Ruptured mycotic thoracoabdominal aortic aneurysms: A report of three cases and a systematic review☆☆☆

Article Outline

• Abstract
• Case reports
  • Case 1
  • Case 2
  • Case 3
• Discussion
  • Pathogenesis
  • Presentation and investigations
  • Antibiotic therapy
  • Technical considerations in surgical planning
  • Elective and emergency surgery
  • Graft material
  • Survival to discharge
  • Long-term follow-up
• Conclusion
• Appendix: Search strategy
• References
• Copyright

Abstract 

We report three cases of ruptured mycotic thoracoabdominal aortic aneurysms (TAAAS) and a review of the literature. Escherichia coli and Streptococcus pneumoniae (2 patients) were the responsible organisms. Surgical management consisted of wide debridement of necrotic tissue and in situ repair with a Dacron graft. Antibiotics were administered intravenously in the hospital and continued orally after discharge for at least 6 weeks, until clinical and laboratory parameters were normalized. A review of the literature showed that Gram-negative microorganisms are found in 47% of mycotic TAAAs. A trend toward increased mortality for these organisms, compared with Gram-positive microorganisms, was observed (P = .09). Lifelong antimicrobial therapy is controversial. No difference in survival or recurrence rate was found between series advocating lifelong therapy and those suggesting prolonged (6 weeks to 12 months) therapy (median follow-up period, 18 and 19 months, respectively). In situ repair with synthetic material can be successful if prompt confirmation of infection is obtained, all possibly infected tissue is resected, and antibiotic therapy based on sensitivity data is administered for a prolonged period. A short-term survival rate as high as 82% can be expected with this strategy, but data on long-term survival rates are limited. Polytetrafluoroethylene-expanded grafts, homografts, and antibiotic-bonded grafts may offer advantages over Dacron grafts, but data are insufficient to draw conclusions. Careful long-term follow-up is an important element of the treatment of these patients. We suggest antibiotic treatment until biochemical parameters of inflammation (white cell count, erythrocyte sedimentation rate, or C-reactive protein) return to normal and a computerized tomography scan every 3 months for 1 year, then annually. (J Vasc Surg 2001;33:861-7.)

Mycotic aneurysms are a life-threatening condition with significant morbidity and mortality. Sir William Osler coined the term mycotic aneurysm in his Gulstonian lectures in 1851,¹ when he described a 30-year-old man who died of a ruptured infected thoracic aneurysm caused by infective endocarditis.

From 0.8% to 3.4% of all aortic aneurysms are mycotic.², ³, ⁴ Thoracoabdominal aortic aneurysms (TAAAs) account for 5% to 10% of aneurysms of the aorta,⁵ and mycotic aneurysms of the thoracoabdominal aorta represent 1.8% of all TAAAs.⁵

Between 1997 and 1999, we operated on three patients with a ruptured mycotic TAAA (4% of all TAAAs treated). We report our experience with the management of this condition and review the literature.

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Case reports 

Case 1 

A 68-year-old man had general symptoms of malaise, severe abdominal and back pain, chills, fever, and confusion. Before his admission, he complained of vague abdominal pain 1 week in duration and underwent a colonoscopy, the results of which were negative for colonic disease. He was a smoker of 50 pack-years with hypertension, diabetes mellitus, and a history of urinary tract infection in the previous 3 months. On physical examination, a pulsatile mass was present in the epigastrium, his temperature was 37.5°C, and his cardiovascular hemodynamics were stable. His white cell count was 14.0 × 10⁹/L, his hemoglobin level was 110 g/L, his platelet count was 234 × 10⁹/L, his creatinine level was 74 μmol/L, and his urea level was 3.6 mmol/L. A saccular, lobulated perivisceral aortic aneurysm 4.5 cm in diameter with a contained rupture at the level of the diaphragmatic crura was shown by means of computed tomographic scans (Fig 1, A ).

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• Fig. 1. 

  Computerized axial tomograms of patients 1, 2, and 3, respectively, display lobulated aneurysm of 4.5 cm with contained rupture (A) , contained extravasation of contrast media from aneurysm at level of diaphragmatic crura (B) , and an 8.2-cm irregular ruptured aneurysm (C) .

