Ann Thorac Surg 2008;86:1672-1674. doi:10.1016/j.athoracsur.2008.04.095
© 2008 The Society of Thoracic Surgeons
Case Reports
Aorto-Atrial Fistula 10 Days After Dissection Repair in Giant Cell Arteritis
Fadia Haddad, MDa,*,
Issam El-Rassi, MDb,
Fadi G. Haddad, MDc,
Rita Nemnoum, MDc,
Victor A. Jebara, MDb
a Department of Anesthesiology and Intensive Care, Hotel Dieu de France Hospital, Beirut, Lebanon
b Department of Cardiac Surgery, Hotel Dieu de France Hospital, Beirut, Lebanon
c Department of Internal Medicine, Hotel Dieu de France Hospital, Beirut, Lebanon
Accepted for publication April 25, 2008.
* Address correspondence to Dr Haddad, Department of Anesthesiology and Intensive Care, Hotel-Dieu de France Hospital, Saint Joseph University, Alfred Naccache St, PO Box 166830, Beirut, Lebanon (Email: fflhlb{at}yahoo.com).
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Abstract
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Active aortitis caused by giant cell arteritis occurs more commonly than has been previously appreciated, and ascending aortic dissection may occur despite medical therapy, without a preexisting aneurysm or ectasia. We report a case of acute type A aortic dissection in a patient already treated for giant cell arteritis. The aortic root was left intact during the initial surgery, but was completely removed 10 days later after an early postoperative aorto-atrial fistula. Complete removal of the aortic root (Bentall procedure) has been advocated in these patients to prevent late additional aortic complications.
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Introduction
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Giant cell arteritis (GCA) is a chronic systemic vasculitis of unknown cause affecting medium-sized and large-sized arteries [1, 2]. Active aortitis caused by GCA occurs more commonly than has been previously appreciated, and steroids are indicated in an effort to control the diffuse inflammatory disease in the aortic wall [3, 4]. However, ascending aortic dissection may occur, despite medical therapy and without a pre-existing aortic aneurysm or ectasia [5]. Although conventional surgery for aortic dissection may be lifesaving, complete removal of the aortic root (Bentall procedure) has been advocated to prevent late additional aortic complications [1].
We hereby report a case of acute type A aortic dissection in a patient already treated for GCA. The aortic root was left intact during the initial surgery, but was completely removed 10 days later after an early postoperative aorto-atrial fistula.
A 66-year-old man was admitted to the hospital with acute type A aortic dissection. Two years earlier he was diagnosed with GCA after right eye blindness. He was treated with prednisone (60 mg daily) and methotrexate (15 mg weekly); steroids were then gradually tapered down to 7.5 mg of prednisone daily and 10 mg of methotrexate weekly. A multi-slice computed tomographic scan was obtained 6 months earlier and ruled out associated aortic disease. Transesophageal echocardiography revealed grade III aortic valve regurgitation due to a prolapse of the noncoronary cusp.
The patient was operated on the same day; the noncoronary sinus of Valsalva was completely dissected, but the rest of the aortic root was intact (Fig 1A). It was decided to separately replace the valve and the ascending aorta and reconstruct the noncoronary sinus. A pericardial bioprosthesis was used to replace the valve. The dissected sinus of Valsalva was reconstructed, using a Teflon felt buttress (Bard Inc, Nogales, AZ) inserted between the two layers of the dissected aortic wall and fixed with pledgeted sutures reaching down to the aortic annulus (Fig 1B). A Dacron tube (Hemashield, Meadox Medical, Oakland, NJ) was then used to replace the ascending aorta. An open anastomosis was performed with a hemi-arch technique. Proximal and distal sutures were reinforced by strips of Teflon felt (Bard Inc). The postoperative course was uneventful. Transthoracic echocardiography was normal.
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Fig 1. (A) The ascending aortic dissection extending down the noncoronary sinus. (B) Reinforcement of the dissected noncoronary sinus by a Teflon patch (Bard Inc, Nogales, AZ) between the two layers. (C) The aorto-atrial fistula (black arrow) below the Teflon patch.
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The aortic wall pathology revealed diffuse aortitis with giant multinucleated cells (Fig 2). On postoperative day 10, while preparing to leave the hospital, the patient complained of shortness of breath with general weakness. Repeat echocardiography showed a high velocity continuous flow from the noncoronary sinus into the right atrium (Fig 3). The patient was then reoperated on within 24 hours; there was a 10-mm communication between the noncoronary sinus and the right atrium. The fistula originated very close to the aortic annulus, below the reinforcing Teflon patch in the wall of the noncoronary sinus (Fig 1C). The prosthetic valve was removed and a Bentall procedure was performed, excluding all the remaining native aortic tissue, with the exception of the two coronary buttons. The tear in the right atrial wall was closed from within the right atrium with interrupted pledgetted sutures. The patient completely recovered after transient renal and liver failure and was discharged home 2 weeks after the second procedure on prednisone (30 mg daily).
