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Ann Thorac Surg 2001;72:1239-1244
© 2001 The Society of Thoracic Surgeons

Original article: cardiovascular

Tubercular pseudoaneurysms of aorta

Shiv Kumar Choudhary, MCha, Anil Bhan, MCha, Sachin Talwar, MSa, Mukesh Goyal, MSa, Sanjeev Sharma, MDa, Panangipalli Venugopal, MCha

a Cardiothoracic Centre, All India Institute of Medical Sciences, New Delhi, India

Accepted for publication February 22, 2001.

Address reprint requests to Dr Bhan, Department of Cardiothoracic & Vascular Surgery, All India Institute of Medical Sciences, Ansari Nagar, New Delhi—110 029, India
e-mail: anil-bhan{at}hotmail.com


   Abstract
Top
 Abstract
Introduction
 Patients, methods, and results
 Comment
 References
 
Background. Tubercular pseudoaneurysm of aorta is a rare but important complication of tuberculosis. With worldwide resurgence of tuberculosis due to increasing incidence of drug-resistant tuberculosis and its association with acquired immunodeficiency syndrome, the tubercular pseudoaneurysm has become a real clinical entity.

Methods. In the past 3 years, 5 young patients (22 to 40 years) presented with tubercular pseudoaneurysm. Site of involvement included ascending aorta, distal aortic arch, proximal descending thoracic aorta, distal descending thoracic aorta, and infrarenal abdominal aorta. Two patients had macroscopic focus of tuberculosis in the nearby vicinity, and all 5 patients had evidence of active/treated pulmonary pericardial tuberculosis. All patients either had received antitubercular therapy previously or were receiving it at the time of presentation. Rapid deterioration in the clinical status was the most marked clinical feature. All patients underwent operation. Graft interposition was performed in 2, patch repair in 2, and direct closure of the rent was performed in 1 patient. All 5 patients received antitubercular therapy in the postoperative period.

Results. All patients survived the operation and were discharged from the hospital. One patient developed recurrence at the original site after 8 months and died at reoperation. The remaining patients are symptom free at 18 to 36 months postoperatively.

Conclusions. A combination of chemotherapy and operation yields gratifying results for the treatment of tubercular pseudoaneurysm.


   Introduction
Top
 Abstract
 Introduction
Patients, methods, and results
 Comment
 References
 
Tubercular involvement of blood vessels is a rare phenomenon. Of the various vascular structures that could be involved, aortic involvement is the rarest [1]. Aortic involvement can be in the form of stenosing arteritis [2, 3] or an aneurysm [1, 4–28]. A tuberculous aneurysm of the aorta is exceedingly rare [9] and since its first description by Kamen [5] in 1895, until December 1999, only 88 such cases could be traced in the English language literature [1, 4–28]. In this report, we present our experience with 5 such patients and a brief review of the English language literature.


   Patients, methods, and results
Top
 Abstract
 Introduction
 Patients, methods, and results
Comment
 References
 
Between October 1997 and March 1999, we operated on 5 patients with tubercular pseudoaneurysm of the aorta. Brief clinical summary of the patients is given in the Table 1.


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  		Table 1. Clinical Summary of the Patients

 
Patient 1
In October 1997, a 22-year-old man, who had been receiving antitubercular treatment for pulmonary tuberculosis, presented with hoarseness and dysphagia. Contrast computerized tomographic (CT) scan revealed extensive mediastinal lymphadenopathy and calcification of some of these lymph nodes. Biopsy from enlarged cervical lymph nodes showed granulomatous lesions suggesting tuberculosis. Soon after this, the patient developed shortness of breath and a repeat CT scan performed 15 days later showed a large (9 cm) saccular pseudoaneurysm arising from the distal aortic arch (Fig 1). The aneurysm was causing severe compression of the left bronchus and the esophagus and had pushed the trachea to the right. Intraarterial digital subtraction angiography corroborated the CT scan findings. Abdominal ultrasound examination showed retroperitoneal and mesenteric lymphadenopathy. The patient underwent operation. Through a left posterolateral thoracotomy, the descending aorta and the pulmonary artery were cannulated and cardiopulmonary bypass was commenced. The patient was cooled down to 18°C and total circulatory arrest was achieved. The aneurysm was incised and the rent in the aortic wall (approximately 1.5 cm) was repaired with a Gore-Tex patch (W. L. Gore and Associates, Flagstaff, AZ). Total circulatory arrest time was 10 minutes and cardiopulmonary bypass time was 72 minutes. The postoperative recovery was uneventful and the patient was discharged from the hospital on the seventh postoperative day.



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  		Fig 1. Computed tomographic scan image of patient 1 showing a large pseudoaneurysm in the descending thoracic aorta (arrowheads). The left lung is partially collapsed. Enlarged mediastinal lymph nodes can also be seen (arrow).

