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1
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Minami R, Nakahodo J, Tabata H, Chiba K, Horiguchi S, Kamisawa T. Epidermoid cyst in an intrapancreatic accessory spleen diagnosed by contrast-enhanced EUS and EUS-FNA (with video). Endosc Ultrasound 2024; 13:49-51. [PMID: 38947112 PMCID: PMC11213605 DOI: 10.1097/eus.0000000000000047] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 07/02/2024] Open
Affiliation(s)
- Ryogo Minami
- Department of Gastroenterology, Tokyo Metropolitan Cancer and Infectious Diseases Center, Komagome Hospital, Tokyo, Japan
| | - Jun Nakahodo
- Department of Gastroenterology, Tokyo Metropolitan Cancer and Infectious Diseases Center, Komagome Hospital, Tokyo, Japan
| | - Hiroki Tabata
- Department of Gastroenterology, Tokyo Metropolitan Cancer and Infectious Diseases Center, Komagome Hospital, Tokyo, Japan
| | - Kazuro Chiba
- Department of Gastroenterology, Tokyo Metropolitan Cancer and Infectious Diseases Center, Komagome Hospital, Tokyo, Japan
| | - Shinichiro Horiguchi
- Department of Pathology, Tokyo Metropolitan Cancer and Infectious Diseases Center, Komagome Hospital, Tokyo, Japan
| | - Terumi Kamisawa
- Department of Gastroenterology, Tokyo Metropolitan Cancer and Infectious Diseases Center, Komagome Hospital, Tokyo, Japan
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2
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Veron Sanchez A, Santamaria Guinea N, Cayon Somacarrera S, Bennouna I, Pezzullo M, Bali MA. Rare Solid Pancreatic Lesions on Cross-Sectional Imaging. Diagnostics (Basel) 2023; 13:2719. [PMID: 37627978 PMCID: PMC10453474 DOI: 10.3390/diagnostics13162719] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/31/2023] [Revised: 08/15/2023] [Accepted: 08/18/2023] [Indexed: 08/27/2023] Open
Abstract
Several solid lesions can be found within the pancreas mainly arising from the exocrine and endocrine pancreatic tissue. Among all pancreatic malignancies, the most common subtype is pancreatic ductal adenocarcinoma (PDAC), to a point that pancreatic cancer and PDAC are used interchangeably. But, in addition to PDAC, and to the other most common and well-known solid lesions, either related to benign conditions, such as pancreatitis, or not so benign, such as pancreatic neuroendocrine neoplasms (pNENs), there are solid pancreatic lesions considered rare due to their low incidence. These lesions may originate from a cell line with a differentiation other than exocrine/endocrine, such as from the nerve sheath as for pancreatic schwannoma or from mesenchymal cells as for solitary fibrous tumour. These rare solid pancreatic lesions may show a behaviour that ranges in a benign to highly aggressive malignant spectrum. This review includes cases of an intrapancreatic accessory spleen, pancreatic tuberculosis, solid serous cystadenoma, solid pseudopapillary tumour, pancreatic schwannoma, purely intraductal neuroendocrine tumour, pancreatic fibrous solitary tumour, acinar cell carcinoma, undifferentiated carcinoma with osteoclastic-like giant cells, adenosquamous carcinoma, colloid carcinoma of the pancreas, primary leiomyosarcoma of the pancreas, primary and secondary pancreatic lymphoma and metastases within the pancreas. Therefore, it is important to determine the correct diagnosis to ensure optimal patient management. Because of their rarity, their existence is less well known and, when depicted, in most cases incidentally, the correct diagnosis remains challenging. However, there are some typical imaging features present on cross-sectional imaging modalities that, taken into account with the clinical and biological context, contribute substantially to achieve the correct diagnosis.
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Affiliation(s)
- Ana Veron Sanchez
- Hôpital Universitaire de Bruxelles, Institut Jules Bordet, 1070 Brussels, Belgium; (I.B.)
| | | | | | - Ilias Bennouna
- Hôpital Universitaire de Bruxelles, Institut Jules Bordet, 1070 Brussels, Belgium; (I.B.)
| | - Martina Pezzullo
- Hôpital Universitaire de Bruxelles, Hôpital Erasme, 1070 Brussels, Belgium
| | - Maria Antonietta Bali
- Hôpital Universitaire de Bruxelles, Institut Jules Bordet, 1070 Brussels, Belgium; (I.B.)
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3
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Lim Y, Leng H, Lee CH, Chhun V, Lee YD. A congenital wandering spleen with a large epithelial cyst: A case report. Clin Case Rep 2022; 10:e6559. [PMID: 36381033 PMCID: PMC9653159 DOI: 10.1002/ccr3.6559] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/15/2022] [Revised: 10/06/2022] [Accepted: 10/15/2022] [Indexed: 11/13/2022] Open
Abstract
We present a case of a 21-year-old female patient who had a wandering spleen with a large cyst. The celiotomy was performed under the impression of torsion of the wandering spleen and leakage from the splenic cyst. The total splenectomy was performed instead of cystectomy and splenopexy.
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Affiliation(s)
- Youhok Lim
- Department of Education & TrainingHebron Medical CenterPhnom PenhCambodia
| | - Hour Leng
- Department of SurgeryHebron Medical CenterPhnom PenhCambodia
| | - Chi Hoon Lee
- Department of Education & TrainingHebron Medical CenterPhnom PenhCambodia
| | - Vireak Chhun
- Department of PathologyHebron Medical CenterPhnom PenhCambodia
| | - Young Don Lee
- Department of SurgeryHebron Medical CenterPhnom PenhCambodia
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Huang JY, Yang R, Li JW, Lu Q, Luo Y. Cavernous hemangioma of an intrapancreatic accessory spleen mimicking a pancreatic tumor: A case report. World J Clin Cases 2022; 10:1973-1980. [PMID: 35317146 PMCID: PMC8891771 DOI: 10.12998/wjcc.v10.i6.1973] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/25/2021] [Revised: 11/02/2021] [Accepted: 01/10/2022] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Intrapancreatic accessory spleen (IPAS) is an uncommon condition, with the majority of cases presenting as solid lesions. Thus, this condition is frequently misdiagnosed as pancreatic solid neoplasm. Moreover, splenic cavernous hemangioma is a rare disorder, whereas lesions with a cystic appearance arising from IPAS have not been reported.
CASE SUMMARY Herein, we present a case involving a 32-year-old male who had a complex cystic lesion in the tail of the pancreas revealed by conventional ultrasound. The lesion was misdiagnosed as a pancreatic cystadenoma because of its confusing anatomic location, as well as due to its peripheral nodular and internal septal enhancement patterns on contrast-enhanced ultrasound. After multidisciplinary discussion, the patient finally underwent laparoscopic pancreatic body and tail resections. Postoperative pathology demonstrated the lesion to be a cavernous hemangioma arising from the IPAS.
CONCLUSION Cavernous hemangioma in the intrapancreatic accessory spleen may mimic pancreatic cystadenoma, which is a condition with the potential to be malignant. Imaging follow-ups or surgical interventions may be helpful for the exclusion of malignant risks in complicated cystic lesions, especially those with parietal and septal enhancements.
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Affiliation(s)
- Jia-Yan Huang
- Department of Medical Ultrasound, West China Hospital of Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Rui Yang
- Department of Medical Ultrasound, West China Hospital of Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Jia-Wu Li
- Department of Medical Ultrasound, West China Hospital of Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Qiang Lu
- Department of Medical Ultrasound, West China Hospital of Sichuan University, Chengdu 610041, Sichuan Province, China
| | - Yan Luo
- Department of Medical Ultrasound, West China Hospital of Sichuan University, Chengdu 610041, Sichuan Province, China
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5
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Zheng X, Zhou B, Sun JQ, Jin M, Yan S. Laparoscopic spleen-preserving distal pancreatectomy for epidermoid cyst in an intrapancreatic accessory spleen: A case report. Medicine (Baltimore) 2021; 100:e26379. [PMID: 34190154 PMCID: PMC8257839 DOI: 10.1097/md.0000000000026379] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/09/2021] [Accepted: 06/02/2021] [Indexed: 01/04/2023] Open
Abstract
RATIONALE Pancreatic tail cystic lesions are increasingly encountered in clinical practice, however, it is difficult to make a correct diagnosis preoperatively because there are many types of pancreatic neoplastic and non-neoplastic cysts. Epidermoid cyst in an intrapancreatic accessory spleen (ECIPAS) is a rare non-neoplastic cyst locating in the pancreatic tail, and it is commonly misdiagnosed as another cystic neoplasm. PATIENT CONCERNS A 51-year-old man was admitted for investigation of abdominal pain. The physical examination and laboratory tests found no abnormalities, except for an elevation of carbohydrate antigen (CA)19-9. Imaging revealed a cystic lesion within the pancreatic tail, and the solid component surrounding the cyst was enhanced similarly to those of the splenic tissue. DIAGNOSIS ECIPAS was diagnosed based on the pathology after surgery. The mass was composed of a cyst and brown solid spleen-like tissue. The microscopic analysis demonstrated that the solid component was accessory splenic tissue, and the cyst wall was lined with a thin stratified squamous epithelium. INTERVENTIONS Laparoscopic spleen-preserving distal pancreatectomy was performed. OUTCOMES The patient was discharged on day 5 postoperatively after an uneventful recovery. CA19-9 returned to normal after surgery. During a 2-years follow-up, there was no evidence of tumor recurrence. LESSONS Although rare ECIPAS should be considered in the differential diagnosis of pancreatic tail cystic lesions, and the typical imaging features might facilitate the preoperative diagnosis. Laparoscopic distal pancreatectomy is a safe and effective approach for treating ECIPAS.
