A 61-year-old woman visited our hospital for bilateral multiple lung nodules and a mass in her thorax. She had a long history of multiple metastatic recurrences of solid pseudopapillary neoplasm (SPN); 24 years previously, the patient had undergone pylorus-preserving pancreaticoduodenectomy for a 9.9 × 8.6 cm mass in the pancreatic head. The tumor was diagnosed as an SPN. Nine years later, metastatic nodules were found on computed tomography in the patient’s liver and peritoneum and were excised. She subsequently underwent an additional eight metastatectomy procedures in diverse organs. For the presented event, the lung nodules were removed. The prevalence of malignant SPN in the general population is 5%–15%. However, multiple metastatic recurrence of malignant SPN is rare; the lung is a particularly rare site of metastasis, found in only three cases in the literature. Here, we describe this exceptional case and provide a literature review.
Citations
Citations to this article as recorded by
Curative Resection for Metastatic Solid Pseudopapillary Neoplasm of Pancreas—a Case Series Aparna M. Jagannathan, Manbha L. Rymbai, Abhilasha Anand, Anoop Paul, Borna Das, Thomas Alex Kodiatte, Frederick L. Vyas, Ravish Sanghi Raju, Philip Joseph Indian Journal of Surgical Oncology.2024; 15(S2): 232. CrossRef
Malignant Solid Pseudopapillary Neoplasm of the Pancreas: An Orthogonal Analysis Andrew M. Fleming, Leah E. Hendrick, Danny Yakoub, Hafeez Abdelhafeez, Jeremiah L. Deneve, Max R. Langham, Evan S. Glazer, Andrew M. Davidoff, Nipun B. Merchant, Paxton V. Dickson, Andrew J. Murphy Annals of Surgical Oncology.2024; 31(1): 475. CrossRef
A Unique Presentation of Solid Pseudopapillary Neoplasm of the Pancreas Requiring Pancreaticoduodenectomy Without Pancreatojejunostomy: A Case Report and Literature Review Alexis L Carmona, Sameh A Fayek Cureus.2024;[Epub] CrossRef
Case report: Clinical analysis and literature review of five cases of metastatic solid pseudopapillary tumor of the pancreas Run Hu, Renjie Gui, Xi Nie, Huaxin Duan Frontiers in Oncology.2024;[Epub] CrossRef
Case of Treatment of Solid Pseudopapillary Pancreatic Tumor F. S. Rakhimova, N. D. Mamashev, O. A. Shimkina, B. Kh. Bebezov Creative surgery and oncology.2023; 13(2): 178. CrossRef
Diagnosis and treatment of solid pseudopapillary neoplasm of the pancreas in children: A report of 18 cases Ayiguzaili Maimaijiang, Haiyun Wang, Wanfu Li, Yaqi Wang Frontiers in Pediatrics.2022;[Epub] CrossRef
Large tumor size, lymphovascular invasion, and synchronous metastasis are associated with the recurrence of solid pseudopapillary neoplasms of the pancreas Goeun Lee, You-Na Sung, Sung Joo Kim, Jae Hoon Lee, Ki-Byung Song, Dae Wook Hwang, Jihun Kim, Sang Soo Lee, Song Cheol Kim, Seung-Mo Hong HPB.2021; 23(2): 220. CrossRef
Solid-Pseudopapillary Neoplasm of the Pancreas: A 63-Case Analysis of Clinicopathologic and Immunohistochemical Features and Risk Factors of Malignancy Hongchun Chen, Yuchen Huang, Ningning Yang, Wentian Yan, Ruxue Yang, Shan Zhang, Panpan Yang, Nan Li, Zhenzhong Feng Cancer Management and Research.2021; Volume 13: 3335. CrossRef
Rare Solid Tumor of the Exocrine Pancreas: A Pictorial Review Marco Dioguardi Burgio, Maxime Ronot, Valérie Vilgrain Seminars in Ultrasound, CT and MRI.2019; 40(6): 483. CrossRef
The Stomach: a Rare Site for Metastatic Solid Pseudopapillary Neoplasm of the Pancreas Prajwala S. Prakash, Dexter Yak Seng Chan, Krishnakumar Madhavan Journal of Gastrointestinal Surgery.2018; 22(4): 759. CrossRef
European evidence-based guidelines on pancreatic cystic neoplasms
Background SIRT7 is one of the histone deacetylases and is NAD-dependent. It forms a complex with ETS-like transcription factor 4 (ELK4), which deacetylates H3K18ac and works as a transcriptional suppressor. Overexpression of SIRT7 and deacetylation of H3K18ac have been shown to be associated with aggressive clinical behavior in some cancers, including hepatocellular carcinoma (HCC). The present study investigated the immunohistochemical expression of SIRT7, H3K18ac, and ELK4 in hepatocellular carcinoma.
