis [12, 13, 14, 15, 16, 17]. Other than images, the patient’s high body mass index, cigarette smoking, family history of heart attack and medical history of hypercholesterolemia all contribute to risk factors for arteriosclerosis. Besides, in this case the deep location and pelvic surroundings made the PA present without a remarkable pulsatile mass or ruptured hemorrhage.
As for treatments of PAs, in recent years, several interventional techniques have been developed to achieve occlusion of PAs, including ultrasound-guided compression, direct percutaneous thrombin injection, transcatheter embolization with coils, glue, gelfoam or sclerosing agents and stent graft placement, showing a shorter hospitalization time, more favorable success rates and minimal morbidity . Although traditional opening surgery may be necessary in some instances, the pendulum has now swung to endovascular therapy .
In this case, the PA is a complex condition as some episodes of giant size, wide neck and bifurcation lesion existed simultaneously. Considering those situations, we tend to opt for combined endovascular managements sequentially. Embolization of his internal artery with coils ensured no recurrence of blood flow into the PA, and then occlusion of the ruptured PA and support of the vessel that supplied blood to his lower extremity with a self-expandable covered stent graft were performed successfully. The result of a 1.5-year follow-up confirmed the combined treatment to be effective and safe.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
The authors gratefully acknowledge the assistance of Qing Xia of the Department of Cardiology of Shanghai Tenth People’s Hospital for help in writing and language review.
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