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Affiliation(s)
- Xiang Zheng
- Department of General Surgery, Second Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310009
| | - Bo Zhou
- Department of General Surgery, Second Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310009
| | - Jing-Qing Sun
- Affiliated Hospital of Stomatology, School of Stomatology, Zhejiang University School of Medicine, and Key Laboratory of Oral Biomedical Research of Zhejiang Province, Hangzhou 310006, Zhejiang Province, China
| | - Ming Jin
- Department of General Surgery, Second Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310009
| | - Sheng Yan
- Department of General Surgery, Second Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310009
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Hiroi S, Hamaoka M, Yamamoto R, Matsugu Y, Nishisaka T, Nakahara H, Itamoto T. A lymphoepithelial cyst in the pancreatic accessory spleen: A case report. Clin Case Rep 2021; 9:e04241. [PMID: 34188926 PMCID: PMC8218320 DOI: 10.1002/ccr3.4241] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/08/2021] [Revised: 04/10/2021] [Accepted: 04/16/2021] [Indexed: 11/16/2022] Open
Abstract
We present the first report of a lymphoepithelial cyst. As additional cases will likely be encountered in the future, our study sets the precedent for future research.
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Affiliation(s)
- Sawako Hiroi
- Department of Gastroenterological SurgeryHiroshima Prefectural HospitalHiroshimaJapan
| | - Michinori Hamaoka
- Department of Gastroenterological SurgeryHiroshima Prefectural HospitalHiroshimaJapan
| | - Rie Yamamoto
- Department of PathologyHiroshima Prefectural HospitalHiroshimaJapan
| | - Yasuhiro Matsugu
- Department of Gastroenterological SurgeryHiroshima Prefectural HospitalHiroshimaJapan
| | | | - Hideki Nakahara
- Department of Gastroenterological SurgeryHiroshima Prefectural HospitalHiroshimaJapan
| | - Toshiyuki Itamoto
- Department of Gastroenterological SurgeryHiroshima Prefectural HospitalHiroshimaJapan
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7
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Ko HJ, Shim JR, Lee TB, Choi BH, Lee JH, Ryu JH, Yang K. Epidermoid cyst in an intrapancreatic accessory spleen in the pancreas head: a case report. BMC Gastroenterol 2020; 20:392. [PMID: 33218300 PMCID: PMC7678289 DOI: 10.1186/s12876-020-01540-4] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/28/2020] [Accepted: 11/16/2020] [Indexed: 02/07/2023] Open
Abstract
Background An epidermoid cyst in an intrapancreatic accessory spleen (ECIPAS) in the pancreas head is an extremely rare condition. The natural course of this condition is not well known, and it is difficult to diagnose before surgery due to the lack of specific imaging findings. Case presentation A tumor was found in the head of the pancreas in a 68-year-old man with abdominal distension and discomfort. Magnetic resonance imaging (MRI) suggested a malignant tumor, such as a colloid cancer. The tumor was removed surgically, with pathologic examination showing that it was an ECIPAS.
Conclusion ECIPAS cannot be easily distinguished from other pancreatic cystic tumors, making it necessary to include ECIPAS in the differential diagnosis of these tumors. Unnecessary surgical resection may be avoided by more accurate preoperative diagnosis based on clinical and imaging characteristics.
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Affiliation(s)
- Hyo Jung Ko
- Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Pusan National University Yangsan Hospital, Yangsan, Gyeongsangnam-do, 50612, Republic of Korea
| | - Jae Ryong Shim
- Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Pusan National University Yangsan Hospital, Yangsan, Gyeongsangnam-do, 50612, Republic of Korea
| | - Tae Beom Lee
- Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Pusan National University Yangsan Hospital, Yangsan, Gyeongsangnam-do, 50612, Republic of Korea
| | - Byung Hyun Choi
- Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Pusan National University Yangsan Hospital, Yangsan, Gyeongsangnam-do, 50612, Republic of Korea
| | - Jung-Hee Lee
- Department of Pathology, Pusan National University Yangsan Hospital, Yangsan, Republic of Korea
| | - Je Ho Ryu
- Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Pusan National University Yangsan Hospital, Yangsan, Gyeongsangnam-do, 50612, Republic of Korea
| | - Kwangho Yang
- Division of Hepato-Biliary-Pancreatic Surgery and Transplantation, Department of Surgery, Pusan National University Yangsan Hospital, Yangsan, Gyeongsangnam-do, 50612, Republic of Korea. .,Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Gyeongsangnam-do, 50612, Republic of Korea.
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8
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Zhou XH, Ma JK, Valluru B, Sharma K, Liu L, Hu JB. Diagnosis and differentiation of mature cystic teratoma of pancreas from its mimics: A case report. Medicine (Baltimore) 2020; 99:e23267. [PMID: 33217853 PMCID: PMC7676578 DOI: 10.1097/md.0000000000023267] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/30/2022] Open
Abstract
INTRODUCTION Mature cystic teratoma originating in the pancreas is very unusual, often observed as an incidental finding during routine examinations or recognized perioperatively as the patients present with very unspecific clinical symptoms. The confirmatory diagnosis of a pancreatic cystic teratoma is generally made by histopathology after surgical excision. So, the preoperative diagnosis is very challenging, especially differentiation from the other pancreatic pathologies. PATIENT CONCERNS A 23-year-old woman was admitted to our hospital with a complaint of mild grade periumbilical abdominal pain. A pancreatic mass was revealed on a preliminary abdominal ultrasound examination. Her medical history was unremarkable with no long-standing illness or malignancy. DIAGNOSIS Mature cystic teratoma in the head of the pancreas. INTERVENTIONS Roux-enY choledochojejunostomy with gastrojejunostomy was performed, excising the tumor from the pancreatic head. OUTCOMES The postoperative course was uneventful; the patient was asymptomatic and has no evidence of recurrence on a 2-year follow up. CONCLUSIONS Pancreatic cystic teratoma is a benign, well-differentiated, and extremely rare congenital tumor. MRI is the choice of imaging modality and phase-GRE or fat suppression is the best technique for pre-operative diagnosis.
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Affiliation(s)
- Xin he Zhou
- Department of Radiology, the First Affiliated Hospital of Dali University
| | - Ji Kong Ma
- Department of Nephrology, Dali Bai Autonomous Prefecture People's Hospital Dali 671000, China
| | - Bimbadhar Valluru
- Department of Radiology, the First Affiliated Hospital of Dali University
| | - Kalyan Sharma
- Department of Radiology, the First Affiliated Hospital of Dali University
| | - Ling Liu
- Department of Radiology, the First Affiliated Hospital of Dali University
| | - Jin Bo Hu
- Department of Radiology, the First Affiliated Hospital of Dali University
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Kato T, Matsuo Y, Ueda G, Aoyama Y, Omi K, Hayashi Y, Imafuji H, Saito K, Tsuboi K, Morimoto M, Ogawa R, Takahashi H, Kato H, Yoshida M, Naitoh I, Hayashi K, Takahashi S, Takiguchi S. Epithelial cyst arising in an intrapancreatic accessory spleen: a case report of robotic surgery and review of minimally invasive treatment. BMC Surg 2020; 20:263. [PMID: 33129283 PMCID: PMC7603683 DOI: 10.1186/s12893-020-00927-0] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/14/2020] [Accepted: 10/25/2020] [Indexed: 01/02/2023] Open
Abstract
Background An epithelial cyst in an intrapancreatic accessory spleen (ECIPAS) is rare. We report a case of ECIPAS that was treated with robot-assisted distal pancreatectomy with splenectomy. Case presentation The case was a 59-year-old woman who was referred to our hospital after a pancreatic tail tumor was found on computed tomography prior to surgery for small bowel obstruction at another hospital. A cystic lesion in the pancreatic tail was discovered and evaluated by magnetic resonance imaging and endoscopic ultrasonography. Based on clinical and radiological features, mucinous cystic neoplasm was included in the differential diagnosis. The patient underwent robot-assisted distal pancreatectomy with splenectomy. The postoperative course was uneventful. Pathological evaluation revealed a 20-mm ECIPAS in the pancreatic tail. Conclusions If a pancreatic tail tumor is present, ECIPAS should be included in the differential diagnosis. However, preoperative diagnosis is difficult, and a definitive diagnosis is often not obtained until after surgery. Surgery should be minimally invasive. Laparoscopic distal pancreatectomy has become a standard surgical procedure because it is minimally invasive. Robot-assisted surgery is not only minimally invasive, but also advantageous, because it has a stereoscopic magnifying effect and allows the forceps to move smoothly. Robot-assisted distal pancreatectomy may be a good option, when performing surgery for a pancreatic tail tumor.
