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Intraductal oncocytic papillary carcinoma with invasion arising from the accessory pancreatic duct
Intraductal oncocytic papillary carcinoma with invasion arising from the accessory pancreatic duct Bunsei Nobukawa, MD, Koichi Suda, MD, Masafumi Suyama, MD, Joe Ariyama, MD, Tomoo Beppu, MD, Shunji Futagawa, MD
Mucin-producing tumors of the pancreas were first reported by Ohhashi and Takagi in 1980. 1 Since then, many cases of intraductal papillarymucinous tumors (IMPTs) of the pancreas, which are similar to mucin-producing tumors of the pancreas, have been reported.2 IMPTs are generally regarded as tumors with a favorable prognosis. However, those with associated infiltration, noted in up to 25% of cases, are often mucinous and clinically indolent.3 Furthermore, some IMPTs exhibit ductal type infiltration and these are always associated with a poor prognosis.4 Intraductal oncocytic papillary neoplasms (IOPNs) of the pancreas have also been reported; these have the potential to develop into invasive carcinoma.5 Most IMPTs arise from the main pancreatic duct and IMPTs arising from the accessory pancreatic duct are relatively rare, there being only 6 reported cases.2,6-10 We present a case of invasive IPON arising from the accessory pancreatic duct. CASE REPORT A 51-year-old Japanese man with dilation of the main pancreatic duct was identified by health screening. He did not have abdominal pain or discomfort. Physical examination and laboratory studies including tumor markers disclosed no abnormal findings. Endoscopically, an enlarged minor papilla was identified and a papillary tumor extending from the orifice into the duodenum was observed. The major papilla was normal. Although a dilated main pancreatic duct was demonstrated by ERCP, the accessory pancreatic dust was not opacified (Fig. 1). Endoscopic duodenal biopsies of the tumor revealed a papillary adenocarcinoma. Pancreatoduodenectomy was performed (Fig. 2). On the cut surface of the resected specimen, most of the pancreatic parenchyma was well preserved because the tumor was located only in the dorsal pancreas near the minor papilla (Fig. 3). Histologically, the tumor had intraductal papillary projections and an
From the First Department of Pathology, Department of Gastroenterology, and Second Department of Surgery, Juntendo University School of Medicine, Tokyo, Japan. Reprint requests: Bunsei Nobukawa, MD, Juntendo University School of Medicine, First Department of Pathology, 2-1-1, Hongo, Bunkyo-ku, Tokyo 113-8421, Japan; fax: 81-3-3812-1056; e-mail: [email protected]. Copyright © 1999 by the American Society for Gastrointestinal Endoscopy 0016-5107/99/$8.00 + 0 37/54/100601 864
GASTROINTESTINAL ENDOSCOPY
Figure 1. Endoscopic retrograde pancreatogram showing a dilated main pancreatic duct but no opacification of the accessory pancreatic duct. oncocytic cytoplasm. The atypical cytology was severe enough to allow a diagnosis of carcinoma (Fig. 4A). The papillary tumor invaded directly into the duodenal wall and was exposed on the duodenal mucosa. Within the pancreatic parenchyma near the accessory pancreatic duct, a mixed mucinous and ductal infiltrative pattern was identified. Lymphogenous metastases (4 of 29, positive/negative nodes) were identified in the peripancreatic lymph nodes (Fig. 4B). Unfortunately, the patient died postoperatively due to leakage of pancreatic juice from the pancreatico-jejunostomy. This complication might have been related to the well-preserved pancreatic parenchyma because the tumor was located only in the dorsal pancreas near the minor papilla. Requests for autopsy were refused.
DISCUSSION Our patient had an invasive IPMT with oncocytic cytoplasms arising from the accessory pancreatic duct near the minor papilla. Abe et al.2 reported a case of IPMT arising in a branch of the accessory pancreatic duct and suggested that tumor secretions possibly accelerated hyperplasia adjacent to the minor papilla. In IPMTs, several histologic features including hyperplasia, adenoma, and carcinoma are often observed in the same lesion, findings that support the concept of intra-lesional progression.11 In our case, the tumor consisted only of malignant cells that were equally oncocytic, with little mucin production. Adsay et al.5 reported the clinical and pathologic features of 11 IOPNs and concluded that these tumors are distinctive pancreatic carcinomas that were usually intraductal but might develop into invasive carcinoma.5 The tumor cells of IOPNs are basophilic on H&E-stained sections and are packed with mitochondria.5 IOPNs have papillary projection like IPMTs, but they have relatively less mucus and are more aggressive than IPMTs. VOLUME 50, NO. 6, 1999
Brief Reports
B Nobukawa, K Suda, M Suyama, et al.
