Anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound-guided fine-needle aspiration: a case report and review of the literature

Anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound-guided fine-needle... Background: Anaplastic carcinoma of the pancreas is a rare pancreatic neoplasm with a poor prognosis. It is classified as a variant of ductal adenocarcinoma, but the clinical features and treatment of it remain unknown because of its rarity and aggressiveness. Endoscopic ultrasonography and endoscopic ultrasound-guided fine- needle aspiration are useful techniques for the diagnosis of pancreatic tumors with high sensitivity and specificity. Case presentation: A 72-year-old Japanese woman presented with a diagnosis of acute pancreatitis, and a cystic lesion with slightly high density area was observed by computed tomography in her pancreatic head. In addition, endoscopic ultrasound revealed a heterogeneous lesion. Endoscopic ultrasound-guided fine-needle aspiration showed pleomorphic atypical cells. We diagnosed anaplastic carcinoma of the pancreas. We resected the lesion, and she has shown no sign of recurrence for > 6 months. There are few reports of anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound-guided fine-needle aspiration and treated by surgery. Our analysis indicates that anaplastic carcinoma of the pancreas is more likely than typical ductal carcinomas to have cystic lesions with the tumor. Conclusions: We report a case of anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound- guided fine-needle aspiration and subsequently resected with a clear margin. We speculate that anaplastic carcinoma of the pancreas is more likely to have cystic changes than pancreatic ductal adenocarcinoma. When we diagnose pancreas tumor as having cystic changes, anaplastic carcinoma of the pancreas should be considered one of the differential diagnoses. Keywords: Anaplastic carcinoma of the pancreas, EUS-FNA, Cystic change * Correspondence: keninoue71@koto.kpu-m.ac.jp Department of Gastroenterology and Hepatology, Fukuchiyama City Hospital, 231 Atsunaka-cho, Fukuchiyama-city, Kyoto 620-8505, Japan Full list of author information is available at the end of the article © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 2 of 6 Background cystic lesion in the pancreatic head. They showed the dila- Anaplastic carcinoma of the pancreas (ACP) is a rare pan- tation of the MPD from the body to the tail of her pan- creatic neoplasm with a poor prognosis [1, 2]. It is classi- creas. We could not identify a connection between the fied as a variant of ductal adenocarcinoma, but the clinical cystic lesion and the MPD. EUS showed the cystic lesion features and treatment of ACP remain unknown because more clearly than other modalities. EUS revealed that the of its rarity and aggressiveness. Endoscopic ultrasonog- cystic lesion consisted of both solid and cystic lesions. The raphy (EUS) and endoscopic ultrasound-guided fine- solid area was shown as a hypoechoic and heterogeneous needle aspiration (EUS-FNA) are useful techniques for the tumor, and the cystic area was shown as an anechoic le- diagnosis of pancreatic tumors with high sensitivity and sion. The EUS also showed that the MPD was dilated to 5 specificity [3, 4]. There are case reports describing ACP, mm, and it was cut off around the mass in the pancreatic but only a few cases of ACP diagnosed by EUS-FNA have head. Endoscopic retrograde cholangiopancreatography been reported. Here, we report the case of a patient with (ERCP) showed > 12-mm-long stenosis of the MPD in the ACP diagnosed by EUS-FNA who subsequently underwent pancreatic head. The stenosis prevented a brush for resection. We also discuss the characteristics of ACP, espe- cytology passing the stricture, and it was not possible to cially in EUS imaging, in a comparison with pancreatic obtain a cytology specimen. ductal adenocarcinoma (PDAC). We performed EUS-FNA (Fig. 2). An echoendoscope (GF-UCT260, Olympus; Tokyo, Japan) with a 22-gauge Case presentation needle was used to obtain cytological material: EchoTip A 72-year-old Japanese woman presented with complaints ProCore® HD Ultrasound Needle (ECHO-HD-22-C; of epigastric pain and nausea. The pain had started 3 days Cook Medical, USA). We carried out two fine-needle as- before admission and gradually worsened. Laboratory test- piration (FNA) punctures, moving ten times in each ing showed a high level of serum amylase (AMY) 838 IU/l puncture. We aspirated the solid area of the tumor, and and pancreatic amylase (P-AMY) 778 IU/l. CA 19-9 was then the cells were histologically revealed as pleo- elevated to 86.4 U/ml, but other tumor markers were nor- morphic atypical cells. Immunohistochemical stains were mal. Computed tomography (CT) scanning showed in- positive for cytokeratin (CK) AE1/AE3 and CK CAM5.2, flammation localized in the pancreatic head and dilatation which confirmed they were epithelial cells. Based on of the main pancreatic duct (MPD) (Fig. 1). In addition, a these findings, we diagnosed the tumor as