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Affiliation(s)
- Tomokatsu Kato
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Yoichi Matsuo
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan.
| | - Goro Ueda
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Yoshinaga Aoyama
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Kan Omi
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Yuichi Hayashi
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Hiroyuki Imafuji
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Kenta Saito
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Ken Tsuboi
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Mamoru Morimoto
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Ryo Ogawa
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Hiroki Takahashi
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
| | - Hiroyuki Kato
- Department of Experimental Pathology and Tumor Biology, Nagoya City University Graduate School of Medical Sciences, Nagoya, Japan
| | - Michihiro Yoshida
- Department of Gastroenterology and Metabolism, Nagoya City University Graduate School of Medical Sciences, Nagoya, Japan
| | - Itaru Naitoh
- Department of Gastroenterology and Metabolism, Nagoya City University Graduate School of Medical Sciences, Nagoya, Japan
| | - Kazuki Hayashi
- Department of Gastroenterology and Metabolism, Nagoya City University Graduate School of Medical Sciences, Nagoya, Japan
| | - Satoru Takahashi
- Department of Experimental Pathology and Tumor Biology, Nagoya City University Graduate School of Medical Sciences, Nagoya, Japan
| | - Shuji Takiguchi
- Department of Gastroenterological Surgery, Nagoya City University Graduate School of Medical Sciences, Kawasumi 1, Mizuho-cho, Mizuho-ku, Nagoya, 4678601, Japan
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10
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Tan HJ, Neo WL, Lee SY, Goh BKP, Kam JH. Epidermal Inclusion Cyst in an Intra-pancreatic Accessory Spleen: a Differential Diagnosis for Pancreatic Cystic Neoplasms and Review of the Literature. J Gastrointest Cancer 2019; 50:308-314. [PMID: 28889365 DOI: 10.1007/s12029-017-0002-2] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022]
Affiliation(s)
- Hiang Jin Tan
- Department of General Surgery, Singapore General Hospital, Bukit Merah, Singapore
| | - Wei Li Neo
- Department of General Surgery, Singapore General Hospital, Bukit Merah, Singapore
| | - Ser Yee Lee
- Department of Hepato-pancreato-biliary and Transplant Surgery, Singapore General Hospital, Bukit Merah, Singapore. .,Duke-NUS Medical School, Singapore, Singapore.
| | - Brian Kim Poh Goh
- Department of Hepato-pancreato-biliary and Transplant Surgery, Singapore General Hospital, Bukit Merah, Singapore.,Duke-NUS Medical School, Singapore, Singapore
| | - Juinn Huar Kam
- Department of Hepato-pancreato-biliary and Transplant Surgery, Singapore General Hospital, Bukit Merah, Singapore.,Duke-NUS Medical School, Singapore, Singapore
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11
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Li BQ, Lu J, Seery S, Guo JC. Epidermoid cyst in intrapancreatic accessory spleen: A systematic review. Pancreatology 2019; 19:10-16. [PMID: 30366677 DOI: 10.1016/j.pan.2018.10.008] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/15/2018] [Revised: 10/16/2018] [Accepted: 10/21/2018] [Indexed: 12/11/2022]
Abstract
BACKGROUND/OBJECTIVES Due to its rarity, epidermoid cyst in intrapancreatic accessory spleen (ECIPAS) is still a diagnostic dilemma during clinical practice. The aim of this review was to summarize the epidemiologic features and management of ECIPAS. METHODS MEDLINE and EMBASE were searched for English articles reporting on ECIPAS up to April 30th, 2018 following the methodology suggested by the PRISMA guidelines. Categorical variables were reported as frequency and percentage. Continuous variables were reported as median (range). RESULTS A total of 56 patients from 47 full articles were included for the final data synthesis. More than half of the ECIPASs (59%) were found incidentally. The female/male ratio was 1.33. ECIPAS is typically a single mono-/multi-lobular cystic lesions in the pancreatic tail with thickened cystic wall or various amount of solid component which had identical density/signal to the spleen on imaging examinations. The cyst is filled with serous or non-serous fluid. Recognition of the surrounding ectopic splenic tissue is the key point to diagnose ECIPAS. However, no preoperative examination was able to make a definite diagnosis. Almost all the patients (96%) received surgical treatment, due to the suspicion of pancreatic malignant or potentially malignant cystic tumor, especially mucinous cystic neoplasm (MCN). CONCLUSIONS Although seldom encountered, ECIPAS should be considered as a differential diagnosis for pancreatic cystic lesions, especially when solid component was detected. As a benign disease, unnecessary surgery should be avoided. Because it is difficult to make a definite diagnosis preoperatively by one single examination, multiple modalities may be required.
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Affiliation(s)
- Bing-Qi Li
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences/Peking Union Medical College, Beijing, China
| | - Jun Lu
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences/Peking Union Medical College, Beijing, China
| | - Samuel Seery
- School of Humanities and Social Sciences, Chinese Academy of Medical Sciences/Peking Union Medical College, Beijing, China
| | - Jun-Chao Guo
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences/Peking Union Medical College, Beijing, China.
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12
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Li BQ, Xu XQ, Guo JC. Intrapancreatic accessory spleen: a diagnostic dilemma. HPB (Oxford) 2018; 20:1004-1011. [PMID: 29843985 DOI: 10.1016/j.hpb.2018.04.004] [Citation(s) in RCA: 17] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/18/2018] [Revised: 04/17/2018] [Accepted: 04/29/2018] [Indexed: 12/12/2022]
Abstract
BACKGROUND As intrapancreatic accessory spleen (IPAS) is rarely encountered during clinical practice, the aim of this review was to summarize the epidemiologic features, the diagnosis and treatment of IPAS. METHODS MEDLINE and EMBASE were searched for articles reporting on IPAS. Categorical variables were reported as frequency and percentage. Continuous variables were reported as median (range). RESULTS A total of 105 patients were included, of which 73% were detected incidentally. The male/female ratio was 1.23. The size of IPAS in patients who had previously undergone splenectomy was larger than that of patients without prior splenectomy (2.5 cm vs 1.5 cm; p = 0.020). No preoperative examination was able to make a definite diagnosis for all IPASs. More than half of the patients (55%) received surgical treatment, most of which (87%) were suspected as pancreatic neuroendocrine tumors (p-net) preoperatively. CONCLUSIONS Although rare, IPAS should be considered in the differential of patients with suspected incidental p-net, especially if there has been a past history of splenectomy. Preoperative diagnosis is important as unnecessary surgery can be avoided. As it is difficult to make a definite diagnosis of IPAS by one single examination, multiple techniques may be required.
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Affiliation(s)
- Bing-Qi Li
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences/Peking Union Medical College, Beijing, China
| | - Xie-Qun Xu
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences/Peking Union Medical College, Beijing, China
| | - Jun-Chao Guo
- Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences/Peking Union Medical College, Beijing, China.
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13
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Ding Q, Ren Z, Wang J, Ma X, Zhang J, Sun G, Zuo C, Gu H, Jiang H. Intrapancreatic accessory spleen: Evaluation with CT and MRI. Exp Ther Med 2018; 16:3623-3631. [PMID: 30250526 DOI: 10.3892/etm.2018.6613] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2017] [Accepted: 03/23/2018] [Indexed: 01/05/2023] Open
Abstract
The aim of the present study was to evaluate the characteristics of computed tomography (CT) and magnetic resonance imaging (MRI), particularly diffusion-weighted imaging (DWI), in the imaging of intrapancreatic accessory spleen (IPAS). The clinical and pathological records of 9 patients, including 8 patients with IPAS and 1 patient with splenosis, were reviewed. The patients had undergone plain and triple-phase enhanced CT scanning (n=9) and MRI scanning (n=8). The lesions of the 8 IPAS patients were located in the pancreatic tail, and were round (n=3), oval (n=4) or triangular (n=1) in shape. The CT and/or MRI densities, signal intensities and dynamic enhanced patterns of the lesions were similar to those of the orthotopic spleen. In DWI images (n=5), the IPAS regions presented high signal intensity (SI), and no significant difference in the apparent diffusion coefficient determined using a b-value of 600 sec/mm2 was identified between the IPAS and orthotopic spleen (P>0.05). One patient with splenosis complicated with cirrhosis had a nodule located in the pancreatic tail with an unenhanced CT value of 65 HU. In MRI examination, with the exception of the dynamic enhancement pattern, the T1-weighted, T2-weighted and DWI signals of splenosis were inconsistent with those of the normal spleen. In conclusion, in pre-contrast and post-contrast-enhanced CT and MRI images, IPAS exhibits similar characteristics to the orthotopic spleen. CT and MRI used in combination with DWI are important in the diagnosis of IPAS.
