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IgG4- related diseases

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https://www.readbyqxmd.com/read/29457382/an-international-multi-specialty-validation-study-of-the-igg4-related-disease-responder-index
#1
Zachary S Wallace, Arezou Khosroshahi, Mollie D Carruthers, Cory A Perugino, Hyon Choi, Corrado Campochiaro, Emma L Culver, Frank Cortazar, Emanuel Dellatorre, Mikael Ebbo, Ana Fernandes, Luca Frulloni, Philip Hart, Omer Karadag, Shigeyuki Kawa, Mitsuhiro Kawano, Myung-Hwan Kim, Marco Lanzillotta, Shoko Matsui, Kazuichi Okazaki, Jay H Ryu, Takako Saeki, Nicolas Schleinitz, Paula Tanasa, Hisanori Umehara, George Webster, Wen Zhang, John H Stone
OBJECTIVE: IgG4-related disease (IgG4-RD) can cause fibro-inflammatory lesions in nearly any organ, leading to organ dysfunction and failure. The IgG4-RD Responder Index (RI) was developed to help investigators assess the efficacy of treatment in a structured manner. We sought to validate the RI in a multi-national investigation. METHODS: The RI guides investigators through assessments of disease activity and damage in 25 domains, incorporating higher weights for disease manifestations that require treatment urgently or that worsen despite treatment...
February 18, 2018: Arthritis Care & Research
https://www.readbyqxmd.com/read/29456677/concurrent-eosinophilia-and-igg4-related-disease-in-a-child-a-case-report-and-review-of-the-literature
#2
Can Chen, Kuang Chen, Xilian Huang, Kaile Wang, Shenxian Qian
The current study presents the case of a 9-year-old Chinese boy who presented with eosinophilia and elevated serum levels of immunoglobulin G4 (IgG4). A bone marrow puncture identified an elevated eosinophil rate of 23% (normal range, <5%), which indicated eosinophilia. However, gene analysis, fluorescent in situ hybridization and other examinations, including bone marrow aspiration, blood routine, auto-antibody tests and parasitic and allergens screening, contradicted a diagnosis of secondary or clonal eosinophilia...
March 2018: Experimental and Therapeutic Medicine
https://www.readbyqxmd.com/read/29452298/treatment-and-outcomes-in-patients-with-igg4-related-disease-using-the-igg4-responder-index
#3
Andreu Fernández-Codina, Blanca Pinilla, Iago Pinal-Fernández, Cristina López, Guadalupe Fraile-Rodríguez, Eva Fonseca-Aizpuru, Iago Carballo, Pilar Brito-Zerón, Carlos Feijóo-Massó, Miguel López-Dupla, Maria Cinta Cid, Fernando Martínez-Valle
BACKGROUND: IgG4-related disease (IgG4-RD) is an autoimmune disease triggering an inflammatory cascade that leads to fibrosis. Outcome measures are limited and treatment options remain underexplored. OBJECTIVES: To assess the variation of the IgG4 responder index (IgG4-RI) in a cohort of IgG4-RD patients and to explore their treatments and outcomes. METHODS: We studied the clinical phenotype, severity of the disease and response to treatment in an ambispective multicenter cohort study including 14 different hospitals in Spain...
February 13, 2018: Joint, Bone, Spine: Revue du Rhumatisme
https://www.readbyqxmd.com/read/29444790/localised-nasopharyngeal-amyloidosis-the-importance-of-postoperative-follow-up
#4
Sanathorn Chowsilpa, Saisawat Chaiyasate, Komson Wannasai, Teerada Daroontum
Localised nasopharyngeal amyloidosis is rare. Findings on physical examination and invasive pattern on CT scan can be misleading as it can resemble nasopharyngeal carcinoma. A 64-year-old man presented with left aural fullness for 6 months. The physical examination showed straw-coloured fluid in the left middle ear and irregular reddish mass at the left side of the nasopharynx. The CT scan showed a lobulated heterogeneous mass at the left side of the nasopharynx involving the left Eustachian tube opening. Pathology report was amyloidosis, thus, surgery was done...
