Ann Thorac Surg 2003;76:935-937
© 2003 The Society of Thoracic Surgeons
Case report
Giant pleural fibroma with an abdominal vascular supply mimicking a pulmonary sequestration
Pradeep Kaul, MD, FRCSa,
Samantha Kay, MDa,
Peter Gaines, MD, FRCPa,
Simon Kim Suvarna, MD, FRCPathb,
David N. Hopkinson, MD, FRCS,
Gaetano Rocco, MD, FECTSa*
a Directorate of Cardiothoracic and Vascular Surgery, Sheffield, United Kingdom
b Department of Histopathology, Northern General Hospital, Sheffield Teaching Hospitals, Sheffield, United Kingdom
Accepted for publication February 14, 2003.
* Address reprint requests to Dr Rocco, The Price-Thomas Thoracic Unit, Northern General Hospital, Herries Rd S5 7AU, Sheffield, United Kingdom
e-mail: gaetano.rocco{at}btopenworld.com
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Abstract
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A 37 -year-old woman was found to have a giant mass in her right chest with an abnormal abdominal vascular supply at preoperative workup. Suspecting a pulmonary sequestration, the lesion was embolized and then removed through a standard thoracotomy. A broad-based, vascularized pedicle connected the mass to the diaphragm. Final pathology demonstrated a solitary fibrous tumor of the pleura with no malignant features. We believe this is the first case of solitary fibrous tumor of the pleura with a direct vascular supply from the abdominal aorta at the level of the renal arteries reported in the literature.
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Introduction
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We report a case of solitary fibrous tumor of the pleura (SFTP) with an unusual vascular supply from the abdominal aorta at the level of the renal artery take-off. On preoperative imaging, the lesion was thought to be a pulmonary sequestration, and an aortogram was done to identify the feeding vessel, which was then embolized prior to thoracotomy to reduce the risk of intraoperative bleeding. Awareness of a possible anomalous abdominal feeding vessel should warrant careful preoperative evaluation of the blood supply to an SFTP.
A 37-year-old woman, with an 18-month history of rheumathoid arthritis presented with a dull ache at the base of her right chest. On the chest roentgenogram she was found to have a mass in her right chest (Fig. 1).
Further investigation included a computed tomographic scan and magnetic resonance imaging, which suggested the diagnosis of a sequestration given the presence of a soft-tissue mass in the right chest with a possible venous drainage through the right inferior pulmonary vein together with a blush of vessels over the right hemidiaphragm (Fig 2).
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Fig 2. Contrast-enhanced computed tomography scan of the thorax demonstrating the mass in the right lower hemithorax with a sizeable vessel crossing its surface.
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An aortogram was performed showing an abnormal arterial supply from the abdominal aorta, just anterior to the right renal artery take-off (Figs 3, 4).
No venous drainage was identified. Two days before surgery a decision was made to embolize the systemic arterial supply in order
to decrease the risk of intraoperative bleeding. A right posterolateral thoracotomy was then performed and the mass was identified as extralobar, completely encapsulated, attached to the lower lobe by means of dense and vascularized adhesions and also to the diaphragm through a single, broad-based, pedicle containing multiple serpiginous vessels.
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Fig 3. (A) and (B) Angiogram of the abdominal aorta and relevant tributaries showing an abnormal vessel originating at the level of the right renal artery (arrows). (C) This vessel bifurcates in the proximity of the diaphragm. Hepatic = hepatic artery; renal = renal artery.
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After division of the adhesions from the surrounding structures, a 2-kg mass was delivered from the chest (Fig 5),
thus obtaining complete re-expansion of the previously compressed right lower and middle lobes.
Pathology showed a solid lesion (measuring 17.5 cm in diameter) with macroscopic foci of cystic degeneration. Microscopically, the lesion was comprised of interweaving fascicles of spindle cells with storiform architecture. Minor cytologic pleomorphism was reported.
Immunohistochemistry showed positive staining solely with vimentin and CD34, implying the mass to be a fibroma.
The postoperative course was uneventful. The patient remains well and asymptomatic 1 year postoperatively.
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Comment
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In retrospect, our case had all the signs of a benign SFTP [1]. The usual differential diagnoses of SFTP do not include pulmonary sequestrations, although the possibility of confusing these two entities has already been reported [2]. In fact, it was the presence in the right lower hemithorax of a soft tissue mass with an abnormal vascular supply on preoperative imaging that called for further investigations. At surgery, a definite venous drainage was not identified, although venous vessels were seen in both the pedicles to the lower lobe and diaphragm. However, the extreme variability of the vascular supply and the common embryological origin between intralobar and extralobar sequestrations hypothesized by some authors [3] may explain the discrepancy between the vascular supply to this mass, consistent with an intralobar sequestration, and also with the macroscopic findings at surgery that led us to diagnose an extralobar sequestration.
Solitary fibrous tumor of the pleura usually arise from the visceral pleura, although an origin from the parietal or the diaphragmatic pleura has been reported in about 20% of the cases [4]. We believe, this is the first time that a direct vascular supply to an SFTP from the abdominal aorta at the level of the renal arteries is reported in the literature. In the past, a case of diaphragmatic SFTP with a blood supply from the inferior phrenic artery was described in a young Japanese woman [5].
Spiral computed tomography has been used to detect abdominal arterial supply to the lung, including branches from the inferior phrenic and hepatic arteries or directly from the aorta [6]. In fact, one reason for the anomalous vascular supply in our patient could be that the SFTP had risen from a previous sequestration [7]. Two additional explanations include: (1) the possibility of a congenitally abnormal supply to the pleura by the abdominal aorta, and (2) the hypothesis of a parasitized vascular supply due to local inflammatory insults leading to neovascularization and deranged vascular inosculation across the diaphragm. The latter seems to be supported by the previous demonstration of an abnormal abdominal blood supply to the pleura related to an old infection [8] and the hypothesis that intralobar sequestrations may be acquired in origin and result from a localized inflammatory process [3].
In conclusion, this case emphasizes the need for a careful preoperative evaluation of the blood supply to an SFTP because of the possibility of anomalous abdominal feeding vessels that can be embolized to decrease the risk of intraoperative bleeding [9].
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Acknowledgments
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The authors wish to thank Drs J. Myers and I. Damjanov for their insights on the histopathologic discussion.
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References
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Abstract
Introduction