A necrotizing infection extending from the lower thoracic to the infrarenal aorta was revealed by means of an emergency thoracoabdominal exploration. The aorta was clamped proximally and entered posteriorly. Occlusion catheters were inserted in the visceral and iliac arteries. The aneurysm was transected perpendicular to the long axis of the aorta, close to the proximal clamp. A proximal anastomosis to a Dacron graft was performed. All anastomoses were performed with running prolene sutures and reinforced with interrupted sutures with pledgets. A Carrel patch containing the take-off of the visceral arteries was excised from the anterior aortic wall and anastomosed to the graft. Flow to the visceral arteries was reestablished (visceral ischemic time, 32 minutes). The distal aorta was then transected close to the bifurcation, and a distal anastomosis was performed. The distal clamp was removed, restoring flow to the lower extremities (total aortic clamp time, 46 minutes). The remainder of the aneurysmal sac was then excised, and extensive debridement of the surrounding tissues was performed, to an extent compatible with the need to respect vital structures and to close surgical wounds. Intravenous metronidazole (500 mg), vancomycin (1 g), and ceftazidime (1 g) were given intraoperatively and continued for 72 hours postoperatively. Cultures of the aneurysmal wall and contents revealed Escherichia coli species. According to available sensitivity reports, 1 g of cefotetan and 400 mg ciprofloxacin were administered intravenously every 12 hours for 6 weeks. Oral ciprofloxacin (500 mg twice daily) was continued for 4 months, until the erythrocyte sedimentation rate (ESR) returned to normal. Prolonged ventilatory support and acute renal failure requiring temporary dialysis characterized the postoperative course, but there were no neurological deficits. After a 4-month hospitalization in acute and rehabilitation settings, the patient was living independently after 26 months' follow-up.

Case 2 

A 61-year-old woman had a 1-week history of low back pain of increasing severity. She had a history of smoking, alcohol abuse, and hypertension. Six weeks before her admission, she had been treated at another institution for left lower lobe pneumonia. She was hemodynamically stable, but severely ill, distressed, and had a temperature of 39°C. Her white cell count was 16 × 10⁹/L, her hemoglobin level was 137 g/L, her platelet count was 285 × 10⁹/L, her creatinine level was 67 μmol/L, and her urea level was 4.2 mmol/L. A multilobulated saccular TAAA, group III with contained extravasation of contrast media, was shown by means of computed tomographic scans (Fig 1, B ). In the emergency department, 80 mg of gentamicin, 500 mg of metronidazole, and 1 g of vancomycin were administered intravenously. A ruptured mycotic aneurysm involving the visceral arteries and the descending thoracic aorta was revealed by means of a thoracoabdominal exploration. Reconstruction with an in situ Dacron graft was performed, in the manner described earlier. Visceral and total clamp times were 40 and 51 minutes, respectively. Blood culture tests were positive for Streptococcus pneumoniae , and the antibiotic regimen was changed to 1 g vancomycin daily and 4.5 g/0.5 g piperacillin-tazobactam four times daily. When the results of microbial sensitivity tests were available, the antibiotic regimen was changed to 2 g of ceftriaxone intravenously daily, until the patient's discharge from the hospital 8 weeks later.

She was discharged from the intensive care unit 7 days after surgery and transferred to a rehabilitation hospital after 7 weeks with normal renal function. She continued to take one tablet of oral trimethoprim-sulfamethoxazole twice daily for 2 months. She had paraparesis, but she was at home, able to walk with the assistance of a walker, and had normal bladder and bowel function, at 22 months' follow-up.

Case 3 

A 67-year-old woman had a sudden onset of abdominal and back pain. She had a history of myocardial infarction, atrial fibrillation, idiopathic pulmonary fibrosis, and multiple myeloma. Two months earlier, a rectovaginal fistula, which was thought to be caused by Crohn's disease, had developed, and a TAAA surrounded by an inflammatory response was disclosed by means of computed tomography. During investigations for this problem, the patient had pneumococcal meningitis, complicated by a syndrome of inappropriate antidiuretic hormone secretion and idiopathic myopathy. She eventually recovered and left the hospital.