an ACP. cystic lesion with a slightly high density area was observed Our patient’s abdominal pain and the elevation of pan- by CT in the pancreatic head. creatic enzyme improved (Fig. 3). CT images suggested Transabdominal ultrasonography and magnetic reson- tumor invasion to the front of her pancreas. Positron ance cholangiopancreatography (MRCP) were performed. emission tomography (PET)-CT and gadolinium- Both ultrasonography and MRCP demonstrated a 14 mm ethoxybenzyl-diethylenetriamine pentaacetic acid (Gd- a b c Fig. 1 a, b CT showed a cystic mass in the pancreatic head (arrow). Slight fat stranding suggested that inflammation was localized to the pancreatic head. The MPD was dilated from the body to the tail (arrow). c The cystic mass was demonstrated as a hypo-echoic area under US (arrow). d MRCP showed that the MPD was obstructed by the cystic mass (arrow). e EUS revealed that the masswas 15 mm and comprise of both solid and cystic components (arrow). f ERP showed the MPD was obstructed for a 12-mm length (arrow). There was no obvious communication between the cystic mass and the MPD Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 3 of 6 been shown by EUS. The cystic lesion was pathologically a confirmed as a pancreatic duct with some blood pooling. Hematoxylin-eosin staining showed spindle cells, pleo- morphic cells, and multinuclear osteoclast-like giant cells (OCGCs), which are characteristic of ACP (Fig. 4). The resection was completed with a clear margin. The final diagnosis was ACP according to Japan Pancreas Society (JPS) classification, staged pathologically T3(pS +)N0 M0, pStageIIA, according to the Union for International Cancer Control (UICC) TNM staging system. After discharge from our hospital, she began oral tegafur/gimeracil/oteracil (S-1) intake as adjuvant chemotherapy. Due to anorexia and diarrhea as adverse effects, however, the S-1 administration was discontin- ued after 2 weeks. Although she declined gemcitabine therapy as an alternative, she has shown no evidence of recurrence 6 months after the resection. Discussion ACP is a variant of ductal adenocarcinoma with poor differentiation. It accounts for only 2–7% of all pancre- atic adenocarcinomas [1]. It is so aggressive that its me- dian survival time is only 5.7 months (n = 18) [2]. Our patient’s ACP showed rapid progression. Although the EUS indicated that the tumor size was 15 mm, it turned out to be 25 mm at the time of surgery at approximately 1 month after the EUS-FNA. This is characteristic of sarcoma-like progression, but ACP is classified as a sub- type of ductal tumors because of the presence of ductal carcinoma cells. ACPs are pathologically classified into three variants: pleomorphic type, spindle cell type, and ACP with OCGCs. ACP with OCGCs has an exception- ally better prognosis than the other subtypes. There are many case reports describing ACP, but only a few cases of ACP diagnosed by EUS-FNA have been re- ported. We performed a literature search with the terms “Anaplastic carcinoma pancreas” and “EUS-FNA”,using PubMed database for the period from 2006 to September 2016. We found seven reports of 19 cases of ACP during that period diagnosed by EUS-FNA (including our patient; Table 1)[5–11]. In most of the cases, the tumors were shown as hypoechoic and heterogeneous by EUS. The fea- Fig. 2 Findings obtained by endoscopic ultrasound-guided fine-needle tures of their components varied; of the components, 30% aspiration. a Endoscopic ultrasound-guided fine-needle aspiration was (3 of 10) were a fully solid mass and 60% (6 of 10) were performed to obtain cytology for the solid mass in the pancreatic head. mixed with solid and cystic lesions. b Histology showed pleomorphic large atypical cells (hematoxylin-eosin, It is reported that ACPs are more likely than PDACs magnitude × 400). c Cytokeratin AE1/AE3 stain was positive, and thus these cells were epithelial cells (cytokeratin AE1/AE3, magnitude × 400) to have a cystic appearance [2, 12]. It is assumed that the aggressiveness of ACP may induce cystic change be- cause of central necrosis or degeneration [2]. Although EOB-DTPA)- enhanced MRI showed no obvious metas- these cystic changes may occur not only in ACP but also tasis. We performed a subtotal stomach-preserving PDAC, such a change occurs in < 1% of PDACs [13]. pancreaticoduodenectomy, and no major complications ACPs should thus be considered a differential diagnosis occurred. On macroscopic examination, the tumor con- in patients with a pancreatic tumor with a cystic lesion. sisted of a yellow nodular mass with a cystic lesion, as had We can also find cystic degeneration in other pancreatic Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 4 of 6 Fig. 3 The patient’s clinical course. This graph shows serum level of amylase and 10-point numerical rating scale on abdominal pain. Both of them improved immediately, except for temporary elevation of serum level of amylase after the endoscopic retrograde cholangiopancreatogra- phy procedure. AMY serum amylase, ERCP endoscopic retrograde