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Ann Thorac Surg 1999;67:224-225
© 1999 The Society of Thoracic Surgeons

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Case Reports

Unsuspected idiopathic mediastinal fibrosis complicating coronary bypass operation

^a Department of Thoracic and Cardiovascular Surgery, Georg-August-University, Göttingen, Germany
^b Department of Pathology, Georg-August-University, Göttingen, Germany

Accepted for publication June 3, 1998.

Address reprint requests to Dr Aleksic, Klinik für Thorax-, Herz und Gefäßchirurgie, Georg-August-Universität Göttingen, Robert-Koch-Str. 40, 37075 Göttingen, Germany
e-mail: (ialeksi{at}gwdg.de)

	Abstract

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A 66-year-old woman was referred for coronary bypass operation to the left anterior descending and the circumflex arteries. She had a systemic inflammatory process of unknown origin. On opening the pericardium, fibrotic plaques encircling the entire ascending aorta were found. Aortic cannulation and proximal anastomosis of the planned vein graft were impossible. The right internal mammary was anastomosed to the left anterior descending artery, the left to the obtuse marginal after femoral arterial cannulation. Pathologic examination revealed idiopathic mediastinal fibrosis.

	Introduction

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Mediastinal fibrosis can be associated with several disease syndromes, including retroperitoneal fibrosis, sclerosing cholangitis, Riedel thyroiditis, and inflammatory pseudotumor of the orbit [1]. All syndromes are believed to be major regional manifestations of a systemic sclerosing disease. When mediastinal fibrosis occurs, common manifestations include the superior vena caval syndrome [2], constrictive pericarditis, and pulmonary hypertension [3]. Most cases of mediastinal fibrosis seen in the United States are caused by fungal infections, primarily histoplasmosis [4]. Mycobacterial infections are less common, and the remainder of cases represent idiopathic forms of mediastinal fibrosis [5]. In idiopathic retroperitoneal fibrosis, which is believed to be similar to mediastinal fibrosis, other possible causes include bacterial infections, autoimmune syndromes, and drug interactions, e.g., methysergide, beta-blockers, methyldopa, and hydralazine [6,7]. We describe the unexpected finding of idiopathic mediastinal fibrosis (Ormond disease) in a woman who had coronary artery bypass grafting and the implications of our intraoperative treatment.

A 65-year-old woman was referred to our department for recurrent stenosis of the circumflex artery and persistent angina after angioplasty. The left anterior descending artery was proximally occluded but perfused via collaterals from the right coronary artery.

Her medical history included tuberculous pleuritis in 1951, ileitis, colitis, and proctitis of unknown origin in 1991. At the time of referral she had chronic systemic inflammatory process of unknown origin with anemia for 6 years and chronic obstructive pulmonary disease.

Laboratory studies showed normal serum creatinine, blood urea nitrogen, uric acid, and creatinine clearance (95 mL/min). Serum electrophoresis demonstrated elevation of 1-, 2-, and -globulins compatible with chronic inflammation. C-reactive protein (67.4 mg/dL), fibrinogen (607 mg/dL), and sedimentation rate (32/60) were elevated. Results of rheumaserology, urinalysis, and liver enzymes were unremarkable. Her chest x-ray was normal except for specific calcified lesions in the right lung attributable to tuberculosis diagnosed in 1951. Abdominal ultrasound did not show hydronephrosis, any echofree mass, or signs of inflammatory abdominal aortic aneurysm.

The initial operative approach was to bypass the left anterior descending artery with the left internal mammary artery and the circumflex artery with a saphenous vein graft to an obtuse marginal branch by using cardiopulmonary bypass. After dissecting the left internal mammary artery and opening the pericardium, a large fibrous plaque around the entire ascending aorta was encountered (Fig 1). The superior vena cava and the innominate vein were both involved. The tissue was woody and aortic cannulation was deemed impossible. Aortic wall thickness was 2.2 cm measured by puncture with an 18-gauge needle. The difficulty in cannulation and anastomosis of the saphenous vein graft to the ascending aorta mandated a change in the operative strategy. Both internal mammary arteries were dissected, and the femoral artery was cannulated. After cardiopulmonary bypass was begun, the right internal mammary artery was anastomosed to the left anterior descending artery, and the left internal mammary artery to the obtuse marginal branch. The operation was completed uneventfully. The postoperative course was uncomplicated and the patient was discharged after 8 days.

View larger version (19K): [in this window] [in a new window]   Fig 1. Intraoperative photograph showing the fibrotic masses encircling the ascending aorta.

View larger version (155K): [in this window] [in a new window]   Fig 2. Histologic section demonstrating bands of hyalinized collagenous fibers with interspersed fibrocytes and fibroblasts, scattered aggregates of lymphocytes, plasma cells, and histiocytes (Trichrom Goldner stain, x100 before 65% reduction).

