Ann Thorac Surg 2009;88:1999-2001. doi:10.1016/j.athoracsur.2009.05.063
© 2009 The Society of Thoracic Surgeons
Case Reports
Preoperative Embolization of Castleman's Disease Using Microspheres
Warren Swee, MD, MPHa,
Ahmed M. Housseini, MDa,c,
J. Fritz Angle, MDa,b,c,d,
David R. Jones, MDb,
Thomas M. Daniel, MDb,
Ulku C. Turba, MDa,
Ehab A. Abdel-Gawad, MDa,d,
Klaus D. Hagspiel, MDa,*
a Department of Radiology, University of Virginia Health System, Charlottesville, Virginia
b Department of Surgery, University of Virginia Health System, Charlottesville, Virginia
c Department of Radiology, Suez Canal University, Ismailia, Egypt
d El-Menia University, Department of Radiology, El-Menia, Egypt
Accepted for publication May 15, 2009.
* Address correspondence to Dr Hagspiel, University of Virginia Health System, 1215 Lee St, Charlottesville, VA 22908 (Email: kdh2n{at}virginia.edu).
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Abstract
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Castleman's disease is a rare form of lymph node hyperplasia most commonly presenting as a solitary hypervascular mediastinal mass. Surgical resection is the treatment of choice, but this can be associated with significant blood loss due to its hypervascularity. We report two cases with a preoperative diagnosis of mediastinal Castleman's disease in whom preoperative embolization with Trisacryl gelatin microspheres (Biosphere Medical, Rockland, MA) was performed. Compared with the literature, a decrease in the amount of perioperative bleeding was noted in both cases.
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Introduction
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Castleman's disease (CD) is a rare lymphoproliferative disorder of unknown cause. Although surgical resection is the treatment of choice, it may be associated with significant blood loss due to inherent hypervascularity [1]. The largest published series reported a mean blood loss of 620 mL for 8 patients who underwent surgical removal of pleural CD [2]. Only four prior reports described the use of preoperative embolization [3–6]. We present two cases of CD treated with open resection after preoperative embolization using microspheres, a newer agent with greater small vessel penetration and improved tumor devascularization.
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Case Reports
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Patient 1
A 15-year-old girl with a history of a 1-week dry cough was found to have a right paratracheal mass on chest roentgenogram. Contrast-enhanced computed tomographic scan of the chest showed a uniformly enhanced (5 x 7 cm) right paratracheal mass (Fig 1A). A core biopsy demonstrated CD of the hyaline vascular type.
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Fig 1. (A) Computed tomographic image shows a homogenously enhanced, well-circumscribed right paratracheal mass. (B) Aortogram demonstrates uniformly enhanced right paratracheal mass. The right internal mammary artery (IMA) (arrow) and bronchial artery (not shown) were the major feeding vessels. (C) Selective angiogram of the right IMA and the right bronchial artery after embolization with microspheres demonstrates cessation of flow of IMA (left) and computed tomographic image (right).
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The patient underwent four cycles of treatment with rituximab (Rituxin; Genentech, San Francisco, CA) to induce regression. Surgical resection was then performed after preoperative embolization.
Thoracic aortography with selective bilateral internal mammary and bronchial artery injections showed a right paratracheal tumor blush (Fig 1B). Feeding vessels included a right medial internal mammary artery (IMA) branch and two right bronchial branches, all of which were embolized with 350 to 500 µm microspheres (Biosphere Medical, Rockland, MA) resulting in tumor devascularization (Fig 1C).
Intraoperatively, the tumor was found to encase the azygos vein and portions of the vagus nerve, which was divided, and the azygos vein was ligated. The tumor was removed without macroscopic evidence for residual tumor. Blood loss was minimal with an estimated 50 cc, and the postoperative course was uncomplicated. The final pathologic diagnosis was hyaline-type CD.
Patient 2
A 46-year-old woman with a diagnosis of hyaline-vascular type CD on open biopsy was transferred to our institution for treatment. A computed tomographic scan demonstrated a strongly enhancing lobulated anterior mediastinal mass (Fig 2A). Her past medical history was significant for thyroid radioablation for Graves' disease 10 years earlier. Surgical removal of the mass after preoperative embolization was planned.
