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Yasrab M, Kwak SJ, Khoshpouri P, Fishman EK, Zaheer A. Misdiagnosis of pancreatic intraductal papillary mucinous neoplasms and the challenge of mimicking lesions: imaging diagnosis and differentiation strategies. Abdom Radiol (NY) 2025; 50:2241-2257. [PMID: 39327307 DOI: 10.1007/s00261-024-04551-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/19/2024] [Revised: 08/20/2024] [Accepted: 08/25/2024] [Indexed: 09/28/2024]
Abstract
The rising prevalence of pancreatic cystic lesions (PCLs), particularly intraductal papillary neoplasms (IPMNs), has been attributed to increased utilization of advanced imaging techniques. Incidental detection of PCLs is frequent in abdominal CT and MRI scans, with IPMNs representing a significant portion of these lesions. Surveillance of IPMNs is recommended due to their malignant potential; however, their overlapping imaging features with benign entities can lead to misdiagnosis, overtreatment, and overutilization of healthcare resources. This paper aims to highlight and differentiate lesions often mistaken for IPMNs, providing insight into their imaging characteristics, diagnostic challenges, and distinctive features while highlighting the incidence of wrong diagnosis for these lesions. These lesions include serous cystadenomas, cystic pancreatic neuroendocrine tumors, mucinous cystic neoplasms, lymphoepithelial cysts, duodenal diverticula, pancreatic schwannomas, chronic pancreatitis, retention cysts, intrapancreatic accessory spleens, pancreatic lipomas, choledochal cysts, and others. Utilizing various imaging modalities, including contrast-enhanced CT, MRI, and EUS, alongside histological and molecular analyses, can aid in accurate diagnosis and appropriate management. Understanding these mimicry scenarios is crucial to avoid unnecessary surveillance, interventions, and the burden they place on both patients and healthcare systems. Improved recognition of these lesions can lead to better patient outcomes and resource allocation.
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Affiliation(s)
- Mohammad Yasrab
- Johns Hopkins Hospital, 1800 Orleans St., Baltimore, MD, 21287, USA.
| | - Stephen J Kwak
- Johns Hopkins Hospital, 1800 Orleans St., Baltimore, MD, 21287, USA.
| | | | - Elliot K Fishman
- Johns Hopkins Hospital, 1800 Orleans St., Baltimore, MD, 21287, USA
| | - Atif Zaheer
- Johns Hopkins Hospital, 1800 Orleans St., Baltimore, MD, 21287, USA
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2
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Ciers P, Vanderhaeghe D, Vansteenkiste F, Moubax K, Vanooteghem S, Vanneste A, Van Moerkercke W. Lymphoepithelial cysts of the pancreas: case report and review of the literature. Acta Chir Belg 2023; 123:550-554. [PMID: 35249466 DOI: 10.1080/00015458.2022.2050032] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/27/2021] [Accepted: 03/02/2022] [Indexed: 11/01/2022]
Abstract
BACKGROUND Lymphoepithelial cysts (LECs) of the pancreas are a rare type of true pancreatic cysts and represent an estimated 0.5% of all pancreatic cystic lesions. They are benign lesions and have no malignant potential. However, they are hard to differentiate from malignant lesions because their imaging and clinical presentation vary greatly. Seeing as these are benign lesions which are increasingly found incidentally during imaging for other indications, correct diagnosis is important to prevent unnecessary intervention and morbidity. CASE REPORT We report the case of a 41-year-old female who presented with abdominal discomfort, bloating and dyspepsia. An abdominal computed tomography (CT) showed a large mass in the left fossa. We describe the diagnostic and therapeutic measures taken in this case. METHODS We reviewed the literature for common features of the LEC. We grouped common imaging and histological features of the LEC of the pancreas to provide easily identifiable characteristics to facilitate diagnosis. For the review, we focused on papers, mostly case reports, presenting these common characteristics. We also reviewed the literature for key topics that should be taken into account when considering therapeutic interventions in a patient with a possible diagnosis of a LEC. CONCLUSION Cysts of the pancreas are increasingly identified due to widespread use and improved resolution of cross-sectional imaging. To obtain the correct diagnosis, it is sometimes necessary to combine advanced imaging, i.e. CT and MRI-imaging, and endoscopic ultrasound with fine needle aspiration (EUS/FNA), while CA 19-9 also has diagnostic value. We summarize all diagnostic characteristics in a table for ease of use. Furthermore we summarized possible therapeutic interventions.