 
The tissue taken from the wall of the aneurysm revealed evidence of granulomatous inflammatory reaction but no Langhans giant cells. However, culture of the same tissue showed the presence of acid-fast bacilli. The patient received antitubercular therapy for 6 months postoperatively. Presently, 36 months after operation, the patient is symptom-free and doing well.

Patient 2
A 40-year-old male hypertensive patient presented in January 1999 with history of severe backache for 3 months, accompanied with weight loss and malaise. Previously he had received irregular antitubercular therapy for pulmonary tuberculosis. Physical examination was unremarkable. Magnetic resonance imaging revealed a large (12 by 8 cm) saccular pseudoaneurysm arising from descending thoracic aorta (Fig 2) and eroding the corresponding vertebral bodies. Intraarterial digital subtraction angiography also showed the pseudoaneurysm. Rest of the aorta was normal.



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  		Fig 2. Magnetic resonance angiogram of patient 2 showing a large pseudoaneurysm in relation to the juxtadiaphragmatic aorta.

 
Patient was operated on through a left posterolateral thoracotomy. Left atriofemoral bypass was established. The aorta was clamped proximally and distally and the aneurysm was incised. The clots and thrombi were removed. A large rent (about 2 cm) was noted in the aorta with atheromatous changes in the aortic wall. After debridement, a 20-mm Vascutek (Sulzer-Vascutek Ltd, Renfrewshire, Scotland) graft was interposed between the proximal and distal aorta. The clamp time was 24 minutes and the bypass time was 30 minutes.

The histopathologic examination of the aneurysm wall revealed features of tuberculosis. Culture was positive for mycobacterium. Antitubercular therapy was started and the patient was discharged. He is asymptomatic and doing well 20 months postoperatively.

Patient 3
A 30-year-old man was admitted in January 1999 with complaints of chest pain and distended neck veins for 3 weeks. He had a history of low-grade fever since September 1998. He was investigated for his fever and was diagnosed as having tubercular pericardial effusion. Antitubercular treatment was started and he became afebrile. During the present admission, a magnetic resonance scan showed a large pseudoaneurysm (about 10 cm) in the ascending aorta (Fig 3) and mediastinal lymphadenopathy. The patient underwent operation. A femorofemoral bypass was established and the patient was cooled down. Median sternotomy was performed. The anterior mediastinum had a "frozen" look due to presence of dense adhesions around the pseudoaneurysm and no structure could be identified separately. At 18°C, total circulatory arrest was achieved and the pseudoaneurysm was incised and opened longitudinally. There was a small rent, about 6 mm, in the anterior wall of the ascending aorta. The rent was directly closed with pledgetted sutures. Caseous material, hemorrhagic debris, and fibrinous flakes were removed from the pseudoaneurysm and part of the aneurysm wall was excised for histopathologic examination. The remaining aneurysm wall was wrapped over the repaired aorta with continuous suture. Circulatory arrest time was 8 minutes and bypass time was 60 minutes. Histopathologic examination showed chronic granulomatous inflammation, but the culture of submitted material did not show any mycobacteria. The patient made an uneventful recovery and was discharged from the hospital on antitubercular therapy. However, 8 months after the initial operation, he developed recurrence of the pseudoaneurysm at the original site. He was reoperated using the similar technique but died as a result of a massive cerebral infarct, probably due to air embolism.



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  		Fig 3. Coronal plane spin-echo magnetic resonance image of patient 3 showing a large pseudoaneurysm arising from the ascending aorta. The aneurysm is filled with thrombus.

 
Patient 4
A 39-year-old male patient, who was being treated for pulmonary tuberculosis, presented in April 1998 with severe backache and abdominal pain for 2 weeks. Contrast enhanced CT scan revealed a leaking aortic aneurysm involving the infrarenal aorta. Patient was operated on urgently. There was a large (10 cm by 8 cm) pseudoaneurysm, arising from the right side of infrarenal abdominal aorta. The pseudoaneurysm was communicating with aorta through a 2.0-cm by 1.0-cm rent. The bodies of L4–5 vertebrae were significantly eroded. The clots and thrombi were removed from the pseudoaneurysm and aortic continuity was established using a 16-mm interposition Vascutek tube graft. Clamp time was 32 minutes.

Histologic examination of the excised tissue showed only thrombus and fresh clot, as the tissue specimen was not adequate, and the culture was also negative for mycobacteria.

The patient made uneventful recovery and completed his antitubercular therapy successfully. He is doing well, 24 months postoperatively.

