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Naoki Konno, Mitsuru Sugimoto, Tadayuki Takagi, Makiko Furuya, Tomoyuki Asano, Shuzo Sato, Hiroko Kobayashi, Kiyoshi Migita, Yoshiaki Miura, Taichi Aihara, Atsushi Komatsuda, Hiromasa Ohira, Hiroshi Watanabe
BACKGROUND: Although increased serum IgG4 level and tissue infiltration of IgG4-positive cells are key events in IgG4-related disease (IgG4RD), and nearly half of IgG4RD patients show hypocomplementemia, the role of IgG4 in the pathogenesis of IgG4RD remains unclear. Many reports show that altered IgG glycosylation, especially IgG with agalactosylated N-linked glycan (G0 N-glycan), have proinflammatory roles including complement activation, implicated in the pathogenesis of various inflammatory diseases...
2018: PloS One
Yayoi Shimazu, Tatsuki Uchiyama, Chisaki Mizumoto, Tomoharu Takeoka, Masaaki Tsuji, Kenjiro Tomo, Koji Takaori, Naoki Sakai, Tomoko Okuno, Tatsuharu Ohno
IgG4-related disease (IgG4RD) is a multi-organ disorder characterized by an elevated serum IgG4 level and IgG4-positive plasma cell infiltration of the affected organs, accompanied by tissue fibrosis and sclerosis. Although it can affect any organ, to our knowledge, no cases involving concurrent autoimmune neutropenia and thrombocytopenia have been reported. A 62-year-old man visited our hospital and was diagnosed with IgG4RD accompanied by interstitial pneumonitis, lymphadenopathy, and interstitial nephritis...
February 28, 2018: Internal Medicine
Takaya Iyoki, Takeshi Maehana, Toshiaki Tanaka, Motohisa Yamamoto, Hiroki Takahashi, Naoya Masumori
About half of idiopathic retroperitoneal fibrosis might be classified as a IgG4-related disease, a newly characterized disease that is especially known to be sensitive to steroid therapy. We developed a new protocol for diagnosis and treatment of retroperitoneal fibrosis, which included aggressive diagnosis of IgG4- related disease. We retrospectively reviewed 22 cases with idiopathic retroperitoneal fibrosis that were diagnosed and treated according to our protocol. Of them, 10 patients (45.5%) had no evidence of IgG4- related disease (non-IgG4RD group), whereas 12 patients (54...
November 2017: Hinyokika Kiyo. Acta Urologica Japonica
Shigeyuki Kawa, Markus Skold, David B Ramsden, Antony Parker, Stephen J Harding
BACKGROUND: IgG4-related disease (IgG4-RD) is an immune-mediated and chronic fibroinflammatory condition that affects almost any organ and often involves multiple organs in the same patient. In this review article, we address the clinical utility of measuring serum immunoglobulin G subclass 4 concentration ([IgG4]) in IgG4-RD diagnosis and in disease monitoring. METHODS: We discuss the latest literature on the relevance of [IgG4] to the investigation and management of IgG4RDs...
September 1, 2017: Clinical Laboratory
Mei Liu, Xiru Li, Ying Li, Zhuo Wang, Liuquan Cheng, Xin Song, Yun Wu
BACKGROUND: A proportion of cases of Rosai-Dorfman disease exhibit some histological features consistent with IgG4-related disease (IgG4RD). Several investigators have discussed whether Rosai-Dorfman disease belongs to the spectrum of IgG4RD or is concurrent with it by coincidence. OBJECTIVE: To elucidate the relationship between the two diseases, we report key features, including IgG4 and amyloid levels, of four cases of Rosai-Dorfman disease in the breast. METHODS: The histological features of the four cases were analyzed and the numbers of IgG4+ plasma cells and IgG4/IgG ratios were evaluated...
June 1, 2017: Asian Pacific Journal of Allergy and Immunology
Renato Cesar Ferreira da Silva, Scott M Lieberman, Henry T Hoffman, Bruno Policeni, Amani Bashir, Richard J H Smith, T Shawn Sato
Immunoglobulin G4-related disease (IgG4RD) is an immune-mediated condition characterized by lymphoplasmacytic infiltrates and fibrosis of affected organs. IgG4RD may affect many different organs either individually or together in a multiorgan condition and, thus, incorporates a wide range of fibroinflammatory phenotypes with shared pathologic features. Although IgG4RD most commonly occurs in late adulthood, it may affect children and adolescents. Only one case of IgG4RD presenting as isolated submandibular gland involvement has been reported in the pediatric population...
March 2017: Radiology Case Reports
Jithin Lal, Suresh Bhat, Siddalingeswar Doddamani, Lekshmi Devi
ImmunoglobulinG4 related disease (IgG4RD) is a systemic fibroinflammatory disease recognized recently. This usually presents with multiorgan involvement. We are reporting a case of a35-year-old male patient with isolated IgG4RD of the testis. This patient presented with right testis pain which responded to conservative treatment. However, later, he reported with hard swelling in the right testis which on imaging was suggestive of malignancy and hence underwent radical orchiectomy. Histopathology with immunohistochemical staining confirmed IgG4RD of the testis...
