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Ann Thorac Surg 1999;68:1881-1885
© 1999 The Society of Thoracic Surgeons

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Current Reviews

Papillary fibroelastoma: increasing recognition of a surgical disease

^a Section of Cardiology, Department of Medicine, Boston Medical Center, Boston, Massachusetts, USA
^b Department of Cardiothoracic Surgery, Boston Medical Center, Boston, Massachusetts, USA
^c Department of Pathology, Boston Medical Center, Boston, Massachusetts, USA
^d Department of Cardiothoracic Surgery, University of Washington School of Medicine, Seattle, Washington, USA

Address reprint requests to Dr Davidoff, Section of Cardiology, Department of Medicine, Boston Medical Center, 88 E Newton St, Boston, MA 02118
e-mail: ravin.davidoff{at}bmc.org

	Abstract

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Papillary fibroelastomas are uncommon benign tumors usually involving the heart valves, which historically have been diagnosed at autopsy. With the advent of echocardiography, however, the number of patients diagnosed in life has increased. Papillary fibroelastomas represent a surgically treatable cause of cerebrovascular and cardiovascular ischemia and infarction making their identification clinically important. We report three unusual cases of papillary fibroelastoma; two patients presenting with symptoms of cerebrovascular ischemia and one presenting with myocardial infarction. We also present a comprehensive review of the literature and provide a compilation of all case reports to date.

	Introduction

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Cerebrovascular ischemia is generally the result of atheroemboli from a central vessel, in situ thrombosis of a major cerebral vessel, or emboli from a cardiac source. Most commonly, cardiac emboli arise from left atrial or left ventricular thrombi. Rarely, a primary cardiac tumor may be the cause of cerebrovascular or cardiac ischemia [1]. Papillary fibroelastomas are the third most common of the primary cardiac tumors after myxomas and lipomas, and the most common tumors of the valvular endothelium represented in autopsy series [2–4]. Before the advent of echocardiography papillary fibroelastomas were found incidentally at autopsy. Initially called "small harmless bodies" [5], they were soon recognized as a cause of sudden death [6–11]. The first patient diagnosed in life with papillary fibroelastoma was an incidental finding at operation for ventricular septal defect repair in 1979 [12]. With the advent of transthoracic and transesophageal echocardiography the number of cases of papillary fibroelastoma reported in life has increased. In recent years papillary fibroelastomas have been implicated in several disease states, which include cerebrovascular events, myocardial ischemia and infarction, and pulmonary embolism (Table 1). Herein we report three patients with papillary fibroelastoma that presented with symptoms of cerebrovascular ischemia, were discovered using echocardiography, and were successfully excised. In 1 patient, the tumor was not associated with the valve and in another, the tumors were multivalvular. Both are rare occurrences. We also present a comprehensive literature review of papillary fibroelastoma and include a compilation of all case reports to date.

	Case reports

Top Abstract Introduction Case reports Comment References

His physical examination was remarkable for a right homonymous hemianopsia on confrontational testing of the visual fields. This was confirmed with visual field mapping. Laboratory examination was unremarkable. The electrocardiogram displayed normal sinus rhythm. A computerized tomogram of the head was normal, without evidence for infarction or bleed.

The patient was admitted with the diagnosis of posterior cerebral infarction and placed on intravenous heparin. A magnetic resonance imaging scan revealed an infarction in the distribution of the left posterior cerebral artery and a small middle cerebral artery infarction. Magnetic resonance angiography showed no evidence for cerebrovascular or carotid artery disease. A transthoracic echocardiogram revealed trace mitral insufficiency with preserved systolic function. A transesophageal echocardiogram disclosed a mobile gelatinous mass at the junction of the left atrial appendage and left atrial body. No definite stalk was identified (Fig 1).

View larger version (66K): [in this window] [in a new window]   Fig 1. Transesophageal echocardiogram demonstrating a friable, broad-based mass attached to the left atrial wall near the junction of the atrial wall and atrial appendage. (LA = left atrium; MV = mitral valve.)