Patient 5
A 32-year-old man, who had successfully completed antitubercular therapy for pulmonary tuberculosis, presented in March 1999 with severe chest pain and dyspnea. Contrast enhanced CT scan and intraarterial digital subtraction angiogram demonstrated a pseudoaneurysm (14 cm by 10 cm) arising from the descending thoracic aorta just distal to the left subclavian artery (Fig 4). The patient was operated on emergently using a left posterolateral thoracotomy. After obtaining proximal and distal aortic control, the aneurysm was incised and opened. The clots and thrombi were evacuated and the rent in the aortic wall (about 8 mm) was repaired with a Gore-Tex patch. Aortic clamp time was 12 minutes. Postoperatively, the patient made an uneventful recovery. Histopathologic examination of the pseudoaneurysm wall showed granulomatous inflammatory reaction with few giant cells. The cultures were negative for mycobacteria. The patient received antitubercular therapy for 6 months, and is now doing well at 18 months.



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  		Fig 4. Intraarterial digital subtraction angiogram of patient 5 showing a large pseudoaneurysm arising from the descending thoracic aorta, just distal to the origin of the left subclavian artery.

 
Follow-up and chemotherapy
All these patients had a contrast enhanced CT scan or intraarterial digital subtraction angiogram before discharge from the hospital. Subsequently, they were followed in the outpatient department and were evaluated clinically. If a patient complained of chest discomfort, pain, or fever, a contrast enhanced CT scan was performed. All patients received antitubercular treatment in the postoperative period. Drugs included rifamipcin (10 mg/kg/d, maximum 600 mg/d), isoniazid (5 mg/kg/d, maximum 300 mg/d), pyrazinamide (30 mg/kg/d, maximum 2gm/d), and ethambutol (25 mg/kg/d). Patients who were culture positive for mycobacteria received all four drugs for 2 months, followed by rifampicin and isoniazid for 4 months. Patients who were culture negative, but were not receiving antitubercular therapy, received the similar regimen. However, patients who were culture negative and were receiving antitubercular therapy received rifampicin and isoniazid for 4 months. In 2 of our patients, culture were positive for mycobacteria, which was sensitive to all first-line antitubercular drugs. We did not encounter any drug-resistant mycobacteria.


   Comment
Top
 Abstract
 Introduction
 Patients, methods, and results
 Comment
References
 
A tuberculous aneurysm of the aorta is a rare phenomenon. Since its first report by Kamen in 1895 [5], we could trace only 88 cases of tuberculous aneurysm in the English language literature. Despite effective chemotherapy, the entity of the tuberculous aneurysm could not be eliminated as it keeps on appearing in the medical literature as sporadic case reports. However, better awareness of this entity and availability of diagnostic facilities and chemotherapy have effectively changed its natural history. Before 1950, most of the reported cases were from autopsies [1, 4, 5, 9]; in later years, antemortem diagnosis and successful treatment became more frequent [5–28].

Almost all the segments of the aorta are reported to be involved. In the reported patients (n = 88), the ascending aorta was involved in 9 (10.2%), aortic arch in 10 (11.4%), descending thoracic aorta in 25 (28.4%), and abdominal aorta was involved in 37 (42.0%) patients. In 1 patient, the thoracoabdominal aorta was involved and in 6 patients (6.9%), the site was not clearly mentioned. However, the areas in close proximity to the mediastinal and para-aortic lymph nodes are more commonly involved [12]. Initially, Haythorn [29] described four types of tubercular involvement in the arterial system. These included (1) miliary tuberculosis of the intima, (2) tubercular polyps attached to the intima, (3) tuberculosis involving several layers of the arterial wall, and (4) aneurysm formation. Recently investigators from India [2, 3] described a stenosing type of tubercular aortoarteritis in young patients secondary to a tubercular focus in the body. Tubercular aneurysms are mostly pseudoaneurysm (87%) and rarely true (9%) or dissecting (4%). In 93% of patients the aneurysm was saccular in shape. It ruptured in neighboring viscera in 65% patients. With better diagnostic modalities, incidence of rupture has decreased in recent decades. Tubercular aneurysms are mostly solitary and only 4 patients with multiple aneurysms have been reported [9, 16, 23, 26]. Calcification is conspicuously absent.

The aorta may be involved in the tubercular process either through the hematogenous route or by way of direct extension of the disease from the neighboring organs [26]. In hematogenous involvement, tubercle bacilli may either lodge in an atheromatous plaque or may involve adventitia and media by way of vasa vasorum. However, this route appears to be uncommon and the far more common mode of involvement of the aorta is extension from a contiguous tuberculous focus. In most of the reports from autopsies [1, 4, 5, 9], a macroscopic contiguous tuberculous focus could be seen. At present, because of early detection and antemortem diagnosis, the contiguous focus may not always be seen by radiologic means. Hence, despite the fact that only 2 of our patients had macroscopic contiguous tuberculous foci, we believe that the aorta was involved as a result of the extension of the disease from the neighboring organs.

