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IgG4 related disease

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https://www.readbyqxmd.com/read/29774578/the-immunobiology-of-rankl-and-myeloid-derived-suppressor-cell-activation-in-igg4-related-sclerosing-cholangitis
#1
Min Lian, Qixia Wang, Xiang Jiang, Jun Zhang, Yiran Wei, Yanmei Li, Bo Li, Weihua Chen, Haiyan Zhang, Qi Miao, Yanshen Peng, Xiao Xiao, Li Sheng, Weici Zhang, Jingyuan Fang, Ruqi Tang, M Eric Gershwin, Xiong Ma
The primary function of myeloid derived suppressor cells (MDSCs) is reflected in their immune modulatory role in several immune-mediated diseases. In IgG4-related disease (IgG4-RD), it has been hypothesized that there are selective regulatory defects that lead to a Th2 bias immune response. Herein we have taken advantage of a large cohort of patients with IgG4-related sclerosing cholangitis (IgG4-SC), the most common extra-pancreatic involvement of IgG4-RD, as well as controls consisting of primary sclerosing cholangitis (PSC), autoimmune hepatitis (AIH) and healthy volunteers, to study MDSC...
May 18, 2018: Hepatology: Official Journal of the American Association for the Study of Liver Diseases
https://www.readbyqxmd.com/read/29768335/igg4-related-disease-of-pulmonary-artery-causing-pulmonary-hypertension
#2
Hui Deng, Sheng Zhao, Yunlong Yue, Yong Liu, Yali Xu, Jin Qian, Xiaorong Ma, Peiliang Gao, Xiaoyan Yao, Xin Jiang, Xiqi Xu, Zhicheng Jing, Yong Wang, Lei Pan, Xinying Xue
IgG4-related disease (IgG4-RD) is recognized as an immune-mediated condition with pathology features of lymphoplasmacytic infiltrate, storiform fibrosis, and obliterative phlebitis, accompanied with or without elevated serum IgG4 concentrations. However, few of pulmonary artery IgG4-RD causing pulmonary hypertension (PH) was reported.The medical records of 3 patients with pulmonary artery IgG4-RD inducing PH were analyzed retrospectively.Imaging findings demonstrated that the lesions of 3 patients located in pulmonary artery, which were initially diagnosed as pulmonary thrombus or malignant tumor...
May 2018: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29767269/-igg4-associated-cholangitis-clinical-presentation-of-an-overlooked-disease-entity
#3
REVIEW
T Herta, J Verheij, U Beuers
IgG4-associated cholangitis (IAC) is the hepatobiliary manifestation of immunoglobulin G4-related disease, which is an immune-mediated fibroinflammatory systemic disorder characterised by often elevated IgG4 serum levels and typical histopathological findings in affected tissues. IAC is frequently (>90%) accompanied by autoimmune pancreatitis type 1 (AIP), which is the pancreatic manifestation of immunoglobulin G4-related disease. In 80-85% of the cases patients with IAC are male, above 50 years of age and present with jaundice and weight loss...
May 16, 2018: Der Internist
https://www.readbyqxmd.com/read/29766732/-remission-of-the-disease-associated-related-with-immunoglobulin-igg4-accompanied-by-multiple-lymphadenopathy-after-treatment-with-rituximab-and-dexamethasone-a-case-report
#4
Zdeněk Adam, Zita Chovancová, Markéta Nová, Pavel Fabian, Zdeněk Řehák, Renata Koukalová, Marek Slávik, Luděk Pour, Marta Krejčí, Aleš Čermák, Zdeněk Král, Jiří Mayer
A disease associated with immunoglobulin IgG4 is a rare unit with very variable symptoms. We describe the course and treatment of the disease in a patient who presented with multiple lymphadenopathy and infiltrates in the area of the retroperitoneum and pelvis and signs of chronic sclerosing pancreatitis. The disease was clinically manifested by a significant loss of weight, but also by a loss of perception of taste and smell. The diagnosis was made based on a high amount of IgG4 expressing plasma cells in the sampled tissue and an increased concentration of immunoglobulins of type IgG and mainly subclass IG4...
2018: Vnitr̆ní Lékar̆ství
https://www.readbyqxmd.com/read/29765543/igg4-related-kidney-disease-the-effects-of-a-rituximab-based-immunosuppressive-therapy
#5
Giacomo Quattrocchio, Antonella Barreca, Andrea Demarchi, Laura Solfietti, Giulietta Beltrame, Roberta Fenoglio, Michela Ferro, Paola Mesiano, Stefano Murgia, Giulio Del Vecchio, Carlo Massara, Cristiana Rollino, Dario Roccatello
IgG4-related disease (IgG4-RD) is a recently recognized disorder, characterized by elevated serum IgG4 concentrations, dense tissue infiltration of IgG4-positive plasma cells and storiform fibrosis. Treatment is usually based on steroids, however, relapses and long-term adverse effects are frequent. We prospectively studied 5 consecutive patients with histologically-proven IgG4-RD and renal involvement, treated with an extended Rituximab protocol combined with steroids. Two doses of intravenous cyclophosphamide were added in 4 patients...