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Affiliation(s)
- Qianjiang Ding
- Department of Radiology, The Affiliated Hospital of Medical School, Ningbo University, Ningbo, Zhejiang 315020, P.R. China
| | - Zhihao Ren
- Department of Radiology, The Affiliated Hospital of Medical School, Ningbo University, Ningbo, Zhejiang 315020, P.R. China
| | - Jianhua Wang
- Department of Radiology, The Affiliated Hospital of Medical School, Ningbo University, Ningbo, Zhejiang 315020, P.R. China
| | - Xiaolong Ma
- Department of Radiology, Changhai Hospital, Second Military Medical University, Shanghai 200433, P.R. China
| | - Jian Zhang
- Department of Nuclear Medicine, Changhai Hospital, Second Military Medical University, Shanghai 200433, P.R. China
| | - Gaofeng Sun
- Department of Nuclear Medicine, Changhai Hospital, Second Military Medical University, Shanghai 200433, P.R. China
| | - Changjing Zuo
- Department of Nuclear Medicine, Changhai Hospital, Second Military Medical University, Shanghai 200433, P.R. China
| | - Hao Gu
- Department of Pathology, Ningbo Second Hospital, Ningbo, Zhejiang 315010, P.R. China
| | - Hui Jiang
- Department of Pathology, Changhai Hospital, Second Military Medical University, Shanghai 200433, P.R. China
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14
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Zhou B, Zhang Q, Zhan C, Ding Y, Yan S. Laparoscopic spleen-preserving pancreatic resection for epidermoid cyst in an intrapancreatic accessory spleen: case report and literature review. Ther Clin Risk Manag 2018; 14:937-944. [PMID: 29805263 PMCID: PMC5960247 DOI: 10.2147/tcrm.s165489] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/29/2022] Open
Abstract
Introduction An epidermoid cyst in an intrapancreatic accessory spleen (ECIPAS) is a rare non-neoplastic cyst, typically occurring in the pancreatic tail. It is difficult to preoperatively differentiate ECIPAS from other types of pancreatic neoplastic cysts. Case presentation We herein report a case of a 32-year-old man with a cystic tumor in the tail of the pancreas. The patient underwent a laparoscopic spleen-preserving distal pancreatectomy, and histological examination revealed the presence of ECIPAS. In addition, we also performed a literature review of 42 case reports of ECIPAS. Conclusion Although the preoperative diagnosis of ECIPAS is relatively difficult, familiarity with the imaging features, the clinical presentation and the location of the cyst could lead to a correct preoperative diagnosis of ECIPAS, which might thereby reduce the number of unnecessary resections.
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Affiliation(s)
- Bo Zhou
- Division of Hepatobiliary and Pancreatic Surgery, Department of Surgery, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou, People's Republic of China
| | - Qiyi Zhang
- Division of Hepatobiliary and Pancreatic Surgery, Department of Surgery, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou, People's Republic of China
| | - Canyang Zhan
- Department of Neonatology, Children's Hospital, School of Medicine, Zhejiang University, Hangzhou, People's Republic of China
| | - Yuan Ding
- Division of Hepatobiliary and Pancreatic Surgery, Department of Surgery, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou, People's Republic of China
| | - Sheng Yan
- Division of Hepatobiliary and Pancreatic Surgery, Department of Surgery, The First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou, People's Republic of China
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15
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Paredes A, Beal EW, Dillhoff ME. Epidermoid cyst within an intrapancreatic accessory spleen. BMJ Case Rep 2018; 2018:bcr-2017-223600. [PMID: 29622708 DOI: 10.1136/bcr-2017-223600] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/26/2022] Open
Abstract
This is a case of an epidermoid cyst in an intrapancreatic accessory spleen at the tail of the pancreas. Concurrent epidermoid cyst within the accessory spleen is an exceedingly rare entity. The patient initially presented with abdominal pain, nausea and vomiting. Imaging studies revealed a 3.6 cm cystic mass in the tail of the pancreas that was concerning for a mucinous cystic neoplasm (MCN). The patient underwent a robotic distal pancreatectomy for a suspected diagnosis of MCN, which following histologically examination was found to be an epidermoid cyst of an intrapancreatic accessory spleen. Further imaging characteristics are needed to characterise and differentiate these lesions from those of malignant potential. Robotic distal pancreatectomy is a safe alternative with many benefits including decreased length of stay and decreased blood loss.
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Affiliation(s)
- Anghela Paredes
- Department of Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA
| | - Eliza Wright Beal
- Department of Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA
| | - Mary E Dillhoff
- Division of Surgical Oncology, Department of Surgery, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA
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16
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Epidermoid Cyst in an Intrapancreatic Accessory Spleen Diagnosed by Typical Radiographic Images and Endoscopic Ultrasound Fine-Needle Aspiration Findings With Contrast Agent. Clin Gastroenterol Hepatol 2018; 16:e13-e14. [PMID: 28300686 DOI: 10.1016/j.cgh.2017.03.001] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/31/2017] [Revised: 03/07/2017] [Accepted: 03/08/2017] [Indexed: 02/07/2023]
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17
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Suzumura K, Hatano E, Okada T, Asano Y, Uyama N, Nakamura I, Hai S, Ichikawa N, Nakasho K, Fujimoto J. Laparoscopic Resection of an Epithelial Cyst in an Intrapancreatic Accessory Spleen. Case Rep Gastroenterol 2017; 11:803-811. [PMID: 29606939 PMCID: PMC5875297 DOI: 10.1159/000484134] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/07/2017] [Accepted: 10/10/2017] [Indexed: 12/15/2022] Open
Abstract
An epithelial cyst in an intrapancreatic accessory spleen (ECIAS) is rare. We herein report a case of a patient with ECIAS who underwent laparoscopic surgery. A 57-year-old woman was referred to our hospital because of a pancreatic tail tumor. She was asymptomatic, and a physical examination revealed no remarkable abnormalities. The levels of the tumor marker carbohydrate antigen 19-9 (CA19-9) and s-pancreas-1 antigen (SPan-1) were elevated. Ultrasonography showed a well-defined homogeneous cystic tumor. Computed tomography showed a well-demarcated cystic tumor in the pancreatic tail. Magnetic resonance imaging showed that the cystic tumor exhibited low intensity on T1-weighted images and high intensity on T2-weighted images. The cystic tumor was diagnosed as mucinous cystic neoplasm preoperatively. The patient underwent laparoscopic spleen-preserving distal pancreatectomy. A histopathological examination revealed the cyst wall to be lined by stratified squamous epithelium within splenic parenchyma, and the ultimate diagnosis was ECIAS. The postoperative course was uneventful, and the patient was discharged on postoperative day 12. ECIAS is very difficult to diagnose preoperatively. Laparoscopic surgery is a safe and minimally invasive procedure for patients with difficult-to-diagnose pancreatic tail tumor suspected of having low-grade malignancy.
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Affiliation(s)
- Kazuhiro Suzumura
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
| | - Etsuro Hatano
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
| | - Toshihiro Okada
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
| | - Yasukane Asano
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
| | - Naoki Uyama
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
| | - Ikuo Nakamura
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
| | - Seikan Hai
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
| | | | - Keiji Nakasho
- Department of Pathology, Hyogo College of Medicine, Nishinomiya, Japan
| | - Jiro Fujimoto
- Department of Surgery, Hyogo College of Medicine, Nishinomiya, Japan
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18
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Rare Case of an Epithelial Cyst in an Intrapancreatic Accessory Spleen Treated by Robot-Assisted Spleen Preserving Distal Pancreatectomy. Case Rep Gastrointest Med 2016; 2016:9475897. [PMID: 27847657 PMCID: PMC5099494 DOI: 10.1155/2016/9475897] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/14/2016] [Accepted: 09/26/2016] [Indexed: 02/07/2023] Open
Abstract
Epithelial cyst in an intrapancreatic accessory spleen (ECIPAS) is exceedingly rare with only 57 cases reported since the first publication in 1980. Comprehensive clinical and diagnostic features remain to be clarified. We present a case of ECIPAS in a 21-year-old Philippine woman who was admitted with right upper quadrant abdominal pain. A cystic lesion in the pancreatic tail was discovered and evaluated by computed tomography and magnetic resonance images. Based on clinical and radiological features a solid pseudopapillary neoplasm was suspected. The patient underwent robot-assisted spleen preserving distal pancreatectomy. Pathological evaluation revealed a 26 mm intrapancreatic accessory spleen with a 16 mm cyst, lined by multilayered epithelium in the tail of the pancreas. The postoperative course was uneventful. Differentiating ECIPAS from (pre)malignant cystic pancreatic neoplasms based on clinical and radiological features remains difficult. When typical radiological signs can be combined with scintigraphy using Technetium-99m labelled colloid or Technetium-99m labelled erythrocytes, which can identify the solid component of the lesion as splenic tissue, it should be possible to make the right diagnosis noninvasively. When pancreatectomy is inevitable due to symptoms or patient preference, minimally invasive laparoscopic or robot-assisted spleen preserving distal pancreatectomy should be considered.
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19
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Fujii M, Yoshioka M, Shiode J. Two Cases of an Epidermoid Cyst Developing in an Intrapancreatic Accessory Spleen Identified during Laparoscopic Distal Pancreatectomy. Intern Med 2016; 55:3137-3141. [PMID: 27803407 PMCID: PMC5140862 DOI: 10.2169/internalmedicine.55.7141] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/18/2022] Open
Abstract
Epidermoid cysts presenting within an intrapancreatic accessory spleen are rare non-neoplastic cysts typically occurring in the pancreatic tail. This entity is difficult to diagnose given there are many types of pancreatic neoplastic cysts. We herein describe two cases of an epidermoid cyst within an intrapancreatic accessory spleen for which we performed a resection by laparoscopic distal pancreatectomy. Epidermoid cysts in an intrapancreatic accessory spleen should therefore be considered in the differential diagnosis of pancreatic tail cystic lesions. Laparoscopic distal pancreatectomy can be a useful, minimally invasive surgical approach for treating these cysts as well as for the treatment of benign or low-grade malignant tumors located in the pancreatic body or tail.