A Figure 2. Gross appearance of resected specimen. The enlarged minor papilla showing invasive papillary tumor (arrow) is exposed from the duodenum. The major papilla seen below the enlarged minor papilla is normal.
B Figure 4. Histologic features of the tumor. A, Marked papillary projections: the tumor cells are cytologically atypical with an oncocytic cytoplasm (H&E, orig. mag. ×200). B, Oncocytic carcinoma metastases in a peripancreatic lymph nodes (H&E, orig. mag. ×4). Figure 3. Magnified view of resected specimen. The papillary tumor arising from the accessory pancreatic duct invaded the near pancreatic parenchyma and was exposed on the duodenal mucosa (H&E, orig. mag. ×1.2). MPD, Main pancreatic duct; APD, accessory pancreatic duct; CBD, common bile duct.
Oncocytic features can be found in some of IPMTs. Intraductal tumors of the pancreas showing invasive growth and progressing to mucinous carcinomas and/or invasive ductal carcinomas had been reported.3,12,13 The number of documented cases of invasive carcinomas with predominant intraductal components, derived from IPMTs, have increased in recent years. IPMTs show various histologic patterns of development and progression. Although IPMTs arising from the accessory pancreatic duct and IOPNs are relatively rare, they might be considered a variant of IPMTs histopathologically. Presented here is a case of intraductal oncocytic papillary carcinoma with invasion arising from the accessory pancreatic duct that is thought to be a variant of IPMT. VOLUME 50, NO. 6, 1999
ACKNOWLEDGMENTS We thank David B. Douglas, PhD, for reviewing the manuscript. REFERENCES 1. Ohhashi K, Takagi K. ERCP and imaging diagnosis of pancreatic cancer [in Japanese with English abstract]. Endoscopy 1980;77:1493-5. 2. Abe H, Kubota K, Takayama T, Kimura W, Makuuchi M. Pancreatic mucin- producing tumor arising in the embryologically dorsal component of the head. Int J Pancreatol 1998; 23:77-80. 3. Adsay V, Conlon K, Brennan M, Klimstra D. Intraductal papillary-mucinous neoplasms of the pancreas (IPMN): an analysis of in situ and invasive carcinomas associated with 23 cases [abstract]. Mod Pathol 1997;10:143A. 4. Kimura W, Sasahira N, Yoshikawa T, Muto T, Makuuchi M. Duct-ectatic type of mucin producing tumors of the pancreas: new concept of pancreatic neoplasia. Hepatogastroenterology 1996;42:692-709 . 5. Adsay NV, Adair CF, Heffess CS, Klimstra DS. Intraductal oncocytic papillary neoplasms of the pancreas. Am J Surg Pathol 1996;20:980-94. 6. Shigeyama K, Nakano S, Takeda I, Kozuka H, Kumada T, Miyazawa Y, et al. Clinical study of 4 cases with mucin proGASTROINTESTINAL ENDOSCOPY
865
Brief Reports
7.
8. 9.
10.
866
ducing carcinoma of the pancreas [in Japanese with English abstract]. J Jpn Pancreatic Soc 1987;2:1-8. Abe Y, Okuyama K, Onoda S, Kohzu T, Ryu M, Isono K. A case report of mucin producing cancer of the pancreas originated from accessory duct [in Japanese]. Surg Diag Treat (Tokyo) 1988;30:1586-9. Takada Y, Tada H. Mucin producing tumor of the pancreas [in Japanese]. Curr Ther 1989;7:1255-64. Horiuchi S, Sato M, Muneta K, Watanabe Y, Ueda N. Two cases of mucin producing cancer of the pancreas [in Japanese]. Surgery (Tokyo) 1992;54:198-200. Sakabe T, Yokoi S, Suzuki M, Niimi Y, Kanda H, Oda T, et al.
GASTROINTESTINAL ENDOSCOPY
An incidental case of mucin producing cancer of the pancreas, originated from accessory duct [abstract in Japanese]. J Jpn Soc Clin Surg 1993;54:350. 11. Fujii H, Inagaki M, Kasai S, Miyokawa N, Tokusashi, Gabrielson E, et al. Genetic progression and heterogeneity in intraductal papillary-mucinous neoplasms of the pancreas. Am J Pathol 1997;151:1447-54. 12. Matsukuma S, Suda K. Small pancreatic adenocarcinomas: clinicopathological analysis with immunohistochemical and histochemical evaluation. Pathol Inter 1996;46:581-8. 13. Milchgrub S, Campuzano M, Casillas J, Albores-Ssaavedra J. Intraductal carcinoma of the pancreas. Cancer 1992;69:651-6.
