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REVIEW
Marco Lanzillotta, Emma Culver, Amita Sharma, Yoh Zen, Wen Zhang, John H Stone, Emanuel Della-Torre
A prompt response to glucocorticoids is a clinical hallmark of IgG4-related disease. However, manifestations characterised by prominent tissue fibrosis on histological examination can be less responsive to glucocorticoid therapy than other types of IgG4-related disease. These manifestations include retroperitoneal fibrosis, fibrosing mediastinitis, Riedel thyroiditis, orbital pseudotumor, and hypertrophic pachymeningitis, among others. To explain this discrepancy, a preliminary distinction into proliferative and fibrotic phenotypes of IgG4-related disease has been proposed on the basis of clinical presentation, pathological features, and response to immunosuppressive therapy...
March 28, 2024: Lancet Rheumatology
#22
REVIEW
Guy Katz, Yasmin Hernandez-Barco, Diego Palumbo, Thomas V Guy, Lingli Dong, Cory A Perugino
IgG4-related disease is an immune-mediated disease that can lead to substantial morbidity and organ damage. Capable of affecting nearly any organ system or anatomic site, and showing considerable overlap in clinical presentation with various other diseases, IgG4-related disease often poses a diagnostic challenge for clinicians. Furthermore, there are no diagnostic biomarkers with high specificity for IgG4-related disease, and histopathological examination is nuanced and requires clinical correlation for accurate diagnosis...
March 28, 2024: Lancet Rheumatology
#23
Yoann Roubertou, Romain Euvrard
No abstract text is available yet for this article.
April 2024: European Heart Journal. Case Reports
#24
REVIEW
Lorenzo Azzi, Francesca Magnoli, Diana Krepysheva, Federico Fontana, Andrea Coppola, Antonella Cappelli, Marta Dani, Paolo Battaglia, Dimitri Rabbiosi
This study aimed to review the lesser-known intraoral manifestations of immunoglobulin G4-related disease (IgG4-RD). In this paper we report an unprecedented case of oral IgG4-RD mimicking angiolymphoid hyperplasia with eosinophilia (ALHE), and another case presenting as plasma cell gingivitis. We then performed a scoping review of published cases of IgG4-RD involving the oral cavity. The following data were collected for each case: age, sex, intraoral site(s) involved, clinical appearance, imaging features, serum IgG4 values, histopathology, treatment, and follow-up duration...
April 3, 2024: Head & Neck
#25
JOURNAL ARTICLE
Yan Qin, Yuhan Jia, Congcong Liang, Rui Fu, Zhaojun Liang, Yanlin Wang, Min Feng, Chong Gao, Jing Luo
BACKGROUND: Detection of IgG subclasses (IgGSc) is vital for the diagnosis and management of disease, especially IgG4-related diseases (IgG4-RD). This study aimed to evaluate the performances of the chemiluminescent immunoassay (CLIA) for detecting IgGSc and diagnosing IgG4-RD by IgGSc. METHODS: A total of 40 individuals with IgG4-RD, 40 with primary Sjogren's syndrome (pSS), and 40 healthy controls (HCs) were enrolled. Serum samples were collected for the simultaneous detection of IgG1, IgG2, IgG3, and IgG4 by the Siemens immunonephelometric assay and the CLIA...
April 2, 2024: Journal of Clinical Laboratory Analysis
#26
JOURNAL ARTICLE
L P Guo, W R Wang, J P Liu, B M Wang, L Zhou
Objective: To deepen understanding of IgG4 -related diseases (RDs), we analyzed the associated lymphocyte subtypes, and explored the pathogenesis and potential immunotherapeutic targets. Methods: Eighty-six patients with IgG4 -RDs were enrolled, and their clinical characteristics, peripheral lymphocyte subtypes, and disease course were analyzed. Results: The mean age of the participants was 36-87(62±11) years; 51 were male (59.3%) and 35 were women (40.7%); and 34.9% had a history of allergy. Follow-up lasted 4...
April 1, 2024: Zhonghua Nei Ke za Zhi [Chinese Journal of Internal Medicine]
#27
JOURNAL ARTICLE
Xun Tao, Siddharth Sukumaran, Gizette Sperinde, Chang Liu, Michelle Irwin Beardsley, Peter Day, Matt Kalo, Ebehiremen Ayewoh, Hao Cai, Yehong Wang, Inyoung Jun, Kyle Hirst, Van Nguyen, Shan Chung, Donna Lee, Annemarie Lekkerkerker, Eric Stefanich
Sialic acid (SA) is crucial for protecting glycoproteins from clearance. Efmarodocokin alfa (IL-22Fc), a fusion protein agonist that links IL-22 to the crystallizable fragment (Fc) of human IgG4, contains 8 N-glycosylation sites and exhibits heterogeneous and variable terminal sialylation biodistribution. This presents a unique challenge for Pharmacokinetic (PK) and Pharmacodynamic (PD) analysis and cross-species translation. In this study, we sought to understand how varying SA levels and heterogeneous distribution contribute to IL-22Fc's complex PKPD properties...