cholangiopancreatography, EUS-FNA endoscopic ultrasound-guided fine-needle aspiration, NRS numerical rating scale tumors such as adenosquamous carcinoma, neuroendo- detecting ACP because of the high resolution of images, crine tumors, solid and pseudopapillary tumors, acinar as has been reported for PDAC. EUS-FNA is useful for cell carcinoma, and so on [14–16]. In our case, ACP making a pathological diagnosis of pancreatic tumors, but might have induced cystic change due to central necrosis there are some risks for dissemination [3, 17]. or degeneration into a pancreatic duct, because the cys- There is no consensus regarding the optimal treatment tic lesion was pathologically a pancreatic duct. for ACPs, due to the lack of data and evidence. It is re- In contrast, CT images of an ACP may show features ported that ACP is rare and aggressive with shorter comparable to those of PDACs. Compared to PDACs, overall survival times than PDAC [2]. This result sug- ACPs are more hypervascular tumors [8]. It is reported gests that an early diagnosis of ACP should be made for that ACPs often present as a mass with peripheral contrast better outcomes. Chemotherapy for ACP is also uncer- enhancement in the portal venous phase [2, 12]. In our tain. There is a report of a patient with ACP who passed patient’s case, a cystic lesion with slightly high density area a chemosensitivity test and was treated with paclitaxel was observed by CT. EUS showed the cystic lesion more [11]. Khashab et al. suggested that in light of the hyper- clearly than other modalities. EUS also might be useful for vascularity of ACPs, investigational therapies with agents a c e b d f Fig. 4 Resected specimen findings. a The tumor was not clearly exposed to the surface of the pancreas. b The tumor consisted of a yellow nodular mass with the cystic lesion in the center of the mass. The cystic lesion was pathologically a pancreatic duct. A pancreatic calculus was found in this specimen. c, d Hematoxylin-eosin staining showed spindle cells and multinuclear giant cells, which are characteristic of anaplastic carcinoma of the pancreas. e, f These cells exhibited immunoreactivity for cytokeratin AE1/AE3. The tumor invaded pancreatic anterior fat tissue Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 5 of 6 Table 1 Reported cases of anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound-guided fine-needle aspiration Author Reference Age/Sex sites Size (mm) Echogenicity Homogeneity Solid or Cystic Pathology Treatment Chopra et al.[6] 89 M Head 20 Hypoechoic Heterogeneous – OGC BSC 64 F Head 31 Hypoechoic Heterogeneous Mixed OGC Surgery Layfield et al.[9] 71 M Head –– – – Pleomorphic BSC 81 F Head –– – – Pleomorphic BSC 59 M Head –– – – Pleomorphic BSC 81 M Head –– – – Pleomorphic BSC 59 M Head –– – – OGC Surgery 64 M Body –– – – Pleomorphic BSC Salla et al.[10] 44 F Tail 50 Hypoechoic Heterogeneous Mixed Pleomorphic – Khashab et al.[8] 60 M Head 43 Hypoechoic Heterogeneous Mixed – Chemotherapy 49 M Body 58 Hypoechoic Heterogeneous Mixed – Surgery 44 M Tail 41 Hypoechoic Heterogeneous Mixed – Chemotherapy 85 M Head 20 Hypoechoic Homogeneous Solid – Surgery 58 M Body 34 Hypoechoic Heterogeneous Solid – NAC + surgery 65 M Head 100 Hypoechoic Heterogeneous Solid – BSC Wakatsuki et al.[11] 58 F Head –– – – Chemotherapy Ergun et al.[7] 60 M Head 32 Hypoechoic Heterogeneous – Chemotherapy Aldaoud et al.[5] 37 F Body 160 Hypoechoic Homogeneous Cystic Pleomorphic Surgery + adj Our case 72 F Head 25 Hypoechoic Heterogeneous Mixed Unclassifiable Surgery Adj adjuvant chemotherapy, BSC best supportive care, F female, M male, NAC Neoadjuvant chemotherapy, OGC osteoclast-like giant cell such as bevacizumab may improve the outcome of pa- Funding Not applicable. tients with ACP [8]. There is no evidence regarding ad- juvant chemotherapy for ACP. We administered S-1 to Availability of data and materials our patient based on a phase 3 trial for PDAC. Not applicable. Authors’ contributions Conclusions All authors were involved in the care of the patient. KO, KI, TO, and KK We report a case of ACP diagnosed by EUS-FNA and contributed equally to drafting, revision, and preparation of the manuscript. KO, KI, and TO performed the EUS-FNA. SN and SK performed the operation. SN has subsequently resected with a clear margin. We speculate followed up this patient as an out-patient. AN performed the pathological that ACPs are more likely to have cystic changes than diagnosis. All authors read and approved the final manuscript. PDAC, and we found that EUS-FNA was a useful tech- Ethics approval and consent to participate nique to detect this characteristic change and make a def- Not applicable. inite diagnosis. When we diagnose the pancreas tumor as having cystic changes, ACP should be considered one of Consent for publication the differential diagnoses. A further accumulation of cases Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written of ACP must be examined to clarify this disease. consent is available for review by the Editor-in-Chief of this