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Autoimmune Neutropenia

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https://www.readbyqxmd.com/read/30079995/expression-of-caspase-1-and-histomorphology-of-lung-after-cladribine-treatment
#1
Marta Lis-Sochocka, Patrycja Chylińska-Wrzos, Ewelina Wawryk-Gawda, Barbara Jodłowska-Jędrych
BACKGROUND: Cladribine is a useful immunosuppressive drug for the treatment of autoimmune diseases, leukemias and multiple sclerosis (MS). Despite the drug having low toxicity, side effects have been reported connected with myelosuppression, neutropenia and severe anemia. OBJECTIVES: The objective of this study was to investigate the influence of cladribine on lung pathomorphology and the expression of caspase 1 using immunohistochemistry method. MATERIAL AND METHODS: The study was conducted on Wistar rats, which were divided into: one control group (C) and one experimental group (E)...
August 6, 2018: Advances in Clinical and Experimental Medicine: Official Organ Wroclaw Medical University
https://www.readbyqxmd.com/read/30078793/-reference-guide-for-adult-chronic-neutropenia
#2
Yasuhisa Yokoyama, Akiyoshi Takami, Yasuo Mori, Shinya Sato, Takahiro Suzuki, Masahiko Gosho, Naoshi Obara, Yasushi Miyazaki, Koichi Akashi, Shigeru Chiba
Chronic neutropenia is a rare hematological entity characterized by isolated neutropenia without anemia or thrombocytopenia. The diagnostic criteria and classification of chronic neutropenia have been more elusive in adult cases than pediatric cases. We herein propose the Reference Guide for Adult Chronic Neutropenia. In this guide, diagnosis of chronic neutropenia is based on recurrent or continuous neutropenia defined by absolute neutrophil count (ANC) less than 1,500/µl for over three or more months. Severity of chronic neutropenia is defined as follows: severe, ANC <500/µl; moderate, ANC between 500 and 1,000/µl; mild, ANC between 1,000 and 1,500/µl...
2018: [Rinshō Ketsueki] the Japanese Journal of Clinical Hematology
https://www.readbyqxmd.com/read/30072561/autoimmune-cytopenias-and-thrombotic-thrombocytopenic-purpura
#3
Charlotte K Brierley, Sue Pavord
The autoimmune cytopenias are a group of disorders resulting primarily from autoantibody-mediated destruction of blood cells, with variable clinical sequelae depending on the severity and lineage affected. Disease presentation ranges from an asymptomatic finding on a routine full blood count to an acutely unwell patient suffering the clinical consequences of severe anaemia, neutropenia or thrombocytopenia. The cytopenia may be primary or secondary to underlying infectious, immune or malignant processes. Thrombotic thrombocytopenic purpura (TTP) is a distinct, rare but potentially life-threatening entity that classically but not invariably presents with a pentad of acute onset haemolytic anaemia, thrombocytopenia, neurological symptoms, renal impairment and fevers...
August 2018: Clinical Medicine: Journal of the Royal College of Physicians of London
https://www.readbyqxmd.com/read/29923432/autoimmune-cytopenias-in-chronic-lymphocytic-leukemia-a-concise-review-and-treatment-recommendations
#4
Tim R De Back, Arnon P Kater, Sanne H Tonino
Chronic lymphocytic leukemia (CLL) is frequently complicated by cytopenias, either due to bone marrow infiltration or autoimmunity, resulting in autoimmune hemolytic anemia (AIHA), immune thrombocytopenia (ITP), pure red cell aplasia (PRCA), or autoimmune neutropenia (AIN). Morbidity due to autoimmune cytopenias (AIC) can be substantial; in addition, infection risk increases and pre-existing infections might deteriorate due to immunosuppressive medication. In the aging population, CLL occurs more frequently and AIC related to CLL represent a growing clinical challenge...
