IgG4-related Diaphragmatic Inflammatory Pseudotumor

A 71-year-old man underwent surgery for a pancreatic neuroendocrine tumor. Follow-up imaging showed swelling of the remnant pancreas, and he was histologically diagnosed with autoimmune pancreatitis based on endoscopic ultrasonography-guided fine-needle aspiration specimens. After two years, a tumor...

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Published inInternal Medicine Vol. 60; no. 13; pp. 2067 - 2074
Main Authors Yoshida, Naoki, Kikuta, Kazuhiro, Miura, Shin, Matsumoto, Ryotaro, Sano, Takanori, Hamada, Shin, Takikawa, Tetsuya, Masamune, Atsushi, Tanaka, Yu, Kume, Kiyoshi, Unno, Michiaki, Hongo, Seiji, Ikeda, Mio
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LanguageEnglish
Published Tokyo The Japanese Society of Internal Medicine 01.07.2021
Japan Science and Technology Agency
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Abstract A 71-year-old man underwent surgery for a pancreatic neuroendocrine tumor. Follow-up imaging showed swelling of the remnant pancreas, and he was histologically diagnosed with autoimmune pancreatitis based on endoscopic ultrasonography-guided fine-needle aspiration specimens. After two years, a tumor appeared on the liver surface. Although we planned to perform laparoscopic partial hepatectomy, the intraoperative findings showed that the tumor was located in the diaphragm. Partial resection of the diaphragm was performed, and the final diagnosis was an immunoglobulin G4-related inflammatory pseudotumor in the diaphragm. To our knowledge, this is the first reported case of an immunoglobulin G4-related diaphragmatic inflammatory pseudotumor.
AbstractList A 71-year-old man underwent surgery for a pancreatic neuroendocrine tumor. Follow-up imaging showed swelling of the remnant pancreas, and he was histologically diagnosed with autoimmune pancreatitis based on endoscopic ultrasonography-guided fine-needle aspiration specimens. After two years, a tumor appeared on the liver surface. Although we planned to perform laparoscopic partial hepatectomy, the intraoperative findings showed that the tumor was located in the diaphragm. Partial resection of the diaphragm was performed, and the final diagnosis was an immunoglobulin G4-related inflammatory pseudotumor in the diaphragm. To our knowledge, this is the first reported case of an immunoglobulin G4-related diaphragmatic inflammatory pseudotumor.
A 71-year-old man underwent surgery for a pancreatic neuroendocrine tumor. Follow-up imaging showed swelling of the remnant pancreas, and he was histologically diagnosed with autoimmune pancreatitis based on endoscopic ultrasonography-guided fine-needle aspiration specimens. After two years, a tumor appeared on the liver surface. Although we planned to perform laparoscopic partial hepatectomy, the intraoperative findings showed that the tumor was located in the diaphragm. Partial resection of the diaphragm was performed, and the final diagnosis was an immunoglobulin G4-related inflammatory pseudotumor in the diaphragm. To our knowledge, this is the first reported case of an immunoglobulin G4-related diaphragmatic inflammatory pseudotumor.A 71-year-old man underwent surgery for a pancreatic neuroendocrine tumor. Follow-up imaging showed swelling of the remnant pancreas, and he was histologically diagnosed with autoimmune pancreatitis based on endoscopic ultrasonography-guided fine-needle aspiration specimens. After two years, a tumor appeared on the liver surface. Although we planned to perform laparoscopic partial hepatectomy, the intraoperative findings showed that the tumor was located in the diaphragm. Partial resection of the diaphragm was performed, and the final diagnosis was an immunoglobulin G4-related inflammatory pseudotumor in the diaphragm. To our knowledge, this is the first reported case of an immunoglobulin G4-related diaphragmatic inflammatory pseudotumor.
ArticleNumber 6589-20
Author Tanaka, Yu
Masamune, Atsushi
Sano, Takanori
Takikawa, Tetsuya
Miura, Shin
Ikeda, Mio
Kikuta, Kazuhiro
Yoshida, Naoki
Kume, Kiyoshi
Hamada, Shin
Unno, Michiaki
Hongo, Seiji
Matsumoto, Ryotaro
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  fullname: Miura, Shin
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  fullname: Matsumoto, Ryotaro
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  organization: Division of Gastroenterology, Tohoku University Graduate School of Medicine, Japan
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  organization: Division of Gastroenterology, Tohoku University Graduate School of Medicine, Japan
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26. ChangChien YC, Kovács I, Hargitai Z, Magyar L. Paratesticular fibrous pseudotumor: a new entity of IgG4-related disease?. Ann Clin Lab Sci 48: 381-385, 2018.
7. Bosman FT, Carneiro F, Hruban RH, Theise ND. WHO World Health Organization classification of tumors and genetics of the digestive system. IARC Press, Lyon, 2010.