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Affiliation(s)
- Philippe Ciers
- Department of Vascular Surgery, Klinikum Westmunsterland GmbH, Bocholt, Germany
| | | | | | - Kim Moubax
- Department of Gastroenterology, AZ Groeninge, Kortrijk, Belgium
| | - Sofie Vanooteghem
- Department of Gastroenterology, OLV van Lourdes Hospital, Waregem, Belgium
| | - Alain Vanneste
- Department of Pathological Anatomy, AZ Groeninge, Kortrijk, Belgium
| | - Wouter Van Moerkercke
- Department of Gastroenterology, AZ Groeninge, Kortrijk, Belgium
- Department of Gastroenterology and Hepatology, University Hospitals Leuven, UZ Leuven, Leuven, Belgium
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Iguchi T, Shimizu A, Kubota K, Notake T, Sugenoya S, Hosoda K, Yasukawa K, Hayashi H, Kobayashi R, Soejima Y. Lymphoepithelial cyst mimicking pancreatic cancer: a case report and literature review. Surg Case Rep 2021; 7:108. [PMID: 33914178 PMCID: PMC8085173 DOI: 10.1186/s40792-021-01191-x] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/20/2021] [Accepted: 04/25/2021] [Indexed: 11/30/2022] Open
Abstract
Background Pancreatic lymphoepithelial cyst (LEC) is a rare nonmalignant cyst consisting of a benign collection of keratinizing squamous epithelial cells with lymphoid tissue. Diagnosing LEC preoperatively is considered difficult because of its non-specific clinical features; therefore, LEC is generally treated the same as a malignant tumor. Case presentation Our case was a 65-year-old man who underwent pancreatoduodenectomy 3 years previously for carcinoma arising from the ampulla of Vater. A pancreatic mass in the remnant pancreatic tail was detected through follow-up abdominal contrast-enhanced computed tomography (CT). This revealed two adjacent ring-enhanced masses that had been in tight contact with the left diaphragm and were enlarged. The tumors had high signal intensity in diffusion-weighted images of magnetic resonance imaging, and fluorodeoxyglucose-positron emission tomography (FDG-PET) showed abnormal uptake (standardized uptake value maximum: 17.4). Therefore, we conducted a partial resection of the remnant pancreas with concomitant resection of the left diaphragm. Microscopically, one of the tumors revealed rare fragments of apparently benign squamous epithelium on a background of keratinous debris, cyst contents, and scattered lymphocytes, and the adjacent mass revealed infiltrated neutrophils. The histopathological diagnosis was an LEC with chronic abscess. The patient recovered uneventfully and was discharged on postoperative day 10. Conclusions We reported a rare case of LEC with chronic abscess that was positively visualized on FDG-PET. When a pancreatic malignancy cannot be excluded, surgical resection is considered inevitable.
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Affiliation(s)
- Takuya Iguchi
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Akira Shimizu
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan.
| | - Koji Kubota
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Tsuyoshi Notake
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Shinsuke Sugenoya
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Kiyotaka Hosoda
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Koya Yasukawa
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Hikaru Hayashi
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Ryoichiro Kobayashi
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
| | - Yuji Soejima
- Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, Nagano, 390-8621, Japan
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Imaging features and pathological evaluation by EUS-FNA enable conservative management in patient of lymphoepithelial cyst of the pancreas: a case report. Clin J Gastroenterol 2020; 14:370-374. [PMID: 33063294 DOI: 10.1007/s12328-020-01240-2] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/29/2020] [Accepted: 09/10/2020] [Indexed: 02/07/2023]
Abstract
Pancreatic lymphoepithelial cysts (LECs) are rare cystic lesions filled with a keratinous substance and lined by squamous epithelium with underlying lymphoid tissue. Because pancreatic LECs are entirely benign, correct preoperative diagnosis is important to avoid unnecessary surgery. However, the imaging features of pancreatic LECs are not specific and preoperative diagnosis has proven difficult. A pancreatic mass was incidentally detected through abdominal ultrasonography in a 63-year-old male presenting without any symptoms. Computed tomography showed an exophytic cystic lesion in the pancreatic head. The lesion had heterogeneous high signal intensity with partial low intensity on T2-weighted magnetic resonance imaging (MRI) and high signal intensity on diffusion MRI. Endoscopic ultrasound (EUS) examination showed an encapsulated cystic lesion with relatively homogenous and highly echoic contents. EUS-guided fine-needle aspiration (EUS-FNA) revealed caseous appearance and rare fragments of apparently benign squamous epithelium on a background of keratinous debris, cyst contents, and scattered lymphocytes. We diagnosed a pancreatic LEC and opted for conservative management without surgery. Pathological evaluation based on images obtained through EUS-FNA showed macro- and microscopic features that were critical to determining the management strategy. In conclusion, the imaging and pathological features of pancreatic LECs can inform preoperative diagnosis, which may enable conservative management.
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Farooq A, Evans JJ, Hagen CE, Hartley CP. Lymphoepithelial cyst of the pancreas: A challenging diagnosis on fine needle aspiration. Ann Diagn Pathol 2020; 49:151603. [PMID: 32949892 DOI: 10.1016/j.anndiagpath.2020.151603] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/30/2020] [Accepted: 08/17/2020] [Indexed: 11/27/2022]
Abstract
Lymphoepithelial cysts (LECs) of the pancreas are rare, benign pancreatic cysts comprising approximately 0.5% of all pancreatic cysts. They occur predominantly in men in the 5th and 6th decades of life. LECs are true cysts lined by stratified squamous epithelium with adjacent subepithelial lymphoid tissue. They range in size from 1.2 to 17 cm (mean size 4.6 cm) and can arise in any part of the pancreas. 1 LEC resembles other benign and malignant pancreatic cysts clinically and radiologically. The cytomorphologic features of LECs have been described in a small number of case reports and it has been indicated that features may overlap with other benign and malignant pancreatic lesions. Herein, we report clinical, radiological, cytological and histopathological features of a pancreatic LEC in a 62-year-old male.
