Lymphoepithelial cyst in the pancreas: A case report

A case of lymphoepithelial cyst in the pancreas was reported. A 64-year-old man without any specific compalints was found to have a cystic lesion in the anterior portion of the pancreas, as revealed by ultrasonography of the abdomen at an annual medical examination in 1988. This was dissected easily from the pancreas. Histologically, it was diagnosed to be a benign lymphoepithelial cyst in the pancreas. Cysts of this type are rare, and their histogenesis is also not well understood.     

Lymphoepithelial cyst of the pancreas associated with elevated CA 19-9 levels

An extremely rare case of a lymphoepithelial cyst associated with persistent elevation of serum carbohydrate antigen (CA) 19-9 levels is described. A 72-year-old man was incidentally found to have a cystic tumor in the uncus of the pancreas and to have a high serum CA 19-9 level. At 2-year follow up, the tumor size had gradually increased and the high CA 19-9 level persisted. Because the malignant potential of the tumor could not be ruled out, a laparotomy was performed. The tumor was excised from the pancreas. On pathological examination, the tumor was proven to be a lymphoepithelial cyst. After the operation, the patient’s serum CA 19-9 level decreased to the normal range. Lymphoepithelial cysts of the pancreas are a rare, benign entity. They are true pancreatic cysts, characterized by a mature, keratinizing, squamous lining surrounded by lymphoid tissue. Because the treatment options and the prognosis of these entities are quite different from those of other cystic neoplasms of the pancreas, preoperative differential diagnosis is the main issue. Thus, although they are rare, lymphoepithelial cysts should be considered in the differential diagnosis of cystic tumors of the pancreas.     

Lymphoepithelial cyst of the pancreas with sebaceous differentiation

We recently encountered a patient with a lymphoepithelial cyst of the pancreas with sebaceous differentiation. We sought to compare the characteristics of this patient with those previously reported in order to foster a keener understanding of this rare clinical entity. After reviewing the present patient's case in detail, we conducted a comprehensive review of the English-language literature and analyzed the clinical characteristics of reported cases of lymphoepithelial cysts. Our patient was an asymptomatic 60-year-old man who presented with an incidental finding of a cystic lesion in the tail of the pancreas documented by computed tomography. The cyst was enucleated, and was found to contain keratinized material. It was lined by squamous epithelium with small sebaceous glands, and surrounded by lymphoid tissue with germinal centers. Of 33 reported cases, only 6 (18%) contained sebaceous glands. In all patients who underwent operation, the cysts were easily resected, and the outcome was favorable. Lymphoepithelial cyst of the pancreas is rare, and may be difficult to differentiate from cystic neoplasms preoperatively. Therefore resection is indicated. The diagnosis, however, can be confirmed by careful histologic review, and the prognosis is excellent. Received: March 8, 1999 / Accepted: October 22, 1999     

Lymphoepithelial cyst of the pancreas

A case of lymphoepithelial cyst (LEC) of the pancreas is presented. A 48-year-old man complaining of general fatigue was found to have a heterogeneous water-dense mass protruding from the surface of the pancreas on plain computed tomography (CT). Dynamic CT disclosed septa within the mass. Magnetic resonance imaging (MRI) showed a hypoiintense mass on T1-weighted imaging, and a hyperintense mass on T2-weighted imaging. MRI with gadolinium enhancement revealed septa within the mass. Endoscopic ultrasonography showed septa and fine echogenic structures within the cystic echoic lesion. Endoscopic retrograde pancreatecraphy showed a normal duct system. Distal pancreatectomy with splenectomy was performed, with a suspected diagnosis of cystic neoplasms of the pancreas. Histopathologic examination disclosed LEC of the pancreas. Our case suggests that LEC should be considered in the differential diagnosis of cystic neoplasms of the pancreas.     

