CASE REPORTS

Angiosarcoma at the site of a Dacron vascular prosthesis: A case report and literature review W a y n e M. Weiss, M D , T h o m a s S. Riles, M D , T h o m a s H . G o u g e , M D , and H o w a r d H . Mizrachi, MD, New York, N.Y. Four of 32 reported sarcomas related to the aorta have arisen around previously placed aortic vascular prostheses suggesting that the graft may have been an etiologic factor. Our recent experience with such an angiosarcoma arising around a Dacron aortic graft prompted a review of the lesion to identify risk factors, diagnostic approaches, and treatment options. The diagnosis of these sarcomas is seldom made before operation. Animal studies have implicated plastic polymers including Dacron as carcinogenic materials capable of inducing sarcoma in 7% to 50% of exposures. Because of the rarity of these tumors and the thousands of vascular implants used over the past 30 years, it is unlikely that this degree of risk can be extrapolated to humans. However, a tumor should be included in the differential diagnosis of any mass or thromboembolic event associated with a vascular prosthesis. (J VAsc Smm 1991;14:87-91.)

The risk o f plastic foreign body carcinogenesis in humans is far less than that seen in murine species. However, the occurrence o f five reported cases o f sarcoma arising in association with synthetic vascular prostheses exemplifies that a small incidence does e x i s t . 2-6

We have recently treated a patient with an angiosarcoma arising around a previously placed aortic Dacron prosthesis. This case is the fifth reported o f a sarcoma in association with a Dacron aortic graft. An additional sarcoma has been reported arising around a femoral Dacron-Teflon prosthesis, s We reviewed the previous reported cases as well as the evidence implicating plastics as carcinogenic to define risk factors as well as current diagnostic and therapeutic modalities. CASE REPORT

A 56-year-old white man underwent an uneventfifl repair of an infrarenal aortic aneurysm on Feb. 7, 1986, with placement of a bifurcated woven double-velour Dacron graft. In late August 1989 he began to complain of left lower From New York UniversityMedical Center, New York. Reprint requests: Thomas S. Riles,MD, 530 First Ave., Suite 6F, New York, NY 10016. 24/4/27350

quadrant and lower back pain with sciatica. In September after radiologic examination of the lumbar spine was nondiagnostic for degenerative disk disease, a differential diagnosis including diverticular disease and pseudoaneurysm led to CT scanning of the abdomen because his symptoms persisted. The scan showed a thickened distal aorta that was interpreted as the previous aortic wall wrap with thrombus interposed between the graft and wrap. Magnetic resonance imaging confirmed that blood flow was isolated to the graft lumen and did not extend into the wrap. The patient's hematologic parameters were normal, and he was afebrile. A graft infection or pseudoaneurysm was considered unlikely, and the patient was observed. His symptoms persisted, and repeat CT scanning 2 months later showed increased size of the mass and left hydroureter with hydronephrosis (Fig. 1). Aortography showed a mild dilation of the graft limb anastomosis to the right lilac artery (Fig. 2). The patient was advised to undergo exploratory laparotomy because of the expansion of the mass, with the main differential diagnosis being pseudoaneurysm, graft infection, or tumor. In December 1989 exploration was undertaken and showed a vacularized mass of tissue surrounding the graft predominantly within but inferiorly beyond the aortic wrap. No evidence of infection or pseudoaneurysm was found. The tissue planes were ill defined and the mass was 87

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Fig. 1. CT scanning demonstrates a mass extrinsic to the contrast enhanced graft lumen. (214)

reoperated on for en block resection of the mass with the previous infrarenal graft and left nephroureterectomy. Local radiation by iodine 125 implant to the aortic bed was used and a new Dacron graft was placed. Seven months after operation the CT scan showed no recurrence of tumor. At 11 months liver metastases were seen. The patient is currently receiving chemotherapy. Pathologic findings

Fig. 2. Aortogram demonstrates a small pseudoaneurysm at the right graft-iliac artery anastomosis. (P)

firmly adherent to both the aortic wrap and prosthesis making the site of origin of the mass unclear. Although clinically suspicious for neoplasm, frozen section revealed only granulation tissue. A resection was not performed. Cultures were negative, and the final histopathologic diagnosis was high-grade angiosarcoma. The patient was