October 2016: Indian Journal of Urology: IJU: Journal of the Urological Society of India
Deep Dutta, Arvind Ahuja, Chitra Selvan
Ig-G4 related disease (IgG4RD) is a heterogeneous disorder with multi-organ involvement recognised as a separate entity at the start of this century only. It is currently one of the hottest areas of clinical and translational research across specialties. Thyroid involvement in IgG4RD is rare, believed to occur in less than 4% of cases, may be isolated or may be associated with other organ involvement. As of today Riedel's thyroiditis, fibrosing variant of Hashimoto's thyroiditis, and few patients of Graves' orbitopathy represent the types of IgG4-related thyroid disease (IgG4RTD)...
2016: Endokrynologia Polska
Rishi Bolia, Sze Yee Chong, Lee Coleman, Duncan MacGregor, Winita Hardikar, Mark R Oliver
We report 3 children who presented with fever and abdominal pain, deranged liver function tests, and on abdominal ultrasound were found to have an enlarged pancreas, substantial abdominal lymphadenopathy, and extrahepatic biliary duct dilatation. After ruling out malignancy, probable immunoglobulin G4-related disease (IgG4RD) associated with autoimmune pancreatitis was considered. This condition was first described in the adults and often mimics pancreatic cancer. It can involve multiple organs, either synchronously or metachronously, and is rarely reported in children...
July 2016: ACG Case Reports Journal
Karla Lais Pêgas, Eduardo Cambruzzi, Gisele Lobato
IgG4-related disease (IgG4RD) is a recent inflammatory process of supposed autoimmune etiology, which is characterized by elevated serum IgG4 levels, dense lymphoplasmacytic infiltration rich in IgG4-positive plasma cells and storiform fibrosis. Tubulointerstitial nephritis is the most common renal manifestation, with different degrees of kidney dysfunction and variable clinical findings. Herein, the authors describe a new case of IgG4 tubulointerstitial nephritis (IgG4TN), and discuss clinic and pathologic criteria...
June 2016: Jornal Brasileiro de Nefrologia: ʹorgão Oficial de Sociedades Brasileira e Latino-Americana de Nefrologia
F Moosig, J H Schirmer, P Lamprecht, J U Holle
Rheumatologist should be familiar with the concept of IgG4-related disease (IgG4-RD). Due to the clinical spectrum IgG4-RD can fall directly within the scope of rheumatology and are often diagnosed primarily by rheumatologists. Furthermore, IgG4RD are relevant differential diagnoses for many other rheumatic conditions. Finally, there are an increasing amount of data suggesting an important role of immunological processes observed in IgG4-RD for other rheumatic diseases.
September 2016: Zeitschrift Für Rheumatologie
Abhijit Chougule, Amanjit Bal
OBJECTIVES: There is marked inconsistency in reporting the key features of IgG4-related inflammatory pseudotumor (IPT) cases. We aimed to analyze the various aspects of IgG4-related IPTs and to test the performance of the consensus criteria for their diagnosis. METHODS: PubMed database was searched for IgG4-related IPT cases. The data regarding patient demographics, clinical presentation, laboratory findings, histopathological features, and treatment response are extracted and are presented here in a descriptive manner...
March 2017: Modern Rheumatology
Davide Gianfreda, Claudio Musetti, Maria Nicastro, Federica Maritati, Rocco Cobelli, Domenico Corradi, Augusto Vaglio
Immunoglobulin-G4 (IgG4)-related disease (IgG4RD) is a fibro-inflammatory disorder characterized by tissue-infiltrating IgG4 plasma cells, and, often, high serum IgG4. Several autoimmune, infectious, or proliferative conditions mimic IgG4RD. Erdheim-Chester disease (ECD) is a rare non-Langerhans cell histiocytosis, characterized by foamy histiocytic infiltration, fibrosis, and chronic inflammation. ECD and IgG4RD manifestations may overlap.A patient presented with huge fibrous retroperitoneal masses causing compression on neighboring structures; the case posed the challenge of the differential diagnosis between IgG4RD and ECD mainly because of a prominent serum and tissue IgG4 response...
May 2016: Medicine (Baltimore)
W Egner, K Swallow, R J Lock, D Patel
Immunoglobulin (Ig)G4 disease can have apparently 'normal' levels of IgG4 due to antigen excess conditions. IgG4 measurement therefore appears falsely low. UK National External Quality Assurance Scheme (UK NEQAS) data and other reports have suggested that this problem occurred despite pre-existing antigen excess detection steps. To determine the clinical relevance of the problem, we examined the prevalence and characteristics of prozoning in our laboratory and patient cohorts. We establish that the prevalence of raised IgG4 in routine IgG4 analysis is low (< 1%) using one of the two routine methods in use in the United Kingdom...