When the patient came to the emergency department, a multilobulated TAAA, group III, 8.2 cm in diameter, with erosion of the vertebral bodies and a contained rupture at the level of the crura of the diaphragm, was disclosed by means of a computed tomography scan (Fig 1, C ). She was in atrial fibrillation, with a temperature of 37°C, a white cell count of 12 × 10⁹/L, a hemoglobin level of 89 g/L, a platelet level of 125 × 10⁹/L, a creatinine level of 68 μmol/L, and a urea level 6.6 mmol/L. Repair was accomplished as in the aforementioned 2 cases, with visceral ischemia and total clamp times of 15 and 32 minutes, respectively.

Vancomycin (1 g), metronidazole (500 mg), and ceftazidime (1 g) were given intravenously at the time of surgery. Postoperatively, cultures of the aneurysmal wall were positive for S pneumoniae , and she was treated with 4.5 g/0.5 g piperacillin-tazobactam every 8 hours and 1 g vancomycin every 12 hours intravenously for 3 weeks. She was discharged home without renal or neurologic complications and treated with one tablet of trimethoprim-sulfamethoxazole twice daily for 6 weeks. At 14 months' follow-up, she was living independently.

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Discussion 

Reports of mycotic TAAAs were identified through a MEDLINE database search from 1966 to November 1999, with the use of Ovid software (Ovid Technologies, New York, NY) and the search strategy outlined in the Appendix.

Pathogenesis 

Mycotic aneurysms involving the thoracoabdominal aorta are rare. In the last three decades, 73 cases have been reported (Table I),⁴, ⁶, ⁷, ⁸, ⁹, ¹⁰, ¹¹, ¹², ¹³, ¹⁴, ¹⁵, ¹⁶, ¹⁷, ¹⁸, ¹⁹, ²⁰, ²¹, ²², ²³, ²⁴, ²⁵, ²⁶, ²⁷, ²⁸, ²⁹, ³⁰, ³¹, ³², ³³, ³⁴, ³⁵, ³⁶ 25 of which ruptured (34%).⁸, ¹⁰, ¹¹, ¹², ¹⁴, ¹⁷, ²¹, ²⁶, ²⁸, ²⁹, ³⁵, ³⁶

Table I. Mycotic thoracoabdominal aortic aneurysms

MI, Myocardial infarction.

Microbial arteritis, the most frequent pathogenic mechanism,⁸, ¹⁴ occurs in arteries already weakened by congenital or acquired disease.³⁷ Primary sources of infection are identifiable in as many as 86% of patients with mycotic abdominal³⁸, ³⁹ and thoracoabdominal aneurysm⁸, ¹⁵; urinary tract infections, gastrointestinal instrumentation, salmonellosis, upper respiratory tract infections, intravenous line sepsis, dental extractions, cellulitis, pneumonia, osteomyelitis, and open infected wounds have all been described. Debilitating diseases or conditions associated with depression of the immune system, including prolonged steroid use, immunosuppressive agents, alcoholism, irradiation, and chronic renal failure, were reported by Chan⁸ in 60% of patients with a mycotic TAAA.

In our review of published cases, bacteriology results of mycotic TAAAs show Gram-negative bacilli in 47% of cases, Gram-positive cocci in 33% of cases, rare organisms in 18% of cases (Candida species, Bacteroides fragilis , mycobacteria, clostridia), and no growth in 2% of cases (Table II).

Table II. Bacteriology of thoracoabdominal aortic aneurysms⁴, ⁶, ⁷, ⁹, ¹⁰, ¹¹, ¹², ¹⁴, ¹⁵, ¹⁶, ¹⁷, ¹⁸, ¹⁹, ²⁰, ²¹, ²², ²³, ²⁴, ²⁵, ²⁶, ²⁷, ²⁸, ²⁹, ³⁰, ³¹, ³³, ³⁴, ³⁵, ³⁶

There was a trend, which did not reach statistical significance, for mycotic aneurysms caused by Gram-negative bacteria to be associated with a higher mortality rate than those caused by Gram-positive bacteria (Fisher exact test, P = .09).