Examination o f the gross specimen revealed an irregular, nodular, soft, tan-red mass measuring 6.0 × 4.0 × 3.5 cm partially surrounding the graft at the aortoiliac bifurcation. T u m o r expanded the space between the graft and the native vessel wrap. It extended into 2 o f 16 paraaortic lymph nodes as well as the surrounding adipose tissue, where the bulk o f the rumor was located. The graft itself was intact, and the lumen was clear (Fig. 3). Microscopic examination revealed a highly cellular neoplasm composed o f irregular nests and cords o f cells within a fine fibrous stroma (Fig. 4). The cells were large and plump with moderate amounts o f amphophilic cytoplasm. The nuclei were round to oval and variably irregular in contour, with vesicular chromatin and prominent nucleoli. Moderate numbers o f mitoses, including atypical forms were noted. Many cells had prominent, single, cytoplasmic rounded spaces suggesting primitive lumen formation. In areas there was obvious formation o f vascular spaces. Neoplastic endothelial cells lined the ir-

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Fig. 3.. Cross section of the Dacron graft distal to the bifurcation. Hemorrhagic and tan nodules (IV) adjacent to the graft and extending into the surrounding adipose tissue (A), the lumen is clear (L).

Fig. 4. Irregularly anastomosing blood-filled channels (C) lincd by highly plcomorphic endothelial ceils (E). (Hematoxylin-eosin stain; original magnification x 184.) regularly shaped and anastomosing blood-filled channels. A prominen~ neutrophilic infiltrate was seen throughout the tumor, and a small focus of necrosis was present. Immunoperoxidase studies for factor VIII, vimentin, and alpha-l-antitrypsin were positive; a study for cytokeratins was negative. Electron microscopy revealed pinocytotic vesicles, abundant intermediate filaments, and partial investment by basal lamina. The final diagnosis was high-grade angiosarcoma.

DISCUSSION

After the description ofVoorhees et al.o of arterial replacement using fle;dble tubes of synthetic "Vinyon N" in 1951, the next 5 years saw the investigation of Teflon, Dacron, and Orlon as potential arterial substitutes. 7 While carrying out studies on hypertension in 1948, Oppenheimer et al.8 noted that rat kidneys wrapped in cellophane caused sarcomas. Further studies in murine species have shown that plastic

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Table I. Clinical and pathologic features of graft associated sarcomas Author

Burns et al. 1972~ O'Connell et al. 1976s Weinberg et aL 19794 Fehrenbacher et al. 1980z Weiss et al. 1990

Graft type and location Primary symptom

TeflonDacron femoral bypass WovenDacron abdominal a o r t a Woven Dacron thoracic aorta WovenDacron abdominal a o r t a Wovendacron abdominal aorta

Thigh m a s s

Histology

Fibrosarcoma

Lower extremity Fibrosarcoma ischemia Hypertension Malignantfibrous histiocytoma Lower extremity Angiosarcoma ischemia Back p a i n Angiosarcoma

films embedded subcutaneously can induce malignant mesenchymal rumors in 7% to 50% of exposures. 9 Primary tumors of the aorta are rare and are infrequently diagnosed before operation or at examination before death. In a recent review Schipper et al. ~° described 28 known cases, and we found four additional cases for a total of 32. 3'1~'13 Vascular insufficiency of the extremities, bowel, or kidneys caused by thomboembolism is the most common clinical presentation of a primary aortic tumor. Less common but specific features are hypertension, back pain, weight loss, and abdominal pain. 2'1° Only four sarcomas have been reported arising in association with Dacron vascular prostheses: three aortic and one femoral (Table I). Wallnofer and ZinnagP ~described a fifth case of an abdominal aortic hemangioendothelial sarcoma in a patient with a history of exposure to polyvinyl chloride and immunosuppression that should be excluded became of this exposure and uncertainty about the type of prosthesis. Our case is similar to the two cases reported by Weinberg and Mini* and Burns et al. s in that a rumor mass surrounded the graft material. O'Connell et al.3 reported a case in which a rumor thrombm obstructed the graft but no rumor could be found extrinsic to the graft, and a true primary site was not established. The patient described by Fehrenbacher et al.2 demonstrated a tumor arising within the graft at the proximal junction of the graft and aortic wall with tumor extending into the perigraft tissues. The three previous aortic cases demonstrated tumor embolization that was not a feature of our case. a'* Oppenheimer et al.9 demonstrated that plastic films embedded subcutaneously in the abdominal walls of murine species would result in sarcomas in the foreign body reaction that they induced. Among many plastics tested, Dacron was shown to be

Time between implant and tumor presentation

Follow-up

10 yr

Alive/well 1.5 yr

4 mo

Deceased 8 mo

14 mo

Operative death

12 yr

Deceased 7 mo

3.5 yr

Alive/well7 mo

tumorigenic. Mesenchymal rumors were induced in 7% to 50% of embeddings, and 85% of these were fibrosarcoma. The latent period varied from 7 months to 21/2 years with most rumors occurring i to 2 years after implantation. The physical form of the plastic influenced carcinogenicity. Films without pores were more carcinogenic than films with pores, and an inverse relationship between pore size and carcinogenicity existed. Also, if the plastic films were ground into powder, carcinogenicity was minimal despite the overall increase in surface area. 9 Oppenheimer et al.9 demonstrated the development of a proliferating fibrous connective tissue sheath around embedded films. When rumors arose, they did so within this sheath that showed progressive cellular atypia that would ultimately become sarcoma in those animals destined to develop a tuxnor.

