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Polyclonal hypergammaglobulinemia

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https://www.readbyqxmd.com/read/30107829/myalgia-caused-by-chronic-myositis-associated-with-plasmacytosis-a-case-report
#1
Taku Hatano, Masashi Takanashi, Hitoshi Tsuchihashi, Shin-Ichi Ueno, Arisa Hayashida, Yutaka Tsukune, Kazuaki Kanai, Yasushi Shimo, Nobutaka Hattori
BACKGROUND: Cutaneous and systemic plasmacytosis are skin disorders characterized by cutaneous polyclonal plasma cell infiltration accompanied by polyclonal hypergammaglobulinemia. Cutaneous plasmacytosis involvement is limited to the skin, mainly on the face and trunk, while systemic plasmacytosis also involves 2 or more organ systems. However, there have been no reports of inflammatory myositis due to plasmacytosis. Here, we report a patient with plasmacytosis who developed myalgia and easy fatigability due to inflammatory myositis...
August 14, 2018: BMC Neurology
https://www.readbyqxmd.com/read/30073990/cutaneous-plasmacytosis-with-mast-cell-infiltration
#2
Sarina Jain, Rohit V Hede, Uday S Khopkar
Cutaneous plasmacytosis is a rare disorder of uncertain etiology, described mainly in patients of Japanese descent. Clinically, it is characterized by multiple pigmented papules and plaques distributed primarily on the trunk. Histopathologically, it is marked by a dense dermal plasma cell infiltrate. Here, we describe a case of cutaneous plasmacytosis in a 55-year-old Indian male who presented with hyperpigmented plaques on the body. Histopathological examination revealed dense superficial and deep perivascular and periappendageal infiltrate composed mainly of plasma cells, lymphoid follicles with reactive germinal centres, perineural distribution of plasma cells, mast cell infiltration and increased dermal small blood vessels...
August 3, 2018: Indian Journal of Dermatology, Venereology and Leprology
https://www.readbyqxmd.com/read/29902238/laty136f-knock-in-mouse-model-for-human-igg4-related-disease
#3
Kazunori Yamada, Masahiko Zuka, Kiyoaki Ito, Keishi Mizuguchi, Yasushi Kakuchi, Tamehito Onoe, Yasunori Suzuki, Masakazu Yamagishi, Shozo Izui, Marie Malissen, Bernard Malissen, Mitsuhiro Kawano
BACKGROUND: The adaptor protein Linker for activation of T cell (LAT) is a key signaling hub used by the T cell antigen receptor. Mutant mice expressing loss-of-function mutations affecting LAT and including a mutation in which tyrosine 136 is replaced by a phenylalanine (LatY136F) develop lymphoproliferative disorder involving T helper type 2 effector cells capable of triggering a massive polyclonal B cell activation that leads to hypergammaglobulinemia G1 and E and to non-resolving inflammation and autoimmunity...
2018: PloS One
https://www.readbyqxmd.com/read/29675946/plasma-proteomics-identifies-a-chemokine-storm-in-idiopathic-multicentric-castleman-disease
#4
Sheila K Pierson, Aaron J Stonestrom, Dustin Shilling, Jason Ruth, Christopher S Nabel, Amrit Singh, Yue Ren, Katie Stone, Hongzhe Li, Frits van Rhee, David C Fajgenbaum
Human Herpesvirus-8 (HHV-8)-negative/idiopathic multicentric Castleman disease (iMCD) is a poorly understood disease involving polyclonal lymphoproliferation with dysmorphic germinal centers, constitutional symptoms, and multi-organ failure. Patients can experience thrombocytopenia, anasarca, reticulin fibrosis, renal dysfunction, organomegaly, and normal immunoglobulin levels, - iMCD-TAFRO. Others experience thrombocytosis, milder effusions, and hypergammaglobulinemia, -iMCD-Not Otherwise Specified (iMCD-NOS)...
July 2018: American Journal of Hematology
https://www.readbyqxmd.com/read/29532268/monoclonal-gammopathy-in-rheumatic-diseases
#5
Yue Yang, Long Chen, Yuan Jia, Yang Liu, Lei Wen, Yaoxian Liang, Yuan An, Shi Chen, Yin Su, Zhanguo Li
To analyze the clinical spectrum, laboratory characteristics, and outcomes of monoclonal gammopathy (MG) in patients with rheumatic diseases. Screening for the presence of MG was performed in 872 inpatients with rheumatic diseases from January 2010 to July 2017. A total of 41 patients were enrolled. Their clinical and biological features in addition to outcomes were described. For each patient with primary Sjögren syndrome (pSS), 2 age- and sex-matched pSS patients without MG were selected as controls. Risk factors for the presence of MG and malignant hematological neoplasias were assessed...