Presentation and investigations 

For the timely diagnosis of a mycotic TAAA, a high index of suspicion is necessary in patients who have fever, abdominal or back pain, and a pulsatile abdominal mass.⁶, ¹⁷ Leukocytosis and elevated erythrocyte sedimentation rate are often present, but are nonspecific findings.⁴⁰ Blood cultures may be negative for bacteremia, particularly when the patient received antibiotic therapy. Features suggestive of infection with contrast-enhanced computed tomography are atypical location of the aneurysm, lack of calcium in the aneurysmal wall, multilobular appearance, multifocal or saccular configuration, periaortic gas, soft tissue reaction, and adjacent vertebral osteomyelitis.⁴¹ In cases of doubt, isotope scanning with Gallium-67 may be useful in localizing the active infectious process and identifying other septic foci.²¹ Angiography, when possible, is used as a means of defining the anatomy of the visceral vessel and facilitating preoperative planning.¹⁸, ²⁷

Antibiotic therapy 

We advocate the intravenous use of two synergistic antibiotics, particularly in Gram-negative infections because of the invasive potential of these microorganisms and the associated poor prognosis. The antibiotics for long-term treatment should be effective against the identified organism, orally administered, and well tolerated. We use ciprofloxacin for mycotic aneurysms caused by Gram-negative infections and trimethoprim-sulfamethoxazole for those caused by Gram-positive infections, because of their bioavailability, tissue penetration, and effectiveness against these organisms. There are no prospective studies addressing the optimal duration of antibiotic therapy after surgical resection. Ten authors⁴, ⁶, ⁸, ⁹, ¹⁰, ¹¹, ¹⁴, ¹⁵, ¹⁷, ²⁰, ²¹, ²⁷ suggest lifelong antibiotic treatment and report a total of 20 patients treated in this way, with a median follow-up period of 18 months. One patient died after 3 months of graft infection, in spite of continuous antibiotic treatment (5% recurrent infection rate).²⁰ In nine series,¹², ¹⁴, ¹⁸, ²², ²³, ²⁸, ²⁹, ³¹, ⁴² antibiotic treatments varying from 6 weeks to 12 months were reported. Data were available only for 10 patients, who were observed for a median of 19 months. No graft infections were reported in these patients. Short-course therapy (13 days) was described in only one patient, who required reoperation for a recurrent graft infection.⁸ The authors supported the discontinuation of antibiotics after 6 weeks to 12 months, suggesting that the important aspects of surgical management are prompt treatment, extensive debridement, and aggressive treatment of the primary focus. We discontinue treatment after a course of antibiotics of at least 6 weeks, and when clinical and laboratory parameters (white cell count and ESR) are normalized. Chiba et al⁴³ also support the use of an index of inflammation (C-reactive protein) to guide the duration of treatment. Antibiotic allergy or intolerance may also require that therapy be discontinued earlier than planned.¹⁸

Technical considerations in surgical planning 

Because of the need to revascularize the viscera, extra-anatomic reconstructions for TAAA are technically challenging. In situ reconstructions have, for this reason, often been preferred.²², ²⁹ For localized, saccular aneurysms, simple excision and repair of the aorta with a synthetic patch provide a third option when adequate debridement can be achieved.²¹

In the current review of the literature, we found six patients with a mycotic TAAA⁷, ¹⁴, ¹⁷, ²⁴, ³⁵ who underwent extra-anatomic bypass grafts, and only two patients survived (33% survival).¹⁴, ¹⁷ In situ reconstruction was used in 60 patients (82%). Of 32 patients for whom followup data were available, 25 survived (78%) after a median follow-up period of 24 months (range, 2-60 months). Causes of early postoperative mortality were sepsis (3 patients, 9%)⁴, ²⁰, ⁴² and rupture of a postoperative pseudoaneurysm (3 patients, 9%).¹⁴, ³³, ³⁴