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Affiliation(s)
- Ayesha Farooq
- Department of Pathology, Medical College of Wisconsin, Milwaukee, WI, USA
| | - John J Evans
- Department of Pathology, Medical College of Wisconsin, Milwaukee, WI, USA
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A Rare Presentation of Pancreatic Lymphoepithelial Cyst: A Case Report and Review. Case Rep Med 2020; 2020:4590758. [PMID: 32099546 PMCID: PMC7040412 DOI: 10.1155/2020/4590758] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/29/2019] [Accepted: 01/18/2020] [Indexed: 11/21/2022] Open
Abstract
Pancreatic lymphoepithelial cyst (LEC) is a rare, benign collection of keratinizing squamous epithelial cells encapsulated by lymphoid tissue. Because of its limited data and nonspecific features that can mimic malignant lesions, LECs can lead to unnecessary operations. A 62-year-old male with a known pancreatic mass presented with abdominal pain. CT scan showed an increased mass in the pancreatic head, and endoscopic ultrasound-guided fine needle aspiration (EUS-FNA) revealed “rare fragments of benign-appearing squamous epithelium in a background of keratin debris, cyst contents, and scattered lymphocytes,” consistent with a lymphoepithelial cyst. Pancreatic LEC is an extremely rare lesion that comprises of only 0.5% of all pancreatic cysts. EUS-FNA has become the mainstay for diagnosing pancreatic LECs. Given the slow growing and benign nature, conservative management and observation is adequate for pancreatic LECs with excellent long-term outcome. With increasing number of imaging ordered by clinicians, it is anticipated that there will be a greater number of incidental pancreatic LECs detected. Thus, EUS-FNA should be utilized more frequently to help distinguish benign pancreatic LECs from premalignant or malignant lesions to avoid surgery.
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7
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Davakis S, Syllaios A, Kyros E, Dimitrokallis N, Orfanos S, Vailas M, Papalampros A, Felekouras E. Lymphoepithelial pancreatic cyst: A rare entity among pancreatic cystic lesions. Report of two cases. Clin Case Rep 2020; 8:132-136. [PMID: 31998503 PMCID: PMC6982493 DOI: 10.1002/ccr3.2574] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/08/2019] [Revised: 10/26/2019] [Accepted: 10/31/2019] [Indexed: 12/18/2022] Open
Abstract
Lymphoepithelial pancreatic cysts are extremely rare benign pancreatic cystic lesions. High suspicion and an individual approach are imperative for the best management of those extremely rare entities.
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Affiliation(s)
- Spyridon Davakis
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
| | - Athanasios Syllaios
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
| | - Eleandros Kyros
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
| | - Nikolaos Dimitrokallis
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
| | - Stamatios Orfanos
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
| | - Michail Vailas
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
| | - Alexandros Papalampros
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
| | - Evangelos Felekouras
- First Department of SurgeryLaiko General HospitalNational and Kapodistrian University of AthensAthensGreece
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8
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Abdelkader A, Hunt B, Hartley CP, Panarelli NC, Giorgadze T. Cystic Lesions of the Pancreas: Differential Diagnosis and Cytologic-Histologic Correlation. Arch Pathol Lab Med 2019; 144:47-61. [PMID: 31538798 DOI: 10.5858/arpa.2019-0308-ra] [Citation(s) in RCA: 52] [Impact Index Per Article: 8.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/18/2022]
Abstract
CONTEXT.— Pancreatic cystic lesions (PCLs) are very common, and their detection is increasing with the advances in imaging techniques. Because of the major implications for management, distinguishing between neoplastic and nonneoplastic PCLs is critical. Neoplastic cysts with potential to progress into cancer include mucinous PCLs (intraductal papillary mucinous neoplasms and mucinous cystic neoplasms) and nonmucinous cysts (solid pseudopapillary tumors, serous cystic neoplasms, and neuroendocrine tumors with cystic degeneration). Nonneoplastic cysts with no risk of malignant transformation include pseudocysts, retention cysts, lymphoepithelial cysts, cystic pancreatic lymphangioma, and duplication cyst/ciliated foregut cysts. The role of endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) cytology with cyst fluid analysis in the diagnosis of PCLs has evolved during the last decade; however, a definitive diagnosis on cytologic specimens is hampered by the sparse cellularity and can be challenging. EUS-FNA can play an important role to differentiate low-risk from high-risk pancreatic cysts and to distinguish between patients with cysts who need clinical follow-up versus those who require surgery. OBJECTIVE.— To provide an integrative approach to diagnose pancreatic cystic lesions using EUS-FNA cytology and cyst fluid analysis, along with clinical, radiologic, histologic, genetic, and molecular characteristics. DATA SOURCES.— The review and analysis of the latest literature describing pancreatic cystic lesions. CONCLUSIONS.— Accurate diagnosis of PCLs requires a multidisciplinary and multimodal team approach, including the integration of clinical findings, imaging, cytology, cyst fluid analysis, and molecular testing.