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Fig 2. (A) Computed tomographic image demonstrates inhomogenously enhanced anterior mediastinal mass. (B) Selective angiogram of the left internal mammary artery demonstrates it to be the major feeding artery of the mediastinal mass (left). Angiogram after embolization with microspheres demonstrates cessation of flow to the mass (right).
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A thoracic aortogram with selective internal mammary and bronchial angiography were performed, which showed that the tumor was supplied by both internal mammary arteries. Embolization of the feeding branches was performed using 500 to 700 µm microspheres to stasis, followed by helical coils, resulting in devascularization of the mass (Fig 2B).
Intraoperatively, the tumor appeared to arise from thymic tissue and encased the phrenic nerves. These portions of the tumor were left unresected. The remaining tumor was removed without complication. The blood loss was an estimated 400 cc. The postoperative course was uncomplicated, and the patient was scheduled to undergo postoperative radiation therapy for the residual tumor. Final pathology of the resected specimen resulted in alternate diagnosis of thymoma with lymphoid B-cell hyperplasia, an entity with histologic similarities to CD.
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Comment
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Castleman's disease was first reported in 1954. Clinically it presents in localized or multicentric forms primarily involving the mediastinum, neck, and mesentery [7].
There are three histologic subtypes: (1) hyaline-vascular, (2) plasma-cell, and (3) a mixed variant. Approximately 85% of localized forms are of the hyaline vascular type, and the remaining 15% are the plasma cell type; mixed types are rare. Localized disease most commonly presents in the mediastinum and is usually discovered incidentally in asymptomatic patients [8].
Symptoms are typically related to mass effect on adjacent structures. The standard treatment of localized forms is surgical resection. However, the tumor may encase or invade adjacent structures preventing complete resection necessitating postoperative radiation. The multicentric subtype is generally of the plasma cell type and presents as a systemic illness with a far more aggressive course, often leading to infectious complications and malignant transformation; this is treated with combination chemotherapy, radiation therapy, and surgical debulking [9].
Computed tomographic imaging typically shows a sharply marginated smooth or lobulated soft tissue mass, with or without lymphadenopathy, and with strong contrast enhancement. An angiography demonstrates a vascular tumor supplied by feeding vessels from the bronchial, internal mammary, or intercostal arteries [10]. Tissue diagnosis is mandatory to avoid mismanagement. A computed tomographic-guided needle biopsy is possible, but it can have low diagnostic accuracy and is also fraught with the risk of uncontrollable hemorrhage [11]. Therefore, some authors recommend thoracoscopic biopsy because of the ability to visualize and control biopsy-induced hemorrhage [12]. Although recent literature has shown that the thoracoscopic resection of CD is possible, open surgery is preferred [13]. It is in these cases that the preoperative embolization was found to be helpful.
Histologically, the hyaline-vascular variant is characterized by the presence of "burned out" germinal centers with mantle zones demonstrating an "onion skin" pattern [1]. Interestingly, in our second case, the final pathology of the resected tumor was thymoma with lymphoid B-cell hyperplasia, whereas two separate readings of the initial biopsy were interpreted as CD. Thymoma with lymphoid B-cell hyperplasia is a rare entity in which the original publication of histopathologic features that were reminiscent of CD was described. Both entities are known to be highly vascularized, and as the primary treatment for both diseases is surgical resection, they share the risk for significant perioperative blood loss [14].
Our cases implemented the use of microspheres, a newer embolic agent preferred nowadays in comparison with polyvinyl alcohol (PVA) by many interventionalists. Microspheres are composed of an acrylic polymer impregnated within porcine gelatin available in sizes of 40 to 1,200 µm. The use of the agent for embolization was first reported in 1996. Several studies have reported their effectiveness for preoperative embolization of CNS meningiomas and for uterine artery embolization to treat uterine fibroids [15]. The older PVA particles have more irregular shapes and can clump, thus possibly occluding micro-catheters or resulting in too proximal occlusion. Microspheres do not clump and thus penetrate deep into target tissues. Two studies have compared the effectiveness of microspheres to PVA in animal models showing microspheres to reduce blood flow more quickly and reliably [15].