journal. Abbreviations Competing interests ACP: Anaplastic carcinoma of the pancreas; AMY: Serum amylase; The authors declare that they have no competing interests. CK: Cytokeratin; CT: Computed tomography; ERCP: Endoscopic retrograde cholangiopancreatography; EUS: Endoscopic ultrasonography; EUS- FNA: Endoscopic ultrasound-guided fine-needle aspiration; FNA: Fine-needle Publisher’sNote aspiration; Gd-EOB-DTPA: Gadolinium-ethoxybenzyl-diethylenetriamine Springer Nature remains neutral with regard to jurisdictional claims in pentaacetic acid; JPS: Japan Pancreas Society; MPD: Main pancreatic duct; published maps and institutional affiliations. MRCP: Magnetic resonance cholangiopancreatography; OCGCs: Osteoclast- like giant cells; P-AMY: Pancreatic amylase; PDAC: Pancreatic ductal Author details adenocarcinoma; PET: Positron emission tomography; S-1: Tegafur/gimeracil/ Department of Gastroenterology and Hepatology, Fukuchiyama City oteracil; UICC: Union for International Cancer Control Hospital, 231 Atsunaka-cho, Fukuchiyama-city, Kyoto 620-8505, Japan. Department of Surgery, Fukuchiyama City Hospital, 231 Atsunaka-cho, Acknowledgements Fukuchiyama-city, Kyoto 620-0056, Japan. Department of Pathology, We are grateful to our patient, who cooperated with the publication of this Fukuchiyama City Hospital, 231 Atsunaka-cho, Fukuchiyama-city, Kyoto report. 620-0056, Japan. Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 6 of 6 Received: 5 June 2017 Accepted: 12 February 2018 References 1. Paal E, Thompson LD, Frommelt RA, Przygodzki RM, Heffess CS. A clinicopathologic and immunohistochemical study of 35 anaplastic carcinomas of the pancreas with a review of the literature. Ann Diagn Pathol. 2001;5:129–40. 2. Strobel O, Hartwig W, Bergmann F, Hinz U, Hackert T, Grenacher L, et al. Anaplastic pancreatic cancer: Presentation, surgical management, and outcome. Surgery. 2011;149:200–8. 3. Chen G, Liu S, Zhao Y, Dai M, Zhang T. Diagnostic accuracy of endoscopic ultrasound-guided fine-needle aspiration for pancreatic cancer: a meta- analysis. Pancreatology. 2013;13:298–304. 4. Hewitt MJ, McPhail MJ, Possamai L, Dhar A, Vlavianos P, Monahan KJ. EUS- guided FNA for diagnosis of solid pancreatic neoplasms: a meta-analysis. Gastrointest Endosc. 2012;75:319–31. 5. Aldaoud N, Joudeh A, Al-Momen S, Alnahawi M, Al-Abbadi MA. Anaplastic Carcinoma Arising in a Mucinous Cystic Neoplasm Masquerading as Pancreatic Pseudocyst. Diagn Cytopathol. 2016;44:538–42. 6. Chopra S, Wu ML, Imagawa DK, Lee J, Gu M. Endoscopic ultrasound-guided fine-needle aspiration of undifferentiated carcinoma with osteoclast-like giant cells of the pancreas: a report of 2 cases with literature review. Diagn Cytopathol. 2007;35:601–6. 7. Ergun M, Aydog G, KayaÇEtin E, Sasmaz N. A case of anaplastic carcinoma of pancreas diagnosed with endoscopic ultrasound-guided fine needle aspiration cytology. Turk J Gastroenterol. 2012;23:828–9. 8. Khashab MA, Emerson RE, DeWitt JM. Endoscopic ultrasound-guided fine- needle aspiration for the diagnosis of anaplastic pancreatic carcinoma: a single-center experience. Pancreas. 2010;39:88–91. 9. Layfield LJ, Bentz J. Giant-cell containing neoplasms of the pancreas: an aspiration cytology study. Diagn Cytopathol. 2008;36:238–44. 10. Salla C, Sakellariou S, Doumani I, Kakiopoulos G, Chatzipantelis P, Karoumpalis I. Neuroendocrine differentiation of anaplastic carcinoma of the pancreas in a case report diagnosed by endoscopic ultrasound-guided fine- needle aspiration cytology. Pancreas. 2009;38:103–5. 11. Wakatsuki T, Irisawa A, Imamura H, Terashima M, Shibukawa G, Takagi T, et al. Complete response of anaplastic pancreatic carcinoma to paclitaxel treatment selected by chemosensitivity testing. Int J Clin Oncol. 2010;15:310–3. 12. Hoshimoto S, Matsui J, Miyata R, Takigawa Y, Miyauchi J. Anaplastic carcinoma of the pancreas: Case report and literature review of reported cases in Japan. World J Gastroenterol. 2016;22:8631–7. 13. Adsay NV. Cystic neoplasia of the pancreas: pathology and biology. J Gastrointest Surg. 2008;12:401–4. 14. Hu S, Hu S, Wang M, Wu Z, Miao F. Clinical and CT imaging features of pancreatic acinar cell carcinoma. Radiol Med. 2013;118:723–31. 15. Jamali M, Serra S, Chetty R. Adenosquamous carcinoma of the pancreas with clear cell and rhabdoid components. A case report. JOP. 2007;8:330–4. 16. Thosani N, Thosani S, Qiao W, Fleming JB, Bhutani MS, Guha S. Role of EUS- FNA-based cytology in the diagnosis of mucinous pancreatic cystic lesions: a systematic review and meta-analysis. Dig Dis Sci. 2010;55:2756–66. 17. Hartwig W,Schneider L, Diener MK, BergmannF,Buchler MW,Werner J. Preoperative tissue diagnosis for tumours of the pancreas. Br J Surg. 2009;96:5–20. 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Anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound-guided fine-needle aspiration: a case report and review of the literature

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Abstract

