Pancreatic metastases are rare, <2% of all pancreatic neoplasia. This is the ﬁrst case of pancreatic metastasis from adaman- tinoma, a rare, low grade and slow growing tumor which is frequently localized in long bones. We describe a case of a 45- year-old woman presenting with increased bilirubin level. Computed tomography and ecoendoscopic ultra sonography revealed a pancreatic head mass. Fine-needle aspiration biopsy was consistent with metastatic adamantinoma. The patient was submitted to a standard pancreaticoduodenectomy. As in the case presented, standard pancreatic resections are safe and feasible options to treat non-pancreatic primary tumor improving patient’s survival and quality of life. We report a case of a female patient who underwent a pan- INTRODUCTION creaticoduodenectomy (PD) for metacronous pancreatic metas- Adamantinoma is rare, low grade and low-growing primary tasis (PM) from tibial adamantinoma (TA). malignant bone tumor of unknown histogenesis: it is fre- quently localized in long bones, especially tibia, and it is a locally aggressive tumor [1, 2]; metastases are present in about CASE REPORT 15−30% of cases, usually to the lungs, bones and nearby lymph nodes ; other metastatic sites are extremely rare and, to our A 45-year-old woman was admitted to the emergency depart- knowledge, cases of pancreatic localization have never been ment (ER) in January 2014 for epigastric pain and jaundice reported yet. (8 mg/dl). In 2012, she underwent surgical amputation of the Received: November 19, 2017. Accepted: January 16, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact firstname.lastname@example.org Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy012/4855836 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 S. Silvestri et al. Figure 1: MDCT scan. (A) CT scan with well-deﬁned hypodense pancreatic head mass of 2 cm of diameter after contrast medium intravenous injection. (B) CT scan which highlight bile duct dilatation. right leg for TA. In 2013, during follow-up, a chest computed tomography (CT) scan detected four bilateral suspected lung metastases therefore she was submitted to a synchronous right superior lobectomy and left apical thoracoscopic lung wedge resection. At histology, it was conﬁrmed that all the lesions were from a metastatic adamantinoma. First the abdominal ultra sonography (AUS) and then CT scan revealed the presence of well-deﬁned hypodense pancreatic head mass of 2 cm of diameter, determining bile duct and Wirsung’s duct dilatation; mesenteric vessels were clearly free of inﬁltration, no liver or lung suspected lesions were detected as well as vol- ume increased lymph nodes (Fig. 1). Ecoendoscopic ultra sonog- raphy (EUS) conﬁrmed the presence of a hypoechoic and well- deﬁned pancreatic head mass (18 × 15 mm) and without sign of vessels inﬁltration (Fig. 2). A ﬁne-needle aspiration (FNA) was per- formed and histopathological examination showed the presence Figure 2: EUS. EUS conﬁrmed the presence of a hypoechoic and well-deﬁned of neoplastic cells with morphological features like the lung ones mass of diameter 18 × 15 mm. and consistent with metastasis from primary adamantinoma. Finally, positron emission tomography conﬁrmed an iso- lated increased standardized uptake value of the radiolabeled from the second to ﬁfth decade of life [1, 3]. The ﬁrst case of tracer in the context of the pancreatic head. adamantinoma was reported by Maier in 1900 and by Fisher in After a complete preoperative work-up and 5 days of oral 1913 [2, 5]. immunonutrition , the patient was submitted to a standard This tumor has marked predilection for the diaphyseal por- PD: during intervention, an hard pancreatic parenchyma was tion of long bones, especially tibia (80–85% of cases), which found, with dilatated Wirsung’s duct. A termino-lateral, hand represents the most characteristic clinical feature and location sewn interrupted stitches pancreatojejunostomy was carried of adamantinoma; other bones that could be involved, in order on as previously described . Post-operative course was of decreasing frequency, are humerus, ulna, femur, ﬁbula, uneventful, and patient was discharged in 10th Post-Operative radius, ribs . Histologically, adamantinoma is composed by Day after PD. epithelial islands in a spindle cell stroma . Final histology conﬁrmed the presence of spindle cell neopla- The clinical manifestations of adamantinoma are often sia in the pancreatic head mass. Immunohistochemical staining indolent and aspeciﬁc. Their onset is insidious, with a gradually was also performed with antibody anti-cytokeratin AE1/AE3 (+), evolving mass associated with dull pain. Usually adamantino- ActineML (+), Vimentine (+), CD99 (+ weak), CD117 (−), VEGFR−2 mas are locally aggressive and extremely low-growing tumors, (−), PDGFR−beta (-), Desmin (−), Citokeratynes 5/6 (−), S100 (−)and with the tendency to metastasize in about 15–30% of cases; p63 (−). The morphological setting and immune-histochemical elective site of metastatic localization is lungs, bones or nearby pattern were in agreement with the clinical suspicion of metasta- lymph nodes. sisfrom adamantinomaasrevealedbythe comparison with the Visceral metastases are extremely rare and, to our knowl- specimens resected during lung resections but especially from edge, cases of PM from adamantinoma have never been those of tibial amputation (Fig. 3). reported yet in literature [2, 5]. More than 4 years after primary intervention and 3 years Pancreatic metastases are