April 20, 2018: Oncotarget
https://www.readbyqxmd.com/read/29762245/igg4-related-disease-simulating-carcinoma-colon-with-diffuse-peritoneal-carcinomatosis-on-18f-fdg-pet-ct
#6
Shelvin Kumar Vadi, Ashwin Singh Parihar, Rajender Kumar, Harmandeep Singh, Bhagwant Rai Mittal, Amanjit Bal, Saroj Kumar Sinha
IgG4-related disease (IgG4-RD) continues to be a diagnostic challenge and a great mimicker of malignancies. We report here a case of young man who presented with subacute intestinal obstruction with initial imaging and clinical features suggestive of carcinoma colon. 18F-FDG PET/CT showed diffuse peritoneal carcinomatosis pattern typically seen with abdominal malignancies. However, the histopathology and the raised IgG4 levels diagnosed it to be IgG4-RD. Although 18F-FDG PET/CT has typical patterns corresponding to the multisystemic involvement of IgG4-RD, the index case did not show any such findings...
May 14, 2018: Clinical Nuclear Medicine
https://www.readbyqxmd.com/read/29755523/hypophyseal-involvement-in-immunoglobulin-g4-related-disease-a-retrospective-study-from-a-single-tertiary-center
#7
Yang Liu, Linjie Wang, Wen Zhang, Hui Pan, Hongbo Yang, Kan Deng, Lin Lu, Yong Yao, Shi Chen, Xiaofeng Chai, Feng Feng, Hui You, Zimeng Jin, Huijuan Zhu
This study aims to outline the clinical features and outcomes of IgG4-related hypophysitis (IgG4-RH) patients in a tertiary medical center. We reviewed clinical manifestations and imaging and pituitary function tests at baseline, as well as during follow-up. Ten patients were included. The mean age at diagnosis of IgG4-RH was 48.4 (16.0-64.0) years. An average of 3 (0-9) extrapituitary organs were involved. Five patients had panhypopituitarism, three had only posterior hypopituitarism, one had only anterior hypopituitarism, and one had a normal pituitary function...
2018: International Journal of Endocrinology
https://www.readbyqxmd.com/read/29755273/orbital-progressive-transformation-of-germinal-centers-as-part-of-the-spectrum-of-igg4-related-ophthalmic-disease-clinicopathologic-features-of-three-cases
#8
M Adelita Vizcaino, Shannon S Joseph, Charles G Eberhart
Progressive transformation of germinal centers (PTGC) is a form of follicular hyperplasia recently associated with immunoglobulin G4-related disease (IgG4-RD), but the ophthalmic manifestations of this combination are poorly described. In this retrospective case series, we present three cases of IgG4-related orbital disease (IgG4-ROD) showing varying degrees of PTGC involving the orbit and lacrimal gland. Three adult women presented with ill-defined lacrimal gland enlargement. Histologic sections showed variable fibrosis and large, irregular lymphoid follicles with prominent mantle zones penetrating the germinal centers, highlighted by Bcl-2 and/or IgD immunostains...
January 2018: Saudi Journal of Ophthalmology: Official Journal of the Saudi Ophthalmological Society
https://www.readbyqxmd.com/read/29754327/the-many-faces-of-igg4-related-disease-report-of-a-case-with-inaugural-recurrent-aortic-aneurism-ruptures-and-literature-review
#9
REVIEW
Mariana Luís, Luísa Brites, Bruno Fernandes, Diogo Jesus, Tânia Santiago, Sara Serra, João Rovisco, Lina Carvalho, José António P da Silva, Armando Malcata
Vascular involvement in IgG4-related disease (IgG4-RD), is a well-recognized feature and large vessel commitment, especially the aorta, can be the only manifestation of the disease. Being a newly recognized disease, its diagnosis and workup still represents a challenge in clinical practice. A 47-year-old-man with two aortic aneurysms ruptures, one at abdominal and the other at thoracic level, was referred to our rheumatology department. The initial analysis of the surgical specimen obtained 3 years earlier revealed a nonspecific aortitis...
May 12, 2018: Rheumatology International
https://www.readbyqxmd.com/read/29742724/a-case-report-of-immunoglobulin-g4-related-sclerosing-cholangitis-with-multiple-relapse
#10
Xiaoqin Dong, Na Huo, Zhao Wu, Guiqiang Wang, He Wang, Hong Zhao
RATIONALE: Immunoglobulin G4-related sclerosing cholangitis (IgG4-SC) is classified as a biliary tract manifestation of immunoglobulin G4-related disease (IgG4-RD). Glucocorticoid is the first-line therapy for most patients, but the optimal starting dose, adequate maintaining dose and withdrawal time remain disputable. PATIENT CONCERNS: An elderly male patient presented to our hospital with neoplasms of the bile duct and pancreas at first visit in December 2011...