Background: Anaplastic carcinoma of the pancreas is a rare pancreatic neoplasm with a poor prognosis. It is classified as a variant of ductal adenocarcinoma, but the clinical features and treatment of it remain unknown because of its rarity and aggressiveness. Endoscopic ultrasonography and endoscopic ultrasound-guided fine- needle aspiration are useful techniques for the diagnosis of pancreatic tumors with high sensitivity and specificity. Case presentation: A 72-year-old Japanese woman presented with a diagnosis of acute pancreatitis, and a cystic lesion with slightly high density area was observed by computed tomography in her pancreatic head. In addition, endoscopic ultrasound revealed a heterogeneous lesion. Endoscopic ultrasound-guided fine-needle aspiration showed pleomorphic atypical cells. We diagnosed anaplastic carcinoma of the pancreas. We resected the lesion, and she has shown no sign of recurrence for > 6 months. There are few reports of anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound-guided fine-needle aspiration and treated by surgery. Our analysis indicates that anaplastic carcinoma of the pancreas is more likely than typical ductal carcinomas to have cystic lesions with the tumor. Conclusions: We report a case of anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound- guided fine-needle aspiration and subsequently resected with a clear margin. We speculate that anaplastic carcinoma of the pancreas is more likely to have cystic changes than pancreatic ductal adenocarcinoma. When we diagnose pancreas tumor as having cystic changes, anaplastic carcinoma of the pancreas should be considered one of the differential diagnoses. Keywords: Anaplastic carcinoma of the pancreas, EUS-FNA, Cystic change * Correspondence: keninoue71@koto.kpu-m.ac.jp Department of Gastroenterology and Hepatology, Fukuchiyama City Hospital, 231 Atsunaka-cho, Fukuchiyama-city, Kyoto 620-8505, Japan Full list of author information is available at the end of the article © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 2 of 6 Background cystic lesion in the pancreatic head. They showed the dila- Anaplastic carcinoma of the pancreas (ACP) is a rare pan- tation of the MPD from the body to the tail of her pan- creatic neoplasm with a poor prognosis [1, 2]. It is classi- creas. We could not identify a connection between the fied as a variant of ductal adenocarcinoma, but the clinical cystic lesion and the MPD. EUS showed the cystic lesion features and treatment of ACP remain unknown because more clearly than other modalities. EUS revealed that the of its rarity and aggressiveness. Endoscopic ultrasonog- cystic lesion consisted of both solid and cystic lesions. The raphy (EUS) and endoscopic ultrasound-guided fine- solid area was shown as a hypoechoic and heterogeneous needle aspiration (EUS-FNA) are useful techniques for the tumor, and the cystic area was shown as an anechoic le- diagnosis of pancreatic tumors with high sensitivity and sion. The EUS also showed that the MPD was dilated to 5 specificity [3, 4]. There are case reports describing ACP, mm, and it was cut off around the mass in the pancreatic but only a few cases of ACP diagnosed by EUS-FNA have head. Endoscopic retrograde cholangiopancreatography been reported. Here, we report the case of a patient with (ERCP) showed > 12-mm-long stenosis of the MPD in the ACP diagnosed by EUS-FNA who subsequently underwent pancreatic head. The stenosis prevented a brush for resection. We also discuss the characteristics of ACP, espe- cytology passing the stricture, and it was not possible to cially in EUS imaging, in a comparison with pancreatic obtain a cytology specimen. ductal adenocarcinoma (PDAC). We performed EUS-FNA (Fig. 2). An echoendoscope (GF-UCT260, Olympus; Tokyo, Japan) with a 22-gauge Case presentation needle was used to obtain cytological material: EchoTip A 72-year-old Japanese woman presented with complaints ProCore® HD Ultrasound Needle (ECHO-HD-22-C; of epigastric pain and nausea. The pain had started 3 days Cook Medical, USA). We carried out two fine-needle as- before admission and gradually worsened. Laboratory test- piration (FNA) punctures, moving ten times in each ing showed a high level of serum amylase (AMY) 838 IU/l puncture. We aspirated the solid area of the tumor, and and pancreatic amylase (P-AMY) 778 IU/l. CA 19-9 was then the cells were histologically revealed as pleo- elevated to 86.4 U/ml, but other tumor markers were nor- morphic atypical cells. Immunohistochemical stains were mal. Computed tomography (CT) scanning showed in- positive for cytokeratin (CK) AE1/AE3 and CK CAM5.2, flammation localized in the pancreatic head and dilatation which confirmed they were epithelial cells. Based on of the main pancreatic duct (MPD) (Fig. 1). In addition, a these findings, we diagnosed the tumor as an ACP. cystic lesion with a slightly high density area was observed Our patient’s abdominal pain and the elevation of pan- by CT in the pancreatic head. creatic enzyme improved (Fig. 3). CT images suggested Transabdominal ultrasonography and magnetic reson- tumor invasion to the front of her