In the four previously published cases of CD embolization, the authors used PVA and gelfoam in one case each. The embolic agent in one case was not specified, and in the last case, microspheres were used [3–6]. Our experience, as well as the case reported by Robert and colleagues [6], suggests that microspheres are a suitable alternative to PVA or gelfoam for the treatment of hypervascular mediastinal tumors. Of note, microspheres smaller than 350 µm should be avoided for bronchial and intercostal artery embolization due to the risk of spinal cord ischemia [6].
Both of our cases were complicated by encasement of mediastinal structures found during operative resection; despite this complication, we believed that optimal resection results with less blood loss than previously published were achieved in both cases [2].
In conclusion, preoperative embolization of Castleman's disease and histologically similar hypervascular mediastinal tumors with Trisacryl gelatin (Biosphere Medical) microspheres may reduce intraoperative bleeding associated with excision of these vascular tumors.
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References
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- Greiner T, Armitage JO, Gross TG. Atypical lymphoproliferative diseases Hematology Am Soc Hematol Educ Program 2000:133-146.
- Ko SF, Ng SH, Hsieh MJ, et al. Castleman disease of the pleura: experience with eight surgically proven cases Ann Thorac Surg 2003;76:219-224.[Abstract/Free Full Text]
- Walter JF, Rottenburg RW, Cannon WB, et al. Giant mediastinal lymph node hyperplasia (Castleman's disease): angiographic and clinical features AJR Am J Roentgenol 1978;130:447-450.[Abstract]
- Williams HR, Millner PA, Coral A. Castleman's disease of the erector spinae muscle Skeletal Radiol 1998;27:637-640.[Medline]
- Safford SD, Lagoo AS, Mahaffey SA. Preoperative embolization as an adjunct to operative management of mediastinal Castleman disease J Pediatr Surg 2003;38:1-3.[Medline]
- Robert JH, Sgourdos G, Kritikos N, Didier D, Terraz S. Preoperative embolization of hypervascular Castleman's disease of the mediastinum Cardiovasc Intervent Radiol 2008;31:186-188.[Medline]
- Castleman B, Iverson L, Mendeudex VP. Localized mediastinal lymph node hyperplasia resmboling thymoma Cancer 1956;9:822-830.[Medline]
- Herrada J, Cabanillas FF. Multicentric Castleman's disease Am J Clin Oncol 1995;18:180-183.[Medline]
- Herrada J, Cabanillas F, Rice L, Manning J, Pugh W. The clinical behavior of localized and multicentric Castleman disease Ann Intern Med 1998;128:657-662.[Abstract/Free Full Text]
- McAdams HP, Rosado-de-Christenson M, Fishback NF, Templeton PA. Castleman disease of the thorax: radiologic features with clinical and histopathologic correlation Radiology 1998;209:221-228.[Abstract/Free Full Text]
- Chronowski GM, Ha CS, Wilder RB, et al. Treatment of unicentric and multicentric Castleman disease and the role of radiotherapy Cancer 2001;92:670-676.[Medline]
- Seirafi PA, Ferguson E, Edwards FH. Thoracoscopic resection of Castleman Disease Chest 2003;123:280-282.[Abstract/Free Full Text]
- Rena O, Casadio C, Maggi G. Castleman's disease: unusual inthrathoracic localization Eur J Cardiothorac Surg 2001;19:519-521.[Abstract/Free Full Text]
- Suster S, Moran CA. Micronodular thymoma with lymphoid B-cell hyperplasia: clinicopathologic and immunohistochemical study of eighteen cases of a distinctive morphologic variant of thymic epithelial neoplasm Am J Surg Pathol 1999;23:955-962.[Medline]
- Pelage J, Laurent A, Wassef M, et al. Uterine Artery Embolization in Sheep: Comparison of Acute Effects with Polyvinyl Alcohol Particles and Calibrated Microspheres Radiology 2002;224:436-445.[Abstract/Free Full Text]
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Abstract
Introduction
