Preoperative Transarterial Chemoembolization of Hepatic Inflammatory Pseudotumor-Like Follicular Dendritic Cell Sarcoma to Downstage to Resection

Hepatic inflammatory pseudotumor (IPT)–like follicular dendritic cell sarcoma (FDCS), usually associated with Epstein-Barr virus, is an exceptionally rare pathologic type of liver malignancy that constitutes <0.1% of all primary hepatic tumors. Diagnosis is hindered in most patients owing to its nonspecific clinical and imaging manifestations. Current treatment options for hepatic IPT-like FDCS include surgical resection, chemotherapy, and radiotherapy. Preoperative bland embolization has been reported for extrahepatic disease to limit intraoperative blood loss (1Sze D.Y. Shelton A.A. SIR 2008 annual meeting film panel case: Castleman disease complicated by follicular dendritic cell sarcoma.J Vasc Interv Radiol. 2008; 19: 1141-1144Abstract Full Text Full Text PDF PubMed Scopus (3) Google Scholar), but locoregional treatment for downstaging to resectability has not been reported. The purpose of this letter is to present the imaging and outcomes of a patient with hepatic IPT-like FDCS treated with drug-eluting embolic transarterial chemoembolization (DEE-TACE) as a bridge therapy, followed by hepatic resection. This report was approved by the authors’ institutional ethics committee. A 24-year-old woman who had a history of Hepatitis B virus infection but did not receive antiviral treatment presented with lumbar back pain initially without other associated symptoms. The results of complete blood cell count, liver function tests, blood urea nitrogen, creatinine, and tumor marker levels were normal. Contrast-enhanced computed tomography (CT) scan revealed a 13 × 10 × 10 cm dominant mass in the right liver (Fig 1). The tumor was a heterogeneous density solid when compared with adjacent liver parenchyma, and the margins of the tumor were well-defined with an extensive range of intratumoral necrosis. She was deemed as a poor surgical candidate because of the huge tumor burden and its unknown etiology. To control the progression of the tumor and reduce its size, the patient underwent DEE-TACE as the initial treatment. Angiography, guided by digital subtraction angiography, was performed before embolization to ascertain the arterial supply of the tumor, which was found to be supplied predominantly by the segment 7 and 8 arteries. No arteriovenous shunting was detected. A 2.4-F microcatheter (Renegade, Boston Scientific, Marlborough, Massachusetts) and a microwire were used to catheterize the tumor-feeding artery for superselective embolization. Drug-eluting microspheres (CalliSpheres, 300–500 μm; Suzhou Hengrui Galisheng Biomedical Technology, Suzhou, China), loaded with 40 mg epirubicin, were used as the chemoembolization agent. One vial of drug-eluting embolics suspended in 20 mL of contrast agent was delivered at a rate of 1 mL/min through the microcatheter into the tumor-supplying artery until near stasis. Subsequently, suspended gelatin sponge particles (diameter: 560–710 μm; Hangzhou Alicon Pharm, Zhejiang, China) were administered until stasis. Postembolization angiography was performed 5 minutes later, to confirm lack of residual tumor hypervascularity (Fig 2a, b). Of note, the patient underwent a percutaneous CT-guided needle biopsy of the tumor using an 18-gauge needle (Cook Medical) prior to the DEE-TACE procedure, which confirmed that the tumor was hepatic IPT-like FDCS by immunohistochemistry. One month after the DEE-TACE procedure, CT imaging revealed that the tumor had undergone considerable necrosis and slightly decreased in size, with dimensions reducing to 11.3 × 8.5 × 8.9 cm. The tumor was distinctly separated from the middle hepatic vein, which had not been clearly visible before DEE-TACE (Fig 3). The patient underwent laparoscopic right hepatectomy 2 months later. During the operation, the tumor and peritumoral regions in the abdomen were carefully inspected. No adverse events occurred during or after surgery. She was discharged 7 days after surgery without any adjuvant therapy. One month later, a follow-up CT scan showed no evidence of residual or recurrent disease (Fig 4). She became pregnant during a 17-month follow-up period, and an ultrasound scan examination at a local hospital showed no signs of tumor recurrence.Figure 4One month after surgical resection, no evidence of residual or recurrent disease was found on follow-up computed tomography imaging.View Large Image Figure ViewerDownload Hi-res image Download (PPT) Almost all of the dozens of previous publications about FDCS have been single case reports, but the association with other findings (ie, myasthenia gravis, hemolytic anemia, pemphigus, intestinal obstruction, and rectal bleeding) seems unique to this entity (2Saygin C. Uzunaslan D. Ozguroglu M. Senocak M. Tuzuner N. Dendritic cell sarcoma: a pooled analysis including 462 cases with presentation of our case series.Crit Rev Oncol Hematol. 2013; 88: 253-271Crossref PubMed Scopus (168) Google Scholar,3Tsunemine H. Akasaka H. Kusama T. et al.Hepatic follicular dendritic cell sarcoma favorably controlled by transcatheter arterial chemoembolization.Intern Med. 2010; 49: 2703-2707Crossref PubMed Scopus (7) Google Scholar). An earlier report found that preoperative embolization before resection may have facilitated surgery in a patient with Castleman disease complicated by FDCS (1Sze D.Y. Shelton A.A. SIR 2008 annual meeting film panel case: Castleman disease complicated by follicular dendritic cell sarcoma.J Vasc Interv Radiol. 2008; 19: 1141-1144Abstract Full Text Full Text PDF PubMed Scopus (3) Google Scholar). However, some patients with good liver function may not qualify for primary resection because of the size and/or vascular invasion. The case described in this letter demonstrated that DEE-TACE caused the tumor to shrink and retract away from the middle hepatic vein. This facilitated and simplified the surgical resection, increased the volume of future liver remnant, allowed for a complete (R0) resection of the tumor, and potentially decreased the risk of recurrence as well. This strategy may offer a new approach for the treatment of this rare disease.