pancreas. Positron ance cholangiopancreatography (MRCP) were performed. emission tomography (PET)-CT and gadolinium- Both ultrasonography and MRCP demonstrated a 14 mm ethoxybenzyl-diethylenetriamine pentaacetic acid (Gd- a b c Fig. 1 a, b CT showed a cystic mass in the pancreatic head (arrow). Slight fat stranding suggested that inflammation was localized to the pancreatic head. The MPD was dilated from the body to the tail (arrow). c The cystic mass was demonstrated as a hypo-echoic area under US (arrow). d MRCP showed that the MPD was obstructed by the cystic mass (arrow). e EUS revealed that the masswas 15 mm and comprise of both solid and cystic components (arrow). f ERP showed the MPD was obstructed for a 12-mm length (arrow). There was no obvious communication between the cystic mass and the MPD Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 3 of 6 been shown by EUS. The cystic lesion was pathologically a confirmed as a pancreatic duct with some blood pooling. Hematoxylin-eosin staining showed spindle cells, pleo- morphic cells, and multinuclear osteoclast-like giant cells (OCGCs), which are characteristic of ACP (Fig. 4). The resection was completed with a clear margin. The final diagnosis was ACP according to Japan Pancreas Society (JPS) classification, staged pathologically T3(pS +)N0 M0, pStageIIA, according to the Union for International Cancer Control (UICC) TNM staging system. After discharge from our hospital, she began oral tegafur/gimeracil/oteracil (S-1) intake as adjuvant chemotherapy. Due to anorexia and diarrhea as adverse effects, however, the S-1 administration was discontin- ued after 2 weeks. Although she declined gemcitabine therapy as an alternative, she has shown no evidence of recurrence 6 months after the resection. Discussion ACP is a variant of ductal adenocarcinoma with poor differentiation. It accounts for only 2–7% of all pancre- atic adenocarcinomas [1]. It is so aggressive that its me- dian survival time is only 5.7 months (n = 18) [2]. Our patient’s ACP showed rapid progression. Although the EUS indicated that the tumor size was 15 mm, it turned out to be 25 mm at the time of surgery at approximately 1 month after the EUS-FNA. This is characteristic of sarcoma-like progression, but ACP is classified as a sub- type of ductal tumors because of the presence of ductal carcinoma cells. ACPs are pathologically classified into three variants: pleomorphic type, spindle cell type, and ACP with OCGCs. ACP with OCGCs has an exception- ally better prognosis than the other subtypes. There are many case reports describing ACP, but only a few cases of ACP diagnosed by EUS-FNA have been re- ported. We performed a literature search with the terms “Anaplastic carcinoma pancreas” and “EUS-FNA”,using PubMed database for the period from 2006 to September 2016. We found seven reports of 19 cases of ACP during that period diagnosed by EUS-FNA (including our patient; Table 1)[5–11]. In most of the cases, the tumors were shown as hypoechoic and heterogeneous by EUS. The fea- Fig. 2 Findings obtained by endoscopic ultrasound-guided fine-needle tures of their components varied; of the components, 30% aspiration. a Endoscopic ultrasound-guided fine-needle aspiration was (3 of 10) were a fully solid mass and 60% (6 of 10) were performed to obtain cytology for the solid mass in the pancreatic head. mixed with solid and cystic lesions. b Histology showed pleomorphic large atypical cells (hematoxylin-eosin, It is reported that ACPs are more likely than PDACs magnitude × 400). c Cytokeratin AE1/AE3 stain was positive, and thus these cells were epithelial cells (cytokeratin AE1/AE3, magnitude × 400) to have a cystic appearance [2, 12]. It is assumed that the aggressiveness of ACP may induce cystic change be- cause of central necrosis or degeneration [2]. Although EOB-DTPA)- enhanced MRI showed no obvious metas- these cystic changes may occur not only in ACP but also tasis. We performed a subtotal stomach-preserving PDAC, such a change occurs in < 1% of PDACs [13]. pancreaticoduodenectomy, and no major complications ACPs should thus be considered a differential diagnosis occurred. On macroscopic examination, the tumor con- in patients with a pancreatic tumor with a cystic lesion. sisted of a yellow nodular mass with a cystic lesion, as had We can also find cystic degeneration in other pancreatic Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 4 of 6 Fig. 3 The patient’s clinical course. This graph shows serum level of amylase and 10-point numerical rating scale on abdominal pain. Both of them improved immediately, except for temporary elevation of serum level of amylase after the endoscopic retrograde cholangiopancreatogra- phy procedure. AMY serum amylase, ERCP endoscopic retrograde cholangiopancreatography, EUS-FNA endoscopic ultrasound-guided fine-needle aspiration, NRS numerical rating scale tumors such as adenosquamous carcinoma, neuroendo- detecting ACP because of the high resolution of images, crine tumors, solid and pseudopapillary tumors, acinar as has been reported