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Affiliation(s)
- Amrou Abdelkader
- From the Department of Pathology, Medical College of Wisconsin, Milwaukee (Drs Abdelkader, Hunt, Hartley, and Giorgadze); and the Department of Pathology, Albert Einstein College of Medicine, The Bronx, New York (Dr Panarelli)
| | - Bryan Hunt
- From the Department of Pathology, Medical College of Wisconsin, Milwaukee (Drs Abdelkader, Hunt, Hartley, and Giorgadze); and the Department of Pathology, Albert Einstein College of Medicine, The Bronx, New York (Dr Panarelli)
| | - Christopher P Hartley
- From the Department of Pathology, Medical College of Wisconsin, Milwaukee (Drs Abdelkader, Hunt, Hartley, and Giorgadze); and the Department of Pathology, Albert Einstein College of Medicine, The Bronx, New York (Dr Panarelli)
| | - Nicole C Panarelli
- From the Department of Pathology, Medical College of Wisconsin, Milwaukee (Drs Abdelkader, Hunt, Hartley, and Giorgadze); and the Department of Pathology, Albert Einstein College of Medicine, The Bronx, New York (Dr Panarelli)
| | - Tamara Giorgadze
- From the Department of Pathology, Medical College of Wisconsin, Milwaukee (Drs Abdelkader, Hunt, Hartley, and Giorgadze); and the Department of Pathology, Albert Einstein College of Medicine, The Bronx, New York (Dr Panarelli)
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9
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Abstract
The pancreas is a complex organ that may give rise to large number of neoplasms and non-neoplastic lesions. This article focuses on benign neoplasms, such as serous neoplasms, and tumorlike (pseudotumoral) lesions that may be mistaken for neoplasm not only by clinicians and radiologists, but also by pathologists. The family of pancreatic pseudotumors, by a loosely defined conception of that term, includes a variety of lesions including heterotopia, hamartoma, and lipomatous pseudohypertrophy. Autoimmune pancreatitis and paraduodenal ("groove") pancreatitis may also lead to pseudotumor formation. Knowledge of these entities will help in making an accurate diagnosis.
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Affiliation(s)
- Olca Basturk
- Memorial Sloan Kettering Cancer Center, New York, NY, USA.
| | - Gokce Askan
- Memorial Sloan Kettering Cancer Center, New York, NY, USA
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10
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Borhani AA, Fasanella KE, Iranpour N, Zureikat AH, Singhi AD, Furlan A, Dasyam AK. Lymphoepithelial cyst of pancreas: spectrum of radiological findings with pathologic correlation. Abdom Radiol (NY) 2017; 42:877-883. [PMID: 27738791 DOI: 10.1007/s00261-016-0932-3] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/29/2022]
Abstract
PURPOSE The purpose of the study was to investigate imaging features as well as pathologic and clinical findings of lymphoepithelial cyst (LEC) of pancreas. MATERIALS AND METHODS Ten patients with surgically resected and pathologically proven LEC, found in a single institution database between 2000 and 2015, were evaluated in a retrospective fashion. Patients' demographics, clinical presentation, co-morbidities, imaging features, cytology and histopathology results, and serum/aspirate biomarkers levels were recorded. RESULTS Eighty percent of patients were male with median age of 59. All lesions were exophytic, with median size of 36 mm. 80% were classified as complex cystic lesions, showing enhancing septa or enhancing rim without measurable enhancing solid component. 80% were located in tail or body. In one patient with MRI, the lesion was mildly T1 hyperintense and markedly T2 hyperintense. All cases were anechoic or hypoechoic on EUS, and majority of them showed posterior acoustic enhancement. Of patients with available fluid aspirate analysis, 3 out of 4 had CEA level > 192 ng/mL and 1 out of 3 had elevated (>250 IU/ml) amylase level. Four out of 7 patients had elevated serum CA 19-9 levels (>37 U/mL); one patient with a value of 361 U/mL had co-existing pancreatic adenocarcinoma. CONCLUSION Round shape, mild complexity, and exophytic location in pancreatic body and tail can be suggestive of LECs. These features however are not specific and may be seen with other cystic pancreatic lesions. CT findings should be used in conjunction with EUS, cytology, and tumor marker studies to secure the diagnosis of LEC.
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Affiliation(s)
- Amir A Borhani
- Division of Abdominal Imaging, University of Pittsburgh School of Medicine, UPMC Presbyterian, Radiology Suite 200 East Wing, 200 Lothrop Street, Pittsburgh, PA, 15213, USA.
