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Immunoglobulin G4 related lung disease

Tetsuyuki Okubo, Yumiko Oyamada, Masaya Kawada, Yo Kawarada, Shuji Kitashiro, Shunichi Okushiba
We report a case of immunoglobulin G4 (IgG4)-related lung disease presenting as a solitary pulmonary nodule with an irregular margin on computed tomography. The nodule showed a high standardized uptake value on positron emission tomography. A malignant pulmonary tumour could not be excluded. Middle lobectomy was performed. Histological analysis revealed marked lymphoplasmacytic infiltration and storiform fibrosis. Immunostaining indicated the presence of IgG4-positive plasma cells. A definitive diagnosis of IgG4-related disease was confirmed...
January 2017: Respirology Case Reports
Shigeto Horita, Hiroshi Fujii, Ichiro Mizushima, Yuhei Fujisawa, Satoshi Hara, Kazunori Yamada, Dai Inoue, Kenichi Nakajima, Kenichi Harada, Mitsuhiro Kawano
We describe an 81-year-old man with immunoglobulin G4-related disease (IgG4-RD) presenting with submandibular gland, lymph node, lung, kidney, aortic wall, and prostate lesions with concomitant gastric cancer. After curative surgical treatment of the gastric cancer, corticosteroid therapy for progressively decreasing renal function was started. Before starting steroid therapy, fluorodeoxyglucose positron emission tomography-computed tomography revealed multiple lesions of IgG4-RD but no metastasis of the cancer...
October 27, 2016: Modern Rheumatology
Melanie C Bois, Hiroshi Sekiguchi, Jay H Ryu, Eunhee S Yi
Pulmonary nodular lymphoid hyperplasia (PNLH) may show heavy lymphoplasmacytic infiltrates with increased immunoglobulin G4 (IgG4)-positive plasma cells as in IgG4-related disease (IgG4-RD). However, it is unclear whether PNLH could be a manifestation of IgG4-RD. A recent study reported the presence of Epstein-Barr virus (EBV) in IgG4-related lymphadenopathy. We postulated that a subset of PNLH might represent IgG4-related lung disease with EBV-positive lymphocytes as has been reported in IgG4-related lymphadenopathy...
January 2017: Human Pathology
Jonathan Dillon, Andrea Dart, Tom Sutherland
Immunoglobulin G4-related disease (IgG4-RD) is a fibroinflammatory condition that was first recognised as a systemic disease in 2003, when patients with autoimmune pancreatitis were found to have extrapancreatic manifestations. Since 2003, IgG4-RD has been described in a diverse range of other organs including the biliary tree, orbits, lacrimal glands, salivary glands, lungs, kidneys, aorta, retroperitoneum, lymph nodes, pachymeninges, prostate and pituitary gland. The disease frequently occurs in the absence of pancreatic involvement...
December 2016: Journal of Medical Imaging and Radiation Oncology
Jay H Ryu, Eunhee S Yi
Immunoglobulin G4-related disease (IgG4-RD) is a systemic fibroinflammatory disease with protean manifestations involving virtually any organ in the body. At initial clinical presentation, 1 or multiple organs may be involved. Initial descriptions focused on pancreatic disease. It has, however, become clear that IgG4-RD can cause an immune-mediated fibroinflammatory process, commonly manifesting as mass-like lesions, in various regions of the body including the thorax where any compartment can be involved. This pathologic process is characterized by infiltration of IgG4+ plasma cells and a propensity to fibrosis leading to organ dysfunction which can be prevented by early diagnosis and corticosteroid therapy...
September 2016: Clinics in Chest Medicine
Satoshi Ikeda, Akimasa Sekine, Tomohisa Baba, Koji Okudela, Tae Iwasawa, Fumikazu Sakai, Kenji Notohara, Kenichi Ohashi, Tamiko Takemura, Takashi Ogura
AIMS: There have been few reports on immunoglobulin-G4 (IgG4)-related interstitial pneumonia (IP), and its clinical features remain unclear. The objective of this study was to assess whether IP with marked IgG4-positive plasma cell infiltration without extrathoracic lesions of IgG4-related disease (RD) should be diagnosed as a subtype of IgG4-RD or a separate entity. METHODS AND RESULTS: All consecutive patients with surgical lung biopsy-proven idiopathic IP with an IgG4/IgG-positive cell ratio of >40% and >50 IgG4(+) plasma cells in a high-power field without extrathoracic lesions of IgG4-RD were reviewed retrospectively...
January 2017: Histopathology
Motohisa Yamamoto, Hiroki Takahashi
Immunoglobulin G4-related disease (IgG4-RD) is a new disease entity that was established in this century. It is a systemic and chronic inflammatory disorder. Autoimmune pancreatitis is a representative condition, but there is clearly involvement from other organs, including the kidneys, the retroperitoneal cavity, the lungs, and the thyroid, lacrimal, salivary, pituitary, and prostate glands. To aid in the resolution of the numerous issues related to IgG4-RD, the authors established a registry system of patients in multicenter institutes with IgG4-RD-the SMART registry...
