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Ann Thorac Surg 2001;72:601-603
© 2001 The Society of Thoracic Surgeons

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Case report

Preoperative embolization in the management of a mediastinal paraganglioma

^a Division of Thoracic Surgery and Interventional Radiology, Centre Hospitalier de l’Université de Montréal, Montréal, Québec, Canada

Accepted for publication July 29, 2000.

Address reprint requests to Dr Ferraro, Department of Surgery, Hotel-Dieu Hospital (CHUM), 3840 St Urbain St, Montréal, Québec, Canada H2W 1T8
e-mail: p.ferraro.chum{at}videotron.ca

	Abstract

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Parangliomas are rare and highly vascular tumors of neuroendocrine cell origin which are treated by complete surgical resection. Preoperative embolization to reduce perioperative bleeding complications, although described in paragangliomas of the neck and carotid body, has never before been described in the case of a mediastinal paraganglioma. The following is a presentation of such a case of mediastinal paraganglioma, in which embolization was used successfully before surgical resection.

	Introduction

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Preoperative embolization has been described by a number of authors in the treatment of paragangliomas of the neck and carotid body [1–3]. These neuroendocrine tumors are highly vascular and perioperative hemorrhagic complications have been frequently reported [4, 5]. We present a case of mediastinal paraganglioma in which preoperative embolization effectively reduced perioperative bleeding and permitted a complete surgical resection.

A 36-year-old patient was referred to our Thoracic Surgery unit after computed tomography (CT) scanning revealed a large mediastinal mass. The patient had been suffering from increased fatigability and muscle weakness. The appearance of thoracolumbar discomfort prompted him to seek medical attention. The preoperative thoracic CT-scan revealed several small adenopathies and a large 7 x 4 cm mass in the middle mediastinum behind the superior vena cava (SVC) and ascending aorta at the level of the carina (Fig 1). BHCG and alpha foeto protein (AFP) levels were normal, as well as routine blood chemistries and coagulation studies.

View larger version (154K): [in this window] [in a new window]   Fig 1. CT Scan of mass found in the middle mediastinum behind the right pulmonary artery at the level of the carina.

On pathologic examination, the biopsy specimen was characteristic of a paraganglioma. A nesting (Zellballen) pattern of round to polygonal tumor cells with abundant, granular eosinophilic cytoplasm, as well as typical neuro-endocrine granulations, was observed. Tumor cells were positive for chromogranin and neuron specific enolase. Peripheral sustentacular cells were positive for S-100 protein.

An I-131-MIBG scintigraphy obtained postoperatively showed intense mediastinal enhancement and no other lesion outside the chest. Urinary levels of 5-HIAA, metanephrine, Noradrenalin (NOR), and adrenalin (AD) were found to be normal. Given the hypervascular nature of the tumor, a consultation in interventional radiology was obtained to assess for the possibility of embolization before definitive surgical resection.

Angiography revealed branches supplying the mediastinal mass from a right bronchial artery, the left and right internal mammary arteries, the left thyrocervical trunk, and an aberrant left bronchial artery (Fig 2A, 2B). Embolization of all of these branches resulted in an almost complete disappearance of the tumor blush. Thoracic aortography was then repeated and revealed no further branches to the tumor mass. The procedure was carried out without complication.

View larger version (52K): [in this window] [in a new window]   Fig 2. (A) Angiogram showing a left bronchial artery and the hypervascular nature of the tumor before embolization. (B) Angiogram after embolization of the left bronchial artery supplying the tumor.

	Comment

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Paragangliomas are rare neoplasms of extraadrenal chromaffin tissue associated with the autonomic nervous system [4]. They have been classified by Glenner and Grimley on the basis of location and histology into branchiomeric (including carotid-body and aorticopulmonary), aorticosympathetic, intravagal, and visceral autonomic tumors [5]. The incidence of malignancy varies from 21% to 76% [4]. There are 149 well described cases of mediastinal paraganglioma [6], with anterior and middle mediastinal paragangliomas being most frequent [7].

In this case, the preoperative working diagnosis was one of lymphoma, and thus, a mediastinoscopy was recommended. When the preoperative workup is suggestive of a paraganglioma, a complete surgical resection should in most cases be attempted without a prior diagnostic biopsy because of the possibility of severe hemorrhage. Complete surgical excision is generally believed to be the treatment of choice for mediastinal paragangliomas [8]. In one review of 79 patients with middle and anterior mediastinal paragangliomas, Lamy and associates reported survival rates of 84.6% with complete resection (mean survival 125 ± 18 months), versus 50% with partial resection or biopsy (mean survival 71 ± 13 months), and an operative mortality of 5.3% [8]. The hypervascular nature and bleeding tendency of paragangliomas are well described [7, 8]. At least 1 intraoperative death has been attributed to hemorrhagic complications [7]. In addition, the relationship of middle and anterior mediastinal paragangliomas to the heart, great vessels and tracheobronchial tree makes surgical resection technically demanding. In previous reports, superselective angiography and subsequent embolization has been described in cases of paragangliomas of the carotid body and certain other paragangliomas occurring in the neck [1–3].

Embolization was usually carried out 1 to 7 days preoperatively, without serious complications [1–3]. Perioperative bleeding and postoperative complications in these patients were significantly decreased [2, 3]. This technique, however, had never been described for mediastinal paragangliomas. In the present case, preoperative embolization significantly reduced the vascularity of the mediastinal tumor, thereby facilitating surgical resection. In order to reduce perioperative hemorrhagic complications, we believe that embolization before surgical excision should be considered in all patients with a bulky mediastinal paraganglioma and in patients in which a difficult surgical exposure is anticipated.

	References

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1. Deruyver D., Mathurin P., Dooms G., Hamoir M. Lateral neck paraganglioma: diagnostic imaging and preoperative embolization. Journal Belge de Radiologie 1993;76:15-19.[Medline]
2. Ward P.H., Liu C., Vinuela F., Bentson J.R. Embolization. An adjunctive measure for removal of carotid body tumors. Laryngoscope 1988;98:1287-1291.[Medline]
3. Smith R.F., Shetty P.C., Reddy D.J. Surgical treatment of carotid paragangliomas presenting unusual technical difficulties. J Vasc Surg 1988;7:631-637.[Medline]
4. O’Riordain D.S., Young W.F., Jr, Grant C.S., Carney J.A., et al. Clinical spectrum and outcome of functional extraadrenal paraganglioma. World J Surg 1996;20:916-922.[Medline]
5. Glenner GG, Grimley PM. Tumors of the extraadrenal paraganglion systems. In: Atlas of tumor pathology (2nd series, fasc. 9). Washington, DC: Armed Forces Institute of Pathology, 1974.
6. Dunn G.D., Brown M.J., Sapsford R.N., et al. Functioning middle mediastinal paraganglioma (pheochromocytoma) associated with intercarotid paragangliomas. Lancet 1986;1(8489):1061-1064.[Medline]
7. Olson J.L., Salyer W.R. Mediastinal paragangliomas (aortic body tumor). A report of four cases and a review of the literature. Cancer 1978;41:2405-2412.[Medline]
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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): André Duranceau Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Rakovich, G. Articles by Duranceau, A. Search for Related Content PubMed PubMed Citation Articles by Rakovich, G. Articles by Duranceau, A. Related Collections Mediastinum