Ann Thorac Surg 2006;82:318-320
© 2006 The Society of Thoracic Surgeons
Case report
Coronary Pseudoaneurysm: An Unreported Complication of Castleman's Disease
S. Chris Malaisrie, MD
a
,
Matthias Loebe, MD, PhD
a
,
Jon-Cecil Walkes, MD
b
,
Michael J. Reardon, MD
b
,
*
a Division of Cardiothoracic Surgery, Michael E. DeBakey, Department of Surgery, Baylor College of Medicine, Houston, Texas
b Methodist DeBakey Heart Center, The Methodist Hospital, Houston, Texas
Accepted for publication July 26, 2005.
* Address correspondence to Dr Reardon, 6560 Fannin St, Suite 1002, Houston, TX 77030 (Email: mreardon{at}tmh.tmc.edu).
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Abstract
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Castleman's disease is considered a benign disease with very few reports of local invasion into adjacent structures. We present the first reported case of myocardial involvement with Castleman's disease. Careful preoperative planning allowed the placement of a left ventricular assist device with eventual heart transplantation after complete resection of the tumor and unsuccessful myocardial revascularization.
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Introduction
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Castleman's disease is a poorly understood lymphoproliferative disorder. The disease typically presents as a solitary lesion in the mediastinum. Early reports have characterized the disease as benign, but later descriptions of a multicentric form of the disease have highlighted its malignant potential. Nevertheless, complete surgical resection remains the recommended treatment. We report a case of recurrent Castleman's disease with invasion into the heart.
A 59-year-old white man was initially found to have a mediastinal mass and axillary lymphadenopathy during a workup for atypical chest pain. Evaluation and biopsy by video-assisted thoracoscopy revealed an anterior mediastinal mass invading the heart. The pathology was consistent with Castleman's disease. A subtotal resection of the mediastinal mass through a left thoracotomy was performed due to invasion of the myocardium. The patient was treated postoperatively with steroids. He presented again with chest pain and an acute myocardial infarction 1 year after his initial diagnosis. Coronary angiogram demonstrated an ectatic left anterior descending coronary artery (LAD) with subtotal occlusion and a thrombus formation. A coronary stent was placed at that time in an unsuccessful attempt to revascularize the myocardium. The patient suffered repeated myocardial infarctions eventually leading to ischemic heart failure. He was evaluated for heart transplantation, but due to the unknown malignant potential of the tumor, he was not deemed eligible. He was referred to our institution for further evaluation and treatment.
Echocardiography showed a left ventricular ejection fraction of 20% to 25%. Computed tomography and magnetic resonance imaging (Fig 1) revealed a mediastinal mass lateral to the pulmonary artery in direct contact with the anterior wall of the heart distal to the bifurcation of the left main coronary artery. Repeat coronary angiography revealed a completely occluded LAD distal to the tumor involvement. Preoperative planning involved resection of the myocardial mass with aortocoronary bypass, but it also included an evaluation for possible heart transplantation.
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Fig 1. Magnetic resonance image of the mediastinal mass (arrow) demonstrating invasion into the myocardium with involvement of the left anterior descending artery.
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The patient underwent exploration through a median sternotomy. A 6-cm mass was found invading the myocardium at the takeoff of the left anterior descending artery. The mass was resected while the patient was on cardiopulmonary bypass. Inspection of the mass revealed a pseudoaneurysm of the LAD and fistula tract to the mediastinal mass with an indwelling coronary stent. Final pathology revealed focal lymphatic aggregates and dilated lymphatic channels without malignant features in the background of fibro-inflammatory changes. The patient underwent aortocoronary bypass to his distal LAD, but failed to separate from cardiopulmonary bypass despite maximal inotropic support and placement of an intraortic balloon pump. A paracorporeal left ventricular assist device (Thoratec VAD System, Thoratec Corporation, Pleasanton, CA) was implanted as a bridge to transplantation. His postoperative course was uneventful, and the patient was mobilized and could fully resume physical activities using the portable driver. Four weeks after left ventricular assist device implantation the patient underwent eventual orthotopic heart transplantation during the same admission and was discharged to cardiac rehabilitation on postoperative day 98 from initial resection. His initial post-transplant hospital course was complicated by Serratia sepsis. With adequate antibiotic therapy he completely recovered, and he continues to be free of disease at 22 months.
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Comment
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Castleman's disease, also described as giant lymph node hyperplasia, angiofollicular lymph node hyperplasia, lymph node hamartoma, and benign lymph node lymphoma, was first described by Castleman and colleagues [1] in 1956. The most common histologic type is hyaline vascular accounting for approximately 90% of cases. Less frequent but more aggressive types are the plasma cell and mixed types [2, 3]. Patients may have a localized or multicentric disease with lymph node involvement typically in the mediastinum [4]. These tumors present with well-circumscribed masses and are believed to have a benign clinical course. Complete surgical resection is the recommended therapy and is considered curative [5]. However, close follow-up is recommended due to reports of recurrences.
