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      Lymphoepithelial cyst of the pancreas: A case report and summary of imaging features of pancreatic cysts

      case-report

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          Highlights

          • A lymphoepithelial cyst (LEC) of the pancreas is a benign and rare lesion.

          • A pancreatic LEC is difficult to be diagnosed and differentiate from the malignancy preoperatively.

          • We summarized the imaging features of pancreatic cysts to differentiate from the malignancy.

          Abstract

          Introduction

          A lymphoepithelial cyst (LEC) of the pancreas is a benign and rare lesion that is difficult to diagnose preoperatively based on imaging studies.

          Presentation of case

          We report a case of a 49-year-old man who presented with weight loss and diarrhea. The serum carcinoembryonic antigen and carbohydrate antigen 19-9 levels were slightly elevated to 6.7 ng/mL (reference value <5.0 ng/mL) and 45 U/mL (reference value <37 U/mL), respectively. Computed tomography showed a large cystic mass with internal septa in the pancreatic tail. The cystic wall and the septa showed enhancement while the cystic contents remained unenhanced. Magnetic resonance imaging (MRI) demonstrated a multiple-ball-like lesion with low signal intensity on T1-weighted image and high signal intensity on T2-weighted image. Diffusion-weighted MRI showed high signal intensity in the central and iso-signal intensity in the peripheral portions of the cystic lesion. The cystic wall and septa showed high signal intensity, and the cystic contents showed low signal intensity on an enhanced MRI. Endoscopic ultrasonography showed a cyst with multiple high-echoic lesions in the pancreatic tail. A mucinous cystic neoplasm and branch duct intraductal papillary mucinous neoplasm were considered among the differential diagnoses, and we performed distal pancreatectomy with concomitant splenectomy and lymphadenectomy for both diagnostic and therapeutic purposes. Histopathological findings revealed that the cystic wall was lined by stratified squamous epithelium, and several lymphoid follicles and a few sebaceous glands were observed within the cystic wall without hair follicles, leading to the final diagnosis of an LEC.

          Conclusion

          We report a rare case of a pancreatic LEC, which was difficult to be diagnosed and summarize the imaging features of pancreatic cysts to differentiate from the malignancy preoperatively.

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          Most cited references19

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          The SCARE Statement: Consensus-based surgical case report guidelines.

          Case reports have been a long held tradition within the surgical literature. Reporting guidelines can improve transparency and reporting quality. However, recent consensus-based guidelines for case reports (CARE) are not surgically focused. Our objective was to develop surgical case report guidelines.
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            Cystic neoplasms of the pancreas.

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              Lymphoepithelial cysts of the pancreas: a report of 12 cases and a review of the literature.

              Lymphoepithelial cyst (LEC) of the pancreas is a rare lesion of undetermined pathogenesis that had been documented almost exclusively in males. The literature on this entity is limited to reports of single or small numbers of cases. Here is presented a clinicopathologic analysis of 12 patients with LEC, 4 of whom were female. The mean age of the patients was 56 years. Four patients presented with abdominal pain and nausea, but in two patients, the cysts were detected incidentally. Only one patient had a history of chronic pancreatitis, and another had a family member with pancreatic cancer. In one patient, a clinical diagnosis of pseudocyst was rendered, and the remaining patients were clinically thought to have cystic neoplasms. None of the patients had any identifiable immunosuppression, HIV positivity, autoimmune disorder (such as Sjogren syndrome) or lymphoma. Seven cysts were located in the head of the pancreas, and 5 were in the tail. The mean size was 4.8 cm (range, 1.2-17 cm). Five LECs were multilocular, three were unilocular; in others, the number of loculi was not recorded. All were "macrocystic" lesions. Two patients had two separate lesions, both in the tail of the pancreas. Histologically, all cases were characterized by cysts, some containing keratin, and lined by mature stratified squamous epithelium surrounded by dense lymphoid tissue, often with prominent follicles. In some areas, the lining epithelium had more cuboidal, flattened, or transitional appearance. Mucinous goblet-like cells were seen in one case. Acute inflammation was not seen. Four cases contained solid lymphoepithelial islands, a feature not previously described in LECs. No squamous metaplasia was identified in the uninvolved pancreatic tissue and no epithelial elements were identified in peripancreatic lymph nodes. In summary, LEC of the pancreas is a rare but distinctive lesion that may be seen in the tail of the organ where most cystic pancreatic neoplasms are encountered. In contrast to the impression from the literature, LECs may also develop in females and, therefore, should be considered in the clinical differential diagnosis of mucinous cystic neoplasms that affect a similar age group. LECs are not associated with the clinical syndromes that are seen with their analogues in the salivary glands.
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                Author and article information

                Contributors
                Journal
                Int J Surg Case Rep
                Int J Surg Case Rep
                International Journal of Surgery Case Reports
                Elsevier
                2210-2612
                30 January 2019
                2019
                30 January 2019
                : 55
                : 192-195
                Affiliations
                [a ]Department of Gastroenterological Surgery, Hiroshima Prefectural Hospital, Japan
                [b ]Department of Gastroenterological and Transplant Surgery, Applied Life Sciences, Institute of Biomedical & Health Sciences, Hiroshima University, Japan
                [c ]Department of Pathology Clinical Laboratory, Hiroshima Prefectural Hospital, Japan
                [d ]Department of Gastroenterology, Hiroshima Prefectural Hospital, Japan
                Author notes
                [* ]Corresponding author at: Department of Gastroenterological Surgery, Hiroshima Prefectural Hospital, 1-5-54, Ujina-kanda, Minami-ku, Hiroshima, 734-8530, Japan. oshita-akihiko@ 123456umin.ac.jp
                Article
                S2210-2612(19)30033-1
                10.1016/j.ijscr.2019.01.022
                6374521
                30763861
                a8aedbd2-740f-4cf9-8c82-08010757a45b
                © 2019 Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd.

                This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/).

                History
                : 26 November 2018
                : 15 January 2019
                : 22 January 2019
                Categories
                Article

                lec, lymphoepithelial cyst,mri, magnetic resonance imaging,mcn, mucinous cystic neoplasm,ipmn, intraductal papillary mucinous neoplasm,ct, computed tomography,cea, carcinoembryonic antigen,ca19-9, carbohydrate antigen 19-9,eus, endoscopic ultrasonography,ercp, endoscopic retrograde cholangiopancreatography,lymphoepithelial cyst,imaging,features,pancreatic,cyst

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