Methods A total of 278 HCC patients were enrolled in this study. Tissue microarray blocks were made from existing paraffin-embedded blocks. Immunohistochemical expressions of SIRT7, H3K18ac and ELK4 were scored and analyzed.
Results High SIRT7 (p = .034), high H3K18ac (p = .001), and low ELK4 (p = .021) groups were associated with poor outcomes. Age < 65 years (p = .028), tumor size ≥ 5 cm (p = .001), presence of vascular emboli (p = .003), involvement of surgical margin (p = .001), and high American Joint Committee on Cancer stage (III&V) (p < .001) were correlated with worse prognoses. In multivariate analysis, H3K18ac (p = .001) and ELK4 (p = .015) were the significant independent prognostic factors.
Conclusions High SIRT7 expression with poor overall survival implies that deacetylation of H3K18ac contributes to progression of HCC. High H3K18ac expression with poor prognosis is predicted due to a compensation mechanism. In addition, high ELK4 expression with good prognosis suggests another role of ELK4 as a tumor suppressor beyond SIRT7’s helper. In conclusion, we could assume that the H3K18ac deacetylation pathway is influenced by many other factors.
Citations
Citations to this article as recorded by
SIRT7: the seventh key to unlocking the mystery of aging Umar Raza, Xiaolong Tang, Zuojun Liu, Baohua Liu Physiological Reviews.2024; 104(1): 253. CrossRef
The Significance of Modified Histone H3 in Epithelial Dysplasia and Oral Cancer Woraphaluck Tachaveeraphong, Ekarat Phattarataratip International Dental Journal.2024; 74(4): 769. CrossRef
Analysis of the Expression and Prognostic Value of SIRTs in Hepatocellular Carcinoma Chuang Qin, Xiaofei Ye, Hongliang Luo, Hu Jin, Qiang Liu, Jiangfa Li International Journal of General Medicine.2024; Volume 17: 2655. CrossRef
Combined single-cell RNA sequencing and mendelian randomization to identify biomarkers associated with necrotic apoptosis in intervertebral disc degeneration Yi Ye, Lun Wan, Jiang Hu, Xiaoxue Li, Kun Zhang The Spine Journal.2024;[Epub] CrossRef
Role of Sirtuins in the Pathogenesis of Rheumatoid Arthritis Agata Poniewierska-Baran, Oliwia Bochniak, Paulina Warias, Andrzej Pawlik International Journal of Molecular Sciences.2023; 24(2): 1532. CrossRef
Role of sirtuins in hepatocellular carcinoma progression and multidrug resistance: Mechanistical and pharmacological perspectives María Paula Ceballos, Ariel Darío Quiroga, Nicolás Francisco Palma Biochemical Pharmacology.2023; 212: 115573. CrossRef
Substrates and Cyclic Peptide Inhibitors of the Oligonucleotide‐Activated Sirtuin 7** Julie E. Bolding, Alexander L. Nielsen, Iben Jensen, Tobias N. Hansen, Line A. Ryberg, Samuel T. Jameson, Pernille Harris, Günther H. J. Peters, John M. Denu, Joseph M. Rogers, Christian A. Olsen Angewandte Chemie International Edition.2023;[Epub] CrossRef
Substrates and Cyclic Peptide Inhibitors of the Oligonucleotide‐Activated Sirtuin 7** Julie E. Bolding, Alexander L. Nielsen, Iben Jensen, Tobias N. Hansen, Line A. Ryberg, Samuel T. Jameson, Pernille Harris, Günther H. J. Peters, John M. Denu, Joseph M. Rogers, Christian A. Olsen Angewandte Chemie.2023;[Epub] CrossRef