February 14, 2018: BMJ Case Reports
https://www.readbyqxmd.com/read/29439708/possible-relationship-between-fibrosis-of-igg4-related-thymitis-and-the-profibrotic-cytokines-transforming-growth-factor-beta-1-interleukin-1-beta-and-interferon-gamma-a-case-report
#5
Atsuko Masunaga, Fumihiro Ishibashi, Eitetsu Koh, Takashi Oide, Yasuo Sekine, Kenzo Hiroshima
BACKGROUND: IgG4-related disease often forms a mass and the affected lesion is clinically removed because the mass cannot be differentiated from a neoplasm. Affected lesions commonly occur in the pancreas, hepatobiliary tract, kidney, and retroperitoneum. However, the lesion rarely occurs in the thymus. A histological worldwide consensus of IgG4-related disease proposed that pathological diagnosis of IgG4-related disease should meet more than two of three major features: 1) dense lymphoplasmacytic infiltration with greater than 40% IgG4+/IgG+ plasma cells, 2) storiform fibrosis; and 3) obliterative phlebitis...
January 17, 2018: Diagnostic Pathology
https://www.readbyqxmd.com/read/29437186/evaluation-of-igg4-plasma-cell-infiltration-in-patients-with-systemic-plasmacytosis-and-other-plasma-cell-infiltrating-skin-diseases
#6
Shintaro Takeoka, Masahiro Kamata, Carren Sy Hau, Mihoko Tateishi, Saki Fukaya, Kotaro Hayashi, Atsuko Fukuyasu, Takamitsu Tanaka, Takeko Ishikawa, Takamitsu Ohnishi, Yuko Sasajima, Shinichi Watanabe, Yayoi Tada
Systemic plasmacytosis is a rare skin disorder characterized by marked infiltration of plasma cells in the dermis. IgG4-related disease is pathologically characterized by lymphoplasmacytic infiltration rich in IgG4+ plasma cells, storiform fibrosis, and obliterative phlebitis, accompanied by elevated levels of serum IgG4. Reports of cases of systemic plasmacytosis with abundant infiltration of IgG4+ plasma cells has led to discussion about the relationship between systemic plasmacytosis and IgG4-related disease...
February 13, 2018: Acta Dermato-venereologica
https://www.readbyqxmd.com/read/29434730/clinical-and-pathological-characteristics-of-igg4-related-interstitial-lung-disease
#7
Xiaoting Lv, Feng Gao, Qicai Liu, Sheng Zhang, Zhihua Huang, Yongping Zhu, Haiyang Zong, Quwen Li, Sanyan Li
IgG4-related interstitial lung disease (IgG4-RILD), which is characterized by increased IgG4 levels, IgG4 + plasma cell infiltration and irregular whorled fibrosis, is a recently described lung disorder that belongs to the group of systemic fibroinflammatory IgG4-related diseases (IgG4-RD). The aim of the present study was to improve the current knowledge regarding the specific clinical and histopathological characteristics of IgG4-RILD and to investigate its underlying immune mechanism in vivo . A total of 7 patients newly diagnosed with IgG4-RILD were enrolled in the present study (4 men and 3 women; mean age, 57 years; range, 29-71 years)...
February 2018: Experimental and Therapeutic Medicine
https://www.readbyqxmd.com/read/29434340/glucocorticoid-receptor-expression-in-resident-and-hematopoietic-cells-in-igg4-related-disease
#8
Taku Iguchi, Koji Takaori, Akiko Mii, Yuki Sato, Yasunori Suzuki, Hajime Yoshifuji, Hiroshi Seno, Osamu Ogawa, Koichi Omori, Kazuhisa Bessho, Satoru Kondo, Tomokazu Yoshizaki, Hitoshi Nakashima, Takao Saito, Tsuneyo Mimori, Hironori Haga, Mitsuhiro Kawano, Motoko Yanagita
Immunoglobulin G4-related disease is a rare immune-mediated disease characterized by the infiltration of IgG4-positive plasma cells and unique storiform fibrosis of multiple organs. The majority of IgG4-related disease patients respond to glucocorticoids, yet the precise mechanism of their action remains unclear. Pathological sections of the submaxillary gland, kidney, and retroperitoneum from 20 patients in total diagnosed with IgG4-related disease were analyzed for glucocorticoid receptor expression and the cell types expressing glucocorticoid receptor...
