Journal of Pathology and Translational Medicine

Case Studies

Gastric IgG4-related disease presenting as a mass lesion and masquerading as a gastrointestinal stromal tumor: Banumathi Ramakrishna, Rohan Yewale, Kavita Vijayakumar, Patta Radhakrishna, Balakrishnan Siddartha Ramakrishna; J Pathol Transl Med. 2020;54(3):258-262. Published online March 4, 2020; DOI: https://doi.org/10.4132/jptm.2020.02.10

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IgG4-related disease of the stomach is a rare disorder, and only a few cases have been reported. We present two cases that were identified over a 2-month period in our center. Two male patients aged 52 and 48 years presented with mass lesion in the stomach, which were clinically thought to be gastrointestinal stromal tumor, and they underwent excision of the lesion. Microscopic examination revealed marked fibrosis, which was storiform in one case, associated with diffuse lymphoplasmacytic infiltration and an increase in IgG4-positive plasma cells on immunohistochemistry. Serum IgG4 level was markedly elevated. Although rare, IgG4-related disease should be considered in the differential diagnosis of gastric submucosal mass lesions.

Citations

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CGB5, INHBA and TRAJ19 Hold Prognostic Potential as Immune Genes for Patients with Gastric Cancer
Bei Ji, Lili Qiao, Wei Zhai
Digestive Diseases and Sciences.2023; 68(3): 791. CrossRef
IgG4-related diseases of the digestive tract
J.-Matthias Löhr, Miroslav Vujasinovic, Jonas Rosendahl, John H. Stone, Ulrich Beuers
Nature Reviews Gastroenterology & Hepatology.2022; 19(3): 185. CrossRef
Clinicopathological characteristics of gastric IgG4‐related disease: Systematic scoping review
Haruki Sawada, Torrey Czech, Krixie Silangcruz, Landon Kozai, Adham Obeidat, Eric Andrew Wien, Midori Filiz Nishimura, Asami Nishikori, Yasuharu Sato, Yoshito Nishimura
Journal of Gastroenterology and Hepatology.2022; 37(10): 1865. CrossRef
Utility of gastric biopsy in diagnosing IgG4‐related gastrointestinal disease
Kaori Uchino, Kenji Notohara, Takeshi Uehara, Yasuhiro Kuraishi, Junya Itakura, Akihiro Matsukawa
Pathology International.2021; 71(2): 124. CrossRef

A Rare Case of Adenosquamous Carcinoma Arising in the Background of IgG4-Related Lung Disease: Sangjoon Choi, Sujin Park, Man Pyo Chung, Tae Sung Kim, Jong Ho Cho, Joungho Han; J Pathol Transl Med. 2019;53(3):188-191. Published online March 11, 2019; DOI: https://doi.org/10.4132/jptm.2019.02.21

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Abstract PDF

IgG4-related disease is a systemic inflammatory disease and is known as IgG4-related lung disease (IgG4-RLD) when it involves the respiratory system. Primary lung cancer arising from a background of IgG4-RLD is very rare. Herein, we report a case of adenosquamous carcinoma arising from the background of IgG4-RLD and presenting as an interstitial lung disease pattern. A 66-year-old man underwent lobectomy under the impression of primary lung cancer. Grossly, the mass was ill-defined and gray-tan colored, and the background lung was fibrotic. Microscopically, tumor cells showed both squamous and glandular differentiation. Dense lymphoplasmacytic infiltration with fibrosis and obliterative phlebitis were seen in the background lung. IgG4 immunohistochemical stain showed diffuse positivity in infiltrating plasma cells. Primary lung adenosquamous carcinoma has not been reported in a background of IgG4-RLD. Due to the rarity of IgG4-RLD, physicians must follow patients with IgG4-RLD over long periods of time to accurately predict the risk of lung cancer.