March 30, 2024: Journal of Pharmaceutical Sciences
#28
JOURNAL ARTICLE
Kevin Muñoz Forti, Gary A Weisman, Kimberly J Jasmer
Salivary gland damage and hypofunction result from various disorders, including autoimmune Sjögren's disease (SjD) and IgG4-related disease (IgG4-RD), as well as a side effect of radiotherapy for treating head and neck cancers. There are no therapeutic strategies to prevent the loss of salivary gland function in these disorders nor facilitate functional salivary gland regeneration. However, ongoing aquaporin-1 gene therapy trials to restore saliva flow show promise. To identify and develop novel therapeutic targets, we must better understand the cell-specific signaling processes involved in salivary gland regeneration...
2024: Journal of Oral Biology and Craniofacial Research
#29
JOURNAL ARTICLE
Hugo Parente, Adriana Carones, Augusto Silva, Bruna Silva, Carlos Costa, Catarina Dantas Soares, Inês Santos, José Miguel Bernardes, Manuel Silvério-António, Rita Pinheiro Torres, Filipa Teixeira
No abstract text is available yet for this article.
2024: ARP Rheumatol
#30
JOURNAL ARTICLE
Using atomistic solution scattering modelling to elucidate the role of the Fc glycans in human IgG4.
Valentina A Spiteri, James Doutch, Robert P Rambo, Jayesh S Bhatt, Jayesh Gor, Paul A Dalby, Stephen J Perkins
Human immunoglobulin G (IgG) exists as four subclasses IgG1-4, each of which has two Fab subunits joined by two hinges to a Fc subunit. IgG4 has the shortest hinge with 12 residues. The Fc subunit has two glycan chains, but the importance of glycosylation is not fully understood in IgG4. Here, to evaluate the stability and structure of non-glycosylated IgG4, we performed a multidisciplinary structural study of glycosylated and deglycosylated human IgG4 A33 for comparison with our similar study of human IgG1 A33...
2024: PloS One
#31
REVIEW
Maurizio Bruschi, Andrea Angeletti, Marco Prunotto, Pier Luigi Meroni, Gian Marco Ghiggeri, Gabriella Moroni, Renato Alberto Sinico, Franco Franceschini, Micaela Fredi, Augusto Vaglio, Andrea Cavalli, Leonardo Scapozza, Jigar J Patel, John C Tan, Ken C Lo, Lorenzo Cavagna, Andrea Petretto, Federico Pratesi, Paola Migliorini, Francesco Locatelli, Giulia Pazzola, Giampaola Pesce, Domenico Giannese, Angelo Manfredi, Giuseppe A Ramirez, Pasquale Esposito, Giuseppe Murdaca, Simone Negrini, Federica Bui, Barbara Trezzi, Giacomo Emmi, Ilaria Cavazzana, Valentina Binda, Paride Fenaroli, Isabella Pisan, Carlomaurizio Montecucco, Domenico Santoro, Francesco Scolari, Federica Mescia, Stefano Volpi, Marta Mosca, Angela Tincani, Angelo Ravelli, Corrado Murtas, Giovanni Candiano, Gianluca Caridi, Edoardo La Porta, Enrico Verrina
Deposition of autoantibodies in glomeruli is a key factor in the development of lupus nephritis (LN). For a long time, anti-dsDNA and anti-C1q antibodies were thought to be the main cause of the kidney damage. However, recent studies have shown that the list of autoantibidies that have renal tropism and deposit in the kidney in LN is increasing and the link between anti-dsDNA and renal pathology is weak due to potential confounders. Aspecific bindings of dsDNA with cationic antibodies and of anti-dsDNA with several renal antigens such as actinin, laminin, entactin, and annexinA2 raised doubts about the specific target of these antibodies in the kidney...
March 27, 2024: Autoimmunity Reviews
#32
Bo Yang, Wei Gou, Naiying Lan, Qing Shao, Weifeng Hu, Cheng Xue, Nanmei Liu
BACKGROUND: Tislelizumab, a humanized IgG4 anti-PD-1 monoclonal antibody has been approved in China and Europe. According to the published clinical research, tislelizumab shows satisfactory safety profile. No severe hepatotoxicity or acute kidney injury were reported. CASE PRESENTATION: We presented a case study of a 74-year-old man who developed acute kidney injury (grade 3) and acute liver injury (grade 4) after being administered tislelizumab for the treatment of esophageal squamous cell carcinoma...
2024: Frontiers in Oncology
#33
Daigo Nishijo, Koki Nakanishi, Masanori Kono, Shogo Shimada, Issei Komuro
Although cardiovascular involvement in immunoglobulin G4-related disease is uncommon, it can lead to life-threatening events. We report a patient with multiple coronary aneurysms that were diagnosed by multimodal imaging. The patient had been treated with prednisolone for more than 15 years for immunoglobulin G4-related disease.