Brand and Brand 1 characterized more precisely the features and risks of foreign body carcinogenesis in animals and man. Using growth behavior, chromosome analysis, and cellular morphologic studies, they were able to identify precancerous cells with tissue culture techniques in foreign body reactions to various implants. The likelihood of isolating these neoplastic cells is directly correlated to the incidence of foreign body tumorigenesis for that given strain or species. Different strains within murine species were found to be more susceptible to oncogenesis with foreign body exposure than others, and various species including humans and guinea pigs are highly resistant, implying a genetic predisposition for those at risk. Experimentally, Brand and Brand 1 studied 27 human foreign body reactions to various implants surgically removed and were unable to isolate precancerous cells from any of these. Additionally, Brand and Brand 1 compiled available reported cases

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of cancer arising at foreign body implantation sites in humans and found that 25% occurred within 15 years and 50% within 25 years of implantation. If a significant etiologic correlation existed, thousands of sarcomas should have already appeared in association with synthetic polymer vascular grafts. Sarcoma in association with vascular prostheses is a rare event without human risk factors, but it must be considered in the differential diagnosis of all masses associated with grafts or thromboembolic events. We recommend CT scanning early in the evaluation of masses to be supplemented with angiography and magnetic resonance imaging to help delineate between :masses extrinsic to the graft, graft infection, or pseudoaneurysm. All emboli recovered should be sent for ]pathologic examination. Biopsy is necessary to confirm the diagnosis, and wide en bloc excision with graft replacement is the treatment of choice. Consultation with medical and radiation oncologists is indicated, because a multimodality approach may offer these patients improved survival. REFERENCES 1. Brand KG, Brand I. Risk assessment of carcinogenesis at implantation sites. Plast Reconstr Surg 1980;66:591-4. 2. Fehrenbacher JW, Bowers W, Strate R, Pittman 1- Angiosarcoma of the aorta associated with a Dacron graft. Ann Thorac Surg 1981;32:297.-301. 3. O'Conuell TX, Fee HJ, Golding A. Sarcoma associated with Dacron prosthetic material. 1 Thorac Cardiovasc Surg 1976; 72:94-6.

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4. Weinberg DS, Mini BS. Primary sarcoma of the aorta associated with a vascular prosthesis. A case report. Cancer I980;46:398-402. 5. Burns AW, Kanhoun S, Tillman L, Satin N, Herrmann JB. Fibrosarcoma occurring at the site of a plastic vascular graft. Cancer 1972;29:66-72. 6. Voorhees AB, Jr, Jaretski A, III, Blakemore AH. Use of tubes constructed from Vingon '~N" cloth in bridging arterial defects. Ann Surg 1952;135:332-336. 7. Wesolowski SA. Foundations of modern vascular grafts. In: Sawyer PN, Kaplitt MJ, eds. Vascular grafts. 1st ed., New York: Appleton-Centui~c-Crofts, I978:27-49. 8. Oppenheimer BS, Oppenheimer ET, Stoup AP. Sarcomas induced in rats by implanting cellophane. Proc Soc Exp Biol Med 1948;67:33-4. 9. Oppenheimer BS, Oppenheimer ET, Stout AP, Willhite M, Danishefsky I. The latent period in carcinogenesis by plastics in rats and its relation to the presarcomatous stage. Cancer 1958;ii:204-i2. 10. Schipper J, van Oostagen IA, den Hollander IC, van Seyen AJ. Aortic mmours: report of a case and review of the literature. Br J Radiol i989;62:35-40. 11. Wallnofer H, Zinnagl N. Harnangiosarkomatose nach polynvinyl-chloridexposition. Med Klin 1977;72:410. 12. Nishikawa H, Mijakoshi S, Nishimura S, Seki A, Honda K. A case of aortic intimal sarcoma manifested with acutely occurring hypertension and aortic occlusion. Heart Vessels I989;5:54-8. 13. Kantelip B, de Riberolles C, Bailly P, Citron B. Primary endotheliosarcoma of the throacic aorta. Pathologica 1989; 81:163-70.

Submitted Sept. 4, 1990; accepted Dec. 13, 1990.

Angiosarcoma at the site of a Dacron vascular prosthesis: a case report and literature review.

Four of 32 reported sarcomas related to the aorta have arisen around previously placed aortic vascular prostheses suggesting that the graft may have b...
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