The patient tolerated the procedure well. There were no complications, and he was discharged on warfarin therapy for a total of 3 months. He has been free of cerebrovascular events since his discharge.

Patient 2
A 54-year-old man was referred to our institution for further evaluation of difficulty speaking and mental confusion. There was no history of cardiac or neurologic disease. He took no medications, smoked one package of cigarettes daily, and no longer drank alcoholic beverages.

Physical examination revealed a blood pressure of 160/85 mm Hg and slurred speech. Laboratory values were within normal limits. The electrocardiogram showed normal sinus rhythm. A computerized tomogram of the head revealed a hypodense lesion in the left basal ganglia. This was confirmed by magnetic resonance imaging. A transthoracic echocardiogram revealed a gelatinous mass with an echolucent center located on the atrial surface of the septal leaflet of the tricuspid valve. A transesophageal echocardiogram confirmed this lesion and identified an additional echodensity on the left coronary leaflet of the aortic valve. These masses were independently mobile of the valve leaflets and there were no clear fronds. He was treated with Coumadin without resolution of the masses, and was referred to operation with the presumptive diagnosis of papillary fibroelastoma. No attempt was made to investigate for pulmonary emboli, although, in retrospect, this may have proved interesting.

At operation a 10-mm (maximum dimension) mass was found on the atrial surface of the septal leaflet of the tricuspid valve (Fig 2). A 6-mm (maximum dimension) mass was identified on the ventricular surface of the left coronary leaflet of the aortic valve at its junction with the right coronary leaflet. Both were exophytic, soft, white, and friable. No other tumors were identified. Pathologic examination of both lesions revealed a collagenous stalk and core, surrounded by a dense mesh of elastic fibers, loose connective tissue matrix, and a single thin layer of cardiac endothelial cells on its surface. These findings were consistent with the diagnosis of papillary fibroelastoma (Fig 3). The patient tolerated the procedure well without postoperative complications. He was treated with warfarin for a total of 3 months, and remains free of cerebrovascular events since his operation.

View larger version (72K): [in this window] [in a new window]   Fig 2. Gross photomicrograph (through a dissecting microscope) of the tricuspid valve papillary fibroelastoma. The two small fragments separated from the main mass attest to the friability of these lesions. The arrowheads point to the previous site of attachment to the tricuspid valve.

View larger version (98K): [in this window] [in a new window]   Fig 3. The low power photomicrograph showing the thin endothelial cells lining the delicate elastic/hyaline-like core. The lower third of the figure shows the stalk merging with the valve tissue. (x20, elastic tissue stain.)

The patient was without significant past medical history and took no medications. He smoked one package of cigarettes daily. He reported that his father had died of a myocardial infarction while in his fifties and his sister had recently undergone coronary artery bypass grafting after myocardial infarction. He denied other cardiac risk factors. His physical examination was remarkable only for bibasilar rales. Laboratory values were notable for a creatinine kinase of 1,595 U/L with an MB fraction of 76 U/L and an index of 5. His electrocardiogram revealed normal sinus rhythm with lateral T wave changes. A radiograph of the chest was clear without evidence of congestive heart failure.

The patient was admitted to the coronary care unit with the diagnosis of myocardial infarction. A cardiac catheterization revealed a total occlusion with abrupt cutoff of the second obtuse marginal branch of the left circumflex artery. In addition, a 95% occlusion of the first diagonal branch of the left anterior descending artery was identified. The remainder of his coronary vessels was described as "pristine." Percutaneous transarterial coronary angioplasty of the first diagonal branch and second obtuse marginal branch was performed with good result. Embolism was suspected. A transthoracic echocardiogram revealed severe apical akinesis with moderately diminished systolic function. There was a mobile echogenic density located on the ventricular surface of the anterior mitral valve leaflet, at the junction of the chordae tendinae. This mass measured 7 mm (maximum dimension) using transesophageal echocardiogram, was round and shaggy without a clear stalk.

