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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 8  |  Issue : 1  |  Page : 110-112

Tubercular aortitis presenting as primary aortoenteric fistula: Report of an uncommon case


1 Department of General Surgery, Postgraduate Institute of Medical Education and Research, Chandigarh, India
2 Department of Internal Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India
3 Department of Pathology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
4 Department of Gastroenterology, Postgraduate Institute of Medical Education and Research, Chandigarh, India

Date of Web Publication12-Mar-2019

Correspondence Address:
Harshal S Mandavdhare
Department of Gastroenterology, Postgraduate Institute of Medical Education and Research, Chandigarh - 160 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmy.ijmy_32_19

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  Abstract 


Tubercular aortitis presenting as primary aortoenteric fistula (AEF) is a rare entity. We present a 78-year-old male who presented with upper gastrointestinal bleed and also had abdominal pain and pulsating abdominal mass and on evaluation was found to have tubercular AEF which was successfully repaired with surgery and the patient recovered with antitubercular therapy along with the surgery. This case highlights the importance of high index of suspicion with early institution of surgical repair along with antitubercular therapy for tubercular AEF with good results.

Keywords: Fistula, surgical repair, tubercular, upper gastrointestinal bleed


How to cite this article:
Savlania A, Sharma V, Rastogi P, Singh H, Sharma V, Mandavdhare HS. Tubercular aortitis presenting as primary aortoenteric fistula: Report of an uncommon case. Int J Mycobacteriol 2019;8:110-2

How to cite this URL:
Savlania A, Sharma V, Rastogi P, Singh H, Sharma V, Mandavdhare HS. Tubercular aortitis presenting as primary aortoenteric fistula: Report of an uncommon case. Int J Mycobacteriol [serial online] 2019 [cited 2019 Jul 21];8:110-2. Available from: http://www.ijmyco.org/text.asp?2019/8/1/110/253965




  Introduction Top


Aortoenteric fistula (AEF), a rare entity, is an abnormal communication between the aorta and gastrointestinal tract that manifests with upper gastrointestinal bleeding. The third part of the duodenum is the usual site of fistula because of its relative fixity and proximity to the aorta.[1] Infection and inflammation of the wall of the aorta or mechanical compression of bowel wall by an enlarging aortic aneurysm are responsible for the formation of primary AEF, while erosion of bowel wall by stent graft following endovascular repair leads to secondary AEF. We report a case of primary AEF due to tubercular aortitis that was managed successfully.


  Case Report Top


A 75-year-old male presented with black tarry stools for 1 week and hematemesis for 2 days. There was no history of nonsteroidal anti-inflammatory drug abuse. There were no comorbidities; he was not a smoker or alcoholic. On clinical examination, he had severe pallor but was hemodynamically stable. A pulsatile mass, approximately 5 cm × 6 cm, with minimal side-to-side mobility was palpable just above the umbilicus. All peripheral pulses were palpable. His hemoglobin was 3.5 gm% and albumin was 2.6 gm%, while the rest of blood profile was normal. Upper gastrointestinal endoscopy revealed an elevated ulcerated lesion in the D2–D3 area with a visible vessel, which due to its shear large size was not amenable for endoscopic intervention [Figure 1]a. On evaluation with contrast-enhanced computed tomography, there was a saccular aneurysm arising from anterior wall of infrarenal aorta which was closely abutting the posterior wall of the duodenum [Figure 1]b and [Figure 1]c. Diagnosis of primary AEF was made, and he was planned for open surgical repair of aneurysm with extra-anatomical right axillounifemoral bypass after optimization with packed red blood cells' transfusion of 3 units. First, axillounifemoral bypass was done (right axillary artery to right common femoral artery bypass with 8-mm polytetrafluoroethylene graft) followed by exploratory laparotomy. The patient had dense adhesions of small and large intestines with multiple granulomas [Figure 2]a. After careful adhesiolysis of infracolic compartment, control of infrarenal aorta and bilateral common iliac arteries were taken under anticoagulation with unfractionated heparin 1 mg/kg. Duodenum was adherent to anterior wall of aneurysm, and on dissection of the duodenum from aneurysm, a 3 cm × 2 cm rent was noticed in the duodenum (eroded by pulsating saccular aneurysm), and it was repaired with interrupted polyglactin 910 (Vicryl 3-0) suture. Aneurysm wall was excised up to healthy aorta proximally and distally followed by closure of both proximal and distal aortic stump with polypropylene 4-0 suture using continuous horizontal mattress followed by over-and-over running suture [Figure 2]b. Abdominal closure was done after thorough lavage of local area followed by closed suction drain placement. The patient recovered well in postoperative period; on postoperative day 5, drain was removed. On histopathological evaluation of the resected specimen, acid-fast bacilli were positive from granuloma from omentum, duodenal wall, and aortic aneurysm wall [Figure 2]c, and hence, the patient was started on standard four drug antitubercular therapy including isoniazid, rifampicin, pyrazinamde and etambutol. The patient is doing fine at 1 month of follow-up with computed tomography (CT) angiogram showing intact stumps of infrarenal aorta, patent axilloright femoral bypass with patent iliac artery bifurcation [Figure 2]d.
Figure 1: (a) Endoscopic images showing an elevated ulcerated lesion protruding into the lumen of duodenum along with a visible vessel, (b) contrast-enhanced computed tomography abdomen showing saccular aneurysm arising from anterior wall of infrarenal aorta, and (c) which is closely abutting the posterior wall of the duodenum

