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Ann Thorac Surg 2000;70:977-979
© 2000 The Society of Thoracic Surgeons

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Case report

Rare presentation of cardiac hemangiomas

^a Department of Thoracic and Cardiovascular Surgery, University Hospital, Bern, Switzerland
^b Institute of Pathology, University Hospital, Bern, Switzerland

Address reprint requests to Dr Kipfer, Department of Thoracic and Cardiovascular Surgery, University Hospital, Freiburgstrasse 10, CH-3010 Bern, Switzerland
e-mail: beat.kipfer{at}insel.ch

	Abstract

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Cardiac hemangiomas are exceptionally rare tumors with an incidence of 1% to 2% of all detected benign heart neoplasms. The clinical appearance of the tumor varies considerably and may mimic other pathological findings of definite heart structures. We report two cases of cardiac hemangiomas presenting with an unusual location and clinical course.

	Introduction

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Tumors of the heart are rare, with the myxomas representing 70% to 80% of all neoplasms [1]. Hemangiomas, with an incidence of 0.8% to 1.6%, present with a variety of symptoms depending on location and extent of the tumor. They may involve cardiac chambers as well as the epicardium. Symptoms result from compression of surrounding structures or embolization [2]. Most neoplasms are discovered at postmortem examination [3, 4]. We report two cases of hemangiomas with different clinical manifestations due to rather unusual locations.

	Case reports

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Case 1
A 74-year-old male was admitted for coronary artery bypass grafting. He had stable angina with T-wave changes and first-degree atrioventricular block evident in the electrocardiogram. Ischemia was demonstrated in the treadmill protocol and triple-vessel disease was found at angiography. The left ventricular ejection fraction was preserved with 65%. At elective surgery, an unexpected mass arising from the anterior wall of the right ventricle displacing the right coronary artery was found. This capsulated and pedunculated tumor was dissected and removed before starting the revascularization. No infiltration of the myocardium occurred and the right coronary artery was preserved. The postoperative course was uneventful. The patient was discharged at day 7 and underwent hip replacement 12 months later.

Case 2
This 62-year old male had five episodes of transient ischemic attacks within 24 months. He was admitted at last with paralysis of the left upper extremity and left-sided facial nerve palsy to our neurological department. Computed tomography scan demonstrated bilateral ischemic lesions in the internal capsule and the cerebellum. Duplex examination showed insignificant changes of both vertebral and basilar arteries. Transesophageal echocardiography revealed aneurismal changes of the interatrial septum with an open foramen ovale and a pedunculated mass of 3.5 x 1.9 cm. The appearance was typical for a myxoma. The suspicion of a myxoma with paradoxiacl embolization was established. The patient was referred to us for surgery. The neurological deficits have meanwhile disappeared and coronary angiography was normal.

At surgery, the interatrial septum was thickened with a bluish appearance. No endocavitary mass as expected was found. The foramen ovale was patent for a 3-mm probe. The interatrial septum was resected and replaced with a PTFE membrane. Frozen section showed no infiltration of the surrounding structures as suspected initially.

After uneventful course, the patient stayed free of any further neurological events. Echocardiography at 7 months excluded any residual tumor and showed competent interatrial septum.

Case 1 histological findings
In Figure 1 the tumor tissue is composed of blood-filled cavernous and arteriovenous vessels within loose connective and adjacent fatty tissue. Patchy areas of chronic inflammation and septal fibrosis are identified. The diagnosis is hemangioma of the arteriovenous and cavernous type with septal fibrosis.

View larger version (152K): [in this window] [in a new window]   Fig 1. Cavernous and arteriovenous vessels within loose connective tissue with patchy areas of chronic inflammation and septal fibrosis.

View larger version (158K): [in this window] [in a new window]   Fig 2. Papillary endothelial hyperplasia and focal infiltration with lymphocytes without signs of malignancy.

	Comment

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The presented cases illustrate well the variety of clinical presentation due to the different localization. While the tumor was found incidentally in the first case, the second patient has suffered from multiple cerebral embolic episodes. The tumor was suspected to be an atrial myxoma, but no endocavitary mass was found at surgery. To our knowledge, we report the first cardiac hemangioma with this complication. Histologically, the tumor shows proliferation of blood vessels that may be either capillaries or large cavernous channels. Papillary endothelial hyperplasia as an isolated entity can easily be misdiagnosed as angiosarcomas [7]. However, lack of mitotic activity, cellular pleomorphism, necrosis, and cellularity distinguishes hemangiomas from malignant lesions.

Careful review of the coronary angiographies can reveal a patch of neovascularity termed as tumor blush, as was suspected in our first case retrospectively. Magnetic resonance imaging and transesophageal echocardiography may help to detect cardiac hemangiomas preoperatively, but we were unable to demonstrate an echo dense mass in one case.

Intracavitary and intramyocardial hemangiomas have a potential risk of recurrence, especially if there has been incomplete resection at initial surgery. Therefore, radical resection of such tumors with extensive reconstruction of intracavitary and septal structures with prosthetic material is recommended [8]. Even asymptomatic epicardial tumors should be removed radically because of the potential of life-threatening pericardial effusion.

	References

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1. Bauer E.P., von Segesser L.K., Carrel T., Laske A., Turina M.I. Frühresultate nach chirurgischer Behandlung von Herztumoren. Schweiz Med Wschr 1991;121:255-258.
2. Brizard C., Latremouille C., Jebara A., Acar C., Fabiani J.N., Carpentier A.F. Cardiac hemangiomas. Ann Thorac Surg 1993;56:390-394.[Abstract/Free Full Text]
3. Basso C., Valente M., Poletti A., Casarotto D., Thiene G. Surgical pathology of primary cardiac and pericardial tumors. Eur J Cardiothorac Surg 1997;12:730-738.[Abstract/Free Full Text]
4. Cooley D.A. Surgical treatment of cardiac neoplasms. Thorac Cardiovasc Surg 1990;38:176-182.
5. Blondeau P. Primary cardiac tumors. Thorac Cardiovasc Surg 1990;38:192-195.
6. Lam K.Y., Chan A.C.L. Tumors of the heart. A 20-year experience with a review of 12,485 consequtive autopsies. Arch Pathol Lab Med 1993;117:1027-1031.[Medline]
7. Burke A., Virmani R. Atlas of tumor pathology. Tumors of the heart and the great vessels. Washington DC: Armed Forces Institute of Pathology, 1996.
8. Abad C., Campo E., Estruch R., et al. Cardiac hemangioma with papillary endothelial hyperplasia. Ann Thorac Surg 1990;49:305-308.[Abstract/Free Full Text]

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