Eur J Cardiothorac Surg 2008;34:1123-1125. doi:10.1016/j.ejcts.2008.07.040
Copyright © 2008, European Association for Cardio-thoracic Surgery. Published by Elsevier. All rights reserved.
An unusual case of calcifying fibrous pseudotumour of the cervicothoracic junction
Antonio Bobbio*,
Antonello Mazzeo,
Paolo Carbognani,
Michele Rusca
Unit of Thoracic Surgery, Department of Surgical Science, University Hospital of Parma, Italy
Received 5 June 2008;
received in revised form 19 July 2008;
accepted 21 July 2008.
* Corresponding author. Address: U.O. Chirurgia Toracica, Azienda Ospedaliera-Universitaria, Viale Gramsci 14, 43100 Parma, Italy. Tel.: +39 03406874733; fax: +39 0521703559. (Email: antonboa{at}hotmail.com).
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Abstract
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Calcifying fibrous pseudotumour is a rare benign tumour of soft tissue origin, occasionally encountered in the thoracic cavity; surgical resection is considered the treatment of choice. Herein we present the case of a young woman who presented with a calcifying fibrous pseudotumour located in the left cervicothoracic junction. At surgery the tumour was found to circumferentially entrap the left subclavian artery and its removal involved segmental artery resection and reconstruction by autologous saphenous vein graft.
Key Words: Calcifying fibrous pseudotumour • Surgery • Artery resection reconstruction
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1. Introduction
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Calcifying fibrous pseudotumour (CFP) is a rare benign tumour of soft tissue origin; the term was coined by Fetsch in 1993 and refers to an earlier definition of ‘childhood fibrous tumour with psammoma bodies’ employed by Rosenthal in 1988 [1,2].
CFP has so far been reported in single cases or small series; although it would appear to be more frequent in the extremities of the human body, several cases have also been described in the thorax, including pleura, mediastinum and chest wall [3–5]; a single case of the lung has also been reported [6]. In the face of suspected or proved CFP, surgery is envisaged and tumour recurrence seems exceptional after complete resection [7].
Herein we describe the case of a woman in whom a calcified tumour of the left thoracic apex had been diagnosed at a young age. At the time of surgery, 14 years after its discovery, the tumour was found to circumferentially encase the left subclavian artery; its resection was achieved by segmental artery resection and reconstruction. Definitive pathological diagnosis was consistent with CFP.
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2. Case report
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A 28-year-old woman was seen for a left apical thoracic mass. Her past medical history was unremarkable except for the incidental discovery on a chest X-ray, 14 years previously, of a calcified mass of the left apex.
She had no symptoms and, at clinical examination, the superior aspect of the tumour was palpable as a firm mass in the left supraclavicular fossa. The thoracic CT scan performed at the time of tumour discovery showed a solid and largely calcified tumour mass of 6 cm at its largest diameter occupying the apex of the left thoracic cavity. The tumour presented with well-defined margins lining the structures of the left superior mediastinum. No other diagnostic or therapeutic procedures had been prescribed over the next 14 years and a new imaging exploration took place because of a change in family practitioner. The new chest X-ray, compared to the 1 of 14 years previously, showed a markedly increased shadow of the mediastinal mass; the contrast enhanced CT scan showed that the tumour had about doubled in size by growing in its intra-thoracic part and the inferior border was now at the level of the carina (Fig. 1
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Fig. 1. Preoperative thoracic CT scan; the horizontal section is at the level of the anonymous vein; the coronal section is at the level of the left subclavian artery.
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Surgery was planned through an anterior thoracic approach and a ‘modified’ hemi-clamshell incision was performed [8]. After section of the costoclavicular ligament and section of the cartilage of the first rib, the tumour had to be dissected from the posterior aspect of the first rib before the lifting of the sterno-costal flap. Once the subclavian vein had been dissected, the retroclavicular tract of the subclavian artery was found to be entirely encircled by the tumour. The tumour had no attachments with other mediastinal structures and, after section of the anterior scalene muscle insertion, the anterior arch of the first rib was removed; the subclavian artery was clamped proximally and distally to its intra-tumoural tract and after segmental artery resection the tumour could be exteriorised. The artery was reconstructed by interposition of an autologous great saphenous vein graft which was inverted and sutured with Prolene° 5/0 (Fig. 2
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Fig. 2. (a) Operative aspect after artery reconstruction with vein graft (arrow). (b) The surgical specimen; the grab is positioned through the lumen of the subclavian artery. (c) Histology by haematoxylin and eosin stains (magnification 10x). Dense collagenous tissue with lymphocyte infiltrate and psammoma bodies.
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Postoperatively the patient presented with mild motor-sensitive deficit of the cubital nerve and she was discharged from hospital 18 days after surgery. On histopathological examination the tumour measured 11.5 cm x 7.5 cm x 5.5 cm and consisted of a densely hyalinised collagenous tissue with low cellularity, lymphoplasmacytic cell infiltrate, several areas of calcification and psammoma bodies (Fig. 2). The intra-tumour segment of subclavian artery measured 3 cm in length (Fig. 2).
Eighteen months after surgery, on thoracic CT scan the vein graft is patent and no sign of tumour recurrence is present.
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3. Discussion
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This case serves to illustrate an unusual clinical and surgical presentation of a CFP located at the cervicothoracic junction. Interestingly, we were able to detail the growth pattern of the tumour, which had about doubled in size during a 14-year period; in spite of its growth, no symptoms had appeared which, looking at previous literature, seems usual in the case of CFP; however, a case of CFP located at the cervicothoracic junction characterised by a thoracic outlet syndrome has been previously reported [3].
The main feature of our case, however, is the surgical finding of a tumour being traversed by an arterial mediastinal vessel; such an anomalous presentation could be due to the origin of the tumour from the wall of the artery. Preoperative imaging by contrasted CT scan could not represent this feature because the presence of diffuse intra-tumoural calcification masked the course of the subclavian artery; a nuclear magnetic resonance imaging would probably have been more useful.
At the time of surgery the CFP excision had to be performed by artery resection because the tumour, extremely firm and dense, could not be dissected from the artery. For artery replacement we opted for an autologous great saphenous vein graft, which was adequate in size and remained patent at follow-up.
In conclusion, a new case of CFP located in the cervicothoracic is added to the previous literature on CFP of the thorax. In this case surgical removal of the CFP was performed by artery resection and reconstruction.
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Footnotes
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Presented at the 16th European Conference on General Thoracic Surgery, Bologna, Italy, June 8–11, 2008.
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References
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- Rosenthal NS, Abdul-Karim FW. Childhood fibrous tumor with psammoma bodies. Clinicopathologic features in two cases. Arch Pathol Lab Med 1988;112:798-800.[Medline]
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- Maeda A, Kawabata K, Kusuzaki K. Rapid recurrence of calcifying fibrous pseudotumor (a case report). Anticancer Res 2002;22:1795-1797.[Medline]
- Rusca M, Carbognani P, Bobbio P. The modified "hemi-clamshell" approach for tumors of the cervicothoracic junction. Ann Thorac Surg 2000;69:1961-1963.[Abstract/Free Full Text]
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Abstract
1. Introduction
