False Aneurysm of the Abdominal Aorta Due to Brucella Suis Patrice B e r g e r o n , M D , Jos6 G o n z a l ~ s - F a j a r d o , MD, N i c o l a Mangialardi, M D , R o b e r t C o u r b i e r , MD, Marseille, France
A 48-year-old man presented with a fissured false aneurysm of the abdominal aorta due to Brucella suis. Clinical findings were lumbosciatic pain, fever, and sudation. Diagnosis was reached through abdominal computed tomographic (CT) scan and arteriograms. An extremely large false aneurysm, thrombosed and perforated posteriorly, was found in the infrarenal aorta. Semiurgent therapy consisted of resection of the aneurysm and prosthetic Dacron graft replacement associated with a transposed omental wrap. Antibiotic therapy was administered for three months. Although bacteriologic specimens were negative, brucellosis was diagnosed because of a positive Wright test and high Brucella antibodies in this patient originating from an endemic area. Six months after surgery he is apparently in good health. (Ann Vasc Surg 1992;6.~)00-000). KEY WORDS:
Aortic aneurysm; false aneurysm; arterial infection; brucellosis,
Brucella suis is a rare c a u s e o f infected a n e u r y s m . W e r e p o r t a case o f brucellar a n e u r y s m o f the a b d o m i n a l a o r t a t r e a t e d with s u c c e s s by resectiongrafting and o m e n t a l w r a p p i n g along with prolonged, specific antibiotic treatment.
CASE REPORT A 48-year-old man was admitted in January 1990 for chronic fissured aneurysm of the abdominal aorta. This patient, living in Corsica, smoked the equivalent of 20 grams of tobacco per day and had undergone percutaneous balloon angioplasty for right femoral artery stenosis in 1986. His present complaints had started six months earlier with bilateral lumbosciatic pain predominating on the left side. Initial radiological and computed tomographic (CT) scans showed that there was inflammation of the L5-SI disc which was treated medically. Pain continued on a
From the Department of Cardiovascular Surgery, HOpital Saint-Joseph, Marseille, France. Reprint requests: P. Bergeron, MD, Service de Chirurgie Cardio-Vasculaire, HOpital Saint-Joseph, 26 boulevard de Louvain, 13008 MarseilIe, France.
chronic basis with recrudescence within the last month prior to admission. Body temperature was 39°-40 ° C associated with abundant sudation. Wright's serodiagnosis was positive. Blood cultures remained negative. Antibiotic therapy with cyclins was commenced. Fever decreased without disappearing completely, but lumbar pain persisted and the patient was admitted to hospital in a neurosurgicat unit. A new contrast-enhanced CT scan documented a small discal hernia and a periaortic, prespinal mass partially enhanced by the contrast medium (Fig. 1), suggesting hematoma of aortic origin. The patient was referred to our unit. Digitalized arteriograms showed that the aortic caliber was normal. Aside from some mural calcifications, a large saccular aneurysm had developed on the posterior aspect of the aorta between the vessel and the spinal column (Fig. 2). A semiurgent operation was decided. Through a midline abdominal approach, an enormous, organized false aneurysm was found on the posterior aspect of an otherwise normal, rather small aorta. Suprarenal clamping of short duration was necessary before adequate clamping of the infrarenal aorta was possible. The aorta was opened and a posterior perforation was found 2 cm distal to the renal arteries. This opening communicated directly with the spine. A large thrombus was found to course along the psoas muscle and diffuse behind the spinal column. No aneurysmal
460
VOLUME 6 No 5 1992
461
AORTIC A N E U R Y S M DUE TO BRUCELLA
-
a
b
Fig. 1. (a, b): Computed tomography demonstrating posterior rupture of aorta.
wall could be isolated. The aorta was resected and replaced by a tubular Dacron graft inserted end-to-end. A fragment of pedicled omentum was passed through the mesocolon and sewn around the aortic graft. Completion arteriograms showed that the anatomic result was excellent. The patient made an uneventful recovery with sweating and pain abated. Cultures of the aortic wall and periaortic tissues remained negative. Serum tests were positive for Brucella suis. Antibrucellar antibody titers were high, attesting to contact with Brucella. Antibiotic treatment with cy-
clins was initiated and continued for three months. The patient was seen again in June 1990 in excellent health.