for PDAC. EUS-FNA is useful for cell carcinoma, and so on [14–16]. In our case, ACP making a pathological diagnosis of pancreatic tumors, but might have induced cystic change due to central necrosis there are some risks for dissemination [3, 17]. or degeneration into a pancreatic duct, because the cys- There is no consensus regarding the optimal treatment tic lesion was pathologically a pancreatic duct. for ACPs, due to the lack of data and evidence. It is re- In contrast, CT images of an ACP may show features ported that ACP is rare and aggressive with shorter comparable to those of PDACs. Compared to PDACs, overall survival times than PDAC [2]. This result sug- ACPs are more hypervascular tumors [8]. It is reported gests that an early diagnosis of ACP should be made for that ACPs often present as a mass with peripheral contrast better outcomes. Chemotherapy for ACP is also uncer- enhancement in the portal venous phase [2, 12]. In our tain. There is a report of a patient with ACP who passed patient’s case, a cystic lesion with slightly high density area a chemosensitivity test and was treated with paclitaxel was observed by CT. EUS showed the cystic lesion more [11]. Khashab et al. suggested that in light of the hyper- clearly than other modalities. EUS also might be useful for vascularity of ACPs, investigational therapies with agents a c e b d f Fig. 4 Resected specimen findings. a The tumor was not clearly exposed to the surface of the pancreas. b The tumor consisted of a yellow nodular mass with the cystic lesion in the center of the mass. The cystic lesion was pathologically a pancreatic duct. A pancreatic calculus was found in this specimen. c, d Hematoxylin-eosin staining showed spindle cells and multinuclear giant cells, which are characteristic of anaplastic carcinoma of the pancreas. e, f These cells exhibited immunoreactivity for cytokeratin AE1/AE3. The tumor invaded pancreatic anterior fat tissue Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 5 of 6 Table 1 Reported cases of anaplastic carcinoma of the pancreas diagnosed by endoscopic ultrasound-guided fine-needle aspiration Author Reference Age/Sex sites Size (mm) Echogenicity Homogeneity Solid or Cystic Pathology Treatment Chopra et al.[6] 89 M Head 20 Hypoechoic Heterogeneous – OGC BSC 64 F Head 31 Hypoechoic Heterogeneous Mixed OGC Surgery Layfield et al.[9] 71 M Head –– – – Pleomorphic BSC 81 F Head –– – – Pleomorphic BSC 59 M Head –– – – Pleomorphic BSC 81 M Head –– – – Pleomorphic BSC 59 M Head –– – – OGC Surgery 64 M Body –– – – Pleomorphic BSC Salla et al.[10] 44 F Tail 50 Hypoechoic Heterogeneous Mixed Pleomorphic – Khashab et al.[8] 60 M Head 43 Hypoechoic Heterogeneous Mixed – Chemotherapy 49 M Body 58 Hypoechoic Heterogeneous Mixed – Surgery 44 M Tail 41 Hypoechoic Heterogeneous Mixed – Chemotherapy 85 M Head 20 Hypoechoic Homogeneous Solid – Surgery 58 M Body 34 Hypoechoic Heterogeneous Solid – NAC + surgery 65 M Head 100 Hypoechoic Heterogeneous Solid – BSC Wakatsuki et al.[11] 58 F Head –– – – Chemotherapy Ergun et al.[7] 60 M Head 32 Hypoechoic Heterogeneous – Chemotherapy Aldaoud et al.[5] 37 F Body 160 Hypoechoic Homogeneous Cystic Pleomorphic Surgery + adj Our case 72 F Head 25 Hypoechoic Heterogeneous Mixed Unclassifiable Surgery Adj adjuvant chemotherapy, BSC best supportive care, F female, M male, NAC Neoadjuvant chemotherapy, OGC osteoclast-like giant cell such as bevacizumab may improve the outcome of pa- Funding Not applicable. tients with ACP [8]. There is no evidence regarding ad- juvant chemotherapy for ACP. We administered S-1 to Availability of data and materials our patient based on a phase 3 trial for PDAC. Not applicable. Authors’ contributions Conclusions All authors were involved in the care of the patient. KO, KI, TO, and KK We report a case of ACP diagnosed by EUS-FNA and contributed equally to drafting, revision, and preparation of the manuscript. KO, KI, and TO performed the EUS-FNA. SN and SK performed the operation. SN has subsequently resected with a clear margin. We speculate followed up this patient as an out-patient. AN performed the pathological that ACPs are more likely to have cystic changes than diagnosis. All authors read and approved the final manuscript. PDAC, and we found that EUS-FNA was a useful tech- Ethics approval and consent to participate nique to detect this characteristic change and make a def- Not applicable. inite diagnosis. When we diagnose the pancreas tumor as having cystic changes, ACP should be considered one of Consent for publication the differential diagnoses. A further accumulation of cases Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written of ACP must be examined to clarify this disease. consent is available for review by the Editor-in-Chief of this journal. Abbreviations Competing interests ACP: Anaplastic carcinoma of the pancreas; AMY: Serum amylase; The authors declare that they have no competing interests. CK: Cytokeratin; CT: Computed tomography; ERCP: Endoscopic retrograde cholangiopancreatography; EUS: Endoscopic