March 20, 2024: JACC. Case reports
#34
JOURNAL ARTICLE
Mayur Parkhi, Debajyoti Chatterjee, Dharambir Kashyap, Ashish Aggarwal, Bishan Radotra
Rosai-Dorfman disease (RDD) is characterized by clonal proliferation of S-100 positive histiocytes and variable emperipolesis. It commonly affects cervical lymph nodes. Central nervous system (CNS) involvement is extremely rare. We attempted to evaluate the Cyclin D1 expression and frequency of KRAS and BRAF mutations in the RDD involving the CNS. All patients with histopathologically diagnosed RDD involving CNS were recruited from 2011 to 2022. All cases were subjected to immunohistochemistry for CD68, CD163, S100, CD1a, GFAP, CD207, EMA, ALK, BRAFV600E, IgG4, IgG, and CyclinD1...
March 28, 2024: Neuropathology: Official Journal of the Japanese Society of Neuropathology
#35
JOURNAL ARTICLE
Kazuma Ino, Yoshiyuki Arinuma, Masashi Akiya, Sabine Kajita, Sosei Okina, Junichi Sakamoto, Tomoki Tanaka, Yu Matsueda, Tatsuhiko Wada, Sumiaki Tanaka, Kenji Oku, Kunihiro Yamaoka
Immunoglobulin (Ig) G4-related disease (IgG4-RD) is a systemic inflammatory disease characterized by elevated serum IgG4, IgG4+ cell infiltration, storiform fibrosis, and obliterative phlebitis. While IgG4-RD can affect various organs, gastrointestinal tract involvement is less common. Here, we report a 70-year-old female with IgG4-RD complicated with diffuse and chronic gastrointestinal inflammation which led to small intestinal perforation. She had been suffering from anorexia, abdominal pain, vomiting, and diarrhea, and hospitalized due to recurrent ileus...
March 28, 2024: Modern rheumatology case reports
#36
Taro Ando, Hiroyuki Nitta, Akira Umemura, Hirokatsu Katagiri, Shoji Kanno, Daiki Takeda, Masao Nishiya, Noriyuki Uesugi, Tamotsu Sugai, Akira Sasaki
INTRODUCTION: Autoimmune pancreatitis (AIP) is recognized as a disease with a good prognosis that responds well to steroids, but the complication of pancreatic ductal adenocarcinoma (PDAC) in AIP is a rare condition. We report a case of PDAC encapsulated by tumor-forming type 1 AIP. CASE PRESENTATION: The patient, a 65-year-old female, was found to have high CA19-9 levels and a pancreatic mass with a diameter of 30 mm on abdominal ultrasonography. Contrast-enhanced computed tomography revealed a 40-mm mass in the tail of the pancreas that had a 27-mm oligemic mass inside it...
2024: Case Reports in Gastroenterology
#37
Alisa Julija Dulko, Irena Butrimiene, Alma Cypiene, Valdas Peceliunas, Donatas Petroska, Ernesta Stankeviciene, Rita Rugiene
Both scleroderma and immunoglobulin G4-related disease (IgG4-RD) are systemic fibro-inflammatory diseases characterised by lymphoplasmacytic infiltrates. IgG4-RD and systemic sclerosis (SSc) may share common pathophysiological mechanisms, but no examples of co-occurrence of the diseases have been found. Autologous haematopoietic stem cell transplantation (AHSCT) is implemented in selected rapidly progressive SSc with a high risk of organ failure. However, existing guidelines are based on clinical trials that do not represent the entire patient population and exclude critically ill patients with no therapeutic alternatives...
March 18, 2024: Medicina
#38
JOURNAL ARTICLE
Takeshi Kaneko, Yasutaka Okita, Yasuhiro Kato, Masashi Narazaki, Atsushi Kumanogo
No abstract text is available yet for this article.
March 27, 2024: QJM: Monthly Journal of the Association of Physicians
#39
JOURNAL ARTICLE
Parag Vijayvergia, Sayan Mukherjee, Lily Singh, Urmila Dhakad
Immunoglobulin G4-related disease (IgG4-RD) is an immune-driven fibroinflammatory disease that presents as tumefactive lesions that not only commonly affects the pancreas, lacrimal and salivary glands, lung, liver and kidney but can also affect any organs. However, involvement of the urinary bladder in IgG4-RD is rarely reported. We describe a case of IgG4-RD involving the urinary bladder mimicking carcinoma and review the published literature-a 39-year-old male presented with complaints of dysuria, urgency and hesitancy...
March 2, 2024: Modern rheumatology case reports
#40
JOURNAL ARTICLE
Xiaoyu Cao, Shaoxiang Li, Jin Wan, Zhibo Yu, Gehong Dong, Wei Zhou
OBJECTIVES: To explore the effectiveness of tofacitinib for immunoglobulin G4-related disease (IgG4-RD) and idiopathic retroperitoneal fibrosis (IRF), and investigate the expression of JAKs in the lesion of these diseases. METHODS: Clinical data of patients with IgG4-RD or IRF who were administered with tofacitinib monotherapy were collected. IgG4-RD responder index (IgG4-RD RI) was assessed. The expression of JAK1, JAK2, JAK3, and TYK2 were analysed with immunohistochemistry staining in three salivary glands specimens of IgG4-RD and one retroperitoneal tissue of IRF...
March 26, 2024: Clinical and Experimental Rheumatology
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