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M. Glascock, S. Carty (2002)
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Hindawi Publishing Corporation Case Reports in Oncological Medicine Volume 2012, Article ID 619428, 4 pages doi:10.1155/2012/619428 Case Report Simultaneous EUS-FNA Diagnosis and TNM Staging of a Pancreatic Neuroendocrine Tumor in a Patient with an Unrecognized MEN Type 1 1 1 1 Francesco Ferrara, Carmelo Luigiano, Antonella Maimone, 1 1 2 3 Marco Bassi, Anna Maria Polifemo, Paola Baccarini, Vincenzo Cennamo, 4 1 Nadia Cremonini, and Carlo Fabbri Unit of Gastroenterology and Digestive Endoscopy, Bellaria and Maggiore Hospitals, AUSL Bologna, Largo Nigrisoli 2, 40133 Bologna, Italy Unit of Pathology, Bellaria Hospital, AUSL Bologna, 40139 Bologna, Italy Unit of Metropolitan Area Digestive Endoscopy, Bellaria Hospital, AUSL Bologna, 40139 Bologna, Italy Unit of Endocrinology, Maggiore Hospital, AUSL Bologna, 40139 Bologna, Italy Correspondence should be addressed to Francesco Ferrara, f.ferrara@ausl.bologna.it Received 9 July 2012; Accepted 12 September 2012 Academic Editors: Y. Aoki and S. B. Chichareon Copyright © 2012 Francesco Ferrara et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. We report the case of a woman who, during oncological followup for bronchial carcinoid (diagnosed in 2005), papillary thyroid carcinoma, and bilateral parathyroid adenoma (simultaneously diagnosed in 2007), performed a pancreatic endoscopic ultrasonography with fine needle agobiopsy (EUS-FNA) for a positron emission tomography (PET) suspicion of pancreatic and hepatic lesions; during the procedure, the pancreatic and liver lesions were confirmed, and a peripancreatic lymph node involvement was found, allowing a complete pTNM staging during the same procedure. 1. Case were resected. Histology confirmed the diagnosis of thyroid papillary cancer and revealed bilateral parathyroid adenoma. A 48-year-old woman, with no family history of neoplastic In September 2008, a biochemical screening revealed diseases, underwent in 2005 to right pulmonary resection for high plasma levels of CgA of 2230 (normal 20–150) and a bronchial carcinoids and was in oncological follow-up for serum gastrin (648 pg/mL). She was then referred to our this reason. hospital. In 2007, during a normal physical examination, a 2 cm A 68Ga-DOTANOC-PET was performed and revealed 3 sized nodule in her left thyroid lobe was palpated. The thy- small areas of hyperaccumulation in pancreatic region and roid function tests were normal. The ultrasonography report one in the liver. revealed a solid hypoechoic nodule measuring 2.5 × 1cm For this reason, an EUS was performed and identified in the left lobe. A fine needle aspiration was attempted 2 nodular hypoechoic lesions of 5 mm with hypervascular under ultrasound guidance and the final cytodiagnosis was Doppler pattern located in the body and in the tail of papillary thyroid carcinoma. the pancreas. During the exploration, also a peripancreatic Following cytodiagnosis, a total thyroidectomy was per- nodal involvement was found and the suspected metastasis formed, but at the time of surgery, during the neck explo- (15 mm) of left liver was confirmed. We performed EUS- ration, the patient was found to have bilateral enlargement guided FNA with a 22G and a 25G needle, with on- of parathyroid glands with nodular aspect and all glands site cytopathologist, and a Pan-NET (T) tumor (Figure 1) 2 Case Reports in Oncological Medicine (a) (b) (c) Figure 1: EUS (a), FNA (b), and cytological (c) images of the neuroendocrine pancreatic tumor. (a) (b) (c) Figure 2: EUS (a), FNA (b), and cytological (c) images of the lymphonodal metastases. with (N) lymph node (Figure 2)and (M)liver (Figure 3) Thyroiddisease canbeobservedinover25% of MEN1 involvement was diagnosed. patients, [3, 4], and it can be detected incidentally during A mutation of the MEN-1 gene was identified and, due parathyroid surgery. to the high level of serum gastrin, a diagnosis of gastrinoma Only three cases of papillary thyroid cancer combined was reached. with MEN1 were reported