Typical presentation includes evidence of tuberculous focus or disseminated tuberculosis (untreated/partially treated) with one or more of the three clinical scenarios: (1) fever and persistent pain related to the location of the aneurysm, (2) hypovolemic shock or other evidence of massive bleeding, or (3) pulsatile, rapidly expanding para-aortic mass. If the diagnosis of tuberculosis is not made previously, a false aneurysm of the aorta associated with a contiguous focus of disease suggests tubercular pseudoaneurysm. Thus, if a diagnosed case of tuberculosis deteriorates suddenly or develops pressure symptoms due to presence of a mass, the possibility of a pseudoaneurysm should be considered and emergent CT scan or magnetic resonance scan should be performed. If a pseudoaneurysm is detected in such circumstances, it should be considered tubercular in origin.

Neither medical nor surgical therapy is curative, if used alone [9]. All the survivors had received a combination of preoperative or postoperative antitubercular chemotherapy and operation [6, 9, 14, 15, 17–22, 24–28]. Once the tubercular aneurysm has been diagnosed, operation should be performed urgently [9, 10, 16]. The antitubercular treatment should be started immediately and should be continued in the postoperative period. Various surgical options have been used by the investigators and include extra-anatomic bypass [11, 21, 27], in situ insertion of an aortic conduit [6, 9, 15, 17, 19, 20, 24] and patch closure [11, 12], or direct closure [10, 11, 28]. Extra-anatomic bypass was only used when preoperative diagnosis of tubercular aneurysm was not certain and the aneurysm was considered to be a nonmycobacterial mycotic aneurysm. In patients in whom clinical diagnosis of the tuberculous aneurysm has already been made, extent of procedure depends on the size of the aortic rent and the condition of the neighboring aortic wall. After removal of clots and debris, the edges of the aortic rent should be freshened and neighboring aorta should be evaluated. Because of clamp-related ischemia and circulatory arrest, the surgeon should rely on visual inspection of the aortic wall to decide on the extent of resection, rather than on time-consuming frozen section and histologic study. If the rent is large or the nearby aorta is not healthy, an interposition graft should be inserted. Although insertion of interposition graft has the advantage of excluding the diseased aorta, if it is technically difficult and there is concern about prolonged ischemia due to aortic clamping or circulatory arrest and the aortic rent is small with healthy surrounding aorta, a patch repair may be an option. Two of our patients, as also reported by other investigators [11, 12], showed good results with patch closure. However, 1 of our patients in whom the aortic rent was closed by direct suture developed a recurrence at the same site. This patient had a frozen mediastinum and the aorta was stiff due to surrounding adhesion. Direct suture closure of the rent was probably a technical mishap. Because of surrounding adhesions and an unyielding aorta, the suture line would have always been in tension. Failure to resect the margins of the rent may have left residual disease. Thus, at present, we believe that direct closure of the rent should not be attempted in such patients, although contradictory reports are available in the literature [10, 11, 28].

We consider the diagnosis of tuberculous pseudoaneurysm a clinical diagnosis. If the patient is not receiving antitubercular chemotherapy, we start with four chemotherapeutic drugs (rifamipcin, isoniazid, pyrizinamide, and ethambutol) even before the histologic or culture reports are available. With use of liquid media and radiometric growth detection, it is possible to obtain results of mycobacterial cultures in 2 weeks. If the culture is positive, the four drugs are continued for 2 months, followed by rifampicin and isoniazid for 4 months. If cultures are negative and the patient has already been receiving antitubercular therapy at the time of hospital admission only rifampicin and isoniazid are continued for 4 months.

These patients should be followed up closely for possible reactivation of the tuberculous process and recurrence [6, 10, 14]. Usually the clinical evaluation is sufficient, but if the patient has chest or abdominal discomfort, pain, or fever, a contrast enhanced CT scan should be obtained.

With worldwide resurgence of tuberculosis due to an increasing incidence of drug-resistant tuberculosis and its association with acquired immunodeficiency syndrome, the incidence of tubercular aneurysm is bound to increase. One should have a high index of suspicion and an aneurysm should be suspected in patients with active tuberculosis if they deteriorate suddenly or if a mass lesion is present. All pseudoaneurysms should undergo histologic and microbial assessment with special consideration to mycobacterial infection. Once the diagnosis is made, operation should be performed urgently even in presence of a small pseudoaneurysm, and all patients should receive preoperative/postoperative chemotherapy.


   References
Top
 Abstract
 Introduction
 Patients, methods, and results
 Comment
 References
 

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