DISCUSSION Infected aneurysms of the abdominal aorta are r e l a t i v e l y r a r e . T h e p r o g n o s i s is g e n e r a l l y p o o r , highlighted by the risk of rupture. The septic con-
a Fig. 2. (a, b): Anteroposterior and lateral aortograms showing false aneurysm of infrarenal aorta.
b
AORTIC A N E U R YSM DUE TO BRUCELLA
462
TABLE I.--Number of cases of Brucella declared in France and in the Bouches-de-Rh6ne department between 1985 and 1991 Year
Symptoms are often atypical in infected aortic aneurysm [8]. The clinical picture is predominately determined by disc inflammation responsible for lumbar or sciatic-type pain. The notion of endemia or family history should suggest Brucella when a sudoalgic syndrome is present. When positive, Wright's serodiagnostic test attests to contact with the Brucella germ. Blood cultures as well as cultures of tissues are, however, usually negative. The aneurysm is generally located behind the aorta, Associated atheroma has been noted in all the reported cases as summarized in Table II. CT scan is essential to diagnosis and was helpful in our patient. Arteriograms delineated the site and the characteristics of the aneurysm. Surgical treatment of brucellar aneurysms should adhere to the same rules as the treatment of other infected aneurysms. Those who recommend resection-prosthetic in situ grafting [9] are in contradiction with those who recommend initial eradication of the infective focus associated with axillofemoral bypass and aortic ligation [t0]. In our opinion, the latter technique is preferable. In the case presented here, however, the operative site was deemed adequate after debridement and a prosthetic graft associated with omental wrapping was implanted. All bacteriological specimens retrieved during operation were negative. Postoperative antibiotics have always been prescribed for at least six weeks by all authors [4,6]. Certain authors advocate lifetime antibiotic therapy. We decided to treat our patient for three months. Six months after operation, he was well without any signs of infection.
Number of cases of brucellosis Bouches-du-Rh6ne France
1985 1986 1987 1988 1989 1990" *Through May 13, 1990.
12 8 23 5 7 2
ANNALS OF VASCULAR SURGERY
226 210 189 204 14 46
text poses specific therapeutic problems. Brucellar aneurysms seem to be exceptional. Only six other cases of abdominal aortic aneurysm due to Brucella were found in the literature. One case was reported by Bennet [1], occurring within the framework of chronic brucellosis. Two cases occurring in men, 45 and 57-years-old, three and four months after the diagnosis of Brucella infection, respectively, were reported by Quilchini and associates [2]. One case was published by Sava and colleagues [3] in which the aneurysm had originated from spondylodiscitis and a nearby cervical lymph node. Fudge and coworkers [4] reported a case concerning multiple aortic aneurysms due to Brucella suis. In the case reported by Gillet and associates [5], partial osteolysis of L4-L5 with spondylodiscitis was found. Tuberculosis, however, was never formally eliminated. The number of cases of Brucella declared in France and in the Bouches-de-Rh6ne department is listed in Table I. As can be seen, the relatively low incidence of Brucella can explain why vascular complications are recorded so infrequently. The mechanism of aortic involvement can be hematogenous migration and fixation of the germ on an atheromatous arterial wall [6,7]. Direct involvement of a healthy aortic wall through an infective focus in the periaortic prespinal space is equally possible. This seems to have been the mechanism involved in our case because of the 2 cm posterior punch-hole perforation in the aorta, the absence of an organized aneurysmal sac, and the destitute aspect of the spinal column and intervertebral disc.
CONCLUSION Even though it is unusual, Brucella infection can be responsible for an aortic aneurysm. Symptoms may be misleading, suggesting lumbar or sciatic pathology. CT scan is essential for diagnosis. Treatment is surgical and consists of resection followed by prosthetic graft replacement associated with omental transposition and prolonged antibiotic therapy.
TABLE II.--Principal characteristics of published case of Brucellar aneurysm of the abdominal aorta Authors (ref) B e n n e t (1) Guilichini (2) Case 1 Case 2 F u d g e (4) S a v a (3) Gillet (5) Present case
Serum
Blood cultures
Culture of aneurysm
Associated lesions
Vertebral erosion
+
-
-
Atheroma
-
Anterior
+ +
.