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Figure 2: (a) Dense adhesions of small and large intestine with multiple granulomas; (b) closure of both proximal and distal aortic stump; (c) panel of photomicrographs depicting the histopathology (a) section from the aortic wall shows disruption and expansion of tunica media and adventitia with chronic inflammatory process (H and E, ×20), (b) higher magnification to show multiple epithelioid cell granulomas composed of Langhans type of giant cells, epithelioid histiocytes, lymphocytes, and plasma cells (H and E, ×400), (c) section from the mesenteric nodule shows many hyalinized granulomas as well as few active granulomas (H and E, ×100), (d) section from the duodenal wall shows ulcerated mucosa and underlying tissues with extensive inflammation and vascularity (H and E, ×100), and (e) Ziehl–Neelsen stain shows acid-fast bacillus (Ziehl–Neelsen stain, ×1000); and (d) follow-up postoperative computed tomography image showing intact stumps of infrarenal aorta, patent axillo-right femoral bypass with patent iliac artery bifurcation

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  Discussion Top


AEFs are rare entities. The most common risk factor for AEF is abdominal aortic aneurysm. Other causes are rare, such as carcinoma, ulcers, infections, foreign body, and radiations.[2] Infective aortitis leading to AEF can be caused by syphilis, tuberculosis, bacterial, and fungal involvement of aorta.[3] Of all the cases of aortic aneurysm, only 0.3% can be attributed to tubercular aortitis.[4]

Tubercular aortitis and consequently AEF can result due to direct intimal implantation of aortic intima by the bacilli, especially in the presence of atherosclerotic changes, hematogenous spread to the adventitia or media through the vasa vasorum, or direct spread from lymph nodal tuberculosis or tubercular paraspinal abscess.[5] The presence of nodular involvement of the omentum and extensive adhesions of bowel loops suggest that our patient might have acquired the infection by contiguous spread.

Tubercular arterial involvement can be of four types – (a) intimal miliary tuberculosis, (b) tubercular polypoids or vegetations attached to the intima, (c) multilayer involvement of the vessel wall, and d) saccular tubercular aneurysm. The fourth type is the most common variant and was seen in the index case as well.[5] Rarely, primary AEFs can be caused without an aneurysmal involvement of the aorta.[6]

Clinical manifestations can be nonspecific systemic manifestations of tuberculosis or pain abdomen, hematemesis, melena, or a pulsatile, palpable abdominal mass. Almost 94% of patients have an initial episode of mild bleed also popularly known as “herald bleed” which was also the case with our patient and which eventually led to the evaluation and successful outcome in our patient.[7]

The sensitivity of upper gastrointestinal endoscopy for AEF is poor. Erosion with an eccentric pulsating mass may be highly suggestive. CT angiography findings suggestive of an AEF may be periaortic gas, gas within the aorta, soft tissue or fluid with thickness >0.5 cm in paraaortic location, focal thickening of bowel wall adjacent to aorta, discontinuity of aortic wall, leakage of contrast into the bowel lumen, loss of fat plane between the aorta and bowel, and tethering of bowel wall toward the aorta.[8] Leakage of contrast into the bowel lumen is a specific finding.

As far as the treatment is considered, involvement of infrarenal aorta and iliac vessels can be effectively managed by ligation of the involved segment and extra-anatomic bypass, usually right axillobifemoral bypass using stent grafts along with antitubercular therapy. However, if there is an involvement of visceral segment of aorta, the involved segment cannot be excluded and in situ repair has to be considered, along with careful reimplantation of all involved side branches. Extensive debridement of infected tissue should be undertaken.[3]


  Conclusion Top


AEF due to tuberculosis is a rare manifestation of a common infection and a rare etiology behind a very common clinical presentation, that is, upper gastrointestinal bleed. Although it is a life-threatening condition, patients can be salvaged if early surgery is undertaken, which needs a high index of suspicion for diagnosis, especially in endemic countries like India, which has the maximum global burden of tuberculosis.

Declaration of patient consent

We certify that we have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Saers SJ, Scheltinga MR. Primary aortoenteric fistula. Br J Surg 2005;92:143-52.  Back to cited text no. 1
    
2.
Na JY, Kim YS, Choi YD, Kim HS, Park JT. Death by aortoesophageal fistula due to disseminated tuberculosis: A case study. Int J Clin Exp Pathol 2015;8:4253-7.  Back to cited text no. 2
    
3.
Müller BT, Wegener OR, Grabitz K, Pillny M, Thomas L, Sandmann W, et al. Mycotic aneurysms of the thoracic and abdominal aorta and iliac arteries: Experience with anatomic and extra-anatomic repair in 33 cases. J Vasc Surg 2001;33:106-13.  Back to cited text no. 3
    
4.
Volini FI, Olfield RC Jr., Thompson JR, Kent G. Tuberculosis of the aorta. JAMA 1962;181:78-83.  Back to cited text no. 4
    
5.
Long R, Guzman R, Greenberg H, Safneck J, Hershfield E. Tuberculous mycotic aneurysm of the aorta: Review of published medical and surgical experience. Chest 1999;115:522-31.  Back to cited text no. 5
    
6.
Kodaira Y, Shibuya T, Matsumoto K, Uchiyama K, Tenjin T, Yamada N, et al. Primary aortoduodenal fistula caused by duodenal tuberculosis without an abdominal aortic aneurysm: Report of a case. Surg Today 1997;27:745-8.  Back to cited text no. 6
    
7.
Leonhardt H, Mellander S, Snygg J, Lönn L. Endovascular management of acute bleeding arterioenteric fistulas. Cardiovasc Intervent Radiol 2008;31:542-9.  Back to cited text no. 7
    
8.
Hagspiel KD, Turba UC, Bozlar U, Harthun NL, Cherry KJ, Ahmed H, et al. Diagnosis of aortoenteric fistulas with CT angiography. J Vasc Interv Radiol 2007;18:497-504.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2]



 

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