July 2018: Clinical Rheumatology
https://www.readbyqxmd.com/read/29289265/utility-of-serum-igg4-levels-in-a-multiethnic-population
#6
Ruyu Qi, Luke Y C Chen, Sujin Park, Robert Irvine, Michael A Seidman, John T Kelsall, David Collins, Vivian Yin, Graham W Slack, Andre Mattman, Eric Lam, Mollie N Carruthers
BACKGROUND: IgG4-related disease (IgG4-RD) is a recently recognized condition defined by characteristic histopathologic findings in affected organs. Serum IgG4 concentration is often but not always elevated. The sensitivity and specificity of serum IgG4 vary greatly across studies and has been anecdotally associated to ethnicity. Our study was conducted to investigate the difference in serum IgG4 levels between Asian and non-Asian patients with IgG4-RD. METHODS: This is a single-center retrospective study of 26 Asian and 10 non-Asian patients with histologically confirmed IgG4-RD...
January 2018: American Journal of the Medical Sciences
https://www.readbyqxmd.com/read/29186262/solitary-ulcerated-plaque-on-the-face-an-unusual-presentation-of-cutaneous-plasmacytosis
#7
Ana Marta António, João Vitor Alves, Ricardo Coelho, Elvira Bártolo
Cutaneous and systemic plasmacytosis is a rare disorder characterized by cutaneous polyclonal plasma cell infiltration frequently associated with polyclonal hypergammaglobulinemia and lymphadenopathy. We report a case of a 67-year-old woman with an inflammatory ulcerated plaque in the left masseter region. A skin biopsy showed dense perivascular infiltrate of mature plasma cells in the dermis without atypia and immunoglobulin light chain restriction. After physical examination and further investigation, we ruled out systemic disease...
May 2017: Anais Brasileiros de Dermatologia
https://www.readbyqxmd.com/read/29176985/mucosal-iga-responses-damaged-in-established-hiv-infection-yet-effective-weapon-against-hiv-transmission
#8
REVIEW
Viraj Kulkarni, Ruth M Ruprecht
HIV infection not only destroys CD4+ T cells but also inflicts serious damage to the B-cell compartment, such as lymphadenopathy, destruction of normal B-cell follicle architecture, polyclonal hypergammaglobulinemia, increased apoptosis of B cells, and irreversible loss of memory B-cell responses with advanced HIV disease. Subepithelial B cells and plasma cells are also affected, which results in loss of mucosal IgG and IgA antibodies. This leaves the mucosal barrier vulnerable to bacterial translocation. The ensuing immune activation in mucosal tissues adds fuel to the fire of local HIV replication...
2017: Frontiers in Immunology
https://www.readbyqxmd.com/read/29089182/the-deadly-dance-of-b-cells-with-trypanosomatids
#9
REVIEW
Sasha Silva-Barrios, Tania Charpentier, Simona Stäger
B cells are notorious actors for the host's protection against several infectious diseases. So much so that early vaccinology seated its principles upon their long-term protective antibody secretion capabilities. Indeed, there are many examples of acute infectious diseases that are combated by functional humoral responses. However, some chronic infectious diseases actively induce immune deregulations that often lead to defective, if not deleterious, humoral immune responses. In this review we summarize how Leishmania and Trypanosoma spp...
February 2018: Trends in Parasitology
https://www.readbyqxmd.com/read/28974340/a-possible-new-morphological-variant-of-mantle-cell-lymphoma-with-plasma-cell-type-castleman-disease-like-features
#10
Takuro Igawa, Rika Omote, Hiaki Sato, Kohei Taniguchi, Katsuya Miyatani, Tadashi Yoshino, Yasuharu Sato
Mantle cell lymphoma (MCL) is an aggressive B-cell lymphoma characterized by overexpression of cyclin D1 resulting from t(11;14)(q13;q32) translocation. Herein, we report 3 cases of MCL with features of plasma cell-type Castleman disease (CD). The 3 patients were all men, ranging from 51 to 74 years in age, and they all presented with systemic lymphadenopathy with anemia, hypoalbuminemia, elevated serum levels of C-reactive protein, and polyclonal hypergammaglobulinemia. Lymph node biopsy specimens of the 3 cases showed histological features of plasma cell-type CD, including atrophic germinal centers and interfollicular plasmacytosis, with no light chain restriction...