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Affiliation(s)
- Masakuni Fujii
- Department of Internal medicine, Okayama Saiseikai General Hospital, Japan
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20
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Kato S, Mori H, Zakimi M, Yamada K, Chinen K, Arashiro M, Shinoura S, Kikuchi K, Murakami T, Kunishima F. Epidermoid Cyst in an Intrapancreatic Accessory Spleen: Case Report and Literature Review of the Preoperative Imaging Findings. Intern Med 2016; 55:3445-3452. [PMID: 27904107 PMCID: PMC5216141 DOI: 10.2169/internalmedicine.55.7140] [Citation(s) in RCA: 17] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/13/2022] Open
Abstract
An epidermoid cyst arising within an intrapancreatic accessory spleen (ECIAS) is rare, and also difficult to correctly diagnose before surgery. It is mostly misdiagnosed as a cystic tumor, such as a mucinous cystic neoplasm or as a solid tumor with cystic degeneration, such as a neuro endocrine tumor. We herein report a case of ECIAS and also perform a literature review of 35 reports of ECIAS. Although the preoperative diagnosis of ECIAS using conventional imaging is relatively difficult to make, careful preoperative examinations of the features on computed tomography and magnetic resonance imaging could lead to a correct preoperative diagnosis of ECIAS which might thereby reduce the number of unnecessary resections.
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Affiliation(s)
- Shin Kato
- Department of Gastroenterology, Okinawa Prefectural Chubu Hospital, Japan
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21
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Kwak MK, Lee NK, Kim S, Han GJ, Seo HI, Park DY, Lee SJ, Kim TU. A case of epidermoid cyst in an intrapancreatic accessory spleen mimicking pancreas neoplasms: MRI with DWI. Clin Imaging 2016; 40:164-6. [DOI: 10.1016/j.clinimag.2015.09.004] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/09/2015] [Accepted: 09/01/2015] [Indexed: 12/15/2022]
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22
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Sinha AK, Kumar A, Kumar B, Kumar P, Sinha R, Bhadani PP. An epidermoid cyst of accessory spleen simulating tumors of the tail of pancreas. JOURNAL OF PEDIATRIC SURGERY CASE REPORTS 2015. [DOI: 10.1016/j.epsc.2015.05.006] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022] Open
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23
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Kumamoto Y, Kaizu T, Tajima H, Kubo H, Nishiyama R, Watanabe M. A rapidly growing epidermoid cyst in an intrapancreatic accessory spleen treated by laparoscopic spleen-preserving distal pancreatectomy: Report of a case. Int Surg 2015; 103:10.9738/INTSURG-D-14-00278.1. [PMID: 26031701 DOI: 10.9738/intsurg-d-14-00278.1] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022] Open
Abstract
Epidermoid cysts arising in an intrapancreatic accessory spleen are exceedingly rare, furthermore the natural course of them is hardly known. We report a case correctly diagnosed with epidermoid cyst in an intrapancreatic accessory spleen, followed by 1 year observation, finally underwent surgical treatment. The patient presented with diarrhea. Contrast-enhanced computed tomography (CT) revealed a pancreatic cyst 20 mm in diameter, surrounded by a solid component showing the same enhancement as the spleen, suggesting the presence of an epidermoid cyst in an intrapancreatic accessory spleen. One year later, back discomfort developed, and a CT scan revealed that the cyst had grown to 38 mm in diameter. To obtain a definitive diagnosis, we performed a laparoscopic spleen-preserving distal pancreatectomy. The histopathological diagnosis was compatible with an epidermoid cyst in an intrapancreatic accessory spleen, which is benign. The postoperative course was uneventful. This case demonstrates that an epidermoid cyst arising in an intrapancreatic accessory spleen can rapidly grow, even if it is benign. Laparoscopic spleen-preserving distal pancreatectomy can be a useful procedure, with the advantages of low invasiveness and organ preservation, for the treatment of benign or low-grade malignant tumors located in the pancreatic body or tail.
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Affiliation(s)
- Yusuke Kumamoto
- c Kitasato University, School of Medicene, Sagamihara, 252-0380, Japan
| | - Takashi Kaizu
- d Department of Surgery, Kitasato University School of Medicine
| | - Hiroshi Tajima
- i Department of Surgery, Kitasato University School of Medicine
| | - Hidefumi Kubo
- j Department of Surgery, Kitasato University School of Medicine
| | - Ryo Nishiyama
- o Department of Surgery, Kitasato University School of Medicine
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24
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Akimori T, Maeda H, Okamoto K, Namikawa T, Usui T, Hanazaki K, Kobayashi M. A case of Peliosis and epithelial cyst of intrapancreatic heterotopic spleen: a differential diagnosis of pancreatic mucinous cystic neoplasm. ANNALS OF CANCER RESEARCH AND THERAPY 2015. [DOI: 10.4993/acrt.23.1] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Affiliation(s)
| | | | - Ken Okamoto
- Cancer Treatment Center, Kochi Medical School
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25
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Foo WC, Wang H, Prieto VG, Fleming JB, Abraham SC. Squamoid cystosis of pancreatic ducts: a variant of a newly-described cystic lesion, with evidence for an obstructive etiology. Rare Tumors 2014; 6:5286. [PMID: 25276318 PMCID: PMC4178271 DOI: 10.4081/rt.2014.5286] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/06/2014] [Accepted: 03/07/2014] [Indexed: 01/01/2023] Open
Abstract
We describe a 40-year-old man who was found to have a cystic mass in the pancreatic tail during workup for weight loss and abdominal discomfort. Although computed tomography scan showed a single cyst associated with dilatation of the main pancreatic duct, gross and histologic examination of the distal pancreatectomy specimen actually revealed a central cyst that was surrounded by multiple smaller cystic spaces. This distinctive appearance was formed from extensive cystic dilatation and squamous metaplasia of the native pancreatic duct system. Further, a traumatic neuroma was discovered near the junction between normal and abnormal parenchyma. We believe that this case represents a variant of the newly-described squamoid cyst of pancreatic ducts which we term squamoid cystosis of pancreatic ducts. The presence of chronic pancreatitis and a traumatic neuroma supports the hypothesis that squamoid cysts are non-neoplastic lesions arising from prior duct obstruction.
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Affiliation(s)
- Wai Chin Foo
- Department of Pathology, M.D. Anderson Cancer Center , Houston, TX, USA
| | - Huamin Wang
- Department of Pathology, M.D. Anderson Cancer Center , Houston, TX, USA
| | - Victor G Prieto
- Department of Pathology, M.D. Anderson Cancer Center , Houston, TX, USA
| | - Jason B Fleming
- Department of Surgical Oncology, M.D. Anderson Cancer Center , Houston, TX, USA
| | - Susan C Abraham
- Department of Pathology, M.D. Anderson Cancer Center , Houston, TX, USA
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26
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Zavras N, Machairas N, Foukas P, Lazaris A, Patapis P, Machairas A. Epidermoid cyst of an intrapancreatic accessory spleen: a case report and literature review. World J Surg Oncol 2014; 12:92. [PMID: 24721745 PMCID: PMC3986436 DOI: 10.1186/1477-7819-12-92] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/02/2013] [Accepted: 03/30/2014] [Indexed: 11/14/2022] Open
Abstract
Background An epidermoid cyst in an intrapancreatic accessory spleen is a rare lesion. Despite advances in radiologic techniques, in most cases it has been diagnosed preoperatively as a possible pancreatic neoplasm. Case presentation Herein, we present a 63-year-old Caucasian woman, diagnosed preoperatively with enhanced-contrast abdominal computed tomography, as having a potential cystic tumor in the tail of the pancreas. The patient underwent a distal pancreatectomy and splenectomy, and the histological examination revealed the presence of an epidermoid cyst of an accessory intrapancreatic spleen. Conclusions Familiarity with the imaging features, the clinical presentation and the location of the cyst are important to consider if this rare entity is to be included in the differential diagnosis of cystic neoplasms of the pancreas.
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Affiliation(s)
- Nick Zavras
- Third Department of Surgery, ATTIKO University Hospital, Athens, Greece.
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27
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Lee CL, Di Y, Jiang YJ, Jin C, Fu DL. Epidermoid cyst of intrapancreatic accessory spleen: A case report and literature review. World J Surg Proced 2013; 3:54-59. [DOI: 10.5412/wjsp.v3.i3.54] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/31/2013] [Revised: 08/14/2013] [Accepted: 09/04/2013] [Indexed: 02/06/2023] Open
Abstract
Epidermoid cyst of intrapancreatic accessory spleen is exceedingly rare; only 30 new cases have been reported in the English literature over the last 30 years. An accurate preoperative diagnosis was made in almost none of them because of the lack of reliable preoperative diagnostic methods. In this report, we present a case diagnosed with fluorine-18 fluorodeoxyglucose positron emission tomography (FDG-PET). A 41-year-old female who had breast cancer was routinely followed up by measuring the concentration of tumor makers. An increasing level of serum carbohydrate antigen 19-9 was detected and a cystic lesion located at the tail of pancreas was found by ultrasonography. A whole body fluorine-18 FDG positron emission tomography was performed because of a high suspicion for either a malignancy of the pancreas or a recurrence of breast cancer. No increased uptake of FDG was noted and therefore the cystic lesion was considered as pancreatic benign disease. Because pancreatic malignancy could not be entirely ruled out, distal pancreatectomy and splenectomy were performed. The final pathological diagnosis was epidermoid cyst of intrapancreatic accessory spleen (ECIAS). The FDG-PET findings matched the histopathology. A literature review reveals that the common clinical manifestations of ECIAS include asymptomatic findings on clinical examination, an occasional increase in tumor makers on laboratory results and occurrence only in the pancreatic tail. It is often misdiagnosed due to its extreme rarity and lack of a specific radiographic sign. There is no evidence of malignancy in ECIAS. Open or laparoscopic spleen preserving distal pancreatectomy is the minimally invasive procedure that would provide the best surgical management for epidermoid cyst of intrapancreatic accessory spleen.