Elective and emergency surgery 

Detailed information on the type of surgery, elective or emergency, was available for 48 patients from the literature. Twenty-three patients had elective surgery, 20 patients with the in situ technique and three patients with extra-anatomic reconstructions. The in-hospital survival rate was 75% for the in situ technique and 33% for the extra-anatomic technique. Twenty-five patients were treated for a ruptured mycotic TAAA. Follow-up data were available for 19 patients; survival was 74% at a median follow-up of 24 months (range, 11-36 months). In situ reconstruction was used in 14 patients (74%), with a mortality rate of 7%; extra-anatomic bypass grafting was used in three patients (16%), with a mortality rate of 66%; and two patients died without surgical reconstruction. The similarity in short-term results in elective and ruptured cases may have occurred because most of the ruptures were contained and patients were hemodynamically stable.

Graft material 

The most common prosthetic material used was Dacron; the next most common were polytetrafluoroethylene-expanded (PTFE) grafts and homografts. The experience with antibiotic-bonded grafts in this setting is limited, and no conclusions can be drawn.²⁸, ⁴⁴, ⁴⁵

Four patients underwent repair of a mycotic TAAA with in situ repair with a PTFE graft.⁶, ²⁶ Three patients survived at a mean follow-up of 20 months, and one patient died of sepsis after surgery. In an in vitro experimental model, PTFE appeared to be more resistant to infections than Dacron grafts.⁴⁶

Homografts were used in 16 patients with a mycotic thoracic aneurysms.²³, ⁴⁷, ⁴⁸ The in-hospital mortality rate was 13%, the rate of recurrent infections was 13%, and one late death was directly related to the presence of a homograft that developed an aortoduodenal fistula. The theoretical advantage of homografts is a higher resistance to infections than synthetic grafts. This is supported by the clinical experience with the use of homograft valves for the treatment of bacterial valve endocarditis.⁴⁹, ⁵⁰ These grafts, however, are costly, require a complex rinsing process before implantation, and may undergo aneurysmal dilatation. The latter complication may be reduced by the use of cryoprecipitated homografts.⁴⁷

Survival to discharge 

The reported overall 74% survival to discharge rate achieved with surgical repair supports this aggressive approach to the management of mycotic TAAAs. Supporting evidence can also be found in the observation that the three patients with a mycotic TAAA who did not undergo surgery all died and in similar mortality data from untreated infrarenal mycotic aneurysms.⁵¹ However, patients selected for surgery would likely have better prognostic features than patients who did not undergo surgery.

Long-term follow-up 

Careful long-term follow-up is an important element in the treatment of these patients. Clinical signs of recurrent infection, such as fever and abdominal or back pain, should prompt further aggressive investigations. Blood cultures, white cell count, erythrocyte sedimentation rate, and C-reactive protein tests may be helpful means of supporting the clinical impression or guiding the duration of the antibiotic treatment. We also perform a computed axial tomography before discharge, every 3 months for 1 year, and then annually. If there are signs of perigraft fluid, a needle aspiration under computerized tomographic guidance can be used as a means of confirming the diagnosis of recurrent infection. Indium-11–labeled white blood cell scans or Gallium-67 isotope imaging have also been used¹¹, ²¹ in this setting.

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Conclusion 

We hope this review will be of use to clinicians caring for patients with this rare problem. Any review of such a problem has two main limitations, the incompleteness of data in the original reports (lack of information on duration of antibiotic treatment and duration of follow up, for example) and the strong probability of publication bias. Improvements in the understanding of the clinical history of such rare conditions in vascular surgery will occur only if we can develop prospective multicenter databases, in which data on consecutive patients can be entered in a standardized way.

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Appendix: Search strategy 

[ANEURYSM or THORACIC AORTIC ANEURYSM or perivisceral aorta or thoracoabdominal aneurysm or thoracoabdominal aneurysm] and [INFECTION or INFECTED ANEURYSM or mycotic].

Capitals are used to indicate preexploded Medical Subject Headings/coding terms, and the lower case is used to indicate text words. The reference lists of all relevant articles, the reference lists of review articles, and the authors' personal files were also searched.

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