| | - Kenneth E Fasanella
- Division of Gastroenterology and Hepatology, University of Pittsburgh School of Medicine, UPMC Presbyterian, 200 Lothrop Street, Pittsburgh, PA, 15213, USA
| | - Negaur Iranpour
- Division of Abdominal Imaging, Department of Radiology, University of Pittsburgh Medical Center, UPMC Presbyterian, Radiology Suite 200 East Wing, 200 Lothrop Street, Pittsburgh, PA, 15213, USA
| | - Amer H Zureikat
- Division of Surgical Oncology, University of Pittsburgh Medical Center, 5150 Centre Ave, Suite 421, Pittsburgh, PA, 15232, USA
| | - Aatur D Singhi
- Department of Pathology, University of Pittsburgh School of Medicine, 200 Lothrop Street, Scaife Hall A616.2, Pittsburgh, PA, 15213, USA
| | - Alessandro Furlan
- Division of Abdominal Imaging, University of Pittsburgh School of Medicine, UPMC Presbyterian, Radiology Suite 200 East Wing, 200 Lothrop Street, Pittsburgh, PA, 15213, USA
| | - Anil K Dasyam
- Division of Abdominal Imaging, University of Pittsburgh School of Medicine, UPMC Presbyterian, Radiology Suite 200 East Wing, 200 Lothrop Street, Pittsburgh, PA, 15213, USA
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12
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An Unusual Suspect: Lymphoepithelial Cyst of the Pancreas. Case Rep Gastrointest Med 2016; 2016:5492824. [PMID: 27847656 PMCID: PMC5101372 DOI: 10.1155/2016/5492824] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/17/2016] [Accepted: 10/11/2016] [Indexed: 11/18/2022] Open
Abstract
Lymphoepithelial cysts (LECs) of the pancreas are benign, rare pancreatic cysts that are found predominantly in men. These cysts can present as a diagnostic conundrum given their rarity and difficulty of distinguishing these cysts from those with malignant potential. We present an incidental case of a LEC in a middle-aged man.
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13
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Tirkes T, Patel A, Cramer H, Evrimler S, DeWitt JM, Al-Haddad M. Lymphoepithelial Cysts of the Pancreas: CT and MRI Features. EUROPEAN MEDICAL JOURNAL 2015. [DOI: 10.33590/emj/22-00053] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/27/2023] Open
Abstract
Objective: Describe CT and MRI features of the lymphoepithelial cyst (LEC) of the pancreas.
Methods: The authors identified 13 cases of LEC by searching their institutional electronic medical archives from 2004 to 2020. All of the patients had CT scans and six patients had both a CT and MRI. The final diagnosis was established either by fine-needle aspiration with cytopathology (n=6) or surgical resection (n=7).
Results: The mean diameter of the cysts was 36 mm (range: 6–93 mm). Almost all of the cysts were exophytic (92%) and solitary (85%), favouring the tail (54%) or body (38%) of the pancreas. LECs were either oval (62%) or round (39%) and had well-defined contours. All LECs showed T2 hyperintensity and T1 hypointensity; however, the signal was heterogeneous. Diffusion-weighted imaging showed restricted diffusion in all cases. On CT, LECs commonly showed complex fluid density (>15 HU) with no visible septation, enhancement or calcification.
Conclusion: LECs have a distinguishing feature on MRI, which is restricted diffusion on diffusion-weighted imaging. This is presumably secondary to the presence of keratin, which can be a helpful feature differentiating LECs from other pancreatic cystic neoplasms. Besides this, LECs predominantly appear as solitary and exophytic lesions, with complex fluid density on CT and heterogeneous hypointense T1 and heterogenous hyperintense T2 signal on MRI.
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Affiliation(s)
- Temel Tirkes
- Department of Radiology, Indiana University School of Medicine, Indianapolis, USA
| | - Aashish Patel
- Department of Radiology, Indiana University School of Medicine, Indianapolis, USA
| | - Harvey Cramer
- Department of Pathology, Indiana University School of Medicine, Indianapolis, USA
| | - Sehnaz Evrimler
- Department of Radiology, Suleyman Demirel University School of Medicine, Isparta, Tϋrkiye
| | - John M. DeWitt
- Division of Gastroenterology, Indiana University School of Medicine, Indianapolis, USA
| | - Mohammad Al-Haddad
- Division of Gastroenterology and Hepatology, Indiana University School of Medicine, Indianapolis, USA
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14
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Jin X, Wu Y. Diagnostic utility of clinical and biochemical parameters in pancreatic head malignancy patients with normal carbohydrate antigen 19-9 levels. Afr Health Sci 2015; 15:123-30. [PMID: 25834540 PMCID: PMC4370120 DOI: 10.4314/ahs.v15i1.17] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/06/2023] Open
Abstract
BACKGROUND Carbohydrate antigen (CA)19-9 that is the most widely used biomarker for pancreatic cancer has certain limitations in diagnosis, which results in a tough job to distinguish pancreatic cancer from benign tumors with normal CA19-9. The aim of this study was to investigate the diagnostic utility of clinical parameters and serum markers in patients with pancreatic head masses but without elevated CA19-9. METHODS Retrospectively, 106 (69 malignant, 37 benign) of 487 patients admitted for pancreatic head masses were enrolled with CA19-9 level of <37u/ml. Clinical parameters and serum biomarkers were assessed. Among the patients with pancreatic head mass, male individuals (p=0.025) and elder individuals (p<0.001) were more likely to have cancer; and cancer patients were more likely to present with abdominal-pain (p=0.023), weight-loss (p=0.013) and jaundice (p<0.001). Serum bilirubin levels among malignancies, including total bilirubin (p<0.001), direct bilirubin (p<0.001) and indirect bilirubin (p<0.001), were considerably higher than those of benign ones. Logistic regression further concluded that age-distribution, abdominal-pain and direct-bilirubin were three independent factors correlating with final diagnosis. However, CEA (p=0.156) was not sufficient enough to exclude pancreatic cancer. CONCLUSIONS In patients with pancreatic head masses and CA19-9 of <37u/ml, age-distribution, abdominal-pain and direct bilirubin might be helpful in differential diagnosis. CEA was insufficient for exclusion of malignancy.