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Abstract
BACKGROUND The aim of this study is to elucidate the characteristics, clinical presentations, and management of epidermoid cysts located in the spleen and the intrapancreatic accessory spleen. METHODS Data pooled for analysis were collected from epidermoid cyst cases encountered in the study institution and sporadic case reports obtained from the English literature. RESULTS There were 115 cases of epidermoid cyst. Seventy eight (67.8 %) were found in the spleen, whereas 37 (32.2 %) were found in the intrapancreatic accessory spleen. Comparing between the two groups, the splenic group showed female predominance, younger age, and larger cystic size. For symptoms, left upper abdominal mass (57.7 %) and left upper abdominal pain (39.0 %) were more commonly found in the spleen group. Meanwhile, the intrapancreatic accessory spleen group had higher percentage of asymptomatic cysts (59.5 %). As for surgical indication, majority of patients in the spleen group underwent surgery for symptomatic relief (75.4 %), while the intrapancreatic accessory spleen group underwent surgery mainly for suspicious malignancy (90.0 %). CONCLUSIONS Surgical treatment for epidermoid cysts of the spleen is considered the optimal treatment for either alleviating the symptoms caused by a bulky cyst or for determining the histopathologic nature of a suspicious malignant cystic neoplasm.
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Affiliation(s)
- Yin-Yin Chen
- Division of General Surgery, Department of Surgery, Taipei Veterans General Hospital, 201 Section 2 Shih-Pai Road, Taipei, 112, Taiwan
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29
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Hong R, Choi N, Sun K, Lim S, Han Y. Epidermoid cyst arising from an intrapancreatic accessory spleen: A case report and review of the literature. Oncol Lett 2013; 5:469-472. [PMID: 23420784 PMCID: PMC3573018 DOI: 10.3892/ol.2012.1061] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/18/2012] [Accepted: 10/10/2012] [Indexed: 11/05/2022] Open
Abstract
We describe an epidermoid cyst arising from an accessory spleen of the pancreas. A 56-year-old female with iron deficiency anemia presented with dizziness. During the clinical workup, a 2×4 cm-sized mass was incidentally detected in the tail of the pancreas in a computed tomography (CT) scan. Under a clinical diagnosis of pancreatic cystic malignant tumor, a distal pancreatectomy was carried out. Grossly, the lesion was composed of a solid and cystic portion. Microscopic analysis revealed that the solid portion was an intrapancreatic accessory spleen and the cystic portion was an epidermoid cyst. An epidermoid cyst in an intrapancreatic accessory spleen is extremely rare and hence difficult to diagnose pre-operatively. Taking into account the possibility of such a cyst in the differential diagnosis of intrapancreatic cystic lesion is recommended.
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Affiliation(s)
- Ran Hong
- Departments of Pathology, College of Medicine, Chosun University, Gwangju 501-759, Republic of Korea
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30
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Laparoscopic resection of an epidermoid cyst within an intrapancreatic accessory spleen: a case report and review article. Surg Laparosc Endosc Percutan Tech 2012; 22:e246-9. [PMID: 22874714 DOI: 10.1097/sle.0b013e31825b3761] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/19/2023]
Abstract
CASE REPORT We report a case of an epidermoid cyst within an intrapancreatic accessory spleen that was treated by laparoscopic excision. A 39-year-old man with no abdominal symptoms was incidentally found to have a cystic pancreatic lesion on computed tomography scan undertaken for suspected deep vein thrombosis. Further computed tomography and magnetic resonance imaging confirmed similar findings and the laparoscopic resection of the distal pancreas and spleen was undertaken as malignancy could not be excluded. Microscopic analysis revealed a well-circumscribed epidermoid cyst within a thin splenic rim in the tail of the pancreas. DISCUSSION Such histologic diagnoses are extremely rare, and this is the 26th case report to our knowledge in English language journals. These lesions should be treated surgically to exclude malignancy. This is the first case reported in the United Kingdom and the first to be excised by pure laparoscopic means, which we believe provides effective and successful surgical management.
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31
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Epidermoid cyst in intrapancreatic accessory spleen: computed tomography findings and clinical manifestation. Abdom Radiol (NY) 2012; 37:828-33. [PMID: 22327420 DOI: 10.1007/s00261-012-9851-0] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/29/2022]
Abstract
OBJECTIVE This study aims to assess retrospectively the imaging features of computed tomography (CT) and clinical characteristics of epidermoid cyst in intrapancreatic accessory spleen (ECIPAS). METHODS Seven consecutive patients with pathologically confirmed ECIPAS were included. CT images and clinical data were analyzed. The CT features emphasized included the location, size, calcification, cystic features, surrounding accessory spleen, density, and enhancement of the lesions. RESULTS Five patients were male and two were female with a mean age of 43.2 years ranging from 25 to 66 years. Most cases were incidentally detected. All lesions were situated in the pancreatic tail, wherein the mean size was 4.4 cm. The cyst appeared unilocular in four cases and multilocular in three cases. An accessory spleen surrounding the cyst was recognized in all seven cases, and the cystic wall of ECIPAS showed a contrast enhancement similar to that of the spleen during multiphasic scans. CONCLUSION ECIPAS is an extremely rare entity. The diagnosis of an ECIPAS should be considered when enhancing the cystic wall of the lesion in the pancreatic tail similar to that of the spleen during multiphasic scans.
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Laparoscopic resection of epidermoid cyst arising from an intrapancreatic accessory spleen: a case report with a review of the literature. Surg Laparosc Endosc Percutan Tech 2012; 21:e275-9. [PMID: 22002295 DOI: 10.1097/sle.0b013e31822dd14a] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/19/2023]
Abstract
We describe a rare case of epidermoid cyst arising in an intrapancreatic accessory spleen that presented as a cystic mass in the tail of the pancreas, and for which laparoscopic distal pancreatectomy was performed successfully. A 36-year-old woman with a cystic mass in the tail of the pancreas, which had been discovered incidentally at a medical checkup, was referred to our department for further examination. Endoscopic retrograde cholangiopancreatography, endoscopic ultrasonography and positron emission tomography demonstrated a multilocular cyst in the tail of the pancreas without any evidence of malignancy, although differential diagnosis was extremely difficult because of the neoplasm-like appearance of the lesion. Therefore, we performed laparoscopic distal pancreatectomy under a preoperative diagnosis of mucinous cystic neoplasm. Postoperative pathologic examination demonstrated an epidermoid cyst arising from a heterotopic spleen within the pancreas. This is the first report of successful laparoscopic distal pancreatectomy for an epidermoid cyst arising in an intrapancreatic accessory spleen. One virtually has no chance to diagnose an epidermoid cyst in an accessory spleen on the basis of preoperative diagnostic workup, and consequently the type of surgical resection (open vs. laparoscopic) would be conditioned by factors other than the clinical entity suspected at the preoperative period.
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Tatsas AD, Owens CL, Siddiqui MT, Hruban RH, Ali SZ. Fine-needle aspiration of intrapancreatic accessory spleen: Cytomorphologic features and differential diagnosis. Cancer Cytopathol 2012; 120:261-8. [DOI: 10.1002/cncy.21185] [Citation(s) in RCA: 30] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/06/2011] [Revised: 11/16/2011] [Accepted: 11/23/2011] [Indexed: 11/09/2022]
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Panagiotopoulos N, Acharya M, Ahmad R, Bansi D, Jiao L. Epithelial inclusion cyst arising within an intra-pancreatic splenunculus. Int J Surg Case Rep 2011; 3:118-20. [PMID: 22288063 DOI: 10.1016/j.ijscr.2011.12.005] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/13/2011] [Revised: 12/18/2011] [Accepted: 12/19/2011] [Indexed: 12/22/2022] Open
Abstract
INTRODUCTION An accessory spleen (splenunculus) may occur in up to 10% of the general population. However, an epithelial inclusion cyst originating within an intra-pancreatic splenunculus is an extremely rare finding, with only twenty-two previous cases described in medical literature. PRESENTATION OF CASE A 51-year-old male presented to our institution for investigation of altered bowel habit. Endoscopic ultrasound examination and CT scanning demonstrated an 18 mm cystic, well-demarcated lesion in the tail of the pancreas, resembling malignancy. Following laparoscopic spleen-preserving distal pancreatectomy, histological analysis confirmed epithelial inclusion cyst arising within an intra-pancreatic splenunculus. DISCUSSION The pre-operative radiological identification of such cystic pancreatic lesions is challenging. Surgical resection is usually performed for clinical suspicion of pancreatic malignancy. CONCLUSION Epithelial inclusion cyst originating within an intra-pancreatic accessory spleen is an extremely rare finding, which can mimic malignancy. Nevertheless, it should be carefully considered in the differential diagnosis of cystic lesions of the tail of the pancreas.