32. Park S, Ro JY, Lee DH, Choi SY, Koo H. Immunoglobulin G4-associated inflammatory pseudotumor of urinary bladder: a case report. Ann Diagn Pathol 17: 540-543, 2013.
28. Sangsoad P, Ramart P, Korpraphong P, Rerkpichaisuth V, Pradniwat K, Treetipsatit J. Female urinary retention from a huge periurethral mass caused by immunoglobulin G4-related disease (IgG4-RD). Urol Case Rep 24: 100844, 2019.
19. Zen Y, Kasahara Y, Horita K, et al. Inflammatory pseudotumor of the breast in a patient with a high serum IgG4 level: histologic similarity to sclerosing pancreatitis. Am J Surg Pathol 29: 275-278, 2005.
22. Rumalla K, Smith KA, Arnold PM. Immunoglobulin G4-related epidural inflammatory pseudotumor presenting with pulmonary complications and spinal cord compression: case report. J Neurosurg Spine 26: 688-693, 2017.
24. Oda R, Okuda K. Thymic inflammatory pseudotumor with multilocular thymic cyst caused by immunoglobulin G4-related disease. 10: 116-119, 2019.
35. Oda R, Okuda K. Pericardial immunoglobulin G4-related inflammatory pseudotumor after right upper lobectomy for lung cancer. Thorac Cancer 11: 3034-3037, 2020.
15. Moon SH, Kim MH, Park DH, et al. Is a 2-week steroid trial after initial negative investigation for malignancy useful in differentiating autoimmune pancreatitis from pancreatic cancer? A prospective outcome study. Gut 57: 1704-1712, 2008.
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1. Yoshida K, Toki F, Takeuchi T, Watanabe S, Shiratori K, Hayashi N. Chronic pancreatitis caused by an autoimmune abnormality. Proposal of the concept of autoimmune pancreatitis. Dig Dis Sci 40: 1561-1568, 1995.
34. Kim Y, Lee HK, Hwang G, Choi IH, Kim HS. Solitary immunoglobulin G4-related inflammatory pseudotumor in the abdomen wall. Korean J Intern Med 32: 933-935, 2017.
12. Hoer J, Steinau G, Fuzesi L, Gunawan B, Schumpelick V. Inflammatory pseudotumor of the diaphragm. Pediatr Surg Int 15: 387-390, 1999.
36. Senda Y, Ikeda Y, Tamauchi S, Yoshikawa N, Kikkawa F, Kajiyama H. A uterine pseudotumor of immunoglobulin G4-related disease. J Obstet Gynaecol Res 47: 430-435, 2021.
6. Chougule A, Bal A. IgG4-related inflammatory pseudotumor: a systematic review of histopathological features of reported cases. Mod Rheumatol 27: 320-325, 2017.
38. Kamisawa T, Okazaki K. Diagnosis and treatment of IgG4-related disease. Curr Top Microbiol Immunol 401: 19-33, 2017.
3. Kamisawa T, Zen Y, Pillai S, Stone JH. IgG4-related disease. Lancet 385: 1460-1471, 2015.
29. Ono K, Shiiba M, Yoshizaki M, et al. Immunoglobulin G4-related sclerosing inflammatory pseudotumors presenting in the oral cavity. J Oral Maxillofac Surg 70: 1593-1598, 2012.
9. Okazaki K, Umehara H. Are classification criteria for IgG4-RD now possible? The concept of IgG4-related disease and proposal of comprehensive diagnostic criteria in Japan. Int J Rheumatol 2012: 357071, 2012.
33. Choi SB, Lim CH, Cha MG, Kang WK. IgG4-related disease of the rectum. Ann Surg Treat Res 90: 292-295, 2016.
39. Masamune A, Nishimori I, Kikuta K, et al. Randomised controlled trial of long-term maintenance corticosteroid therapy in patients with autoimmune pancreatitis. Gut 66: 487-494, 2017.
40. Carruthers MN, Topazian MD, Khosroshahi A, et al. Rituximab for IgG4-related disease: a prospective, open-label trial. Ann Rheum Dis 74: 1171-1177, 2015.
21. Ferreira NR, Vaz R, Carmona S, et al. IgG4-related disease presenting with an epidural inflammatory pseudotumor: a case report. J Med Case Rep 10: 61, 2016.
16. Witkiewicz AK, Kennedy EP, Kennyon L, Yeo CJ, Hruban RH. Synchronous autoimmune pancreatitis and infiltrating pancreatic ductal adenocarcinoma: case report and review of the literature. Hum Pathol 39: 1548-1551, 2008.
17. Oh HC, Kim JG, Kim JW, et al. Early bile duct cancer in a background of sclerosing cholangitis and autoimmune pancreatitis. Intern Med 47: 2025-2028, 2008.