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Affiliation(s)
- Xiaoli Jin
- Department of Surgery, Sir Run Run Shaw Hospital College of Medicine, Zhejiang University, 3 Qingchun Road East, Hangzhou, Zhejiang Province 310016, P.R. China
| | - Yulian Wu
- Department of Surgery, Second Affiliated Hospital, College of Medicine, Zhejiang University, 88 Jiefang Road, Hangzhou, Zhejiang Province 310009, P.R. China
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Terakawa H, Makino I, Nakagawara H, Miyashita T, Tajima H, Kitagawa H, Fujimura T, Inoue D, Kozaka K, Gabata T, Ohta T. Clinical and radiological feature of lymphoepithelial cyst of the pancreas. World J Gastroenterol 2014; 20:17247-17253. [PMID: 25493042 PMCID: PMC4258598 DOI: 10.3748/wjg.v20.i45.17247] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/25/2014] [Revised: 06/15/2014] [Accepted: 07/30/2014] [Indexed: 02/06/2023] Open
Abstract
A lymphoepithelial cyst (LEC) of the pancreas is a rare benign lesion. Because patients with LEC of the pancreas have a good prognosis, it is important that these lesions are accurately differentiated from other more aggressive pancreatic neoplasms for an appropriate treatment strategy. Previous studies have reported that a definitive diagnosis of LEC often cannot be obtained based solely on the findings of preoperative imaging (e.g., Computed tomography or Magnetic resonance imaging). In this study, we reviewed four cases of pancreatic LECs to investigate the feature of LECs. We reviewed these cases with regard to symptoms, imaging findings, surgical procedures, and other clinical factors. We found that LEC was associated with unique characteristics on imaging findings. A preoperative diagnosis of LEC may be possible by comprehensively evaluating its clinical and imaging findings.
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Abstract
OBJECTIVES Lymphoepithelial cyst (LEC) of the pancreas is an unusual and benign cystic tumor. Accurate preoperative diagnosis is difficult; hence, most of pancreatic LECs are resected. The aim was to describe clinicopathological features of pancreatic LEC to guide appropriate management. METHODS We retrospectively collected data about LEC patients treated in our department between 1987 and 2012 and added cases from review of the literature during the same period. RESULTS One hundred seventeen cases (3 from our institution and 114 from literature review) were identified. Most patients were men (78%). The discovery was generally fortuitous. Serum CA19-9 was elevated in half of the cases. No specific radiological feature was identified. Fine needle aspiration and cytologic analysis allowed a correct preoperative diagnosis in 21% of the patients, showing presence of squamous cells, lymphocytes, and keratinous debris. Half of them were treated conservatively, whereas other patients underwent surgery. Neither malignant transformation nor recurrence after resection was observed. CONCLUSIONS The LEC of the pancreas is a rare benign tumor that could be treated conservatively. Fine needle aspiration is the only tool that can achieve a diagnosis without resection. If no certain diagnosis can be made, surgery is warranted to rule out a malignant differential diagnosis.
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Konstantinidis IT, Kambadakone A, Catalano OA, Sahani DV, Deshpande V, Forcione DG, Wargo JA, Fernandez-del Castillo C, Lillemoe KD, Warshaw AL, Ferrone CR. Lymphoepithelial cysts and cystic lymphangiomas: Under-recognized benign cystic lesions of the pancreas. World J Gastrointest Surg 2014; 6:136-141. [PMID: 25068011 PMCID: PMC4110531 DOI: 10.4240/wjgs.v6.i7.136] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/10/2014] [Revised: 05/05/2014] [Accepted: 07/14/2014] [Indexed: 02/06/2023] Open
Abstract
AIM: To identify their diagnostic and prognostic clinical characteristics in a large series.
METHODS: Retrospective review of clinicopathologic and imaging characteristics of patients diagnosed with lymphoepithelial cysts and cystic lymphangiomas of the pancreas at Massachusetts General Hospital.
RESULTS: Twelve patients were identified between 1/1/1997 and 8/1/2007. Their median age was 55.5 years (range 19-78 years), and 6 were females. The lesion was incidentally discovered in half of the patients. Contrast enhanced computed tomography demonstrated that the cysts had thin walls, without calcifications, pancreatic duct dilation or pancreatic parenchyma invasion. Endoscopic ultrasound with fine needle aspiration (EUS/FNA) confirmed the diagnosis of a lymphoepithelial cyst in 3 patients, one of whom was spared an operation and continues to do well after 6 years. Eleven patients had a resection: 3 pancreaticoduodenectomies, 7 distal pancreatectomies, and 1 enucleation. The median size of the cysts was 3 cm (range 2-20 cm). At a median follow-up of 57 mo no recurrences or other pancreas-related conditions occurred.
CONCLUSION: Lymphoepithelial cysts and cystic lymphangiomas of the pancreas can be diagnosed with a combination of contrast-enhanced computed tomography scans and EUS/FNA. If the lesion is asymptomatic, an operation might be avoided.