	Comment

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Mediastinal fibrosis can be self-limiting but also cause serious complications. Approximately 40% of patients are asymptomatic and the disease is discovered incidentally. In the remainder, symptoms reflect the progressive restriction of mediastinal structures by the fibrosing process and might mimic a malignant process. Thin-walled structures such as the superior vena cava are most commonly affected. Mediastinal fibrosis was described as the most frequent cause of superior vena caval obstruction [8]. In most patients the superior vena cava syndrome will improve over time because of developing venous collaterals. Superior vena cava bypass has been recommended in patients who do not improve [8]. Patients might also present with cough, dyspnea, chest pain, fever, or dysphagia. Cough, dyspnea, and hemoptysis are the most frequent symptoms. Pulmonary hypertension has been described occasionally [3]. Our patient did not have any signs of superior vena cava syndrome. Intermittent coughing and dyspnea were attributed to underlying chronic obstructive pulmonary disease.

The typical feature on routine chest x-ray is a widened superior mediastinal shadow. However, 18% of patients with mediastinal fibrosis had a normal appearing chest x-ray in one study [9], as was the case in this patient. This patient did not have evidence of retroperitoneal fibrosis or sclerosing cholangitis, which had been excluded by abdominal ultrasound and normal hepatic and renal laboratory tests. It is possible that the patient was operated on at a stage when retroperitoneal involvement simply was not detected.

Revascularization in patients with mediastinal fibrosis represents a unique operative challenge, as the fibrous tissue, typically described as woody, obscures normal tissue planes [5]. The thickness of the ascending aorta made aortic cannulation and proximal saphenous vein graft anastomosis virtually impossible. In such patients complete arterial revascularization is an important alternative approach, as both internal mammary arteries might be spared from the mediastinal involvement. Other surgeons might have chosen minimally invasive approaches to avoid both proximal aortic anastomosis and cardiopulmonary bypass. However, the obscured tissue planes make exposure difficult, if not impossible, through a small incision. Furthermore, coronary artery bypass without cardiopulmonary bypass and cardioplegic arrest on more than one coronary artery has not been reported widely yet. Additionally, no long-term follow-up data on any minimally invasive coronary bypass are available at present. In conclusion, when faced with the unexpected finding of mediastinal fibrosis after median sternotomy, revascularization with both internal mammary arteries and femoral arterial cannulation yields the optimum benefit for the patient in terms of total arterial revascularization and safety for performing the anastomosis to the obtuse marginal branch.

	References

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1. Stewart T.W., Jr, Friberg T.R. Idiopathic retroperitoneal fibrosis with diffuse involvement: further evidence of systemic idiopathic fibrosis. South Med J 1984;77:1185-1187.[Medline]
2. Miller R.E., Sullivan F.J. Superior vena caval obstruction secondary to fibrosing mediastinitis. Ann Thorac Surg 1973;15:483-492.[Abstract/Free Full Text]
3. Arnett E.N., Bacos J.M., Macher A.M., et al. Fibrosing mediastinitis causing pulmonary arterial hypertension without pulmonary venous hypertension. Clinical and necropsy observations. Am J Med 1977;63:634-643.[Medline]
4. Goodwin R.A., Nickell J.A., DesPrez R.M. Mediastinal fibrosis complicating healed primary histoplasmosis and tuberculosis. Medicine 1972;51:227-246.[Medline]
5. Urschel H.C., Jr, Razzuk M.A., Netto G.J., Disiere J., Chung S.Y. Sclerosing mediastinitis: improved management with histoplasmosis titer and ketoconazole. Ann Thorac Surg 1990;50:215-221.[Abstract/Free Full Text]
6. Koep L., Zuidema G.D. The clinical significance of retroperitoneal fibrosis. Surgery 1977;81:250-257.[Medline]
7. Brooks M.T., Magill H.L., Hanna S.L., et al. Pediatric case of the day: idiopathic retroperitoneal fibrosis. Radiographics 1990;10:1096-1100.[Medline]
8. Doty D.B., Doty J.R., Jones K.W. Bypass of superior vena cava: fifteen years’ experience with spiral vein graft for obstruction of superior vena cava caused by benign disease. J Thorac Cardiovasc Surg 1990;99:889-895.[Abstract]
9. Parish J.M., Marschke R.F., Jr, Dines D.E., Lee R.E. Etiologic considerations in superior vena cava syndrome. Mayo Clin Proc 1981;56:407-413.[Medline]

This article has been cited by other articles:

				P. Saxena and P. J. Tesar Vascular obstruction related to mediastinal fibrosis: an interesting clinical entity. J. Thorac. Cardiovasc. Surg., November 1, 2007; 134(5): 1379 - 1379. [Full Text] [PDF]

				P. Saxena and P. J. Tesar Mediastinal Fibrosis Causing Myocardial Ischemia Ann. Thorac. Surg., December 1, 2005; 80(6): 2368 - 2370. [Abstract] [Full Text] [PDF]

This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Ivan Aleksic Thomas Busch Horia Sîrbu Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Aleksic, I. Articles by Dalichau, H. Search for Related Content PubMed PubMed Citation Articles by Aleksic, I. Articles by Dalichau, H.