November 2017: Pathology, Research and Practice
https://www.readbyqxmd.com/read/28611626/angioimmunoblastic-t-cell-lymphoma-mimicking-drug-reaction-with-eosinophilia-and-systemic-symptoms-dress-syndrome
#11
Joanna Mangana, Emmanuella Guenova, Katrin Kerl, Mirjana Urosevic-Maiwald, Valerie C Amann, Cornelia Bayard, Reinhard Dummer, Lars E French
Angioimmunoblastic T-cell lymphoma (AITCL) is a rare, aggressive lymphoma which derives from follicular helper T cells, commonly affecting the elderly population. It accounts for 2% of all non-Hodgkin lymphomas, with a reported 5-year overall survival rate of less than 30%. Very often, the clinical picture of AITCL encompasses systemic symptoms such as generalized lymphadenopathy, hepatosplenomegaly, skin rash, anemia, and polyclonal hypergammaglobulinemia. Here we report on the case of a female patient who presented with clinical features resembling drug reaction with eosinophilia and systemic symptoms (DRESS syndrome) prior to the definitive diagnosis of AITCL...
January 2017: Case Reports in Dermatology
https://www.readbyqxmd.com/read/28458319/overlap-of-igg4-related-disease-and-multicentric-castleman-s-disease-in-a-patient-with-skin-lesions
#12
Hiroki Mochizuki, Manako Kato, Takakazu Higuchi, Ryosuke Koyamada, Satoru Arai, Sadamu Okada, Hikaru Eto
A 59-year-old man presented with multiple dark red erythemas with induration, anemia, and polyclonal hypergammaglobulinemia. A skin biopsy revealed the infiltration of lymphocytes and plasma cells and he was initially diagnosed with multicentric Castleman's disease (MCD). Glucocorticoid treatment was only partially effective. Four years later, the patient's bilateral lacrimal glands gradually became enlarged and a biopsy revealed dense lymphocyte and plasma cell infiltration with an IgG4+/IgG+ plasma cell ratio of 70%...
2017: Internal Medicine
https://www.readbyqxmd.com/read/27884455/-acid-sphingomyelinase-deficiency-niemann-pick-disease-type-b-in-adulthood-a-retrospective-multicentric-study-of-28-adult-cases
#13
MULTICENTER STUDY
O Lidove, N Belmatoug, R Froissart, C Lavigne, I Durieu, K Mazodier, C Serratrice, C Douillard, C Goizet, P Cathebras, G Besson, Z Amoura, A Tazi, M Gatfossé, S Rivière, T Sené, M T Vanier, J-M Ziza
INTRODUCTION: Acid sphingomyelinase deficiency (ASMD) is an autosomal recessive disease with a clinical spectrum ranging from a neurovisceral infantile form (Niemann-Pick disease type A) to a chronic visceral form also encountered in adults (Niemann-Pick disease type B, NP-B). METHODS: Retrospective multicentric analysis of French adult patients with ASMD over the period 1985-March 2015. Clinical, biological, and imaging data were analyzed. RESULTS: Twenty-eight patients (19 males, 9 females) were analyzed...
May 2017: La Revue de Médecine Interne
https://www.readbyqxmd.com/read/27840753/mercaptopurine-treatment-in-an-adult-man-with-orbital-and-intracranial-rosai-dorfman-disease
#14
Valentina Arnao, Marianna Riolo, Giovanni Savettieri, Paolo Aridon
Background. Rosai-Dorfmann disease (RDD) is a rare, idiopathic non-Langerhans cell histiocytosis, affecting children and young adults, that commonly presents as painless, massive cervical lymphadenopathy with fever, weight loss, and polyclonal hypergammaglobulinemia. Cervical lymphadenopathy and extranodal involvement are the main presentations. On the contrary, ophthalmic involvement and localisation in the central nervous system are rare. Case Report. An old man was admitted to our hospital for first seizure...
2016: Case Reports in Neurological Medicine
https://www.readbyqxmd.com/read/27824644/rapid-fatal-acute-peripheral-t-cell-lymphoma-associated-with-igg-plasma-cell-leukemia-and-iga-hypergammaglobulinemia
#15
Nives Jonjić, Irena Seili Bekafigo, Dora Fučkar Čupić, Ksenija Lučin, Antica Duletić Načinović, Toni Valković
Simultaneous occurrence of T-cell and B-cell neoplasms is rare, and etiologic relationships between these 2 malignancies are poorly understood. We describe the case of a 66-year-old woman who was admitted to the hospital because of fever, hemoptysis, lymphadenopathy, and skin rash. Enlarged lymph nodes in axillary, pectoral, paratracheal, and periportal regions as well as slight hepatomegaly and splenomegaly were confirmed. A peripheral blood smear revealed rouleaux formation and numerous circulating plasma cells, with plasmacytoid lymphocytes...
