Case report
Hepatobiliary & Pancreatic Diseases International
Pancreatic Castleman disease treated with laparoscopic distal pancreatectomy Filip Čečka, Alexander Ferko, Bohumil Jon, Zdeněk Šubrt, Petra Kašparová and Rudolf Repák Hradec Králové, Czech Republic
BACKGROUND: Castleman disease is an uncommon lymphoproliferative disorder most frequently occurring in the mediastinum. Abdominal forms are less frequent, with pancreatic localization of the disease in particular being extremely rare. Only seventeen cases have been described in the world literature.
Introduction
T
umors in the body and tail of the pancreas are often asymptomatic or may present with vague and indistinctive abdominal pain. A list of the most common diagnoses of masses in the body and tail METHOD: This report describes an interesting and unusual of the pancreas include adenocarcinoma, cystic tumors, case of pancreatic Castleman disease treated with laparoscopic and functioning or non-functioning neuroendocrine resection. tumors. Other findings such as Castleman disease are RESULTS: A 48-year-old woman presented with epigastric pain. CT scan showed a well-encapsulated mass on the ventral border quite rare. The authors report a rare case of pancreatic of the pancreas. Endosonography with fine needle aspiration Castleman disease treated with laparoscopic distal biopsy was performed. Biopsy showed lymphoid elements and pancreatectomy. structures of a normal lymph node. The patient was treated with laparoscopic distal pancreatectomy. The pancreas was transected with a Ligasure device and the pancreatic stump was secured with a manual suture. One year after surgery the patient was complaint-free and showed no signs of recurrence of the disease.
Case report
A 48-year-old woman presented with epigastric pain. The pain was temporary with prompt relief after CONCLUSIONS: Laparoscopic distal pancreatectomy is a analgesia. Contrast enhanced CT scan (Fig. 1) revealed feasible and safe method for the treatment of lesions in the body a well-encapsulated mass 37×37 mm on the ventral and tail of the pancreas. Transection of the pancreas with a border of the pancreas. The CT scan also showed Ligasure device offers the advantages of low bleeding and low suspicion of infiltration of the gastric wall, indicating risk of pancreatic fistula. an origin from the muscular layer of the gastric wall (Hepatobiliary Pancreat Dis Int 2013;12:332-334) (e.g. gastrointestinal stromal tumor (GIST)). Endosonography showed a mass 38×26 mm located between the KEY WORDS: Castleman disease; pancreas and gastric wall (Fig. 2). However, it could not pancreas; laparoscopic distal pancreatectomy; Ligasure
Author Affiliations: Department of Surgery (Čečka F, Ferko A, Jon B and Šubrt Z), Fingerland Department of Pathology (Kašparová P) and Second Department of Internal Medicine (Repák R), Faculty of Medicine and University Hospital Hradec Králové, Sokolská 581, 500 05 Hradec Králové, Czech Republic; Department of Field Surgery, Military Health Science Faculty, Hradec Králové, Defence University Brno, Třebešská 1575, 500 01 Hradec Králové, Czech Republic (Šubrt Z) Corresponding Author: Filip Čečka, MD, PhD, Department of Surgery, Faculty of Medicine and University Hospital Hradec Králové, Sokolská 581, 500 05 Hradec Králové, Czech Republic (Tel: 420-737-163931; Fax: 420-495-832026; Email: fi
[email protected]) © 2013, Hepatobiliary Pancreat Dis Int. All rights reserved. doi: 10.1016/S1499-3872(13)60053-3
Fig. 1. Contrast enhanced CT scan showing a well-encapsulated mass of 37×37 mm on the ventral border of the pancreas. The black arrow points at the tumor.
• Hepatobiliary Pancreat Dis Int,Vol 12,No 3 • June 15,2013 • www.hbpdint.com
Pancreatic Castleman disease
confirm the origin of the tumor. Fine-needle aspiration biopsy showed lymphoid elements, structures of a normal lymph node and structures of clear cylindrical epithelium without dysplasia, possibly originating from a benign mucinous tumor. After preoperative diagnosis, a differential diagnosis was made between a cystic tumor of the pancreas, GIST originating from the gastric wall, or an enlarged lymph node. Laparoscopic exploration and resection of the tumor were decided.