A solid cystic tumor of the pancreas with ossification and possible malignancy, coexisting nonfusion of the pancreatic ducts

We report the case of a 34-year-old woman with a solid cystic tumor (SCT) of the pancreas accompanied by ossification and possible malignancy, coexisting nonfusion of the pancreatic ducts. There was a 24 x 29 x 33-mm mass with a prominent calcified lesion in the tail of the pancreas detected by abdominal ultrasonography, computed tomography, and magnetic resonance imaging. There were no distal metastases detected. Endoscopic retrograde pancreatography revealed nonfusion of the pancreatic ducts. The resected tumor consisted of solid and cystic components. The tumor was not encapsulated and included a severely ossified lesion inside. On microscopy, the tumor cells were small, eosinophilic, and proliferated in a solid or pseudo-papillary pattern. The tumor cells infiltrated into the surrounding normal pancreas parenchyma and invaded part of the mesentery. The immunostaining was positive for alpha-1-antitrypsin, neuron-specific enolase, vimentin, and chromogranin A. In the literature, only a few cases of SCT of the pancreas described ossification. As far as we know, only three cases of SCT of the pancreas, which demonstrated nonfusion of the pancreatic ducts, have been reported. Thus, SCT of the pancreas with ossification, possible malignancy, and coexisting nonfusion of the pancreatic ducts is extremely rare.     

Solitary true cyst of the pancreas in an adult: report of a case

Background: Solitary true cyst of the pancreas is rare in adults, and the differential diagnosis of cystic lesions of the pancreas is challenging. Aim of the Study: To describe a solitary true cyst of the pancreas in an adult and discuss the differential diagnosis. Methods: A 50 yr old woman presented with a mass lesion in the right upper quadrant of the abdomen. Abdominal computed tomography showed a cystic lesion, with a maximum diameter of 12 cm, between the inferior surface of the liver and the ascending colon. The cyst was homogenous and had smooth edges. On magnetic resonance imaging, a unilocular cyst was seen that was low intensity on T1-weighted images and very high intensity on T2-weighted images. No connection between the cyst and the pancreatic ductal system was demonstrated on endoscopic retrograde cholangiopancreatography. Laparotmy was performed with a presumptive diagnosis of cystic tumor of the pancreas. Results: On pathologic examination, the cyst was serous and was lined with a single layer of normal cuboidal epithelium that was periodic acid Schiff stain negative. Meticulous examination failed to identify honeycomb-like microcysts characteristic of serous cystadenoma. The final diagnosis was a solitary true cyst of the pancreas. Conclusion: The differential diagnosis of solitary true cyst from other cystic lesion of the pancreas usually is based on histology. Particular care must be taken to distinguish this lesion from macrocystic serous cystadenoma.     

Mucinous cystadenoma of the pancreas 17 years after excision of gallbladder because of a choledochal cyst

A 56-year-old woman who had undergone excision of the gallbladder because of a choledochal cyst had a tumorous lesion of the pancreas identified by upper abdominal ultrasonography, but an operation was not carried out, because there was no apparent increase in the cystic mass and no elevation of serum tumor markers. In October 2001, she was admitted to our hospital to check for malignancy because of elevated levels of the tumor marker Dupan-2. Abdominal enhanced computed tomography and upper abdominal ultrasonography revealed a large multilocular cystic mass in the body to tail of the pancreas. Endoscopic retrograde cholangiopancreatography showed elongation of the common duct that communicates with the common bile duct and the main pancreatic duct, indicating an anomalous arrangement of the biliary and pancreatic duct system. No apparent communications between the cystic mass and the main pancreatic duct were observed. In January 2002, the patient underwent a spleen-preserving distal pancreatectomy, and histopathological and immunohistochemical examinations led to the diagnosis of pancreatic mucinous cystadenoma with ovarian-like stroma. The mucinous cystadenoma was detected 17 years after the operation for the choledochal cyst. To the best of our knowledge, no documented case reports of mucinous cystadenoma of the pancreas associated with a choledocal cyst have been reported to date. We present here the first case report of pancreatic mucinous cystadenoma occurring in the body to tail of the pancreas, associated with a choledocal cyst.     

Epidermoid cyst in an intrapancreatic accessory spleen: a case report

Ectopic splenic tissue in the abdominal cavity is a common entity, with a reported incidence of 10% in the general population. However, an intrapancreatic accessory spleen is a rare disease, and moreover cyst formation in it is exceedingly rare. A 58-year-old woman with a 25-mm multilocular cyst in the tail of the pancreas detected incidentally by ultrasonography was admitted for further evaluation. Because malignancy could not be ruled out, a spleen-preserving distal pancreatectomy was performed. The cut surface of the surgical specimen showed a multilocular cyst surrounded by brown solid tissue resembling normal spleen. Pathological examination revealed it was stratified squamous epithelium and was surrounded by splenic tissue. The final pathological diagnosis was epidermoid cyst in an accessory spleen in the pancreas. This cyst has no characteristic features on diagnostic imaging. Consequently, it is not possible to make a definite preoperative diagnosis in most cases. Epidermoid cyst in intrapancreatic splenic tissue is another lesion to be considered in the differential diagnosis of pancreatic tail tumors.     