Upon further questioning, the patient recalled a transient period of right hand numbness 3 days before admission. It resolved within 30 minutes and he had no further symptoms. A noncontrast, computerized tomogram of the head revealed a recent infarction involving the left frontal operculum and insular cortex without bleed. The patient was seen in consultation with Cardiothoracic Surgery. He was taken urgently to the operating room with the presumptive diagnosis of papillary fibroelastoma.

At operation a 7-mm (maximum dimension), soft, white, friable mass was identified on the ventricular surface of the anterior leaflet of the mitral valve, between the chordae tendinae. It was resected without difficulty and the mitral valve was repaired. Pathologic examination confirmed the diagnosis of papillary fibroelastoma. The patient was placed on warfarin for a total of 3 months. He has remained free of cardiovascular or cerebral events since his operation.

	Comment

Top Abstract Introduction Case reports Comment References

 
Primary cardiac tumors have an estimated incidence of 0.0017% to 0.33% in autopsy series, and an estimated echocardiographic incidence of 0.019% in one clinical series [13]. Papillary fibroelastomas are the third most common of the primary cardiac tumors after atrial myxomas and lipomas, accounting for approximately 7% of cases [2–4]. Although they may be found anywhere in the heart, approximately 80% are found on the valvular endocardium. They represent the most common of the tumors of the heart valves accounting for 70% to 80% of patients [2–4, 14]. In autopsy series, they are found with nearly equal frequency on both sides of the heart and on all heart valves. They are usually singular in number. Multiple tumors have rarely been reported, with only seven such cases in the literature [15–21]. Grossly, they have been compared to a sea anemone because of their numerous and delicate papillary fronds. They are soft, white to tan, and friable, often with adherent thrombus, which explains their propensity to embolize [2, 14, 22].

The etiology of papillary fibroelastoma is unknown. One explanation is that they result from mechanical trauma as they are more common in elderly patients with long-standing heart disease, including rheumatic disease, and are rarely seen in children [2]. Other theories suggest that they represent neoplasm, hamartomas, and inflammatory nodules [2, 23]. Multiple terms have been used to describe this tumor including fibroma, papillary fibroma, papillary tumors, cardiac papilloma, myxofibroma, fibroelastic hamartoma, and valvular papilloma. They are believed by most investigators to be an entity separate from Lambl’s excrescences [2, 12, 24, 25]. Most authorities agree that papillary fibroelastoma is the correct term as it best describes the gross and histologic appearance of the tumor [2, 14].

Previous case reports have underrepresented the actual numbers of papillary fibroelastomas reported in life. Our review of the literature revealed 97 cases of papillary fibroelastoma (including our 3 patients) diagnosed in life and confirmed at operation (Table 1). In 63 of these patients, the symptoms that prompted investigation using echocardiography. In the remaining 34 patients the diagnosis was found incidentally in life either by echocardiography or operation performed for other indications. The most common presenting symptom of papillary fibroelastoma was cerebrovascular in origin, with 53% patients presenting with transient ischemic attack or stroke. The next most common presenting symptoms were of cardiac origin. Myocardial infarction was diagnosed in 13% by changes in the electrocardiogram and serial creatine kinase enzyme determination. Angina pectoris was diagnosed in 8% based on clinical grounds and changes in the electrocardiogram. Other presenting symptoms were dyspnea in 8%, myocardial infarction coexistent with transient ischemic attack, as in our third patient, in 5%, and presumed subacute bacterial endocarditis in 1 of 62 patients. There were two cases of pulmonary emboli, one case of syncope, and one case found during investigation of a cardiac murmur. We were unable to determine the presenting symptoms in 3 patients.