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Endometrioid Carcinomas of the Ovaries and Endometrium Involving Endocervical Polyps: Comprehensive Clinicopathological Analyses
Jihee Sohn, Yurimi Lee, Hyun-Soo Kim
Diagnostics.2022; 12(10): 2339. CrossRef
A Case of IgG4-related Disease Composed of a Paravertebral Tumor Alone with Multiple Lung Cancers
Mutsumi Ozasa, Toyomitsu Sawai, Yosuke Harada, Sumako Yoshioka, Nobuko Matsuo, Hiroshi Mukae
Haigan.2021; 61(3): 213. CrossRef
Serous Carcinoma of the Endometrium with Mesonephric-Like Differentiation Initially Misdiagnosed as Uterine Mesonephric-Like Adenocarcinoma: A Case Report with Emphasis on the Immunostaining and the Identification of Splice Site TP53 Mutation
Sangjoon Choi, Yoon Yang Jung, Hyun-Soo Kim
Diagnostics.2021; 11(4): 717. CrossRef
A Case of IgG4-related Thyroiditis Diagnosed by Total Thyroidectomy
Daiki Sakamoto, Masao Yagi, Hiroshi Iwai
Practica Oto-Rhino-Laryngologica.2021; 114(7): 547. CrossRef
Mesonephric-like Differentiation of Endometrial Endometrioid Carcinoma: Clinicopathological and Molecular Characteristics Distinct from Those of Uterine Mesonephric-like Adenocarcinoma
Sujin Park, Go Eun Bae, Jiyoung Kim, Hyun-Soo Kim
Diagnostics.2021; 11(8): 1450. CrossRef
Mesonephric-like Adenocarcinoma of the Uterine Corpus: Comprehensive Immunohistochemical Analyses Using Markers for Mesonephric, Endometrioid and Serous Tumors
Hyunjin Kim, Kiyong Na, Go Eun Bae, Hyun-Soo Kim
Diagnostics.2021; 11(11): 2042. CrossRef
Not Cancer After All: Two Rare Cases of IgG4-Related Lung Disease
Josué Pinto, Carla Damas, António Morais
Archivos de Bronconeumología.2020; 56(1): 53. CrossRef
Not Cancer After All: Two Rare Cases of IgG4-Related Lung Disease
Josuèc) Pinto, Carla Damas, António Morais
Archivos de Bronconeumología (English Edition).2020; 56(1): 52. CrossRef
Axillary lymphadenopathy with IgG4 positive plasma cell infiltration as differential diagnosis of metastatic lung adenocarcinoma
Yutaro Ito, Masanori Harada, Namio Kagoo, Tsutomu Kubota, Koshiro Ichijyo, Eisuke Mochizuki, Masahiro Uehara, Shun Matsuura, Masaru Tsukui, Naoki Koshimizu
Respiratory Medicine Case Reports.2020; 31: 101196. CrossRef

Isolated Mass-Forming IgG4-Related Cholangitis as an Initial Clinical Presentation of Systemic IgG4-Related Disease: Seokhwi Kim, Hyunsik Bae, Misun Choi, Binnari Kim, Jin Seok Heo, Ho Seong Kim, Seung Hee Choi, Kee-Taek Jang; J Pathol Transl Med. 2016;50(4):300-305. Published online January 11, 2016; DOI: https://doi.org/10.4132/jptm.2015.12.01

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Abstract PDF

IgG4-related disease (IgG4-RD) may involve multiple organs. Although it usually presents as diffuse organ involvement, localized mass-forming lesions have been occasionally encountered in pancreas. However, the same pattern has been seldom reported in biliary tract. A 61-year-old male showed a hilar bile duct mass with multiple enlarged lymph nodes in imaging studies and he underwent trisectionectomy under impression of cholangiocarcinoma. Gross examination revealed a mass-like lesion around hilar bile duct. Histopathologically, dense lymphoplasmacytic infiltration and storiform fibrosis were identified without evidence of malignancy. Immunohistochemical stain demonstrated rich IgG4-positive plasma cell infiltration. Follow-up imaging studies disclosed multiple enlarged lymph nodes with involvement of pancreas and perisplenic soft tissue. The lesions have been significantly reduced after steroid treatment, which suggests multi-organ involvement of systemic IgG4-RD. Here, we report an unusual localized mass-forming IgG4-related cholangitis as an initial presentation of IgG4-RD, which was biliary manifestation of systemic IgG4-related autoimmune disease.