February 12, 2018: Modern Pathology: An Official Journal of the United States and Canadian Academy of Pathology, Inc
https://www.readbyqxmd.com/read/29434076/differences-in-gut-microbiota-profiles-between-autoimmune-pancreatitis-and-chronic-pancreatitis
#9
Shin Hamada, Atsushi Masamune, Tatsuhide Nabeshima, Tooru Shimosegawa
Host-derived factors alter gut microenvironment, and changes in gut microbiota also affect biological functions of host. Alterations of gut microbiota have been reported in a wide variety of diseases, but the whole picture of alterations in pancreatic diseases remains to be clarified. In particular, the gut microbiota may be affected by malnutrition or impaired exocrine pancreas function that is associated with pancreatic diseases. We here conducted comprehensive analysis of gut microbiota in patients with type 1 autoimmune pancreatitis (AIP), a pancreatic manifestation of the systemic IgG4-related disease, and chronic pancreatitis (CP)...
2018: Tohoku Journal of Experimental Medicine
https://www.readbyqxmd.com/read/29429186/-igg4-related-sclerosing-disease-with-initial-ear-symptom-a-case-report
#10
M Hu, W Wang, Y Cheng
No abstract text is available yet for this article.
February 7, 2018: Zhonghua Er Bi Yan Hou Tou Jing Wai Ke za Zhi, Chinese Journal of Otorhinolaryngology Head and Neck Surgery
https://www.readbyqxmd.com/read/29420193/clinicoserological-factors-associated-with-response-to-steroid-treatment-and-recurrence-in-patients-with-igg4-related-ophthalmic-disease
#11
Ji Wook Hong, Sunah Kang, Min Kyung Song, Chan Joo Ahn, Ho-Seok Sa
PURPOSE: To investigate the factors associated with response to steroid treatment and recurrence in patients with IgG4-related ophthalmic disease (ROD). METHODS: Twenty-eight patients with biopsy-proven IgG4-ROD treated between March 2010 and January 2017 were included in this retrospective study. Clinical features, serum IgG4 levels, systemic involvement, treatments and treatment outcome, factors associated with response to treatment and recurrence were assessed...
February 2, 2018: British Journal of Ophthalmology
https://www.readbyqxmd.com/read/29417330/igg4-related-disease-mimicking-crohn-s-disease-a-case-report-and-review-of-literature
#12
Fabiana Ciccone, Antonio Ciccone, Mirko Di Ruscio, Filippo Vernia, Gianluca Cipolloni, Gino Coletti, Giuseppe Calvisi, Giuseppe Frieri, Giovanni Latella
No abstract text is available yet for this article.
February 8, 2018: Digestive Diseases and Sciences
https://www.readbyqxmd.com/read/29416818/clinical-features-and-prognostic-factors-of-ten-patients-with-renal-failure-caused-by-igg4-related-retroperitoneal-fibrosis
#13
Wei Zhang, Feng Xue, Cui Wang, Leping Shao
IgG4-related retroperitoneal fibrosis (IgG4-RPF) is a newly recognized entity which often mimics cancer. We aimed to investigate the clinical features and the causes of renal failure, as well as to explore the factors affecting the prognosis of renal function by analysis of the clinical data of patients with IgG4-RPF. We reviewed clinical features of 10 patients with renal failure caused by IgG4-RPF, which was confirmed by pathology review and clinic-pathologic correlations. All patients were male, and the mean age at onset was 64...
January 5, 2018: Oncotarget
https://www.readbyqxmd.com/read/29415331/igg4-related-sclerosing-oesophagitis-in-a-9-year-old-girl
#14
Lucie Bienfait, Nathalie Nagy, Imad El Moussaoui, Pierre Lingier, Issam El Nakadi, Pieter Demetter, Laurine Verset
Immunoglobulin G4 (IgG4)-related disease is a fibroinflammatory disorder characterised by a histopathological pattern of dense lymphoplasmocytic inflammation, fibrosis, obliterative phlebitis, and demonstration of dominance of IgG4-positive plasmocytes. Recognition of the disease entity is vital due its frequent misdiagnosis as a malignant tumour, which can result in unnecessary treatment measures including surgery. Despite recent insights into the mechanisms behind IgG4-related disease, a review of the literature yields only 13 (adult) cases of IgG4-related disease occuring within the oesophagus...
February 7, 2018: Histopathology
https://www.readbyqxmd.com/read/29413316/igg4-related-disease-of-the-central-and-peripheral-nervous-systems
#15
REVIEW
Mahmoud A AbdelRazek, Nagagopal Venna, John H Stone
IgG4-related disease can involve nearly any organ system, including the central and peripheral nervous systems. The pathology findings are consistent from organ to organ, but careful clinicopathological correlation is necessary to establish the diagnosis. Many non-neurological and neurological inflammatory conditions, previously regarded as idiopathic in nature, are now recognised to fall within the spectrum of IgG4-related disease. The condition is highly treatable, but probably remains substantially under-recognised...