May 2018: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29741204/striatal-hypermetabolism-in-a-case-of-igg4-related-disease
#11
Abdelhamid Biyi, Omar Ait Sahel, Abderrahim Mejjad, Yasser Benameur, Salah Nabih Oueriagli, Abderrahim Doudouh
A 77 years-old man with lung and bone involvement of proven IgG4-related disease complained of tree month's memory deficits. Brain MRI was normal. 18F-FDG whole body PET/CT showed in addition to the bone lesions, a marked symmetrical striatal hypermetabolism in contrast with cortical hypometabolism. Despite steroid treatment, the patient's clinical status declined rapidly and he died two months later. Striatal hypermetabolism has been reported in autoimmune limbic encephalitis, but to the best of our knowledge, this is the first description in a case of IgG4-related disease...
2018: Nuclear Medicine Review. Central & Eastern Europe
https://www.readbyqxmd.com/read/29738364/a-newly-recognized-histologic-pattern-of-igg4-related-lymphadenopathy-expanding-the-morphologic-spectrum
#12
Ying-Ren Chen, Yi-Ju Chen, Ming-Chung Wang, L Jeffrey Medeiros, Kung-Chao Chang
Immunoglobulin (Ig)G4-related sclerosing disease is a fibroinflammatory disorder characterized by tumor-forming lesions at multiple anatomic sites and by increased serum levels of IgG4. IgG4-related lymphadenopathy, defined as lymphadenopathy developing in patients with IgG4-related sclerosing disease, is known to manifest in 5 histologic patterns: (1) multicentric Castleman disease-like; (2) reactive follicular hyperplasia; (3) interfollicular plasmacytosis with immunoblasts; (4) progressive transformation of germinal centers-like; and (5) inflammatory pseudotumor-like...
May 4, 2018: American Journal of Surgical Pathology
https://www.readbyqxmd.com/read/29737040/marked-accumulation-of-fdg-and-inflammatory-cells-expressing-glucose-transporter-3-in-igg4-related-autoimmune-hepatitis
#13
Toshihiro Araki, Teruko Arinaga-Hino, Hironori Koga, Jun Akiba, Tatsuya Ide, Yoshinobu Okabe, Reiichiro Kuwahara, Keisuke Amano, Makiko Yasumoto, Toshihiro Kawaguchi, Tomoya Sano, Reiichiro Kondou, Seiji Kurata, Keiichi Mitsuyama, Takuji Torimura
Immunoglobulin G (IgG) 4 related-autoimmune hepatitis (AIH) is a recently proposed subtype that responds well to steroid treatment; however, its pathogenesis remains unclear. We report here a 65-year-old Japanese female with skin itching and lip swelling. She had liver injury with jaundice, which persisted despite stopping of anti-allergic agents. Blood chemistry revealed highly elevated serum IgG and IgG4 (535 mg/dL) levels, and positive anti-nuclear antibody. The diagnosis of AIH was based on liver biopsy...
May 7, 2018: Hepatology Research: the Official Journal of the Japan Society of Hepatology
https://www.readbyqxmd.com/read/29736618/leptomeningeal-form-of-immunoglobulin-g4-related-hypertrophic-meningitis-with-perivascular-spread-a-case-report-and-review-of-the-literature
#14
Jasmina Boban, Selin Ardalı, Majda M Thurnher
PURPOSE: Immunoglobulin G4 (IgG4)-related disease represents a spectrum of fibro-inflammatory disorders that affects various organ systems, including the central nervous system. METHODS: Here we present the case of lgG4-related hypertrophic meningitis with exclusively leptomeningeal involvement and spread via perivascular spaces. RESULTS: A 58-year-old male patient presented with complex partial seizures. Initial computed tomography examination showed left frontal sulcal hyperdensity...
May 7, 2018: Neuroradiology
https://www.readbyqxmd.com/read/29736428/early-treatment-for-igg4-related-disease-may-prevent-cognitive-impairment-caused-by-cerebral-vasculitis-a-case-report-and-review-of-the-literature
#15
Toshihiko Usami, Shoji Kawashima, Yoshino Ueki, Takanari Toyoda, Kenji Okita, Noriyuki Matsukawa
IgG4-related disease (IgG4-RD) is a recently recognized disease entity. A 74-year-old male presented with transient headache. He was diagnosed IgG4-RD by pancreatic biopsy at the age of 72. Magnetic Resonance Imaging (MRI) showed disseminated cerebral microbleeds and microinfarctions in time and space. It suggested cerebral vasculitis, however any causative factor were not confirmed. IgG4-RD rarely causes cerebral vasculitis. This might be a first case of an asymptomatic cerebral vasculitis due to IgG4-RD. Patient was started on oral prednisolone, and no neurological or neuropsychological symptom was clinically observed...