20. Chougule A, Bal A, Das A, Singh G. IgG4 related sclerosing mastitis: expanding the morphological spectrum of IgG4 related diseases. Pathology 47: 27-33, 2015.
8. Shimosegawa T, Chari ST, Frulloni L, et al. International consensus diagnostic criteria for autoimmune pancreatitis: guidelines of the International Association of Pancreatology. Pancreas 40: 352-358, 2011.
13. Deshpande V, Zen Y, Chan JK, et al. Consensus statement on the pathology of IgG4-related disease. Mod Pathol 25: 1181-1192, 2012.
4. Masamune A, Kikuta K, Hamada S, et al. Nationwide epidemiological survey of autoimmune pancreatitis in Japan in 2016. J Gastroenterol 55: 462-470, 2020.
2. Nagpal SJS, Sharma A, Chari ST. Autoimmune Pancreatitis. Am J Gastroenterol 113: 1301, 2018.
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References_xml – reference: 11. Miyabe K, Zen Y, Cornell LD, et al. Gastrointestinal and extra-intestinal manifestations of IgG4-related disease. Gastroenterology 155: 990-1003.e1001, 2018.
– reference: 24. Oda R, Okuda K. Thymic inflammatory pseudotumor with multilocular thymic cyst caused by immunoglobulin G4-related disease. 10: 116-119, 2019.
– reference: 25. Hart PA, Moyer AM, Yi ES, Hogan MC, Pearson RK, Chari ST. IgG4-related paratesticular pseudotumor in a patient with autoimmune pancreatitis and retroperitoneal fibrosis: an extrapancreatic manifestation of IgG4-related disease. Hum Pathol 43: 2084-2087, 2012.
– reference: 30. Virk JS, Stamatoglou C, Kwame I, Salama A, Sandison A, Sandhu G. IgG4-sclerosing pseudotumor of the trachea: a case report and review of the literature. Arch Otolaryngol Head Neck Surg 138: 864-866, 2012.
– reference: 13. Deshpande V, Zen Y, Chan JK, et al. Consensus statement on the pathology of IgG4-related disease. Mod Pathol 25: 1181-1192, 2012.
– reference: 32. Park S, Ro JY, Lee DH, Choi SY, Koo H. Immunoglobulin G4-associated inflammatory pseudotumor of urinary bladder: a case report. Ann Diagn Pathol 17: 540-543, 2013.
– reference: 17. Oh HC, Kim JG, Kim JW, et al. Early bile duct cancer in a background of sclerosing cholangitis and autoimmune pancreatitis. Intern Med 47: 2025-2028, 2008.
– reference: 3. Kamisawa T, Zen Y, Pillai S, Stone JH. IgG4-related disease. Lancet 385: 1460-1471, 2015.
– reference: 4. Masamune A, Kikuta K, Hamada S, et al. Nationwide epidemiological survey of autoimmune pancreatitis in Japan in 2016. J Gastroenterol 55: 462-470, 2020.
– reference: 10. Kamisawa T, Funata N, Hayashi Y, et al. A new clinicopathological entity of IgG4-related autoimmune disease. J Gastroenterol 38: 982-984, 2003.
– reference: 5. Patnana M, Sevrukov AB, Elsayes KM, Viswanathan C, Lubner M, Menias CO. Inflammatory pseudotumor: the great mimicker. Am J Roentgenol 198: W217-227, 2012.
– reference: 21. Ferreira NR, Vaz R, Carmona S, et al. IgG4-related disease presenting with an epidural inflammatory pseudotumor: a case report. J Med Case Rep 10: 61, 2016.
– reference: 1. Yoshida K, Toki F, Takeuchi T, Watanabe S, Shiratori K, Hayashi N. Chronic pancreatitis caused by an autoimmune abnormality. Proposal of the concept of autoimmune pancreatitis. Dig Dis Sci 40: 1561-1568, 1995.
– reference: 33. Choi SB, Lim CH, Cha MG, Kang WK. IgG4-related disease of the rectum. Ann Surg Treat Res 90: 292-295, 2016.
– reference: 16. Witkiewicz AK, Kennedy EP, Kennyon L, Yeo CJ, Hruban RH. Synchronous autoimmune pancreatitis and infiltrating pancreatic ductal adenocarcinoma: case report and review of the literature. Hum Pathol 39: 1548-1551, 2008.
– reference: 23. Noh D, Park CK, Kwon SY. Immunoglobulin G4-related sclerosing disease invading the trachea and superior vena cava in mediastinum. Eur J Cardiothorac Surg 45: 573-575, 2014.
– reference: 12. Hoer J, Steinau G, Fuzesi L, Gunawan B, Schumpelick V. Inflammatory pseudotumor of the diaphragm. Pediatr Surg Int 15: 387-390, 1999.