October 2016: Clinical and Experimental Immunology
Hong Ryeol Cheong, Bong Eun Lee, Geun Am Song, Gwang Ha Kim, Sung Gyu An, Won Lim
Immunoglobulin G4 (IgG4)-related disease (IgG4RD) is a relatively recently recognized entity that is histopathologically characterized by an extensive infiltration of lymphocytes and IgG4-positive plasma cells with dense fibrosis. IgG4RD is now known to affect any organ system, and a few cases of gastrointestinal lesions have also been reported. However, solitary IgG4RD of the stomach is still very rare. Furthermore, as it can mimic malignant conditions, it is important to recognize this disease to avoid unnecessary surgery...
March 2016: Clinical Endoscopy
Mitsuru Sugimoto, Hiroshi Watanabe, Tomoyuki Asano, Shuzo Sato, Tadayuki Takagi, Hiroko Kobayashi, Hiromasa Ohira
OBJECTIVE: To investigate which IgG subclasses contribute to the activation of the complement pathway in IgG4-related disease (IgG4RD) patients with hypocomplementemia. METHODS: Sera of IgG4RD patients were analyzed for the binding ability of IgG subclasses to complement component 1q (C1q). Polyethylene glycol (PEG) precipitates containing immune complexes (ICs) in sera of IgG4RD patients were analyzed for IgG subclass composition by Western blotting. PEG precipitates containing ICs (PEG-ICs) in sera of patients were also analyzed for their ability to consume complement in normal human serum (NHS) using a total complement hemolytic (CH50) assay and a commercial kit to measure the complement capacity of all three individual complement pathways...
2016: Modern Rheumatology
Abhijit Chougule, Amanjit Bal, Ashim Das, Gurpreet Singh
IgG4 related disease (IgG4RD) is a recently recognised condition characterised by mass forming lesions associated with storiform fibrosis, obliterative phlebitis, lymphoplasmacytic infiltrate rich in IgG4 positive plasma cells and elevated serum IgG4 levels. Although rare, mammary involvement has been reported as IgG4 related sclerosing mastitis, the morphological counterpart of a growing family of IgG4 related diseases. A total of 17 cases belonging to mass forming benign inflammatory breast lesions such as plasma cell mastitis, granulomatous lobular mastitis, non-specific mastitis and inflammatory pseudotumour were investigated as a possible member of IgG4 related sclerosing mastitis...
January 2015: Pathology
Fang Fang, Yanming Li, Mingjun Sun, Songtao Hu, Zheng Wang, Dongge Liu, Chen Wang
OBJECTIVE: To evaluate the clinical and pathological features of IgG4-related disease (IgG4RD). METHODS: The clinical data, laboratory profiles, radiological, pathological and therapeutic features of eight cases of IgG4RD were analyzed. This cohort included two cases of common bile duct and partial hepatectomy specimens, two of submandibular gland excision specimens, one from lung biopsy specimen, one from open lung biopsy specimen, one from renal biopsy specimen, and one from renal excision specimen...
September 2014: Zhonghua Bing Li Xue za Zhi Chinese Journal of Pathology
Clio P Mavragani, George E Fragoulis, Dimitra Rontogianni, Maria Kanariou, Haralampos M Moutsopoulos
OBJECTIVE: To determine IgG4 levels in a cohort of consecutive patients with primary Sjögren's syndrome (SS) and other autoimmune diseases and explore whether they associate with distinct clinical, serologic, and histopathologic features. METHODS: Serum IgG4 levels were measured in 133 primary SS patients and 49 healthy donors (HDs). Seventy-four lupus and 54 rheumatoid arthritis (RA) patients served as disease controls. Immunohistochemical IgG4 analysis was performed in paraffin-embedded minor salivary gland (MSG) tissues...
May 2014: Arthritis Care & Research
Naoko Ito, Kunimasa Yagi, Mitsuhiro Kawano, Yukiko Mori, Satoko Okazaki, Daisuke Chujo, Yoshiyu Takeda, Junji Kobayashi, Masakazu Yamagishi
IgG4-related disease (IgG4RD) is a newly recognized systemic disease characterized by the elevation of serum IgG4 levels and abundant IgG4-positive plasma cell infiltration into the involved organs. Few data exist regarding the relationship between diabetes or glucose intolerance and IgG4RD in the absence of obvious type 1 autoimmune pancreatitis (AIP). Therefore, we are characterizing pancreatic endocrine function in IgG4RD patients with no signs of type 1 AIP. 28 patients (12 men, mean age 62.1 years old) were diagnosed as having IgG4RD from serum IgG4 levels, histopathology and images...
2014: Endocrine Journal
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