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Fig 2. Pathology of the aortic wall showing granulomatous infiltration with giant cells (white arrows). (Hematoxylin & eosin stain, x200.)
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Fig 3. (A) Right ventricle. (B) Right atrium and a high-velocity flow through the aorto-right atrial fistula.
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Comment
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The GCA or Horton disease is a chronic systemic vasculitis that may affect the aorta. As many as 15% of patients with GCA might have angiographic evidence of aortic involvement [4], and this figure may be significantly higher when newer specific diagnostic modalities, such as integrated positron emission tomography and computed tomography are considered [6].
Compared with the general population, patients with GCA have a 17-fold risk to develop a thoracic aortic aneurysm, despite adequate medical therapy [1]. Even in the absence of pre-existing aortic aneurysms or ectasia, patients with GCA can present acute aortic complications, such as aortic dissection and aorto-atrial fistula [4, 5]. It has been hypothesized that these complications are due either to the regression of an aortic wall granuloma (spontaneously, or secondary to immunosuppressive therapy), or to spontaneous aortic rupture, as has been reported in other inflammatory aortitis such as rheumatoid arthritis and Behcet's disease [3, 5].
Surgical repair of these aneurysms should aim at the removal of the entire diseased aortic root using a Bentall type procedure, as recommended by Gelsomino and colleagues [1] in their series, including 10 patients with confirmed GCA. The only 2 patients that did not undergo a Bentall procedure had to be reoperated on later for dilatation of the remaining native sinuses. Valve-sparing operations that exclude the diseased aortic wall or prevent its continued expansion may also be indicated in these patients to eliminate the possibility of future dissection or rupture of the aorta. Among these are the Yacoub, David, and Florida sleeve procedures [7]. Our patient would not have probably declared an aorto-atrial fistula on postoperative day 10 if the aortic root been replaced during the first operation. However, it remains questionable whether this dreadful complication was the direct consequence of GCA. Most reported aorto-atrial fistulas have been described years after cardiac and aortic surgery or trauma [8]. This is the first reported case occurring early after surgery. Moreover, the noncoronary sinus of Valsalva wall was completely dissected in our patient, and this part of the aorta is in intimate contact with the right atrial wall; continued periprosthetic leakage, tension on the suture line, rupture of a residual fragile area of the aortic root, and early postoperative adhesions may have all contributed to the formation of the fistula [5, 8].
In conclusion, patients with GCA are at a higher risk for catastrophic aortic complications. Surgery should be aggressive in removing all the diseased aortic tissue to prevent future aortic dilatation, dissection, or rupture.
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References
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- Gelsomino S, Romagnoli S, Gori F, et al. Annuloaortic ectasia and giant cell arteritis Ann Thorac Surg 2005;80:101-105.[Abstract/Free Full Text]
- Nuenninghoff DM, Hunder GG, Christianson TJ, McClelland RL, Matteson EL. Incidence and predictors of large artery complication (aortic aneurysm, aortic dissection, and/or large-artery stenosis) in patients with giant cell arteritis: a population-based study over 50 years Arthritis Rheum 2003;48:3522-3531.[Medline]
- Crean PA, Reid PS, Buchanan J. Aorto-atrial fistula and rheumatoid arthritis Ann Rheum Dis 1983;42:203-205.[Abstract/Free Full Text]
- Kerr LD, Chang YJ, Spiera H, Fallon JT. Occult active giant cell aortitis necessitating surgical repair J Thorac Cardiovasc Surg 2000;120:813-815.[Free Full Text]
- Melua A, Campbell N, McCluskey D, MacGowan SW. Aorto-atrial fistula without aneurysm formation in Behcet's disease Heart 1998;80:200-201.[Abstract/Free Full Text]
- Kwon CM, Hong YH, Chun KA, et al. A case of silent giant cell arteritis involving the entire aorta, carotid artery, and brachial artery screened by integrated PET/CT Clin Rheumatol 2007;26:1959-1962.[Medline]
- Hess Jr PJ, Klodell CT, Beaver TM, Martin TD. The Florida sleeve: a new technique for aortic root remodeling with preservation of the aortic valve and sinuses Ann Thorac Surg 2005;80(2):748-750.[Abstract/Free Full Text]
- Chung DA, Page AJF, Coulden RA, Nashef SAM. Aorto-atrial fistula after operated type A dissection Eur J Cardiothoracic Surg 2000;17:617-619.[Abstract/Free Full Text]
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Abstract
Introduction