Lymphoepithelial cyst in the pancreas: a case report

A case of lymphoepithelial cyst in the pancreas was reported. A 64-year-old man without any specific complaints was found to have a cystic lesion in the anterior portion of the pancreas, as revealed by ultrasonography of the abdomen at an annual medical examination in 1988. This was dissected easily from the pancreas. Histologically, it was diagnosed to be a benign lymphoepithelial cyst in the pancreas. Cysts of this type are rare, and their histogenesis is also not well understood.     

Intracystic hemorrhage of a simple liver cyst mimicking a biliary cystadenocarcinoma

Simple liver cysts are rarely complicated by intracystic hemorrhage. We encountered a case of simple liver cyst that was morphologically similar to biliary cystadenocarcinoma, which was complicated by asymptomatic intracystic hemorrhage and successfully treated by right lobectomy. A large cystic lesion of the liver was detected in a 57-year-old woman during a mass screening health check. Abdominal ultrasonography (US) revealed that the cystic lesion, containing many hyperechoic papillary structures, occupied almost the entire region of the right hepatic lobe. In addition, a round mural nodule, measuring approximately 5 cm in diameter, was detected in the cystic wall. Abdominal computed tomography (CT) revealed that the inner part of the cystic lesion showed homogeneous low density, but CT did not show the round nodule detected by US. On T1-weighted sequence of magnetic resonance imaging (MRI), the lesion showed homogeneous high signals, together with a low-signal tumorous lesion in the cystic wall. T2-weighted sequence of MRI showed unhomogeneous high signals, together with high signals in the tumorous part. These findings did not exclude the possibility of a malignant cystic tumor, such as biliary cystadenocarcinoma. Therefore, right lobectomy was performed. Histological examinations of resected tissue specimens revealed that the lesion was a liver cyst containing a large amount of blood clot, and that the tumorous lesion detected by US and MRI was a large mass of blood clot which was partly liquefied. This case indicates the diagnostic importance of the morphological discordance between CT and US or MRI findings for liver cyst containing a large amount of blood clot. Received: May 7, 2001 / Accepted: December 27, 2001 Reprint requests to: Y. Kitajima     

Lymphoepithelial cyst of the pancreas: a new case report

We report the case of a lymphoepithelial cyst of the pancreas discovered by chance on imaging in a 54-year old man. CT-scan showed a 10 cm hypodense multilocular cystic tumor of the pancreatic isthmus. Fine-needle aspiration did not provide further information. Due to the lack of preoperative diagnosis and mostly because it was not known if the cyst was malignant or benign, the patient underwent a cephalic duodenopancreatectomy. Lymphoepithelial cyst of the pancreas is a rare benign lesion which is difficult to diagnose before surgery. Histologically, the cyst wall is lined by mature keratinizing squamous epithelium and a distinct surrounding lymphoid tissue layer. The cysts are filled with keratin plugs that are not always visualized on imaging. Cytological and histological analysis of fine-needle aspiration material if the sample material is sufficient may help avoid extensive surgery.     

Epidermoid cyst originating from an intrapancreatic accessory spleen

We report a rare case of an epidermoid cyst originating from an intrapancreatic accessory spleen, in a 40-year-old Japanese man with no clinical symptoms. A cystic tumor in the pancreatic tail was detected incidentally by abdominal ultrasonography. The patient was referred to the KKR Tachikawa Hospital for further examination of the tumor. Preoperative imaging findings suggested that the tumor was an epidermoid cyst originating from an intrapancreatic accessory spleen. On both pre-and post-contrast computed tomography and magnetic resonance images, the solid compartment of the tumor had the same X-ray attenuation and intensity as the spleen. Upon surgical excision, the mass consisted of solid and cystic components that were macroscopically evident on the preoperative images. Microscopic analysis revealed that the solid component was an accessory spleen in the pancreatic tail, whereas the cystic component was lined with stratified epithelium representative of an epidermoid cyst. This is the thirteenth report (in English) of an epidermoid cyst originating from an intrapancreatic accessory spleen, and the first case to be diagnosed prior to surgery.     