March 2018: ENeurologicalSci
https://www.readbyqxmd.com/read/29736303/hypothyroidism-in-patients-with-autoimmune-pancreatitis
#16
Ryoko Shimizuguchi, Terumi Kamisawa, Yuka Endo, Masataka Kikuyama, Sawako Kuruma, Kazuro Chiba, Taku Tabata, Satomi Koizumi
AIM: To examine thyroid function and clinical features of hypothyroidism in autoimmune pancreatitis (AIP) patients. METHODS: We examined thyroid function in 77 patients with type 1 AIP (50 males, 27 females; median age 68 years, range 33-85) diagnosed according to the Japanese diagnostic criteria for AIP 2011. We compared clinical and serological findings between patients with and without various categories of hypothyroidism. The change in hypothyroidism after steroid therapy was also examined...
May 6, 2018: World Journal of Gastrointestinal Pharmacology and Therapeutics
https://www.readbyqxmd.com/read/29735503/immunoglobulin-g4-related-disease-of-the-paranasal-sinuses
#17
Anthony Bashyam, Sidhartha Nagala, Fawzia Tahir, Showkat Mirza
Immunoglobulin G4-related disease (IgG4-RD) is becoming increasingly documented. It was first described in relation to autoimmune pancreatitis. Features of the disease include tissue infiltration by IgG4 plasma cells with associated fibrosis and the growth of pseudotumours. A 71-year-old woman presented with increasing right cheek swelling and mild proptosis. Ten years earlier, she had a similar presentation and was diagnosed with an inflammatory pseudotumour. Examination revealed a lesion in the right nasal cavity...
May 7, 2018: BMJ Case Reports
https://www.readbyqxmd.com/read/29732477/effectiveness-of-rituximab-containing-treatment-regimens-in-idiopathic-multicentric-castleman-disease
#18
Yujun Dong, Lu Zhang, Lin Nong, Lihong Wang, Zeyin Liang, Daobin Zhou, David C Fajgenbaum, Hanyun Ren, Jian Li
Human herpes virus type 8 (HHV-8)-negative, idiopathic multicentric Castleman disease (iMCD) is a rare lymphoproliferative disease often involving constitutional symptoms, cytopenias, and multiple organ system dysfunction. In China, the majority of MCD cases are HHV-8 negative. Given that siltuximab, the only FDA-approved treatment for iMCD is not available in China; rituximab- and cyclophosphamide-containing regimens are often used in the treatment of Chinese iMCD patients. To evaluate the efficacy of rituximab in this rare and heterogeneous disease, clinical and pathological data from 27 cases of iMCD were retrospectively analyzed from two large medical centers in China...
May 7, 2018: Annals of Hematology
https://www.readbyqxmd.com/read/29709946/a-refractory-case-of-secondary-membranous-nephropathy-concurrent-with-igg4-related-tubulointerstitial-nephritis
#19
Hiroyuki Arai, Naohiro Toda, Ryo Kamimatsuse, Keisuke Nishioka, Shuichiro Endo, Shinichi Akiyama, Shoichi Maruyama, Takeshi Matsubara, Hideki Yokoi, Motoko Yanagita
A 58-year-old man with type 1 autoimmune pancreatitis was referred to nephrologists for severe proteinuria. Laboratory data revealed a high serum IgG4 level, hypoalbuminemia, and massive proteinuria, which were compatible with nephrotic syndrome. The renal pathological findings confirmed the diagnosis of secondary membranous nephropathy concurrent with IgG4-related tubulointerstitial nephritis. Despite the improvement of interstitial markers, the proteinuria was refractory to prednisolone, requiring cyclosporine to achieve complete remission...
April 27, 2018: Internal Medicine
https://www.readbyqxmd.com/read/29707337/an-initial-exploration-for-comprehensive-assessment-of-igg4-related-lung-disease-analyses-on-the-cases-enrolled-from-a-systematic-review
#20
An Wang, Jie Fan, Xiaofeng Chen, Shaohua Wang
Background: The existence of two diagnostic systems, the Boston and Japan criteria, for immunoglobulin G4-related disease (IgG4-RD) confuse the medical practice. We aimed to develop a comprehensive assessment based on the weight of each diagnostic item in the existing criteria to improve the diagnostic efficiency of Boston criteria. Methods: We assessed the patients enrolled by a systematic review of the literatures using the Boston criteria, Japan criteria and a tentative comprehensive assessment respectively, and evaluated the efficiency of each system and their consistency...
March 2018: Journal of Thoracic Disease
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