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Enlarging lymphoepithelial cyst of the pancreas during 12 months of observation: report of a case. Surg Today 2014; 45:101-4. [DOI: 10.1007/s00595-014-0960-y] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/09/2012] [Accepted: 08/20/2013] [Indexed: 10/25/2022]
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19
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Magnetic resonance imaging of cystic pancreatic lesions in adults: an update in current diagnostic features and management. ACTA ACUST UNITED AC 2013; 39:48-65. [DOI: 10.1007/s00261-013-0048-y] [Citation(s) in RCA: 26] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/17/2022]
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20
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A case of lymphoepithelial cyst of pancreas with unique “cheerios-like” appearance in EUS. Clin J Gastroenterol 2012; 5:388-92. [DOI: 10.1007/s12328-012-0336-z] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/20/2012] [Accepted: 09/19/2012] [Indexed: 02/03/2023]
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21
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Bédat B, Genevay M, Dumonceau JM, Frossard JL, Forget J, Morel P, Berney T. Association between lymphoepithelial cysts of the pancreas and HIV infection. Pancreatology 2011; 12:61-4. [PMID: 22487477 DOI: 10.1016/j.pan.2011.12.002] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
BACKGROUND & AIMS To report the association of lymphoepithelial cysts (LEC) of the pancreas with Human Immunodeficiency Virus (HIV) infection. An association between LEC and HIV infection is already established in the parotid gland (PG). METHODS Report of the first two cases of LEC of the pancreas associated with HIV infection and comparison of the clinical and histopathological aspects of LECs of the pancreas and of the PG. RESULTS LECs of the pancreas were discovered by CT imaging in 2 patients with a history of HIV infection. Notably, LEC completely resolved in one patient after initiation of antiretroviral therapy. CONCLUSION This is the first report of an association of LEC of the pancreas and HIV infection. In the presence of LEC of the pancreas, we propose a systematic screening for HIV infection and associated lesions in the PG. Antiretroviral therapy should be initiated in untreated patients. Surgery should be reserved for symptomatic patients in whom medical therapy has failed.
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Affiliation(s)
- Benoît Bédat
- Department of Surgery, Division of Visceral Surgery, Geneva University Hospitals, Rue Gabrielle-Perret-Gentil, 4, 1211 Geneva 14, Switzerland.
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22
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Pancreatic Cystic Lesions of Nonmucinous Type. Surg Pathol Clin 2011; 4:553-77. [PMID: 26837488 DOI: 10.1016/j.path.2011.03.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/22/2022]
Abstract
Cystic lesions of nonmucinous type can arise within the pancreas or can develop from adjacent structures and appear to involve the pancreas. In addition, some typically solid masses can become cystic or can present as cystic lesions. Nonmucinous cysts can be neoplastic, inflammatory, reactive, or congenital. The vast majority of neoplastic nonmucinous cysts are benign. Because of the difficulty in determining the neoplastic potential of a pancreatic cyst preoperatively, many non-neoplastic cysts are resected. This article reviews the surgical pathology of nonmucinous cysts.
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23
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Nam SJ, Hwang HK, Kim H, Yu JS, Yoon DS, Chung JJ, Kim JH, Kim KW. Lymphoepithelial cysts in the pancreas: MRI of two cases with emphasis of diffusion-weighted imaging characteristics. J Magn Reson Imaging 2011; 32:692-6. [PMID: 20815068 DOI: 10.1002/jmri.22260] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/11/2023] Open
Abstract
Pancreatic lymphoepithelial cysts (LECs) are rare pancreatic cystic lesions filled with keratinized material, lined by mature, keratinizing squamous epithelium and surrounded by lymphoid tissue containing few lymphoid follicles. We report two cases of surgically confirmed pancreatic LECs showing a profound restriction of water molecules on diffusion-weighted (DWI) magnetic resonance imaging (MRI). For pancreatic cystic lesions showing lack of molecular motion on DWI with or without thin marginal enhancement on contrast material-enhanced imaging, LECs consisting of internally keratinized materials with restricted diffusion should be considered in differential diagnoses even though they cannot always be easy to distinguish from other focal pancreatic lesions containing mucin, blood clot, or nonliquefactive necrosis.
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Affiliation(s)
- Se Jin Nam
- Department of Radiology, Yonsei University College of Medicine, Gangnam Severance Hospital, Seoul, Republic of Korea
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Selected case from the Arkadi M. Rywlin International Pathology Slide Seminar: benign lymphoepithelial cyst, head of pancreas. Adv Anat Pathol 2011; 18:98-100. [PMID: 21169743 DOI: 10.1097/pap.0b013e3182026d8f] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
Pancreatic lymphoepithelial cysts are more common in men, can occur anywhere in the pancreas, are sharply demarcated from surrounding tissues, and range in size from 1.2 to 17 cm. Patients are usually middle aged, presenting symptoms include abdominal pain, nausea, vomiting, and diarrhea, although many tumors are asymptomatic and are discovered incidentally on organ imaging or at autopsy. An elevated serum carbohydrate-associated antigen 19-9 may wrongly suggest a mucinous neoplasm. The diagnosis can be made preoperatively with a combination of organ imaging, fine needle aspiration biopsy, or ultrasound-guided Trucut biopsies. Cysts can be unilocular, bilocular, or multilocular, have walls up to 0.6 cm thick which are lined by squamous epithelium, occasional columnar mucinous cells, and small foci of sebaceous cells. The epithelium is surrounded by a dense rim of lymphoid tissue with scattered lymphoid follicles. Invaginations of the epithelium into the lymphoid tissue, reminiscent of a Warthin tumor, are occasionally observed. The pathogenesis is unknown. Pancreatic lymphoepithelial cysts are cured by conservative resection but if they are asymptomatic and are diagnosed before surgery, no treatment is necessary.