November 2016: Applied Immunohistochemistry & Molecular Morphology: AIMM
https://www.readbyqxmd.com/read/27790959/igg4-related-syndrome-another-multiorgan-disease-in-the-interest-field-of-internal-medicine
#16
REVIEW
Antonino Tuttolomondo, Irene Simonetta, Domenico Di Raimondo, Tiziana Di Chiara, Roberta Conigliaro, Francesca Corpora, Valerio Vassallo, Antonio Pinto
BACKGROUND: IgG4-related disease is a rare, clinical and pathologic disease entity of unknown etiology. Its main features are increased serum concentrations of IgG4 > 1,35 g/l, lymphocyte and IgG4+plasma-cell infiltration within tissues, fibrosis or sclerosis. The classical presentation of IgG4-RSD is pancreatitis which is combined with the involvement of biliary ducts in 74 percent of patients. Extrapancreatic manifestations include: abdominal or mediastinal lymphadenopathy; the involvement of salivary glands and lacrimal glands, kidneys, lung, retroperitoneum...
2016: Current Pharmaceutical Design
https://www.readbyqxmd.com/read/27364300/a-case-of-lethal-idiopathic-plasmacytic-lymphadenopathy-with-polyclonal-hypergammaglobulinemia-a-medical-challenge-for-the-forensic-pathologist
#17
Lucia Tattoli, Biagio Solarino, Oronzo Schiraldi, Giancarlo Di Vella
A rare case of lethal idiopathic plasmacytic lymphadenopathy (IPL) with polyclonal hyperimmunoglobulinemia with chronic renal failure is described. A 40-year-old woman who had suffered from upper airways disease was admitted to the Emergency Room with acute renal failure and hypergammaglobulinemia. She developed pericardial effusion, a pruritic rash, splenomegaly and fell into a coma after 6 days. Multiple myeloma, infection, collagenopathy, and coagulopathy were ruled out. Finally, a form of malignant hypergammapathy was suspected...
July 2016: Journal of Forensic Sciences
https://www.readbyqxmd.com/read/27243459/disruption-of-splenic-lymphoid-tissue-and-plasmacytosis-in-canine-visceral-leishmaniasis-changes-in-homing-and-survival-of-plasma-cells
#18
Joselli Silva-O'Hare, Isabela Silva de Oliveira, Thaís Klevorn, Valter A Almeida, Geraldo G S Oliveira, Ajax M Atta, Luiz Antonio R de Freitas, Washington L C Dos-Santos
Visceral leishmaniasis (VL) is a disease caused by Leishmania infantum, which is transmitted by phlebotomine sandflies. Dogs are the main urban reservoir of this parasite and the disease presents similar characteristics in both humans and dogs. In this paper, we investigated the potential pathways involved in plasma cell replacement of normal cell populations in the spleen, with respect to disease severity in dogs from an endemic area for visceral leishmaniasis. To this end, canine spleen samples were grouped into three categories: TYPE1SC- (non-infected dogs or without active infection with organized white pulp), TYPE1SC+ (infected dogs with organized white pulp) or TYPE3SC+ (infected animals with disorganized white pulp)...
2016: PloS One
https://www.readbyqxmd.com/read/27160753/a-case-report-of-igg4-related-syndrome-and-literature-review
#19
Antonino Tuttolomondo, Irene Simonetta, Domenico Di Raimondo, Roberta Conigliaro, Francesca Corpora, Valerio Vassallo, Antonio Pinto
BACKGROUND: IgG4-related disease is a rare, clinical and pathologic disease entity of unknown etiology. Its main features are increased serum concentrations of IgG4 > 1,35 g/l, lymphocyte and IgG4+plasma-cell infiltration within tissues, fibrosis or sclerosis. The classical presentation of IgG4-RSD is pancreatitis which is combined with the involvement of biliary ducts in 74 percent of patients. Extrapancreatic manifestations include: abdominal or mediastinal lymphadenopathy; the involvement of salivary glands and lacrimal glands, kidneys, lung, retroperitoneum...
May 10, 2016: Current Pharmaceutical Design
https://www.readbyqxmd.com/read/27057027/primary-cutaneous-plasmacytosis-masquerading-as-hidradenitis-suppurativa
#20
Tarang Goyal, Anupam Varshney, Vijay Zawar, Veena Sharma
Isolated cutaneous plasmacytosis (CP) is a rare entity with few cases reported in world literature. CP masquerading as hidradenitis suppurativa like presentation is a unique case with some features differentiating it clinically from it which were further confirmed by histopathology and immunostaining. Our case showed hyperplasia of mature plasma cells and polyclonal hypergammaglobulinemia, immunostaining for CD138 positivity and kappa: lambda ratio more than 3:1. Extensive clinical and laboratory investigations failed to reveal any underlying pathology, presence of any underlying disease accompanying the hypergammaglobulinemia and/or plasma cell proliferation...
March 2016: Indian Journal of Dermatology
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