Atheroma
+
+ +
B. suis B. a b o r t u s
+
Atheroma
-
-
-
+
+ +
-
-
Atheroma Atheroma
+ +
Posterior Anterior Posterior Posterior Posterior Posterior
+ .
.
.
Site
VOLUME 6 No 5 - 1992
AORTIC A N E U R YSM DUE TO BRUCELLA
REFERENCES 6. 1. BENNET DE. Primary mycotic aneurysms of the aorta. Arch Surg 1967;94:758-765. 2. QUILICHINI F, TOURNIGAND P, MERCIER C. Les andvrysmes rompus de l'aorte abdominale. J Chir 1974;108: 351-368. 3. SAVA P, CAMELOT G, MIGUET JP. An6vrysme de l'aorte abdominale et brucellose:/i propos d'un nouveau cas op6r6 avec succ~s. Ann Chir Thorac Cardiovasc 1977;16: 221-225. 4. FUDGE TL, OCHSNER JL, ANCALMON N, et al. Surgical resection of multiple aortic aneurysms due to brucella suis. Surgery 1977;81:236-238. 5. GILLET M, SAVA P, CASSOU M, et al. Andvrysmes
n u n
7. 8 .
9.
10.
463
infectieux de l'aorte sous-rdnale: rdflexions ~t propos de deux cas d'6tiologie brucellienne. Chirurgie 1983;109:168-172. KIEFFER E. An6vrysmes infectieux de l'aorte. Rev Prat 1979;29:777-783. BASTIN R, BR1CAIRE F, FROTTIER J, et al. Art6riopathies infectieuses. Ann Med lnt 1983;134:475-478. RAFFI F, PATRA P, DUPON H. Aspects cliniques et bactdriologiques des an6vrysmes infectieux. Med Mal l n f 1986;12:783-788. WALKER WE, COOLEY DA, DUNCAN JM, et al. The management of aortoduodenal fistula by in situ replacement of the infected abdominal aortic graft. Ann Surg 1987;205: 727-732. BERGERON P, ESPINOZA H, RUDONDY P, et al. Secondary aortoduodenal fistulas: value of initial axillofemoral bypass. Ann Vasc Surg 1991;5:4--7.
A 48-year-old man presented with a fissured false aneurysm of the abdominal aorta due to Brucella suis. Clinical findings were lumbosciatic pain, fever, and sudation. Diagnosis was reached through abdominal computed tomographic (CT) scan and arteriograms. An extremely large false aneurysm, thrombosed and perforated posteriorly, was found in the infrarenal aorta. Semiurgent therapy consisted of resection of the aneurysm and prosthetic Dacron graft replacement associated with a transposed omental wrap. Antibiotic therapy was administered for three months. Although bacteriologic specimens were negative, brucellosis was diagnosed because of a positive Wright test and high Brucella antibodies in this patient originating from an endemic area. Six months after surgery he is apparently in good health. (Ann Vasc Surg 1992;6.~)00-000). KEY WORDS:
Aortic aneurysm; false aneurysm; arterial infection; brucellosis,
Brucella suis is a rare c a u s e o f infected a n e u r y s m . W e r e p o r t a case o f brucellar a n e u r y s m o f the a b d o m i n a l a o r t a t r e a t e d with s u c c e s s by resectiongrafting and o m e n t a l w r a p p i n g along with prolonged, specific antibiotic treatment.
CASE REPORT A 48-year-old man was admitted in January 1990 for chronic fissured aneurysm of the abdominal aorta. This patient, living in Corsica, smoked the equivalent of 20 grams of tobacco per day and had undergone percutaneous balloon angioplasty for right femoral artery stenosis in 1986. His present complaints had started six months earlier with bilateral lumbosciatic pain predominating on the left side. Initial radiological and computed tomographic (CT) scans showed that there was inflammation of the L5-SI disc which was treated medically. Pain continued on a
From the Department of Cardiovascular Surgery, HOpital Saint-Joseph, Marseille, France. Reprint requests: P. Bergeron, MD, Service de Chirurgie Cardio-Vasculaire, HOpital Saint-Joseph, 26 boulevard de Louvain, 13008 MarseilIe, France.