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Affiliation(s)
- Nikolaos Panagiotopoulos
- Department of Surgery and Cancer, Imperial College London, Hammersmith Hospital Campus, Du Cane Road, London W12 0HS, United Kingdom
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Abstract
OBJECTIVES Intrapancreatic accessory spleen is a benign lesion that mimics hypervascular or cystic pancreatic neoplasm. A comprehensive clinicopathologic analysis has not yet been reported. METHODS We described the clinicopathologic characteristics of 12 cases of pathologically proven intrapancreatic accessory spleen, among which 6 had internal epidermoid cysts. Immunohistochemistry was done to clarify the origin of epidermoid cysts. RESULTS Most cases were incidentally detected in young adults. Two-thirds of cases with intra-lesional cysts showed elevated serum carbohydrate antigen 19-9 levels. Radiologically, heterogeneously enhancing a solid portion similar to the spleen was a helpful, but not convincing, feature. Grossly, a well-circumscribed dark red mass with or without cysts in the pancreatic tail was characteristic. Microscopically, small foci of pancreatic tissue were embedded within the splenic tissue. Epidermoid cysts consisted of modified squamous epithelium, some of which had intracellular mucin. Cytologic smears showed large aggregates of benign spindle cells that were reactive against CD8. Immunohistochemical staining of the cystic epithelium suggested its pancreatic ductal origin. CONCLUSIONS Intrapancreatic accessory spleen with or without epidermoid cyst should be considered as differential diagnoses when well-enhanced solid or cystic tumors are found in the pancreatic tail. Radiologic suspicion and preoperative aspiration or biopsy might minimize the need for unnecessary surgery.
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Yamanishi H, Kumagi T, Yokota T, Koizumi M, Azemoto N, Watanabe J, Mizuno Y, Sugita A, Abe M, Ikeda Y, Matsuura B, Hiasa Y, Onji M. Epithelial cyst arising in an intrapancreatic accessory spleen: a diagnostic dilemma. Intern Med 2011; 50:1947-52. [PMID: 21921374 DOI: 10.2169/internalmedicine.50.5340] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/23/2023] Open
Abstract
An epithelial cyst in an intrapancreatic accessory spleen (ECIAS) is a rare disease that is commonly misdiagnosed preoperatively. To identify the clinical and imaging features of ECIAS, we reviewed the relevant medical literature. Twenty-one cases of ECIAS were identified, including our own. The cases were mainly diagnosed as mucinous cystic neoplasm (MCN) preoperatively based on clinical and imaging features, such as, a woman in middle age; elevation of serum CA19-9 levels; location in the tail of the pancreas; and a solid component resembling a mural nodule. ECIAS is another lesion to be considered in the differential diagnosis of MCN.
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Affiliation(s)
- Hirofumi Yamanishi
- Gastroenterology and Metabology, Ehime University Graduate School of Medicine, Japan
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Hori S, Nara S, Shimada K, Ojima H, Kanai Y, Hiraoka N. Serous cystic neoplasm in an intrapancreatic accessory spleen. Pathol Int 2010; 60:681-4. [PMID: 20846266 DOI: 10.1111/j.1440-1827.2010.02579.x] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
Abstract
Serous cystic neoplasm (SCN) of the pancreas is a benign epithelial neoplasm, except in extremely rare malignant cases. Development of SCN in tissues other than the pancreas has been never reported. Here we present the first reported case of SCN in an intrapancreatic accessory spleen (IPAS). A 54-year-old female patient with von Hippel-Lindau (VHL) syndrome was found to have pancreatic tail mass. Pathologically the 25-mm solid mass was an IPAS showing proliferation of clear cuboidal tumor cells without atypia, forming numerous small cysts. The tumor cells were rich in cytoplasmic glycogen and distributed in the splenic tissue almost diffusely. Immunohistochemically, tumor cells were positive for cytokeratins, MUC6, and neuron-specific enolase, and negative for neuroendocrine markers. From these findings, we diagnosed the lesion as SCN in IPAS. This tumor is suggested to develop as a VHL-associated SCN from coexisting pancreatic tissue in IPAS rather than as a metastatic tumor.
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Affiliation(s)
- Shutaro Hori
- Pathology Division, National Cancer Center Research Institute, Tokyo, Japan
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Epidermoid cyst in intrapancreatic accessory spleen: radiological findings including superparamagnetic iron oxide-enhanced magnetic resonance imaging. J Comput Assist Tomogr 2010; 34:217-22. [PMID: 20351508 DOI: 10.1097/rct.0b013e3181c1b2bd] [Citation(s) in RCA: 24] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/19/2023]
Abstract
It is crucial to differentiate between nonneoplastic and neoplastic cysts of the pancreas by images. An epidermoid cyst in intrapancreatic accessory spleen (IPAS) is a rare pancreatic cyst that should be managed nonsurgically as well as other nonneoplastic cyst. However, the imaging features of an epidermoid cyst in IPAS have not been organized. We therefore reviewed articles describing cases of epidermoid cyst in IPAS, with 6 additional cases to explore the imaging findings.
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Laparoscopic resection of an epidermoid cyst originating from an intrapancreatic accessory spleen: report of a case. Surg Today 2009; 40:72-5. [PMID: 20037845 DOI: 10.1007/s00595-009-4006-9] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/18/2008] [Accepted: 02/19/2009] [Indexed: 02/07/2023]
Abstract
A 67-year-old man underwent an investigation of epigastric pain and weight loss. Preoperative imaging findings suggested the presence of a tumor, which developed as an epidermoid cyst and originated from an intrapancreatic accessory spleen; however, the possibility of malignancy could not be ruled out. We therefore performed a laparoscopic-assisted distal pancreatectomy with a splenectomy for both diagnostic and treatment purposes. Five laparoscopic ports were created. After the spleen and pancreatic tail were dissected from the retroperitoneum laparoscopically, they were pulled out through a 7-cm left subcostal incision and resected with an endoscopic linear stapler. The operative time was 227 min and the blood loss was 400 ml. The postoperative course was uneventful. The final pathological diagnosis was in agreement with the preoperative diagnosis. This case demonstrates that the minimally invasive approach of laparoscopic surgery can be used safely and successfully for difficult-to-diagnose pancreatic tumors. This is the first report describing a laparoscopic resection of an epidermoid cyst originating from an intrapancreatic accessory spleen.
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40
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Kadota K, Kushida Y, Miyai Y, Katsuki N, Hayashi T, Bando K, Shibuya S, Haba R. Epidermoid cyst in an intrapancreatic accessory spleen: three case reports and review of the literatures. Pathol Oncol Res 2009; 16:435-42. [PMID: 19949910 DOI: 10.1007/s12253-009-9229-y] [Citation(s) in RCA: 24] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/27/2009] [Accepted: 11/16/2009] [Indexed: 01/19/2023]
Abstract
The development of an epidermoid cyst in an intrapancreatic accessory spleen is an extremely rare lesion, with only 17 cases being reported in the English literature. All such cases were located in the pancreatic tail, some of which showed carbohydrate antigen 19-9 (CA19-9) immunoreactivity in the lining of the epithelium. A few of them indicated an elevation of the serum CA19-9 level. Here we report three cases of an epidermoid cyst in an intrapancreatic accessory spleen. Cases 1 and 2 were 57-year-old and 70-year-old women, while case 3 was a 37-year-old man. All three cases were asymptomatic. Serum CA19-9 levels showed within normal limits (case 1), slightly elevated (case 2), and clearly elevated (case 3). They underwent a distal pancreatectomy with splenectomy (cases 1 and 2) and without splenectomy (case 3). Grossly, the surgical specimen was a well-demarcated, multiple (case 1) or solitary (cases 2 and 3) cystic mass in the pancreatic tail. A high level of fluid CA 19-9 was detected in case 1. Microscopically, the cystic walls were lined with squamous and cuboidal epithelium, which were surrounded by normal splenic tissue and hyalinized fibrous tissue. The lining squamous epithelium was revealed as nonkeratinizing (Cases 1 and 2) or keratinizing (Case 3). Immunohistochemically, CA19-9 was positive in the monolayer and surface layer of the cuboidal epithelium, but negative for the keratinizing squamous epithelium. As for the histogenesis, it is suggested that the cystic lining of the epithelium may derive from the pancreatic duct which protrudes into the accessory spleen.
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Affiliation(s)
- Kyuichi Kadota
- Department of Diagnostic Pathology, University Hospital, Faculty of Medicine, Kagawa University, 1750-1 Ikenobe, Miki-cho, Kagawa, Japan.