A giant retention cyst of the pancreas (cystic dilatation of dorsal pancreatic duct) associated with pancreas divisum

We describe a rare case of pancreas divisum associated with a giant retention cyst (cystic dilatation of the dorsal pancreatic duct), presumably formed following obstruction of the minor papilla. The patient was treated by pancreatico(cysto)jejunostomy. A 50-year-old man was admitted with complaints of increasing upper abdominal distension and body weight loss. There was no previous history of pancreatitis, gallstones, drinking, or abdominal injury. An elastic-hard tumor-like resistance was palpable in the upper abdomen. Computed tomography and ultrasound (US) examinations revealed a giant cystic lesion expanding from the pancreas head to the tail. Endoscopic retrograde cholangiopancreatography findings showed a looping pancreatic duct which drained only the head and uncinate process of the pancreas to the main papilla. A US-guided puncture to the cystic lesion revealed that the lesion continued to the main pancreatic duct in the tail of pancreas. The lesion was connected to a small cystic lesion, which was located inside the minor papilla, and ended there. The amylase level in liquid aspirated from the cyst was 37 869 IU/l, and the result of cytological examination of the liquid showed class II. A pancreatico(cysto)jejunostomy was performed, with the diagnosis being pancreas divisum associated with a retention cyst following obstruction of the minor papilla. The histological findings of a specimen from the cyst wall revealed that the wall was a pancreatic duct covered with mildly inflammatory duct epithelium; there was no evidence of neoplasm. The patient is currently well, and a CT examination 2 years after the operation showed disappearance of the cyst and normal appearance of the whole pancreas. Received: April 24, 2001 / Accepted: September 14, 2001     

Case of lymphoepithelial cyst of the pancreas]

We recently experienced a case of lymphoepithelial cyst of the pancreas, which is a rare benign pancreatic cystic tumor. A 59-year-old-man was pointed out as having a pancreatic tumor based on abdominal ultrasonography findings obtained during a medical check-up. Thereafter, abdominal ultrasonography, CT, and MRI studies revealed the presence of a multi-cystic tumor in the pancreas body to tail measuring 45 mm in size which had no infiltrating tendency. The operative findings showed a yellowish-white colored cystic tumor covered with a thin capsule, which could be easily extirpated from the pancreas. The dissected tumor, which contained a curd-like material and a condensed milky liquid, was diagnosed to be a lymphoepithelial cyst of the pancreas based on the pathological findings. Similar reports of lymphoepithelial cysts arising in the pancreas have recently been increasing.     

Very high CEA level in a large pancreatic cyst: Is it a surgical indication by itself?

BackgroundPancreatic mucinous cystic lesions might develop malignancy if untreated, or could harbor malignancy at the time of the diagnosis. Many reports stated that cyst fluid carcinoembryonic antigen is an accurate diagnostic marker of pancreatic mucinous cysts.MethodsA man with a incidental pancretic cystic lesion of 35 mm in diameter was admitted to our Department. CT and EUS did not reveal solid components, main duct was not dilated and cyst fluid CEA was very high (1445 ng/ml).ResultsThe patient underwent a pancreatoduodenectomy and the surgical specimen showed a pseudocyst with columnar mucinous epithelium, consistent with low-grade PanIN.ConclusionsIs it possible that the mucinous epithelium of panIN was responsible for the unexpectedly high CEA value?Clinicians should be aware of the usefulness of the CEA level in cystic fluid but even a very high CEA value should not be considered by itself to be evidence of a mucinous lesion.     

Undifferentiated (anaplastic) carcinoma of the pancreas showing sarcomatous change and neoplastic cyst formation

Summary Conclusion Pancreatic undifferentiated (anaplastic) carcinoma contained two components: sarcomatous change of spindle-cell type and a neoplastic cyst. Immunohistochemical analysis suggested that both the sarcomatous and the cystic portions were of epithelial origin. This case showed a “dual” differentiation both into the sarcomatous change and into the cystic lesion. Background Pancreatic sarcoma or sarcomatous change is very rate. The origin of the sarcomatous change is unknown. On the other hand, pancreatic adenocarcinoma sometimes shows necrosis and forms a cystic lesion during its growth, but a neoplastic cyst formation is very rare except for cystadenomas. Methods We report a case of pancreatic undifferentiated (anaplastic) carcinoma associated with sarcomatous change and neoplastic cyst formation. Clinicopathological and immunohistochemical analyses were performed. Results A 75-yr-old male was admitted because of low back pain and body weight loss. He died 7 d after admission presenting obstructive jaundice. Autopsy disclosed that the large mass was a poorly differentiated ductal adenocarcinoma, which mostly consisted of sarcomatous components of spindle-cell type. The cystic lesion was a neoplastic cyst with the wall composed of epithelial tumor cells. There was no necrosis or hemorrhage in the cystic cavity. Immunohistochemical analyses suggested that both the tumor and the cystic lesion were of epithelial origin. Sarcomatous changes were recognized also in the metastatic lesions in the liver and lymph nodes. The tumor is considered to be labeled undifferentiated (anaplastic) carcinoma of the pancreas.     