Fig. 2. Endosonography showing a mass of 38×26 mm located between the pancreas and gastric wall. The needle for the aspiration biopsy is visible in the tumor.
During the laparoscopic exploration, a lesser sac was opened through division of the gastrocolic ligament. A tumor was found on the ventral border of the pancreas; it did not cohere to the gastric wall (Fig. 3). Laparoscopic resection was made of the tail of the pancreas with the tumor. The pancreas between the body and tail was transected with a Ligasure device. Afterwards, the pancreatic stump was secured with a manual suture. In the postoperative course, a type A pancreatic fistula (according to International Study Group on Pancreatic Fistula) was observed.[1] No other complications in the postoperative course were noted and the patient was discharged from the hospital on the seventh postoperative day. A final histological examination showed a finding typical for Castleman disease: an enlarged intrapancreatic lymphatic node with angiofolicular hyperplasia – hyaline-vascular type (Fig. 4). The patient is now being followed up at our department and is complaint-free, showing no signs of recurrence of the disease one year after the surgery.
Discussion
Castleman disease is a relatively rare disorder characterized by benign proliferation of lymphoid tissue. Its precise incidence is unknown.[2] It was first described by Castleman et al in 1954.[3] There are two histological types of the disease.[4] The more frequent type is the hyaline-vascular type (85%-90% of cases), which is characterized by abnormal lymphoid follicles, numerous vessels, and wide fibrous septa. The disease is usually asymptomatic. The plasma-cell type is less frequent (10%-15%); it is characterized by large follicles with intervening sheets of plasma cells and few vessels. Castleman disease commonly occurs in the mediastinum (60%-70% of cases). Abdominal forms Fig. 3. Laparoscopic intraoperative image. The white arrow points are less frequent (10%-17%), and the majority of them at the tumor on the ventral border of the pancreas. are retroperitoneal.[5] Other locations are less common. Pancreatic Castleman disease is extremely rare, with only 17 cases having been described in the literature so far.[2, 4, 6-20] Little is known about the etiology of the disease.[2, 21] A recent report[22] suggest the association of Castleman disease with HHV-8 and HIV infections. However, the sources of immune activation in the HHV-8 and HIV negative patients are still unidentified. Other exogenous and endogenous factors may induce IL-6 secretion from B-lymphocytes. Local production of IL-6 may contribute to the characteristic B-cell proliferation [23] Moreover, in patients with Fig. 4. Histologically, the surgical specimen showing a lymphatic node and vascularization. multicentric Castleman disease, systemic symptoms with angiofolicular hyperplasia-hyaline-vascular type of Castleman may result from increased production and circulation of disease (Hematoxylin-eosin staining, original magnification ×40). Hepatobiliary Pancreat Dis Int,Vol 12,No 3 • June 15,2013 • www.hbpdint.com •
Hepatobiliary & Pancreatic Diseases International
IL-6.[23] The main issue in pancreatic Castleman disease lies in the establishment of clinical diagnosis. Imaging methods (ultrasonography, CT and MRI) have been shown to be helpful in the diagnosis; however, localized Castleman disease may be clinically and radiographically indistinguishable from other lymphoid and non-lymphoproliferative disorders.[4, 9] Castleman disease is not usually included in the list of possible diagnosis, one of the reasons may be its low incidence. CT scan often shows a solid mass with well-defined margins. Dense enhancement immediately after the application of contrast medium is seen. Percutaneous fine-needle aspiration biopsy is often not diagnostic in Castleman disease,[5, 21] which was true for our patient as well. The histological diagnosis of the disease is based on cell architecture, and therefore requires the study of the entire surgical specimen. Complete surgical resection is curative in unicentric forms of Castleman disease [24, 25] with very good prognosis after complete resection. Contributors: ČF proposed the study. JB and ŠZ wrote the first draft. FA, KP and RR collected and analyzed the data. All authors contributed to the design and interpretation of the study. ČF is the guarantor. Funding: The work was supported by Research Project MZO 00179906 from the Ministry of Health Care, Czech Republic. Ethical approval: Not needed. Competing interest: No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article.
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