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Sewkani A, Purohit D, Singh V, Jain A, Varshney R, Varshney S. Lymphoepithelial cyst of the pancreas: a rare case report and review of literature. Indian J Surg 2010; 72:427-32. [PMID: 22131649 DOI: 10.1007/s12262-010-0152-y] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/26/2009] [Accepted: 09/02/2010] [Indexed: 11/25/2022] Open
Abstract
UNLABELLED Lymphoepithelial cysts are rare pancreatic lesions of undetermined pathogenesis. The literature on this entity is limited to case reports or small series. We describe a case of 66 year male, incidentally diagnosed as lymphoepithelial cyst of pancreas that was managed by enucleation. This is the first case report of lymphoepithelial cyst from India. An extensive Medline search was carried out for lymphoepithelial cyst of pancreas. Till date less than 100 cases were identified in available literature. All these cases (including our case) were analyzed. This entity has uniform and distinctive clinicopathological features. About half of the reported cases were asymptomatic with most of the lesions diagnosed incidentally. Majority of patients presents with non-specific symptoms making preoperative diagnosis difficult. Lymphoepithelial cyst of the pancreas is a rare benign lesion, which is difficult to diagnose preoperatively. High index of suspicion and preoperative fine needle aspiration cytology may help in making diagnosis and avoiding surgery in asymptomatic patients. ELECTRONIC SUPPLEMENTARY MATERIAL The online version of this article (doi:10.1007/s12262-010-0152-y) contains supplementary material, which is available to authorized users.
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Pancreatic lymphoepithelial cysts express CEA and can contain mucous cells: potential pitfalls in the preoperative diagnosis. Mod Pathol 2010; 23:1467-76. [PMID: 20802468 DOI: 10.1038/modpathol.2010.144] [Citation(s) in RCA: 42] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/30/2023]
Abstract
Pancreatic lymphoepithelial cysts are rare benign cysts that cannot be reliably differentiated from neoplastic mucinous cysts preoperatively. Although elevated cyst fluid carcinoembryonic antigen (CEA) levels support a diagnosis of a mucinous cyst, the finding of increased CEA levels in lymphoepithelial cysts prompted this study. Nine resected lymphoepithelial cysts were examined for expression of CEA, carbohydrate antigen (CA) 19-9, CK7, p63, PAS-D and a panel of mucins. The pathology data were correlated with clinical information, including serum, cyst fluid and imaging studies. By computed tomography scan, although most lymphoepithelial cysts appeared cystic, 23% were described as masses. The endoscopic ultrasound findings were variable, but the lymphoepithelial cysts tended to be hypoechoic cystic lesions or masses. On cytology, 44% of the cysts had squamous cells, 67% had glandular cells and 56% had atypical cells. The cysts were resected because of size ≥3 cm (89%), symptoms (44%) and/or elevated cyst fluid CEA levels (33%). The cyst fluid CEA levels in the three cysts tested were >450 ng/ml. Histopathologically, all cysts were lined by mature, stratified squamous-type cells and produced keratin. Mucous cells were present in 78% of the cysts. The immunohistochemical profile of the squamous lining was CK7+, p63+, MUC1+, MUC4+, MUC2-, MUC5AC- and MUC6-. Even though lymphoepithelial cysts are lined by squamous-type epithelium, all our resected lymphoepithelial cysts expressed CEA and/or CA19-9, many contained mucous cells, and three exhibited markedly elevated cyst fluid CEA levels. Although cyst fluid CEA levels >200 ng/ml support the diagnosis of mucinous neoplasms, this study emphasizes the need for clinicians and pathologists to recognize that lymphoepithelial cysts can mimic neoplastic mucinous cysts clinically, radiographically and on cyst fluid CEA analysis.
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Lymphoepithelial cysts of the pancreas: the use of endoscopic ultrasound-guided fine-needle aspiration in diagnosis. CANADIAN JOURNAL OF GASTROENTEROLOGY = JOURNAL CANADIEN DE GASTROENTEROLOGIE 2010; 24:348-50. [PMID: 20559575 DOI: 10.1155/2010/456867] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Abstract
Lymphoepithelial cysts (LECs) are rare non-neoplastic lesions that can appear as a complex cyst or a mass in the pancreas. Cytology from endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) can be helpful in making a diagnosis with the aim of avoiding unnecessary surgical resection. A case involving a 51-year-old woman with lower abdominal pain who was found to have a multiloculated cystic lesion at the junction of the pancreatic body and tail is described. Cytology from EUS-FNA was consistent with a pancreatic LEC. The lesion was managed conservatively and follow-up imaging of the cyst over the following two years was unchanged. The patient remains clinically well. Cytology from EUS-FNA can help distinguish LECs from cystic neoplasms, thus preventing radical surgical resection of this benign pancreatic cyst.
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