February 2018: Lancet Neurology
https://www.readbyqxmd.com/read/29398045/-management-of-orbital-inflammation-in-internal-medicine-proposal-for-a-diagnostic-work-up
#16
S Abad, F Héran, C Terrada, P Bielefeld, D Sène, S Trad, D Saadoun, P Sève
Inflammatory orbitopathies relate to an inflammatory state originating within the orbit and its adnexes, except the inner ocular globe. Orbital inflammation (OI) may be either localized manifestation of a proven or like autoimmune disease, or local response from immune system against infectious, structural or tumoral antigens. We review the clinical manifestations of OI, which provide helpful clues to the diagnosis and describe the inflammatory, infectious and neoplastic conditions classically associated with OI...
February 1, 2018: La Revue de Médecine Interne
https://www.readbyqxmd.com/read/29394821/-a-case-of-an-igg4-related-inflammatory-pseudotumor-of-the-liver-showing-enlargement-that-was-difficult-to-differentiate-from-hepatic-cancer
#17
Kei Adachi, Kazuhiko Hashimoto, Ryoji Nonaka, Jeong-Ho Moon, Yujiro Fujie, Shoichiro Fujita, Keiko Kojima, Jun Hanai, Shingi Imaoka, Tadashi Ohnishi
A 60-year-old man was admitted for a liver mass(S3), which rapidly increased in size during intraductal papillary mucinous neoplasm(IPMN)follow-up. Although EOB-MRIwas performed, the mass could not be accurately diagnosed as hepatic cancer. Thus, we performed a lateral segmentectomy. In the resected specimen, a solid tumor mass was clearly bound in segment 3 of the liver. Since histopathology revealed no malignant cells and many IgG4-positive cells, we confirmed the diagnosis as IgG4-related inflammatory pseudotumor of the liver...
November 2017: Gan to Kagaku Ryoho. Cancer & Chemotherapy
https://www.readbyqxmd.com/read/29391913/the-utility-of-fdg-pet-ct-in-igg4-related-disease-with-a-focus-on-coronary-artery-involvement
#18
Hian L Huang, Warren Fong, Wee M Peh, Kasat A Niraj, Winnie W Lam
Purpose: Our case series aims to study the growing use of FDG PET/CT in diagnostic evaluation and follow up of IgG4-RD with emphasis on patients presenting with coronary artery involvement. Methods: We conducted a search on the nuclear medicine and rheumatology service databases and identified patients with histologically proven IgG4-RD with FDG PET/CT performed at the Singapore General Hospital. The radiological, clinical, and laboratory findings of these patients were analyzed retrospectively...
February 2018: Nuclear Medicine and Molecular Imaging
https://www.readbyqxmd.com/read/29390345/a-case-of-immunoglobulin-g4-related-lung-disease-with-bilateral-diffuse-infiltration-a-case-report
#19
Ailing Liu, Qianwen Zhang, Bo Liu, Ning Xu, Aijun Li
RATIONALE: Immunoglobulin (Ig) G4-related disease (IgG4-RD) is a rare and chronic progressive autoimmune disease. It is a novel clinical entity characterized by elevated serum IgG4 concentration and tissue infiltration by IgG4 plasma cells. IgG4-related lung disease (IgG4-RLD) has been described as interstitial pneumonia and inflammatory pseudotumor, with various abnormal radiographic patterns. We report a case of IgG4-related lung disease with bilateral diffuse infiltration. PATIENT CONCERNS: A 65-year-old woman was admitted to our hospital because of cough, sputum, and fever...
December 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/29388171/combined-membranous-nephropathy-and-tubulointerstitial-nephritis-as-a-rare-renal-manifestation-of-igg4-related-disease-a-case-based-literature-review
#20
Wei Zhang, Jeffrey H Glaze, David Wynne
IgG4-related disease (IgG4-RD) is a newly recognized immune-mediated multisystemic disease characterized by a fibro-inflammatory condition with tissue infiltration of IgG4-positive plasma cells and often associated with elevated serum IgG4 levels. Typical renal involvement of IgG4-RD presents as tubulointerstitial nephritis (TIN), membranous or membranoproliferative nephropathy. We are presenting a case with combined IgG4 membranous nephropathy and TIN, as well as a literature review on pathophysiology, diagnosis and treatment of IgG4-RD...
February 1, 2018: CEN Case Reports
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