Epigenomic interplay in tumor heterogeneity: Potential of epidrugs as adjunct therapy Suvasmita Rath, Diptesh Chakraborty, Jyotsnarani Pradhan, Mohammad Imran Khan, Jagneshwar Dandapat Cytokine.2022; 157: 155967. CrossRef
Distinct histone H3 modification profiles correlate with aggressive characteristics of salivary gland neoplasms Aroonwan Lam-Ubol, Ekarat Phattarataratip Scientific Reports.2022;[Epub] CrossRef
Acetyl-CoA: An interplay between metabolism and epigenetics in cancer Yang Hao, Qin Yi, Xu XiaoWu, Chen WeiBo, Zu GuangChen, Chen XueMin Frontiers in Molecular Medicine.2022;[Epub] CrossRef
Sirtuins (SIRTs) As a Novel Target in Gastric Cancer Agata Poniewierska-Baran, Paulina Warias, Katarzyna Zgutka International Journal of Molecular Sciences.2022; 23(23): 15119. CrossRef
Novel oncogenes and tumor suppressor genes in hepatocellular carcinoma Fang Wang, Peter Breslin S J, Wei Qiu Liver Research.2021; 5(4): 195. CrossRef
Acute high folic acid treatment in SH-SY5Y cells with and without MTHFR function leads to gene expression changes in epigenetic modifying enzymes, changes in epigenetic marks, and changes in dendritic spine densities Daniel F. Clark, Rachael Schmelz, Nicole Rogers, Nuri E. Smith, Kimberly R. Shorter, Lorenzo Chiariotti PLOS ONE.2021; 16(1): e0245005. CrossRef
The E-Twenty-Six Family in Hepatocellular Carcinoma: Moving into the Spotlight Tongyue Zhang, Danfei Liu, Yijun Wang, Mengyu Sun, Limin Xia Frontiers in Oncology.2021;[Epub] CrossRef
Upregulation of histone acetylation reverses organic anion transporter 2 repression and enhances 5-fluorouracil sensitivity in hepatocellular carcinoma Yingying Wang, Qianying Zhu, Haihong Hu, Hong Zhu, Bo Yang, Qiaojun He, Lushan Yu, Su Zeng Biochemical Pharmacology.2021; 188: 114546. CrossRef
HCG11 up-regulation induced by ELK4 suppressed proliferation in vestibular schwannoma by targeting miR-620/ELK4 Ruiqing Long, Zhuohui Liu, Jinghui Li, Yuan Zhang, Hualin Yu Cancer Cell International.2021;[Epub] CrossRef
Downregulation of circular RNA circPVT1 restricts cell growth of hepatocellular carcinoma through downregulation of Sirtuin 7 via microRNA‐3666 Yong Li, Haitao Shi, Jia Yuan, Lu Qiao, Lei Dong, Yan Wang Clinical and Experimental Pharmacology and Physiology.2020; 47(7): 1291. CrossRef
Clinicopathological and molecular analysis of SIRT7 in hepatocellular carcinoma Masae Yanai, Morito Kurata, Yutaka Muto, Hiroto Iha, Toshinori Kanao, Anna Tatsuzawa, Sachiko Ishibashi, Masumi Ikeda, Masanobu Kitagawa, Kouhei Yamamoto Pathology.2020; 52(5): 529. CrossRef
MicroRNA-148b Inhibits the Malignant Biological Behavior of Melanoma by Reducing Sirtuin 7 Expression Levels Rui Sun, Meiliang Guo, Xiaojing Fan, Qinqin Meng, Dingfen Yuan, Xinrong Yang, Kexiang Yan, Hui Deng, Fengjie Sun BioMed Research International.2020; 2020: 1. CrossRef
H3K18Ac as a Marker of Cancer Progression and Potential Target of Anti-Cancer Therapy Marta Hałasa, Anna Wawruszak, Alicja Przybyszewska, Anna Jaruga, Małgorzata Guz, Joanna Kałafut, Andrzej Stepulak, Marek Cybulski Cells.2019; 8(5): 485. CrossRef
Sirtuin7 has an oncogenic potential via promoting the growth of cholangiocarcinoma cells Wenzhi Li, Zhe Sun, Chen Chen, Lin Wang, Zhimin Geng, Jie Tao Biomedicine & Pharmacotherapy.2018; 100: 257. CrossRef