chronic basis with recrudescence within the last month prior to admission. Body temperature was 39°-40 ° C associated with abundant sudation. Wright's serodiagnosis was positive. Blood cultures remained negative. Antibiotic therapy with cyclins was commenced. Fever decreased without disappearing completely, but lumbar pain persisted and the patient was admitted to hospital in a neurosurgicat unit. A new contrast-enhanced CT scan documented a small discal hernia and a periaortic, prespinal mass partially enhanced by the contrast medium (Fig. 1), suggesting hematoma of aortic origin. The patient was referred to our unit. Digitalized arteriograms showed that the aortic caliber was normal. Aside from some mural calcifications, a large saccular aneurysm had developed on the posterior aspect of the aorta between the vessel and the spinal column (Fig. 2). A semiurgent operation was decided. Through a midline abdominal approach, an enormous, organized false aneurysm was found on the posterior aspect of an otherwise normal, rather small aorta. Suprarenal clamping of short duration was necessary before adequate clamping of the infrarenal aorta was possible. The aorta was opened and a posterior perforation was found 2 cm distal to the renal arteries. This opening communicated directly with the spine. A large thrombus was found to course along the psoas muscle and diffuse behind the spinal column. No aneurysmal
460
VOLUME 6 No 5 1992
461
AORTIC A N E U R Y S M DUE TO BRUCELLA
-
a
b
Fig. 1. (a, b): Computed tomography demonstrating posterior rupture of aorta.
wall could be isolated. The aorta was resected and replaced by a tubular Dacron graft inserted end-to-end. A fragment of pedicled omentum was passed through the mesocolon and sewn around the aortic graft. Completion arteriograms showed that the anatomic result was excellent. The patient made an uneventful recovery with sweating and pain abated. Cultures of the aortic wall and periaortic tissues remained negative. Serum tests were positive for Brucella suis. Antibrucellar antibody titers were high, attesting to contact with Brucella. Antibiotic treatment with cy-
clins was initiated and continued for three months. The patient was seen again in June 1990 in excellent health.
DISCUSSION Infected aneurysms of the abdominal aorta are r e l a t i v e l y r a r e . T h e p r o g n o s i s is g e n e r a l l y p o o r , highlighted by the risk of rupture. The septic con-
a Fig. 2. (a, b): Anteroposterior and lateral aortograms showing false aneurysm of infrarenal aorta.
b
AORTIC A N E U R YSM DUE TO BRUCELLA
462
TABLE I.--Number of cases of Brucella declared in France and in the Bouches-de-Rh6ne department between 1985 and 1991 Year
Symptoms are often atypical in infected aortic aneurysm [8]. The clinical picture is predominately determined by disc inflammation responsible for lumbar or sciatic-type pain. The notion of endemia or family history should suggest Brucella when a sudoalgic syndrome is present. When positive, Wright's serodiagnostic test attests to contact with the Brucella germ. Blood cultures as well as cultures of tissues are, however, usually negative. The aneurysm is generally located behind the aorta, Associated atheroma has been noted in all the reported cases as summarized in Table II. CT scan is essential to diagnosis and was helpful in our patient. Arteriograms delineated the site and the characteristics of the aneurysm. Surgical treatment of brucellar aneurysms should adhere to the same rules as the treatment of other infected aneurysms. Those who recommend resection-prosthetic in situ grafting [9] are in contradiction with those who recommend initial eradication of the infective focus associated with axillofemoral bypass and aortic ligation [t0]. In our opinion, the latter technique is preferable. In the case presented here, however, the operative site was deemed adequate after debridement and a prosthetic graft associated with omental wrapping was implanted. All bacteriological specimens retrieved during operation were negative. Postoperative antibiotics have always been prescribed for at least six weeks by all authors [4,6]. Certain authors advocate lifetime antibiotic therapy. We decided to treat our patient for three months. Six months after operation, he was well without any signs of infection.
Number of cases of brucellosis Bouches-du-Rh6ne France
1985 1986 1987 1988 1989 1990" *Through May 13, 1990.