– reference: 14. Khosroshahi A, Wallace ZS, Crowe JL, et al. International consensus guidance statement on the management and treatment of IgG4-related disease. Arthritis Rheumatol 67: 1688-1699, 2015.
– reference: 22. Rumalla K, Smith KA, Arnold PM. Immunoglobulin G4-related epidural inflammatory pseudotumor presenting with pulmonary complications and spinal cord compression: case report. J Neurosurg Spine 26: 688-693, 2017.
– reference: 26. ChangChien YC, Kovács I, Hargitai Z, Magyar L. Paratesticular fibrous pseudotumor: a new entity of IgG4-related disease?. Ann Clin Lab Sci 48: 381-385, 2018.
– reference: 38. Kamisawa T, Okazaki K. Diagnosis and treatment of IgG4-related disease. Curr Top Microbiol Immunol 401: 19-33, 2017.
– reference: 31. Lynnhtun K, Achan A, Lam V. IgG4 related pseudotumour (calcifying fibrous tumour) of adrenal gland. Pathology 45: 519-521, 2013.
– reference: 39. Masamune A, Nishimori I, Kikuta K, et al. Randomised controlled trial of long-term maintenance corticosteroid therapy in patients with autoimmune pancreatitis. Gut 66: 487-494, 2017.
– reference: 8. Shimosegawa T, Chari ST, Frulloni L, et al. International consensus diagnostic criteria for autoimmune pancreatitis: guidelines of the International Association of Pancreatology. Pancreas 40: 352-358, 2011.
– reference: 20. Chougule A, Bal A, Das A, Singh G. IgG4 related sclerosing mastitis: expanding the morphological spectrum of IgG4 related diseases. Pathology 47: 27-33, 2015.
– reference: 36. Senda Y, Ikeda Y, Tamauchi S, Yoshikawa N, Kikkawa F, Kajiyama H. A uterine pseudotumor of immunoglobulin G4-related disease. J Obstet Gynaecol Res 47: 430-435, 2021.
– reference: 15. Moon SH, Kim MH, Park DH, et al. Is a 2-week steroid trial after initial negative investigation for malignancy useful in differentiating autoimmune pancreatitis from pancreatic cancer? A prospective outcome study. Gut 57: 1704-1712, 2008.
– reference: 2. Nagpal SJS, Sharma A, Chari ST. Autoimmune Pancreatitis. Am J Gastroenterol 113: 1301, 2018.
– reference: 9. Okazaki K, Umehara H. Are classification criteria for IgG4-RD now possible? The concept of IgG4-related disease and proposal of comprehensive diagnostic criteria in Japan. Int J Rheumatol 2012: 357071, 2012.
– reference: 19. Zen Y, Kasahara Y, Horita K, et al. Inflammatory pseudotumor of the breast in a patient with a high serum IgG4 level: histologic similarity to sclerosing pancreatitis. Am J Surg Pathol 29: 275-278, 2005.
– reference: 29. Ono K, Shiiba M, Yoshizaki M, et al. Immunoglobulin G4-related sclerosing inflammatory pseudotumors presenting in the oral cavity. J Oral Maxillofac Surg 70: 1593-1598, 2012.
– reference: 40. Carruthers MN, Topazian MD, Khosroshahi A, et al. Rituximab for IgG4-related disease: a prospective, open-label trial. Ann Rheum Dis 74: 1171-1177, 2015.
– reference: 35. Oda R, Okuda K. Pericardial immunoglobulin G4-related inflammatory pseudotumor after right upper lobectomy for lung cancer. Thorac Cancer 11: 3034-3037, 2020.
– reference: 6. Chougule A, Bal A. IgG4-related inflammatory pseudotumor: a systematic review of histopathological features of reported cases. Mod Rheumatol 27: 320-325, 2017.
– reference: 18. Ikeda R, Kurakami K, Ohta N, et al. Malignancies in patients with IgG4-related diseases in head and neck regions. Tohoku J Exp Med 249: 285-290, 2019.
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Snippet A 71-year-old man underwent surgery for a pancreatic neuroendocrine tumor. Follow-up imaging showed swelling of the remnant pancreas, and he was histologically...
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SubjectTerms autoimmune pancreatitis
Case Report
Diaphragm
Hepatectomy
IgG4
IgG4-related disease
Immunoglobulin G
Immunoglobulin G4
Immunoglobulins
Inflammation
inflammatory pseudotumor
Internal medicine
Laparoscopy
Neuroendocrine tumors
Pancreas
pancreatic neuroendocrine tumor
Pancreatitis
Pseudotumors
Surgery
Title IgG4-related Diaphragmatic Inflammatory Pseudotumor
URI https://www.jstage.jst.go.jp/article/internalmedicine/60/13/60_6589-20/_article/-char/en
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https://pubmed.ncbi.nlm.nih.gov/PMC8313910
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