Identification of cancer‐related potential biomarkers based on lncRNA–pseudogene–mRNA competitive networks Cheng Wu, Yunzhen Wei, Yinling Zhu, Kun Li, Yanjiao Zhu, Yichuan Zhao, Zhiqiang Chang, Yan Xu FEBS Letters.2018; 592(6): 973. CrossRef
SIRT7 suppresses the epithelial-to-mesenchymal transition in oral squamous cell carcinoma metastasis by promoting SMAD4 deacetylation Wenlu Li, Dandan Zhu, Shuaihua Qin Journal of Experimental & Clinical Cancer Research.2018;[Epub] CrossRef
Sirtuin 7: a new marker of aggressiveness in prostate cancer Romain Haider, Fabienne Massa, Lisa Kaminski, Stephan Clavel, Zied Djabari, Guillaume Robert, Kathiane Laurent, Jean-François Michiels, Matthieu Durand, Jean-Ehrland Ricci, Jean-François Tanti, Frédéric Bost, Damien Ambrosetti Oncotarget.2017; 8(44): 77309. CrossRef
Benign perivascular epithelioid cell tumor (PEComa) of the lung is a rare benign neoplasm, a sclerosing variant of which is even rarer. We present a case of 51-year-old man who was diagnosed with benign sclerosing PEComa by percutaneous fine needle aspiration cytology and biopsy. The aspirate revealed a few cell clusters composed of bland-looking polygonal or spindle cells with fine granular or clear cytoplasm. Occasional fine vessel-like structures with surrounding hyalinized materials were seen. The patient later underwent wedge resection of the lung. The histopathological study of the resected specimen revealed sheets of polygonal cells with clear vacuolated cytoplasm, variably sized thin blood vessels, and densely hyalinized stroma. In immunohistochemical studies, reactivity of tumor cells for human melanoma black 45 and Melan-A further supported the diagnosis of benign sclerosing PEComa. To the best of our knowledge, this is the first case of benign sclerosing PEComa described in lung.
Citations
Citations to this article as recorded by
Cytopathology of rare gastric mesenchymal neoplasms: A series of 25 cases and review of literature Carla Saoud, Peter B. Illei, Momin T. Siddiqui, Syed Z. Ali Cytopathology.2023; 34(1): 15. CrossRef
Retroperitoneal Sclerosing Angiomyolipoma with Long-Term Follow up: A Case Report with Unique Clinicopathologic and Genomic Profile Liwei Jia, Vandana Panwar, Michelle Parmley, Elena Lucas, Ivan Pedrosa, Payal Kapur International Journal of Surgical Pathology.2022; 30(1): 86. CrossRef
Perivascular epithelioid cell tumor of the lung: A case report and literature review Shaofu Yu, Shasha Zhai, Qian Gong, Xiaoping Hu, Wenjuan Yang, Liyu Liu, Yi Kong, Lin Wu, Xingxiang Pu Thoracic Cancer.2022; 13(17): 2542. CrossRef
Cytopathology of extra-renal perivascular epithelioid cell tumor (PEComa): a series of 7 cases and review of the literature Sintawat Wangsiricharoen, Tatianna C. Larman, Paul E. Wakely, Momin T. Siddiqui, Syed Z. Ali Journal of the American Society of Cytopathology.2021; 10(2): 175. CrossRef
Clear cell sugar tumour: a rare tumour of the lung Sarah Page, Matthew S. Yong, Alka Sinha, Pankaj Saxena ANZ Journal of Surgery.2020;[Epub] CrossRef
Perivascular Epithelioid Cell Tumors (PEComas) of the Orbit Panagiotis Paliogiannis, Giuseppe Palmieri, Francesco Tanda, Antonio Cossu Journal of Pathology and Translational Medicine.2017; 51(1): 7. CrossRef