VOLUME 50, NO. 6, 1999
Mucin-producing tumors of the pancreas were first reported by Ohhashi and Takagi in 1980. 1 Since then, many cases of intraductal papillarymucinous tumors (IMPTs) of the pancreas, which are similar to mucin-producing tumors of the pancreas, have been reported.2 IMPTs are generally regarded as tumors with a favorable prognosis. However, those with associated infiltration, noted in up to 25% of cases, are often mucinous and clinically indolent.3 Furthermore, some IMPTs exhibit ductal type infiltration and these are always associated with a poor prognosis.4 Intraductal oncocytic papillary neoplasms (IOPNs) of the pancreas have also been reported; these have the potential to develop into invasive carcinoma.5 Most IMPTs arise from the main pancreatic duct and IMPTs arising from the accessory pancreatic duct are relatively rare, there being only 6 reported cases.2,6-10 We present a case of invasive IPON arising from the accessory pancreatic duct. CASE REPORT A 51-year-old Japanese man with dilation of the main pancreatic duct was identified by health screening. He did not have abdominal pain or discomfort. Physical examination and laboratory studies including tumor markers disclosed no abnormal findings. Endoscopically, an enlarged minor papilla was identified and a papillary tumor extending from the orifice into the duodenum was observed. The major papilla was normal. Although a dilated main pancreatic duct was demonstrated by ERCP, the accessory pancreatic dust was not opacified (Fig. 1). Endoscopic duodenal biopsies of the tumor revealed a papillary adenocarcinoma. Pancreatoduodenectomy was performed (Fig. 2). On the cut surface of the resected specimen, most of the pancreatic parenchyma was well preserved because the tumor was located only in the dorsal pancreas near the minor papilla (Fig. 3). Histologically, the tumor had intraductal papillary projections and an
From the First Department of Pathology, Department of Gastroenterology, and Second Department of Surgery, Juntendo University School of Medicine, Tokyo, Japan. Reprint requests: Bunsei Nobukawa, MD, Juntendo University School of Medicine, First Department of Pathology, 2-1-1, Hongo, Bunkyo-ku, Tokyo 113-8421, Japan; fax: 81-3-3812-1056; e-mail: [email protected]. Copyright © 1999 by the American Society for Gastrointestinal Endoscopy 0016-5107/99/$8.00 + 0 37/54/100601 864
GASTROINTESTINAL ENDOSCOPY
Figure 1. Endoscopic retrograde pancreatogram showing a dilated main pancreatic duct but no opacification of the accessory pancreatic duct. oncocytic cytoplasm. The atypical cytology was severe enough to allow a diagnosis of carcinoma (Fig. 4A). The papillary tumor invaded directly into the duodenal wall and was exposed on the duodenal mucosa. Within the pancreatic parenchyma near the accessory pancreatic duct, a mixed mucinous and ductal infiltrative pattern was identified. Lymphogenous metastases (4 of 29, positive/negative nodes) were identified in the peripancreatic lymph nodes (Fig. 4B). Unfortunately, the patient died postoperatively due to leakage of pancreatic juice from the pancreatico-jejunostomy. This complication might have been related to the well-preserved pancreatic parenchyma because the tumor was located only in the dorsal pancreas near the minor papilla. Requests for autopsy were refused.
DISCUSSION Our patient had an invasive IPMT with oncocytic cytoplasms arising from the accessory pancreatic duct near the minor papilla. Abe et al.2 reported a case of IPMT arising in a branch of the accessory pancreatic duct and suggested that tumor secretions possibly accelerated hyperplasia adjacent to the minor papilla. In IPMTs, several histologic features including hyperplasia, adenoma, and carcinoma are often observed in the same lesion, findings that support the concept of intra-lesional progression.11 In our case, the tumor consisted only of malignant cells that were equally oncocytic, with little mucin production. Adsay et al.5 reported the clinical and pathologic features of 11 IOPNs and concluded that these tumors are distinctive pancreatic carcinomas that were usually intraductal but might develop into invasive carcinoma.5 The tumor cells of IOPNs are basophilic on H&E-stained sections and are packed with mitochondria.5 IOPNs have papillary projection like IPMTs, but they have relatively less mucus and are more aggressive than IPMTs. VOLUME 50, NO. 6, 1999
Brief Reports
B Nobukawa, K Suda, M Suyama, et al.