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41
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Kim SH, Lee JM, Han JK, Lee JY, Kim KW, Cho KC, Choi BI. Intrapancreatic accessory spleen: findings on MR Imaging, CT, US and scintigraphy, and the pathologic analysis. Korean J Radiol 2008; 9:162-74. [PMID: 18385564 PMCID: PMC2627219 DOI: 10.3348/kjr.2008.9.2.162] [Citation(s) in RCA: 82] [Impact Index Per Article: 4.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022] Open
Abstract
Although the tail of the pancreas is the second most common site of an accessory spleen, intrapancreatic accessory spleen (IPAS) has rarely been noted radiologically. However, as the imaging techniques have recently advanced, IPAS will be more frequently detected as an incidental pancreatic nodule on CT or MRI. Because accessory spleens usually pose no clinical problems, it is important to characterize accessory spleens as noninvasively as possible. An IPAS has similar characteristics to those of the spleen on the precontrast and contrast-enhanced images of all the imaging modalities. In particular, inhomogeneous enhancement of an IPAS in its early phases may be a diagnostic clue. Superparamagnetic iron oxide (SPIO)-enhanced MRI and Levovist-enhanced US, and the mechanisms of which are theoretically similar to that of Tc-99m scintigraphy, can be used as alternative tools to confirm the diagnosis of IPAS. An IPAS shows a significant signal drop similar to the spleen on the SPIO-enhanced T2 or T2*-weighted imaging and prolonged enhancement on the delayed hepatosplenic phase of contrast-enhanced US. We review and illustrate the differential points between IPAS and hypervascular pancreatic tumors in this manuscript.
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Affiliation(s)
- Se Hyung Kim
- Department of Radiology, Seoul National University Hospital, Seoul, Korea
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42
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Itano O, Shiraga N, Kouta E, Iri H, Tanaka K, Hattori H, Suzuki F, Otaka H. Epidermoid cyst originating from an intrapancreatic accessory spleen. ACTA ACUST UNITED AC 2008; 15:436-9. [PMID: 18670847 DOI: 10.1007/s00534-007-1243-4] [Citation(s) in RCA: 28] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/08/2006] [Accepted: 03/05/2007] [Indexed: 02/05/2023]
Abstract
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.
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Affiliation(s)
- Osamu Itano
- Department of Surgery, KKR Tachikawa Hospital, Tokyo, Japan
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43
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Giant epidermoid cyst within an intrapancreatic accessory spleen mimicking a cystic neoplasm of the pancreas: case report and review of the literature. Pancreas 2008; 36:98-100. [PMID: 18192891 DOI: 10.1097/mpa.0b013e3181359e36] [Citation(s) in RCA: 29] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/19/2023]
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Ru K, Kalra A, Ucci A. Epidermoid cyst of intrapancreatic accessory spleen. Dig Dis Sci 2007; 52:1229-32. [PMID: 17385039 DOI: 10.1007/s10620-006-9376-x] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/02/2006] [Accepted: 04/05/2006] [Indexed: 02/07/2023]
Affiliation(s)
- Kun Ru
- Department of Pathology Box 802, New England Medical Center, Tufts University School of Medicine, Boston, Massachusetts 02111, USA.
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45
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Affiliation(s)
- J A A Awe
- Surgical Outpatient Department, Northern Area Armed Forces Hospital, King Khalid Military City, Hafr Al-Batin, Saudi Arabia.
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46
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Hashimoto T, Sugino T, Fukuda T, Hoshi N, Ogura G, Watanabe K, Satoh Y, Hasegawa T, Ohishi A, Haga J, Miyata M, Suzuki T. Multiple epithelial cysts of the spleen and on the splenic capsule, and high serum levels of CA19-9, CA125 and soluble IL-2 receptor. Pathol Int 2004; 54:349-54. [PMID: 15086840 DOI: 10.1111/j.1440-1827.2004.01630.x] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Abstract
An 18-year-old woman with abdominal pain was diagnosed as having splenic cysts by computed tomography scan. She had high serum levels of CA19-9 (2886.8 U/mL; normal value, <35 U/mL), CA125 (131.1 U/mL; normal value, <35 U/mL) and soluble IL-2 receptor (1490 U/mL; normal range, 220-530 U/mL). The resected spleen weighed 1050 g, was 14 x 28 cm, and had more than 10 macroscopic cysts up to 10.3 x 9.5 cm. There were numerous microscopic cysts in the spleen and several on the splenic capsule. The levels of CA19-9 and CA125 in the cyst fluid were 2165550 U/mL and 160400 U/mL, respectively. After the surgery, the serum levels of the tumor markers decreased gradually. The inside of the largest cyst was mainly covered by granulation tissue with a focal lining of epithelial cells, and the other macroscopic cysts had stratified squamous epithelium. The microscopic splenic cysts and cysts on the splenic capsule were lined by either attenuated single-layered or multilayered epithelial cells. The lining epithelial cells of these cysts were positive for epithelial membrane antigen and cytokeratins. CA19-9 and CA125 were detected in the lining cells of the splenic cysts. In the present case, it is suspected that the splenic cysts were derived from the capsular lining cells that showed migration from the capsule or formed microcysts on the splenic capsule, as in the case of ovarian inclusion cysts.
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Affiliation(s)
- Takeaki Hashimoto
- Department of Pathology, Fukushima Medical University, School of Medicine, Fukushima, Japan
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47
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Watanabe H, Yamaguchi Y, Ohtsubo K, Mouri H, Motoo Y, Yamashita K, Minamoto T, Gabata T, Sawabu N. Epidermoid cyst of the intrapancreatic accessory spleen producing CA19-9. Dig Endosc 2004. [DOI: 10.1111/j.1443-1661.2004.00347.x] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/23/2023]
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Peres LC, Barbosa GHTDS, Careta RS, Nassif CM, de Pina-Neto JM, Giuliani LR, Martinhago CD, Gomy I. Splenopancreatic field abnormality is not unique to trisomy 13. Pediatr Dev Pathol 2004; 7:91-4. [PMID: 15255041 DOI: 10.1007/s10024-003-6067-x] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
Abstract
Splenopancreatic fusion is an uncommon finding, usually only seen as part of the splenopancreatic field abnormality associated with trisomy 13. It may present itself either as ectopic splenic tissue in the cauda pancreatis, as ectopic pancreatic tissue in the spleen or accessory spleen, or as fusion of the cauda pancreatis and splenic hilum. In this study, we report four unrelated congenital anomaly cases presenting trisomy 21, osteocraniostenosis syndrome, isolated congenital heart defect, and oligohydramnios sequence due to prune belly syndrome, in which fusion was observed. This demonstrates that, although it may be more common in trisomy 13, this phenomenon should not be interpreted as pathognomonic to that syndrome.
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Affiliation(s)
- Luiz Cesar Peres
- Department of Pathology, Ribeirão Preto Medical School, University of São Paulo, Av. Bandeirantes, 3900, Ribeirão Preto, SP 14049-900, Brazil.
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49
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Adsay NV, Hasteh F, Cheng JD, Bejarano PA, Lauwers GY, Batts KP, Klöppel G, Klimstra DS. Lymphoepithelial cysts of the pancreas: a report of 12 cases and a review of the literature. Mod Pathol 2002; 15:492-501. [PMID: 12011254 DOI: 10.1038/modpathol.3880553] [Citation(s) in RCA: 105] [Impact Index Per Article: 4.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/17/2023]
Abstract
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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Affiliation(s)
- N Volkan Adsay
- Department of Pathology, Karmanos Cancer Institute, Wayne State University School of Medicine, Detroit, Michigan, USA.
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Horibe Y, Murakami M, Yamao K, Imaeda Y, Tashiro K, Kasahara M. Epithelial inclusion cyst (epidermoid cyst) formation with epithelioid cell granuloma in an intrapancreatic accessory spleen. Pathol Int 2001; 51:50-4. [PMID: 11148465 DOI: 10.1046/j.1440-1827.2001.01155.x] [Citation(s) in RCA: 42] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
The histologic characteristics of a case of epidermoid cyst formation with an epithelioid cell granuloma that developed in intrapancreatic accessory spleen were investigated, with the aim of clarifying its origin as well as etiologic factors. The patient, a 48-year-old male, was found to have a cystic lesion in the tail of the pancreas and renal cell carcinoma (clear cell carcinoma) during a medical check up. The pancreatic mass appeared to be a so-called "mucinous tumor" on imaging, and combined resection of the body and tail of the pancreas and the spleen was performed together with a left nephrectomy. The lesion in the tail of the pancreas was then demonstrated to have accessory splenic tissue with cyst formation in its central region. The cystic wall was covered with stratified squamous epithelium and ductal epithelium with squamous metaplasia, and pancreatic islet cells were evident at various sites within the accessory spleen. Formation of epithelioid cell granuloma tissue was observed around the cysts. The epithelium of the cyst was positive for CA19-9 and negative for antibodies against mesothelial cells, whereas the pancreatic island cells were positive for insulin. These results suggested that cystic epithelium in the accessory spleen could be derived from pancreatic duct epithelium. Frequent recurrence of chronic inflammation and epitheloid cell granuloma formation may have resulted from an aberration of the ectopic remaining pancreatic tissue in the spleen.
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Affiliation(s)
- Y Horibe
- Department of Pathology, Clinical Laboratory, Fujita Health University Second Teaching Hospital, Oto-Bashi, Nakagawa-ward, Nagoya-city, Aichi 454-8509, Japan.
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