in the literature and seems that She started therapy with octreotide LAR followed by these cases did not correlate to MEN1 [4–6]. receptor radiometabolic therapy with radiolabelled somato- Probably for this reason, the diagnosis of MEN1 was not statin analogues (177 Lu-DOTATATE) and she is presently made before. alive. About 20% of MEN-1 patients succumb to malignant tumors and malignant Pan-NET are unequivocally the most frequent cause of death [7]. Imaging modalities such as CT and MRI have enabled 2. Discussion detection of Pan-NETs. Overall sensitivity for CT ranges from 64 to 82%, with lesser sensitivity for tumors <1cm in Multiple endocrine neoplasia type 1 (MEN-1) syndrome is a size. Similarly, MRI offers superb imaging of the pancreas, rare disease, inherited as an autosomal dominant trait with with sensitivity of up to 90% in Pan-NETs [8, 9]. an estimated prevalence of 0.01–2.5/100000 [1]. Nuclear medicine studies have been developed to target MEN-1 syndrome is characterized by parathyroid hyper- specific receptors on Pan-NET tumor cells with radiolabeled plasia, neuroendocrine pancreatoduodenal tumors, and receptor-binding peptides. The most common of these tests pituitary adenomas. Less commonly, MEN-1 patients can is somatostatin receptor scintigraphy (SRS). The sensitivity develop bronchial, gastrointestinal, and thymic carcinoids, of SRS for detecting gastrinomas is as high as 75–100%; in benign thyroid and adrenocortical tumors, lipomas, angiofi- contrast, SRS is able to detect insulinomas in approximately bromas, skin collagenomas, and ependymomas of the central only half of the times [10]. nervous system [2]. Case Reports in Oncological Medicine 3 (a) (b) (c) Figure 3: EUS (a), FNA (b), and cytological (c) images of the liver metastases. Endoscopic ultrasound (EUS) is an extremely valuable [2] J. M. Glascock and S. E. Carty, “Multiple endocrine neoplasia type 1: fresh perspective on clinical features and penetrance,” tool in the diagnosis and management of these tumors. The Surgical Oncology, vol. 11, no. 3, pp. 143–150, 2002. important role of EUS in the detection of Pan-NETs was first [3] Q. Dong, L. V. Debelenko, S. C. Chandrasekharappa et al., described in 1992 [11]: EUS demonstrated a sensitivity of “Loss of heterozygosity at 11q13: analysis of pituitary tumors, 82% and a specificity of 92% in the detection of islet cell lung carcinoids, lipomas, and other uncommon tumors in tumors in patients whith previously undetected tumors by subjects with familial multiple endocrine neoplasia type 1,” US and CT. Since then, EUS has been increasingly used in Journal of Clinical Endocrinology and Metabolism, vol. 82, no. the localization of Pan-NETs [12]. EUS is particularly useful 5, pp. 1416–1420, 1997. in the detection of smaller insulinomas. The average size of [4] D. Desai, L. A. McPherson, J. P. T. Higgins, and R. J. Weigel, insulinomas at initial diagnosis is 6–10 mm, with 90% of “Genetic analysis of a papillary thyroid carcinoma in a patient cases under 2 cm [13]. with MEN 1,” Annals of Surgical Oncology,vol. 8, no.4,pp. A recent study on 52 patients undergoing EUS for 342–346, 2001. detection of a suspected insulinoma (based on clinical and [5] A. O. Vortmeyer, I. A. Lubensky, M. Skarulis et al., “Multiple laboratory findings) reported a sensitivity of 89.5% and endocrine neoplasia type 1: atypical presentation, clinical accuracy of 83.7% based on surgical findings. The sensitivity course, and genetic analysis of multiple tumors,” Modern of EUS for detection of lesions in pancreatic head, body, and Pathology, vol. 12, no. 9, pp. 919–924, 1999. tail was 92.6, 78.9, and 40.0%, respectively [14]. [6] H.J.Kim,J.S.Park, C. S. Kimetal., “A case of multiple endocrine neoplasia type 1 combined with papillary thyroid The detection rates for pancreatic gastrinomas by EUS carcinoma,” Yonsei Medical Journal, vol. 49, no. 3, pp. 503–506, are similar to that of insulinomas, approximately 75–94% [15]. [7] G.M.Doherty,J.A.Olson,M.M.Frisella, T. C. Lairmore,S. Pan-NETs may be pathologically evaluated by FNA A. Wells, and J. A. Norton, “Lethality of