A Figure 2. Gross appearance of resected specimen. The enlarged minor papilla showing invasive papillary tumor (arrow) is exposed from the duodenum. The major papilla seen below the enlarged minor papilla is normal.
B Figure 4. Histologic features of the tumor. A, Marked papillary projections: the tumor cells are cytologically atypical with an oncocytic cytoplasm (H&E, orig. mag. ×200). B, Oncocytic carcinoma metastases in a peripancreatic lymph nodes (H&E, orig. mag. ×4). Figure 3. Magnified view of resected specimen. The papillary tumor arising from the accessory pancreatic duct invaded the near pancreatic parenchyma and was exposed on the duodenal mucosa (H&E, orig. mag. ×1.2). MPD, Main pancreatic duct; APD, accessory pancreatic duct; CBD, common bile duct.
Oncocytic features can be found in some of IPMTs. Intraductal tumors of the pancreas showing invasive growth and progressing to mucinous carcinomas and/or invasive ductal carcinomas had been reported.3,12,13 The number of documented cases of invasive carcinomas with predominant intraductal components, derived from IPMTs, have increased in recent years. IPMTs show various histologic patterns of development and progression. Although IPMTs arising from the accessory pancreatic duct and IOPNs are relatively rare, they might be considered a variant of IPMTs histopathologically. Presented here is a case of intraductal oncocytic papillary carcinoma with invasion arising from the accessory pancreatic duct that is thought to be a variant of IPMT. VOLUME 50, NO. 6, 1999
ACKNOWLEDGMENTS We thank David B. Douglas, PhD, for reviewing the manuscript. REFERENCES 1. Ohhashi K, Takagi K. ERCP and imaging diagnosis of pancreatic cancer [in Japanese with English abstract]. Endoscopy 1980;77:1493-5. 2. Abe H, Kubota K, Takayama T, Kimura W, Makuuchi M. Pancreatic mucin- producing tumor arising in the embryologically dorsal component of the head. Int J Pancreatol 1998; 23:77-80. 3. Adsay V, Conlon K, Brennan M, Klimstra D. Intraductal papillary-mucinous neoplasms of the pancreas (IPMN): an analysis of in situ and invasive carcinomas associated with 23 cases [abstract]. Mod Pathol 1997;10:143A. 4. Kimura W, Sasahira N, Yoshikawa T, Muto T, Makuuchi M. Duct-ectatic type of mucin producing tumors of the pancreas: new concept of pancreatic neoplasia. Hepatogastroenterology 1996;42:692-709 . 5. Adsay NV, Adair CF, Heffess CS, Klimstra DS. Intraductal oncocytic papillary neoplasms of the pancreas. Am J Surg Pathol 1996;20:980-94. 6. Shigeyama K, Nakano S, Takeda I, Kozuka H, Kumada T, Miyazawa Y, et al. Clinical study of 4 cases with mucin proGASTROINTESTINAL ENDOSCOPY
865
Brief Reports
7.
8. 9.
10.
866
ducing carcinoma of the pancreas [in Japanese with English abstract]. J Jpn Pancreatic Soc 1987;2:1-8. Abe Y, Okuyama K, Onoda S, Kohzu T, Ryu M, Isono K. A case report of mucin producing cancer of the pancreas originated from accessory duct [in Japanese]. Surg Diag Treat (Tokyo) 1988;30:1586-9. Takada Y, Tada H. Mucin producing tumor of the pancreas [in Japanese]. Curr Ther 1989;7:1255-64. Horiuchi S, Sato M, Muneta K, Watanabe Y, Ueda N. Two cases of mucin producing cancer of the pancreas [in Japanese]. Surgery (Tokyo) 1992;54:198-200. Sakabe T, Yokoi S, Suzuki M, Niimi Y, Kanda H, Oda T, et al.
GASTROINTESTINAL ENDOSCOPY
An incidental case of mucin producing cancer of the pancreas, originated from accessory duct [abstract in Japanese]. J Jpn Soc Clin Surg 1993;54:350. 11. Fujii H, Inagaki M, Kasai S, Miyokawa N, Tokusashi, Gabrielson E, et al. Genetic progression and heterogeneity in intraductal papillary-mucinous neoplasms of the pancreas. Am J Pathol 1997;151:1447-54. 12. Matsukuma S, Suda K. Small pancreatic adenocarcinomas: clinicopathological analysis with immunohistochemical and histochemical evaluation. Pathol Inter 1996;46:581-8. 13. Milchgrub S, Campuzano M, Casillas J, Albores-Ssaavedra J. Intraductal carcinoma of the pancreas. Cancer 1992;69:651-6.
VOLUME 50, NO. 6, 1999