ultrasonography; EUS- FNA: Endoscopic ultrasound-guided fine-needle aspiration; FNA: Fine-needle Publisher’sNote aspiration; Gd-EOB-DTPA: Gadolinium-ethoxybenzyl-diethylenetriamine Springer Nature remains neutral with regard to jurisdictional claims in pentaacetic acid; JPS: Japan Pancreas Society; MPD: Main pancreatic duct; published maps and institutional affiliations. MRCP: Magnetic resonance cholangiopancreatography; OCGCs: Osteoclast- like giant cells; P-AMY: Pancreatic amylase; PDAC: Pancreatic ductal Author details adenocarcinoma; PET: Positron emission tomography; S-1: Tegafur/gimeracil/ Department of Gastroenterology and Hepatology, Fukuchiyama City oteracil; UICC: Union for International Cancer Control Hospital, 231 Atsunaka-cho, Fukuchiyama-city, Kyoto 620-8505, Japan. Department of Surgery, Fukuchiyama City Hospital, 231 Atsunaka-cho, Acknowledgements Fukuchiyama-city, Kyoto 620-0056, Japan. Department of Pathology, We are grateful to our patient, who cooperated with the publication of this Fukuchiyama City Hospital, 231 Atsunaka-cho, Fukuchiyama-city, Kyoto report. 620-0056, Japan. Oka et al. Journal of Medical Case Reports (2018) 12:152 Page 6 of 6 Received: 5 June 2017 Accepted: 12 February 2018 References 1. Paal E, Thompson LD, Frommelt RA, Przygodzki RM, Heffess CS. A clinicopathologic and immunohistochemical study of 35 anaplastic carcinomas of the pancreas with a review of the literature. Ann Diagn Pathol. 2001;5:129–40. 2. Strobel O, Hartwig W, Bergmann F, Hinz U, Hackert T, Grenacher L, et al. Anaplastic pancreatic cancer: Presentation, surgical management, and outcome. Surgery. 2011;149:200–8. 3. Chen G, Liu S, Zhao Y, Dai M, Zhang T. Diagnostic accuracy of endoscopic ultrasound-guided fine-needle aspiration for pancreatic cancer: a meta- analysis. Pancreatology. 2013;13:298–304. 4. Hewitt MJ, McPhail MJ, Possamai L, Dhar A, Vlavianos P, Monahan KJ. EUS- guided FNA for diagnosis of solid pancreatic neoplasms: a meta-analysis. Gastrointest Endosc. 2012;75:319–31. 5. Aldaoud N, Joudeh A, Al-Momen S, Alnahawi M, Al-Abbadi MA. Anaplastic Carcinoma Arising in a Mucinous Cystic Neoplasm Masquerading as Pancreatic Pseudocyst. Diagn Cytopathol. 2016;44:538–42. 6. Chopra S, Wu ML, Imagawa DK, Lee J, Gu M. Endoscopic ultrasound-guided fine-needle aspiration of undifferentiated carcinoma with osteoclast-like giant cells of the pancreas: a report of 2 cases with literature review. Diagn Cytopathol. 2007;35:601–6. 7. Ergun M, Aydog G, KayaÇEtin E, Sasmaz N. A case of anaplastic carcinoma of pancreas diagnosed with endoscopic ultrasound-guided fine needle aspiration cytology. Turk J Gastroenterol. 2012;23:828–9. 8. Khashab MA, Emerson RE, DeWitt JM. Endoscopic ultrasound-guided fine- needle aspiration for the diagnosis of anaplastic pancreatic carcinoma: a single-center experience. Pancreas. 2010;39:88–91. 9. Layfield LJ, Bentz J. Giant-cell containing neoplasms of the pancreas: an aspiration cytology study. Diagn Cytopathol. 2008;36:238–44. 10. Salla C, Sakellariou S, Doumani I, Kakiopoulos G, Chatzipantelis P, Karoumpalis I. Neuroendocrine differentiation of anaplastic carcinoma of the pancreas in a case report diagnosed by endoscopic ultrasound-guided fine- needle aspiration cytology. Pancreas. 2009;38:103–5. 11. Wakatsuki T, Irisawa A, Imamura H, Terashima M, Shibukawa G, Takagi T, et al. Complete response of anaplastic pancreatic carcinoma to paclitaxel treatment selected by chemosensitivity testing. Int J Clin Oncol. 2010;15:310–3. 12. Hoshimoto S, Matsui J, Miyata R, Takigawa Y, Miyauchi J. Anaplastic carcinoma of the pancreas: Case report and literature review of reported cases in Japan. World J Gastroenterol. 2016;22:8631–7. 13. Adsay NV. Cystic neoplasia of the pancreas: pathology and biology. J Gastrointest Surg. 2008;12:401–4. 14. Hu S, Hu S, Wang M, Wu Z, Miao F. Clinical and CT imaging features of pancreatic acinar cell carcinoma. Radiol Med. 2013;118:723–31. 15. Jamali M, Serra S, Chetty R. Adenosquamous carcinoma of the pancreas with clear cell and rhabdoid components. A case report. JOP. 2007;8:330–4. 16. Thosani N, Thosani S, Qiao W, Fleming JB, Bhutani MS, Guha S. Role of EUS- FNA-based cytology in the diagnosis of mucinous pancreatic cystic lesions: a systematic review and meta-analysis. Dig Dis Sci. 2010;55:2756–66. 17. Hartwig W,Schneider L, Diener MK, BergmannF,Buchler MW,Werner J. Preoperative tissue diagnosis for tumours of the pancreas. Br J Surg. 2009;96:5–20. Submit your next manuscript to BioMed Central and we will help you at every step: • We accept pre-submission inquiries � Our selector tool helps you to find the most relevant journal � We provide round the clock customer support � Convenient online submission � Thorough peer review � Inclusion in PubMed and all major indexing services � Maximum visibility for your research Submit your manuscript at www.biomedcentral.com/submit

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Journal of Medical Case ReportsSpringer Journals

Published: May 31, 2018

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