A recent retrospective series from the Mayo clinic [13] reported 18 patients with surgically resected and pathologically proven papillary fibroelastoma. We were unable to ascertain whether any of these patients had previously been reported. We have included these patients in Table 1. The researchers also identified 34 patients with the presumptive diagnosis of papillary fibroelastoma based on their own echocardiographic criteria. We did not include these 34 patients in our compilation of cases, as the diagnosis of papillary fibroelastoma was not confirmed [13]. In addition, we reasoned that any cases that may have been confirmed would have been included among the pathologically proven specimens. We report 3 new patients, which extend the clinical spectrum of disease caused by these rare tumors. Our first 2 patients presented with symptoms of cerebrovascular ischemia, the most common presenting symptom for papillary fibroelastoma. In our third case, the patient also had symptoms of cerebrovascular ischemia, a documented recent cerebrovascular infarction, and an acute myocardial infarction. In our first patient, the tumor was located in the left atrium. This patient had symptomatic papillary fibroelastoma involving the left atrium and a papillary fibroelastoma involving the left atrium [21, 26]. Our second patient had symptomatic papillary fibroelastoma involving multiple valves [15, 19] and involving both left and right-sided cardiac valves. Our last patient had papillary fibroelastoma presenting with myocardial infarction, and symptoms of cerebrovascular ischemia [27, 28]. All reported patients have involved the mitral valve.

The identification of papillary fibroelastoma is important because it represents a surgically correctable cause of cardiac embolic phenomena. Surgical excision is recommended regardless of symptoms. There is one case report of a patient with an aortic valve mass initially believed to represent a thrombus [29]. The patient was treated with warfarin for 7 months and experienced six transient ischemic attacks before undergoing surgical excision. At operation, she was found to have a papillary fibroelastoma. After excision, she reported no further cerebrovascular symptoms. In another case report [30], a patient with suspected papillary fibroelastoma identified with echocardiography was placed on warfarin and followed. Two weeks later she returned with symptoms of a cerebrovascular ischemia. A 3-mm papillary fibroelastoma was resected and she had no further neurologic complaints at her 1-year follow up.

Surgical excision with reconstitution or replacement of the valve is the recommended therapy for primary cardiac tumors, regardless of size and symptoms, given the risk for embolism [27, 29, 30–33]. In one series 10 of 12 patients (91%) undergoing operation for valvular papillary fibroelastoma were treated with excision of the tumor and valve repair. Only 2 of 12 patients (9%) required valve replacement [13]. Surgical resection should be performed as soon as possible to avoid recurrent embolism. In case of hemorrhagic stroke, the risk of systemic anticoagulation required during cardiopulmonary bypass probably outweighs the risk of recurrent embolism, and operation should be postponed for several weeks. There is no change in surgical approach, cannulation, cardiopulmonary bypass technique, or surgical resection of this tumor, compared with other cardiac tumors such as atrial myxoma. It should be emphasized, however, that papillary fibroelastoma is a friable tumor, and aggressive manipulation may result in fragmentation and further embolism. The use of preoperative transesophageal echocardiography to determine the exact site of attachment and its intraoperative use to guide excision has been recommended [22, 34]. The intraoperative use of dental mirrors and video-assisted thoracoscopy has also proven helpful in identifying these tumors [35]. No study looking specifically at the operative risks and postoperative course of papillary fibroelastoma excision has been performed. After operation, it is common practice to place these patients on warfarin. If valve repair or replacement is required, standard treatment guidelines regarding Coumadin use apply. The literature provides no firm evidence-based guidelines regarding use and duration of anticoagulation in the absence of valve repair or replacement. However, data on follow-up of these patients is lacking.

In conclusion, papillary fibroelastoma is an uncommon but increasingly recognized cause of embolic phenomena. Prompt identification allows for surgical excision, which is seemingly curative, safe, and well tolerated. In addition, embolic papillary fibroelastoma should be entertained as a cause for otherwise unexplained cardiovascular and cerebrovascular ischemia and infarction.

	References

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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): Gabriel S. Aldea Oz M. Shapira Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Howard, R. A. Articles by Davidoff, R. Search for Related Content PubMed PubMed Citation Articles by Howard, R. A. Articles by Davidoff, R.