This case demonstrates the invasive nature of what is believed to be a benign disease. Reports of chest wall [6], vertebral body [7], pulmonary artery, and bronchial adherence have been reported [8], but no cases of myocardial invasion have been described. Although complete resection is the recommended treatment of Castleman's disease, initial subtotal resection of the tumor in this patient was performed due to invasion of the myocardium. The patient was appropriately followed and treated with steroids under the direction of an oncologist, but the residual disease progressed to recurrence of the lymph node hyperplasia with invasion into a coronary artery as well as the myocardium. This seemed to have caused an inflammatory response ultimately leading to the formation of a pseudoaneurysm of the LAD and fistulization into the tumor. The pseudoaneurysm pre-dated the use of percutaneous coronary intervention and the subsequent placement of an intracoronary stent across the fistula neck was unsuccessful.
The presentation of a coronary pseudoaneurysm in association with tumor invasion is similarly an uncommon occurrence [9]. The most frequent cause is traumatic injury from catheter-based interventions [10]. Clinical presentation is varied, but it usually includes angina pectoris and myocardial infarction. Due to the risk of thrombosis or rupture, these pseudoaneurysms should not be left untreated. No standard therapy exists, but options include placement of covered stents, embolization with spring coils, and operative resection with bypass [11].
In this case, repeated myocardial infarctions in the anterior and septal myocardium resulted in poor left ventricular function. The patient was not eligible for heart transplantation due to the unknown malignant potential of his disease. Previous cases of malignant degeneration have been described in limited disease [12], but malignant degeneration is more typical in multicentric disease [13]. Therefore the patient underwent repeat resection to permit pathologic diagnosis and confirmation of the benign nature of the tumor. Failure to separate from cardiopulmonary bypass was anticipated due to his poor ventricular function. Although the LAD was bypassed in an attempt to revascularize the anterior wall of the heart, early recognition of the eventual need for a cardiac assist device allowed for the implantation of a paracorporeal left ventricular assist device prior to end-organ dysfunction.
This is the first reported case of Castleman's disease with myocardial invasion. The unique combination of tumor involvement with the left anterior descending artery causing ischemic heart failure from repeated myocardial infarctions with the use of a left ventricular assist device bridge highlights the importance of careful preoperative planning in this complex case.
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References
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- Keller AR, Hochholzer L, Castleman B. Hyaline-vascular and plasma-cell types of giant lymph node hyperplasia of the mediastinum and other locations Cancer 1972;29:670-683.[Medline]
- Shahidi H, Myers JL, Kvale PA. Castleman's disease Mayo Clin Proc 1995;70:969.[Medline]
- Gaba AR, Stein RS, Sweet DL, Variakojis D. Multicentric giant lymph node hyperplasia Am J Clin Pathol 1978;69:86-90.[Medline]
- Samuels LE, et al. Castleman's diseasesurgical implications. Surg Rounds 1997;20:449.
- Rena O, Casadio C, Maggi G. Castleman's diseaseunusual intrathoracic localization. Eur J Cardio-thorac Surg 2001;19:519-521.[Abstract/Free Full Text]
- Stokes SH, Griffith RC, Thomas PR. Angiofollicular lymph node hyperplasia (Castleman's disease) associated with vertebral destruction Cancer 1985;56:876-879.[Medline]
- Kim JH, Jun TG, Sung SW, et al. Giant lymph node hyperplasia (Castleman's disease) in the chest Ann Thorac Surg 1995;59:1162-1165.[Abstract/Free Full Text]
- Sawamura T, Takiya H, Yamada T, et al. A case of cardiac angiosarcoma with a pseudoaneurysm formed in the right coronary artery Jpn J Thorac Cardiovasc Surg 1994;47:565-568.
- Swaye PS Fisher LD, Litwin P, et al. Aneurysmal coronary artery disease Circulation 1983;67:134-138.[Abstract/Free Full Text]
- Aquel RA, Zoghbi GJ, Iskandrian A. Spontaneous coronary artery dissection, aneurysms, and pseudoaneurysmsa review. Echocardiography 2004;21:175-182.[Medline]
- Vasef M, Katzin WE, Mendelsohn G, Reydman M. Report of a case of localized Castleman's disease with progression to malignant lymphoma Am J Clin Pathol 1992;98:633-636.[Medline]
- Weisenburger DD, Nathwani BN, Winberg CD, Rappaport H. Multicentric angiofollicular lymph node hyperplasiaa clinicopathologic study of 16 cases. Hum Pathol 1985;16:162-172.[Medline]
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Abstract
Introduction
