1. Case details
A 60-yr-old white man presented with painless hematuria to his family doctor and was referred to a urologist. He had a 15–pack-year smoking history. Abdominal examination was unremarkable. Laboratory tests noted normal renal function (creatinine 76 μmol/l) and prostate-specific antigen (2.3 μg/l). Transurethral resection of bladder tumor (TURBT) revealed two pT1 grade3 bladder tumors. Computed tomography (CT) scan of the abdomen found no abnormalities in the upper tract, no enlarged lymph nodes, and an unremarkable liver. Furthermore, a juxtarenal aneurysm of the aorta of 4.5 cm in diameter was identified. The consulted vascular surgeon advised a biannual check-up. Two weeks later, bacillus Calmette-Guérin (BCG) instillations were initiated. After the first instillation, the patient had 3 d of general malaise without fever. After the second instillation, the patient felt extremely tired. After the third instillation, the patient remained tired and had liver function disturbances. BCG instillations were temporarily interrupted, and the patient was referred to our hospital.
Re-TURBT revealed a single pTa grade 3 tumor. Biopsies of the prostatic urethra were negative. A single postoperative mitomycin C instillation within 24 h after surgery was administered. Four weeks later, another 6-wk course of BCG commenced. During this treatment, the patient experienced night sweating and lost 10 kg of body weight. Cystoscopy and cytology were negative 3 mo after TURBT. Another three weekly instillations of BCG were given.
Three weeks later, the patient presented at the emergency unit complaining of general malaise and back pain. Abdominal examination revealed a nontender pulsatile mass in the midline. A CT scan showed a contained rupture of the aorta that had increased in diameter to 8.9 cm (Fig. 1). Because the patient was hemodynamically stable, a successful endovascular aneurysm repair procedure was performed the next day, with no endoleak at the end of the procedure. Postoperatively, the patient had fever up to 39.5 °C. Blood cultures showed a gram-positive rod that appeared to be Staphylococcus epidermidis in one of the two culture bottles, most probably contamination. Vancomycin antibiotics were given for a few days. When polymerase chain reaction and culture for mycobacteria were negative and further cultures for syphilis and Q fever were also negative, vancomycin was stopped.
Infrarenal aneurysm of abdominal aorta with two penetrating ulcers, lateral and dorsolateral.
At 6 mo after TURBT, three more weekly instillations of BCG were given. One month later, the patient presented again to the emergency unit. He was chronically unwell, was extremely tired, and had night sweating and back pain. The patient had lost 5 kg of body weight. He had a temperature of 37.5 °C. Abdominal examination was unremarkable, distal pulses were palpable in both lower extremities, and both feet were warm; however, the psoas sign was positive on the left side. CT scan showed fluid collection in the left psoas muscle. A fluorodeoxyglucose positron emission tomography–CT scan showed fluid collection around the prosthesis and a psoas abscess (Fig. 2). The fluid collection was drained with CT guidance, and 45 ml of fluid was removed. Culture of the fluid yielded mycobacteria subsequently identified as BCG strain Mycobacterium bovis. Bacterial and fungal cultures were negative. Tuberculostatics were initiated for 6 mo: rifampicin 600 mg, isoniazid 300 mg, and ethambutol 1200 mg daily. BCG instillations were stopped.
Fluorodeoxyglucose positron emission tomograpy–computed tomography scan. Endovascular graft in situ. Intense staining of fluid collection is visible around the prosthesis and a psoas abscess.
Sonography of the aorta 3 wk after drainage of the abscess showed no fluid collection. Cystoscopy and urine cytology at 9 and 12 mo after TURBT remained negative.
Mycotic aneurysms are localized irreversible arterial dilatations due to endothelial destruction by an infection, most commonly found in the abdominal aorta and femoral artery . The typical triad of symptoms—pulsatile mass, fever, and abdominal pain—were present in our patient . Combined surgical and medical approaches have greater survival than medical approaches alone, although Witjes et al described a patient with a popliteal aneurysm that was successfully treated with only a medical approach  and .
Assuming that the routes of infection are similar to M tuberculosis, three different mechanisms could explain an arterial infection by M bovis and ; (1) direct intimal colonization during hematogenous dissemination, especially in the setting of an altered arterial wall by atherosclerosis; (2) metastatic implantation of the bacteria through adventitial vasa vasorum; or (3) local vascular extension of an infected site such as contiguous lymphadenitis. Because of the presence of a preexisting aneurysm, the first hypothesis seems the most appropriate for our patient.
The incidence of mycotic aneurysms in BCG-treated patients is unknown, and it is uncertain whether patients with known aortic aneurysms should receive prophylactic antimycobacterial therapy prior to BCG instillation . Twenty cases of patients treated with intravesical BCG complicated by mycotic abdominal aortic aneurysm have been reported in the literature to date . The International Bladder Cancer Group recommends the permanent cessation of BCG therapy plus the institution of two or more antimicrobial agents (fluoroquinolones, isoniazid, rifampicin) if high-grade fever persists (>38.5 °C for >48 h) . In severe systemic BCG reactions, discontinuation of BCG therapy; initiation of high-dose fluoroquinolones and corticosteroids; and daily isoniazid, rifampicin, and ethambutol for a duration of 6 mo are advised. BCG is not responsive to pyrazinamide  and .
Standard management of primary aortic infection involves local debridement of infected aortic tissue with ligation of the aorta, combined with extra-anatomic bypass, but in situ repair with a prosthetic graft or an endovascular graft appears to be a safe treatment in the setting of the relatively low virulence of M bovis infection  and .
To prevent life-threatening aortic ruptures, it is important that primary care physicians and urologists are aware of the symptoms of a mycotic abdominal aortic aneurysm, even several months or years after BCG therapy .
Conflicts of interest
The authors have nothing to disclose.
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a Department of Urology, VU University Medical Center, Amsterdam, The Netherlands
b Department of Vascular Surgery, VU University Medical Center, Amsterdam, The Netherlands
Corresponding author. Department of Urology, VU Medical Center, De Boelelaan 1117, Postbus 7057, Amsterdam, 1007MB, The Netherlands.
© 2016 European Association of Urology, Published by Elsevier B.V.