12 8 23 5 7 2
ANNALS OF VASCULAR SURGERY
226 210 189 204 14 46
text poses specific therapeutic problems. Brucellar aneurysms seem to be exceptional. Only six other cases of abdominal aortic aneurysm due to Brucella were found in the literature. One case was reported by Bennet [1], occurring within the framework of chronic brucellosis. Two cases occurring in men, 45 and 57-years-old, three and four months after the diagnosis of Brucella infection, respectively, were reported by Quilchini and associates [2]. One case was published by Sava and colleagues [3] in which the aneurysm had originated from spondylodiscitis and a nearby cervical lymph node. Fudge and coworkers [4] reported a case concerning multiple aortic aneurysms due to Brucella suis. In the case reported by Gillet and associates [5], partial osteolysis of L4-L5 with spondylodiscitis was found. Tuberculosis, however, was never formally eliminated. The number of cases of Brucella declared in France and in the Bouches-de-Rh6ne department is listed in Table I. As can be seen, the relatively low incidence of Brucella can explain why vascular complications are recorded so infrequently. The mechanism of aortic involvement can be hematogenous migration and fixation of the germ on an atheromatous arterial wall [6,7]. Direct involvement of a healthy aortic wall through an infective focus in the periaortic prespinal space is equally possible. This seems to have been the mechanism involved in our case because of the 2 cm posterior punch-hole perforation in the aorta, the absence of an organized aneurysmal sac, and the destitute aspect of the spinal column and intervertebral disc.
CONCLUSION Even though it is unusual, Brucella infection can be responsible for an aortic aneurysm. Symptoms may be misleading, suggesting lumbar or sciatic pathology. CT scan is essential for diagnosis. Treatment is surgical and consists of resection followed by prosthetic graft replacement associated with omental transposition and prolonged antibiotic therapy.
TABLE II.--Principal characteristics of published case of Brucellar aneurysm of the abdominal aorta Authors (ref) B e n n e t (1) Guilichini (2) Case 1 Case 2 F u d g e (4) S a v a (3) Gillet (5) Present case
Serum
Blood cultures
Culture of aneurysm
Associated lesions
Vertebral erosion
+
-
-
Atheroma
-
Anterior
+ +
.
Atheroma
+
+ +
B. suis B. a b o r t u s
+
Atheroma
-
-
-
+
+ +
-
-
Atheroma Atheroma
+ +
Posterior Anterior Posterior Posterior Posterior Posterior
+ .
.
.
Site
VOLUME 6 No 5 - 1992
AORTIC A N E U R YSM DUE TO BRUCELLA
REFERENCES 6. 1. BENNET DE. Primary mycotic aneurysms of the aorta. Arch Surg 1967;94:758-765. 2. QUILICHINI F, TOURNIGAND P, MERCIER C. Les andvrysmes rompus de l'aorte abdominale. J Chir 1974;108: 351-368. 3. SAVA P, CAMELOT G, MIGUET JP. An6vrysme de l'aorte abdominale et brucellose:/i propos d'un nouveau cas op6r6 avec succ~s. Ann Chir Thorac Cardiovasc 1977;16: 221-225. 4. FUDGE TL, OCHSNER JL, ANCALMON N, et al. Surgical resection of multiple aortic aneurysms due to brucella suis. Surgery 1977;81:236-238. 5. GILLET M, SAVA P, CASSOU M, et al. Andvrysmes
n u n
7. 8 .
9.
10.
463
infectieux de l'aorte sous-rdnale: rdflexions ~t propos de deux cas d'6tiologie brucellienne. Chirurgie 1983;109:168-172. KIEFFER E. An6vrysmes infectieux de l'aorte. Rev Prat 1979;29:777-783. BASTIN R, BR1CAIRE F, FROTTIER J, et al. Art6riopathies infectieuses. Ann Med lnt 1983;134:475-478. RAFFI F, PATRA P, DUPON H. Aspects cliniques et bactdriologiques des an6vrysmes infectieux. Med Mal l n f 1986;12:783-788. WALKER WE, COOLEY DA, DUNCAN JM, et al. The management of aortoduodenal fistula by in situ replacement of the infected abdominal aortic graft. Ann Surg 1987;205: 727-732. BERGERON P, ESPINOZA H, RUDONDY P, et al. Secondary aortoduodenal fistulas: value of initial axillofemoral bypass. Ann Vasc Surg 1991;5:4--7.