rare, <2% of all pancreatic neopla- after PD, the patient is alive and disease free. sia. Primary tumors which metastasizemorefrequentlytothe pancreas are melanoma, clear cell renal carcinoma, breast and colon cancer [6, 7]. At diagnosis of PM, most patients have wide- DISCUSSION spread disease and only in selected cases, it is possible to observe Adamantinoma is a rare neoplasm accounting only for 0.3–1% the presence of oligo-metastatic or solitary pancreatic localiza- of all primary malignant bone tumors, and it mostly occurs tion. Symptoms and signs are similar for both primary and Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy012/4855836 by Ed 'DeepDyve' Gillespie user on 16 March 2018 Solitary pancreatic head metastasis from tibial adamantinoma 3 Figure 3: Immunohistochemical staining. (A) Hematoxylin and eosin stain (H&E stain). (B and C) H&E stain with magniﬁcation 4× and 20×.(D) Anti-cytokeratin AE1/ AE3 + (5×). (E) Vimentine + (4×). (F) ActineML + (4×). (G) CD99 + weak (10×). (H) CD117 − (2×). (I) Desmin – (4×). secondary pancreatic tumors and differential diagnosis may be (Turin) for the clinical supervision as well as for proactive very difﬁcult; PM are usually asymptomatic and detected during support. the follow-up, but it is frequent the presentation with obstructive jaundice, pain and weight loss which are clinically indistinguish- CONFLICT OF INTEREST STATEMENT able from primary pancreatic cancer as in the case presented. If None declared. imaging is not able to reliably differentiate primary pancreatic tumors from metastatic lesions, EUS–FNA represents an import- ant diagnostic tool. In fact FNA adds the possibility to make com- CONSENT STATEMENT parison of cytology from a metastatic pancreatic mass to a Written informed consent was obtained from the patient for previous cytology or histology from primary tumor; ﬁnally, the the publication of this case report. application of immunocytochemistry may be helpful to orientate once more the differential diagnosis and to set the deﬁnitive diag- nostic and therapeutic algorithm [8, 9]. REFERENCES Although the pancreatic gland is an uncommon site of 1. Kitsoulis P, Charchanti A, Paraskevas G, Marini A, Karatzias metastases, the potential beneﬁt of pancreatic resection is well G. Adamantinoma. Acta Orthop Belg 2007;73:425–31. known. As reported by several authors, standard pancreatic 2. Roque P, Mankin HJ, Rosenberg A. Adamantinoma: an resections (PD or distal pancreatectomy) are safe and feasible unusual bone tumour. Chir Organi Mov 2008;92:149–54. options to treat non-pancreatic primary tumor, since they 3. Szendroi M, Antal I, Arató G. Adamantinoma of long bones: improve patient’s survival as well as quality of life. As a matter a long-term follow-up study of 11 cases. Pathol Oncol Res POR of fact, Bassi et al. showed the importance of performing stand- 2009;15:209–16. ard pancreatic resections instead of atypical resection, with 4. Silvestri S, Franchello A, Deiro G, Galletti R, Cassine D, Campra lower rate of post-operative complication and local recurrence. D, et al. Preoperative oral immunonutrition versus standard Moreover, if negative surgical margins are achievable, in preoperative oral diet in well nourished patients undergoing selected patients, the presence of limited extra pancreatic dis- pancreaticoduodenectomy. Int J Surg Lond Engl 2016;31:93–9. ease together with PM is not always a contraindication to pan- 5. Papagelopoulos PJ, Mavrogenis AF, Galanis EC, Savvidou creatic surgery . OD, Inwards CY, Sim FH. Clinicopathological features, diag- This is the ﬁrst reported case of PM from adamantinoma. nosis, and treatment of adamantinoma of the long bones. Although rare for its biological features, metastatic involve- Orthopedics 2007;30:211–5. quiz 216–7. ment of the pancreas is a possible occurrence. As reported for 6. Zerbi A, Pecorelli N. Pancreatic metastases: an increasing other cases of PM, in absence of widespread metastatic disease, clinical entity. World J Gastrointest Surg 2010;2:255–9. curative pancreatic resections are recommended to improve 7. Sperti C, Pasquali C, Liessi G, Pinciroli L, Decet G, Pedrazzoli quality of life and survival. S. Pancreatic resection for metastatic tumors to the pan- creas. J Surg Oncol 2003;83:161–6. discussion 166. ACKNOWLEDGEMENTS 8. Ardengh JC, Lopes CV, Kemp R, Venco F, de Lima-Filho ER, The authors thank A. Fiore, D. Cassine, MD and D. Campra, MD, Santos JS dos. Accuracy of endoscopic ultrasound-guided of 4th Division of Surgery, AOU City of Health and Science ﬁne-needle aspiration in the suspicion of pancreatic Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy012/4855836 by Ed 'DeepDyve' Gillespie user on 16 March 2018 4 S. Silvestri et al. metastases. BMC Gastroenterol [Internet] 2013 Dec [cited 2017 A meta-analysis and systematic review. Pancreas 2013;42: Jan 15];13:63 http://bmcgastroenterol.biomedcentral.com/ 20–6. articles/10.1186/1471-230X−13−63. 10. Bassi C, Butturini G, Falconi M, Sargenti M, Mantovani W, 9. Puli SR, Bechtold ML, Buxbaum JL, Eloubeidi MA. How good Pederzoli P. High recurrence rate after atypical resection for is endoscopic ultrasound-guided ﬁne-needle aspiration in pancreatic metastases from renal cell carcinoma. Br J Surg diagnosing the correct etiology for a solid pancreatic mass?: 2003;90:555–9. 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Journal of Surgical Case Reports – Oxford University Press
Published: Feb 1, 2018
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