August 2016: Seminars in Liver Disease
Aleksandra Szczawinska-Poplonyk, Irena Wojsyk-Banaszak, Katarzyna Jonczyk-Potoczna, Anna Breborowicz
BACKGROUND: Immunoglobulin G4-related disease (IgG4-RD) is a multiorgan fibroinflammatory condition with lymphoplasmacytic infiltrates containing abundant IgG4-positive plasma cells. The immunopathogenesis of the disease and the potential role of triggering autoantigens or infectious factors have not been clearly defined. Immunoglobulin G4-related lung disease is a new and emerging condition in pediatric patients and to date, there have been only two reports regarding pulmonary manifestation of IgG4-RD in children recently published...
June 8, 2016: Italian Journal of Pediatrics
Clémence Delteil, Anne-Laure Chesnais, Anne-Marie Tasei, Carole Cohen-Pansieri, Nidal Naffaa, Guillaume Granier
We report the case of a 61-year-old man with a pulmonary nodule discovered while staging the metastasis of a squamous cell carcinoma. No primary cancer was found. Histological examination of the resected specimen (lingula) was in favor of IgG4-related inflammatory pseudotumor. Histological criteria were described in 2012, combining a dense lymphoplasmacitic infiltrate rich in IgG4-positive plasma cell, storiform fibrosis and obliterative phlebitis. IgG4/IgG plama cell ratio>40 % is mandatory for histological diagnosis of IgG4-related disease...
June 2016: Annales de Pathologie
Hiroki Tashiro, Koichiro Takahashi, Tomomi Nakamura, Kazutoshi Komiya, Shinya Kimura, Naoko Sueoka-Aragane
BACKGROUND: Immunoglobulin G4-related disease is characterized by infiltration of immunoglobulin G4-positive plasmacytes in various organs. The radiological findings of lung involvement of immunoglobulin G4-related disease include hilar and mediastinal lymphadenopathies, thickness of bronchovascular bundles, peribronchovascular consolidation, and lung nodules. Although a pathological approach is needed to diagnose immunoglobulin G4-related disease, it is ordinarily diagnosed by biopsy from one lesion even if there are multiple lesions...
May 9, 2016: Journal of Medical Case Reports
Gregory D Scott, David A Sauer, Kirsten M Woolf, Mithran S Sukumar, Brandon H Tieu
We describe a patient presenting with bilateral radiologically similar lung lesions initially diagnosed as immunoglobulin (Ig) G4-related disease from biopsy of one lesion, but radiographic changes 6 months later prompted biopsy of the second lesion and showed adenocarcinoma. No case of lung IgG4-related disease and a distant lung malignancy has been previously reported. This is notable because lung IgG4-related disease often manifests in multiple thoracic locations but is diagnosed from a representative biopsy specimen...
May 2016: Annals of Thoracic Surgery
Frank Schneider, Kristen L Veraldi, Marc C Levesque, Thomas V Colby, Eunhee S Yi
We report a case of immunoglobulin(Ig)G4-related disease with the radiologic and histopathological manifestations resembling usual interstitial pneumonia (UIP). The patient was a 62-year-old man who presented with progressive dyspnea of insidious onset. High resolution computed tomography of the chest showed lower-lobe predominant peripheral reticulation and traction bronchiectasis but no honeycomb change. Microscopic examination of the surgical lung biopsy showed characteristic features of UIP including architectural distortion by fibrosis with peripheral and paraseptal accentuation, scattered fibroblast foci and microscopic honeycomb change...
2016: Open Rheumatology Journal
Yasutaka Onishi, Yasuharu Nakahara, Katsuya Hirano, Shin Sasaki, Tetsuji Kawamura, Yoshiro Mochiduki
A 67-year-old man with a history of asbestos exposure and rounded atelectasis complained of cough and swelling in the left submandibular region. Computed tomography showed an increase in size of the right lower lung lobe lesion, which was recognized as the pre-existing rounded atelectasis, as well as swelling of the pancreas and submandibular glands. Biopsy from a submandibular gland and the pulmonary lesion led to a diagnosis of immunoglobulin G4-related disease (IgG4-RD). IgG4-RD is a recently recognized disease that was first reported as an autoimmune disease; however, some reports have indicated another pathogenesis of an allergic nature that is characterized by type 2 helper T cell (Th2) inflammation...
March 2016: Respirology Case Reports
Xuefeng Sun, Hongrui Liu, Ruie Feng, Min Peng, Xiaomeng Hou, Ping Wang, Hanping Wang, Wenbing Xu, Juhong Shi
BACKGROUND: Immunoglobulin G4-related disease (IgG4-RD) is a fibroinflammatory disorder that may involve single or multiple organs. Biopsy-proven lung involvement of this disease is occasionally reported, but not well understood. METHODS: Patients with the diagnosis of biopsy-proven IgG4-related lung disease (IgG4-RLD) from Peking Union Medical College Hospital between January 2011 and July 2015 were retrospectively analyzed. Age, sex, clinical symptoms, laboratory findings, pulmonary function test results, chest CT tests, positron emission tomography (PET) examinations, treatments and prognoses were retrieved from medical records and analyzed...