Lymphoepithelial cyst of the pancreas: report of a case

The aim of this report is to describe the clinical and pathological features of a lympoepithelial cyst of the pancreas and to recommend fine-needle aspiration biopsy for the differential diagnosis of this lesion. A 55-year-old man was incidentally diagnosed as having a pancreatic tumor by abdominal ultrasonography. A hypoechoic cystic lesion was detected on the surface of the pancreatic body. A computed tomography revealed a cystic lesion surrounded by a smooth, flat wall. Magnetic resonance imaging revealed that the lesion was a low-intensity mass on the T1-image and a slightly high-intensity mass on the T2-image. The lesion was diagnosed as a benign cystic tumor, and enucleation of the tumor was scheduled. The fine needle aspiration biopsy was performed after laparotomy, and cytology of the contents was negative for malignant cells. The postoperative pathological diagnosis was a lymphoepithelial cyst of the pancreas. This cyst is an unusual but benign mass that requires minimal surgery.     

Intrapancreatic gastric duplication cyst mimicking pancreatic cystic tumor

Cystic lesions of the pancreas are sometimes difficult to diagnose. We report a case of a gastric duplication cyst (GDC) of the pancreas in an adult. A 45-year-old woman was admitted to our department for the investigation of anemia. Abdominal ultrasonography revealed a large cystic lesion, measuring about 40 × 70 mm with calcification in the tail of the pancreas. Contrast-enhanced computed tomography of the abdomen revealed a non-enhanced cystic lesion with non-enhanced wall. Endoscopic retrograde pancreatography revealed a mild extended main pancreatic duct and an aberrant pancreatic duct, but there was no communication with the cyst. We could not deny the malignancy, so distal pancreatectomy was performed. The pathological examination revealed that the inner wall of the cyst consisted of columnar epithelium and smooth muscle layer. Immunohistochemical analysis revealed the columnar epithelium to be immunopositive for cytokeratin 7 (CK7) and immunonegative for cytokeratin 20 (CK20) and the glands to be immunonegative for CK 7 and immunopositive for CK 20. Therefore, a diagnosis of GDC of the pancreas was made. On imaging, a GDC usually mimics a pancreatic pseudocyst or a cystic neoplasm of the pancreas. Therefore, even though it is rare, a GDC should be considered in the differential diagnosis of cystic tumors of the pancreas.     

Lymphoepithelial cyst of the pancreas]

A 66-year-old woman presented with abdominal pain and weight loss. Ultrasonography and computed tomography demonstrated a cystic lesion of the pancreas. After surgical resection, the patient's symptoms disappeared. Microscopic examination of the cyst lining showed mature, keratinizing squamous epithelium, surrounded by lymphoid tissue. Only three cases of this type of lesion, called "lymphoepithelial cyst", have been described previously. Histogenesis of this particular type of cyst is not well know, but can be histologically differentiated from other pancreatic cysts.     

A case of a mucin-producing bile duct tumor diagnosed over the course of 6 years

We report a case of a mucin-producing intraductal papillary neoplasm of the intrahepatic bile duct (M-IPNB) diagnosed over a period of 6 years. A 64-year-old man underwent follow-up evaluations for an abdominal aortic aneurysm at our hospital. In 2009, a computed tomography (CT) scan revealed a simple hepatic cyst in segment 3 of the liver. Annual CT scans initially showed almost no change in the size or shape of the cyst. The cystic lesion, which measured 5 cm in 2014, had increased to 11 cm by 2015, and a solid component was detected within the cyst. A biliary cystic tumor was suspected and we performed a left lateral hepatectomy. Pathological examination showed that the papillary lesion in the cyst included adenocarcinoma and adenoma components. We diagnosed M-IPNB in 2015. Identification of the solid component of the cyst, as well as an increase in cyst diameter in the image analyses, was critical for diagnosis of M-IPNB.