Citations

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Pathologic interpretation of endoscopic ultrasound–guided fine needle aspiration cytology/biopsy for pancreatic lesions
Haeryoung Kim, Kee-Taek Jang
Journal of Pathology and Translational Medicine.2020; 54(5): 367. CrossRef
Multivisceral IgG4-related disease presenting as recurrent massive gastrointestinal bleeding: a case report and literature review
Xuexue Deng, Ronghua Fang, Jianshu Zhang, Rongqiong Li
BMC Gastroenterology.2018;[Epub] CrossRef
Recent advances in understanding and managing IgG4-related disease
Anna R. Wolfson, Daniel L. Hamilos
F1000Research.2017; 6: 185. CrossRef

Review

Overview of IgG4-Related Tubulointerstitial Nephritis and Its Mimickers: Hyeon Joo Jeong, Su-Jin Shin, Beom Jin Lim; J Pathol Transl Med. 2016;50(1):26-36. Published online December 14, 2015; DOI: https://doi.org/10.4132/jptm.2015.11.09

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Abstract PDF

Tubulointerstitial nephritis (TIN) is the most common form of renal involvement in IgG4-related disease. It is characterized by a dominant infiltrate of IgG4-positive plasma cells in the interstitium and storiform fibrosis. Demonstration of IgG4-positive plasma cells is essential for diagnosis, but the number of IgG4-positive cells and the ratio of IgG4-positive/IgG-positive plasma cells may vary from case to case and depending on the methods of tissue sampling even in the same case. IgG4-positive plasma cells can be seen in TIN associated with systemic lupus erythematosus, Sjögren syndrome, or anti-neutrophil cytoplasmic antibody–associated vasculitis, which further add diagnostic confusion and difficulties. To have a more clear view of IgG4-TIN and to delineate differential points from other TIN with IgG4-positive plasma cell infiltrates, clinical and histological features of IgG4-TIN and its mimickers were reviewed. In the rear part, cases suggesting overlap of IgG4-TIN and its mimickers and glomerulonephritis associated with IgG4-TIN were briefly described.

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IgG4-related kidney disease: Clinicopathologic features, differential diagnosis, and mimics
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Die Nephrologie.2023; 18(4): 249. CrossRef
Concurrent anti-neutrophil cytoplasmic antibody-associated glomerulonephritis and IgG4-associated tubulointerstitial nephritis with C3 glomerulonephritis
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Medicine.2020; 99(5): e18857. CrossRef
A case of eosinophilic granulomatosis with polyangiitis as a mimicker of IgG4-related disease
Ryuichiro Kanda, Satoshi Kubo, Kazuhisa Nakano, Akio Kawabe, Aya Nawata, Kentaro Hanami, Shingo Nakayamada, Yoshiya Tanaka
Modern Rheumatology Case Reports.2020; 4(2): 278. CrossRef
Renal tubular acidosis as the initial presentation of Sjögren’s syndrome
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BMJ Case Reports.2019; 12(8): e230402. CrossRef
Immunoglobulin G4-related kidney diseases: An updated review
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Systemic lupus erythematosus in a patient with an organic lesion of the central nervous system: practicaldifferential diagnosis
E. V. Lebedeva, M. V. Novoseltsev, A. N. Lvov, I. V. Khamaganova
Klinicheskaya dermatologiya i venerologiya.2018; 17(6): 21. CrossRef
Concurrent IgG4-related tubulointerstitial nephritis and IgG4 myeloperoxidase-anti-neutrophil cytoplasmic antibody positive crescentic glomerulonephritis
Tao Su, Li Yang, Zhao Cui, Su-xia Wang, Ming-hui Zhao
Medicine.2017; 96(20): e6707. CrossRef
IgG4-Related Kidney Disease: Report of a Case Presenting as a Renal Mass
Daniele Bianchi, Luca Topazio, Gabriele Gaziev, Valerio Iacovelli, Pierluigi Bove, Alessandro Mauriello, Enrico Finazzi Agrò
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