multiple endocrine during the EUS examination. Three recent studies reported neoplasia type I,” World Journal of Surgery,vol. 22, no.6,pp. sensitivities of 61–84% and overall accuracy of up to 92.5% 581–587, 1998. of EUS-FNA in establishing the diagnosis of Pan-NETs [16– [8] T.C.Noone,J.Hosey,F.Zeynep, andR.C.Semelka,“Imaging 18]. Additionally, FNA may detect and confirm the presence and localization of islet-cell tumours of the pancreas on CT of malignant lymph nodes and liver metastases previously and MRI,” Best Practice and Research, vol. 19, no. 2, pp. 195– unseen on CT imaging [18, 19]. Recently, Lewis et al. [20] 211, 2005. further confirmed in 52 patients the high accuracy of EUS- [9] Y. M. Ku, S. S. Shin, C. H. Lee, and R. C. Semelka, “Magnetic FNA in preoperative assessment in MEN-1, and Barbe et al. resonance imaging of cystic and endocrine pancreatic neo- [21] established a complementary role for MRI and EUS. plasms,” Topics in Magnetic Resonance Imaging, vol. 20, no. 1, To the best of our knowledge, this is the first report where pp. 11–18, 2009. EUS-FNA has allowed not only the localization but also a [10] E. P. Tamm, E. E. Kim, and C. S. Ng, “Imaging of neu- complete diagnosis with pTNM staging of Pan-NETs. roendocrine tumors,” Hematology/Oncology Clinics of North America, vol. 21, no. 3, pp. 409–432, 2007. [11] T. Rosc ¨ h, C. J. Lightdale, J. F. Botet et al., “Localization of pancreatic endocrine tumors by endoscopic ultrasonography,” References New England Journal of Medicine, vol. 326, no. 26, pp. 1721– 1726, 1992. [1] D.H.Schussheim, M. C. Skarulis,S.K.Agarwal et al., [12] T. Ishikawa, A. Itoh, H. Kawashima et al., “Usefulness of EUS “Multiple endocrine neoplasia type 1: new clinical and basic combined with contrast-enhancement in the differential diag- findings,” Trends in Endocrinology and Metabolism, vol. 12, no. nosis of malignant versus benign and preoperative localization 4, pp. 173–178, 2001. 4 Case Reports in Oncological Medicine of pancreatic endocrine tumors,” Gastrointestinal Endoscopy, vol. 71, no. 6, pp. 951–959, 2010. [13] G. Akerstro ¨m andP.Hellman,“Surgeryonneuroendocrine tumours,” Best Practice & Research Clinical Endocrinology & Metabolism, vol. 21, no. 1, pp. 87–109, 2007. [14] R. Sotoudehmanesh, A. Hedayat, N. Shirazian et al., “Endo- scopic ultrasonography (EUS) in the localization of insuli- noma,” Endocrine, vol. 31, no. 3, pp. 238–241, 2007. [15] A. M. McLean and P. D. Fairclough, “Endoscopic ultrasound in the localisation of pancreatic islet cell tumours,” Best Practice and Research, vol. 19, no. 2, pp. 177–193, 2005. [16] S. A. Pais, K. Mcgreevy, J. K. Leblanc et al., “Utility of EUS- FNA in the diagnosis of pancreatic neuroendocrine tumors: correlation with histopathology in 76 patients,” Gastrointesti- nal Endoscopy, vol. 65, no. 5, p. AB304, 2007. [17] E. W. Holt,E.A.Macklin, andW.R.Brugge, “Variables affecting the accuracy of EUS-guided FNA in the diagnosis of focal pancreatic masses,” Gastrointestinal Endoscopy, vol. 67, no. 5, pp. AB218–AB219, 2008. [18] M. Atiq, M. S. Bhutani, M. Bektas et al., “EUS-FNA for pancreatic neuroendocrine tumors: a tertiary cancer center experience,” Digestive Diseases and Sciences,vol. 57, no.3,pp. 791–800, 2012. [19] J. M. O’Neil, M. A. Al-Haddad, J. Leblanc et al., “Endoscopic ultrasound morphology features and initial detection of metastatic liver lesions from primary pancreatic adenocarci- noma and pancreatic neuroendocrine carcinoma,” Gastroin- testinal Endoscopy, vol. 67, no. 5, pp. AB217–AB218, 2008. [20] M. A. Lewis, G. B. Thompson, and W.F. Young Jr, “Pre- operative assessment of the pancreas in multiple endocrine neoplasia type 1,” World Journal of Surgery,vol. 36, no.6,pp. 1375–1381, 2012. [21] C. Barbe, A. Murat, B. Dupas et al., “Magnetic resonance imaging versus endoscopic ultrasonography for the detection of pancreatic tumours in multiple endocrine neoplasia type 1,” Digestive and Liver Disease, vol. 44, no. 3, pp. 228–234, 2012. 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Published: Oct 10, 2012
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