January 25, 2016: BMC Pulmonary Medicine
Yunyun Fei, Juhong Shi, Wei Lin, Yu Chen, Ruie Feng, Qingjun Wu, Xin Gao, Wenbing Xu, Wen Zhang, Xuan Zhang, Yan Zhao, Xiaofeng Zeng, Fengchun Zhang
To investigate clinical and radiological features of IgG4-related disease (IgG4-RD) patients with intrathoracic involvement. A prospective cohort study was performed and IgG4-RD patients were enrolled from January 2011 to March 2015 in Peking Union Medical College Hospital, in which the clinical and radiological characteristics of IgG4-RD patients with intrathoracic involvement were summarized. Out of total 248 cases with IgG4-RD, 87 cases had intrathoracic lesions, including 58 male cases and 29 female cases, with average age of 54...
December 2015: Medicine (Baltimore)
Anxo Martínez-de-Alegría, Sandra Baleato-González, Roberto García-Figueiras, Anaberta Bermúdez-Naveira, Ihab Abdulkader-Nallib, José A Díaz-Peromingo, Carmen Villalba-Martín
Immunoglobulin G4 (IgG4)-related disease is a relatively recently proposed clinical-pathologic entity that is characterized by fibro-inflammatory lesions rich in IgG4-positive plasma cells and, often but not always, elevated serum IgG4 concentrations. IgG4-related disease was recognized as a systemic disease in 2003, when extrapancreatic manifestations were identified in patients with autoimmune pancreatitis. Since then, the disease has been reported as affecting virtually every organ system and has been identified in the biliary tree, salivary and lacrimal glands, periorbital tissues, lungs, lymph nodes, thyroid gland, kidneys, prostate gland, testicles, breasts, and pituitary gland...
November 2015: Radiographics: a Review Publication of the Radiological Society of North America, Inc
Sho Saeki, Yuko Horio, Susumu Hirosako, Hidenori Ichiyasu, Kazuhiko Fujii, Hirotsugu Kohrogi
Paragonimiasis is a parasitic pleuropulmonary infection caused by eating raw crustaceans and wild boar meat and this infection is endemic in Asia. We herein report two cases of pulmonary P aragonimus westermani infection associated with elevated levels of serum immunoglobulin (Ig) G4 and dense infiltration of IgG4-positive plasma cells in the lung lesions. Treatment with praziquantel resolved the pulmonary lesions and decreased the serum levels of IgG4. IgG4-related disease is a systemic disease occasionally involving the lungs and leads to increased serum levels of IgG4...
September 2015: Respirology Case Reports
Praveen K Jinnur, Eunhee S Yi, Jay H Ryu, Vivek N Iyer
BACKGROUND: Immunoglobulin (Ig) G4-related disease, previously referred to as IgG4-related sclerosing disease or hyper-IgG4 disease, may occur in the lung, involving alveolar parenchyma, airways, and pleura. Various pulmonary manifestations of IgG4-related disease have been reported, but to the best of our knowledge a cavitating lung disease has not been reported previously. CASE REPORT: We describe a 60-year-old man who presented with hemoptysis and cavitating lung disease with clinical, laboratory, and histopathologic findings compatible with IgG4-related disease...
2015: American Journal of Case Reports
Sven Höhne, Monika Milzsch, Johannes Adams, Christian Kunze, Rainer Finke
AIMS AND BACKGROUND: Inflammatory pseudotumor (IPT) and inflammatory myofibroblastic tumor (IMT) represent different entities. However, it is only in recent years that this has been taken into increasing consideration. Some authors still use both terms synonymously or interchangeably. Inflammatory myofibroblastic tumor is a real neoplasm because of the proliferation of myofibroblastic cells. Inflammatory pseudotumor is a more inflammatory reactive or regenerative entity and shows an overlapping with immunoglobulin G4-related disease...
May 2015: Tumori
Azusa Tokue, Tetsuya Higuchi, Yukiko Arisaka, Takahito Nakajima, Hiroyuki Tokue, Yoshito Tsushima
PURPOSE: The aim of the current study was to evaluate the utility of F-18-fluoro-2-deoxy-D-glucose (F-18 FDG) positron emission tomography/computed tomography (PET/CT) in assessing patients diagnosed with immunoglobulin G4 (IgG4)-related disease (IgG4-RD) with inflammation of the head and neck glands. METHODS: We reviewed the records of 17 patients (16 men and 1 woman) with IgG4-RD exhibiting inflammation of the head and neck glands (lacrimal or salivary glands; LSG) who were diagnosed by excisional biopsy of the LSG...
July 2015: Annals of Nuclear Medicine
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