July 14, 2018: Expert Review of Hematology
https://www.readbyqxmd.com/read/29889673/a-case-of-severe-pembrolizumab-induced-neutropenia
#5
Barbacki Ariane, Peter G Maliha, Marie Hudson, David Small
Immune checkpoint inhibitors have revolutionized cancer therapy. Given their mechanism of action, immune-related adverse events have been associated with their use. We present the first documented case of pembrolizumab-induced grade IV neutropenia. A 73-year-old women known for myositis, Crohn's disease, and hypothyroidism and diagnosed with PD-L1 positive stage IV pulmonary adenocarcinoma is treated with Pembrolizumab. She develops grade IV neutropenia 2 weeks after her second infusion. She is therefore hospitalized and treated initially with corticosteroids, granulocyte colony-stimulating factor, and intravenous immunoglobulins...
June 8, 2018: Anti-cancer Drugs
https://www.readbyqxmd.com/read/29739087/human-neutrophil-alloantigen-genotype-frequencies-among-syrian-population
#6
Barbara Hauck-Dlimi, Mohammed Damrah, Susanne Achenbach, Stephan Ott, Robert Zimmermann, Juergen Zingsem, Julian Strobel
BACKGROUND: Human neutrophil antigens (HNA) are able to provoke allo- and autoimmune antibodies which lead to reactions like autoimmune and neonatal neutropenia. However, until now no data about HNA genotype distribution in Syrian population exists. The aim of this study was to determine the HNA allele frequencies in the largest group asking for asylum in Germany since 2015. Allele frequencies were compared to data from German blood donors. Therefore, we calculated the risk of alloimmunization and associated transfusion reactions, as well as the risk of developing neonatal neutropenia for newborns of mixed race couples...
April 1, 2018: Clinical Laboratory
https://www.readbyqxmd.com/read/29721441/indolent-t-cell-large-granular-lymphocytic-leukaemia-in-a-dog-presenting-with-severe-neutropenia-and-an-absence-of-lymphocytosis
#7
James Elliott, Elizabeth Villiers
In humans, large granular lymphocytic leukaemia (LGLL) is a low-grade, indolent lymphoproliferative disorder of large granular lymphocytes (LGL) associated with autoimmune disorders; including rheumatoid arthritis and single or multiple cytopenias; particularly neutropenia. Therapy largely centres around immunosuppression which aims to resolve the immune-mediated secondary pathology, often without eradicating the neoplastic clone. The most effective agents appear to be cyclophosphamide, cyclosporine and methotrexate...
2018: Open Veterinary Journal
https://www.readbyqxmd.com/read/29683950/atypical-autoimmune-hematological-disorders-in-a-patient-with-kabuki-syndrome
#8
Ara C Almécija, Vanesa Pérez, María Baro, Pilar Guerra-García, Jose L Vivanco
Kabuki syndrome is a rare genetic disorder characterized by congenital anomalies and developmental delay. It is often associated with impaired immune response and autoimmune abnormalities. We report the clinical case of a girl with Kabuki syndrome who developed autoimmune neutropenia, not previously reported, followed by hemolytic anemia and autoimmune thrombocytopenia.
April 20, 2018: Journal of Pediatric Hematology/oncology
https://www.readbyqxmd.com/read/29665800/mda-5-associated-rapidly-progressive-interstitial-lung-disease-with-recurrent-pneumothoraces-a-case-report
#9
Safi Alqatari, Peter Riddell, Sinead Harney, Michael Henry, Grainne Murphy
BACKGROUND: Clinically hypomyopathic dermatomyositis is a rare disease that is important to recognize, investigate and treat early as it is associated with poor prognosis. In a proportion of patients, myositis specific antibodies could be negative, but with high clinical suspicion, myositis associated antibodies should be ordered. Anti-MDA-5 antibodies was reported in literature to be associated with severe and rapidly progressive interstitial lung disease, with few case reports of pneumothorax and/or pneumomediastinum...
April 17, 2018: BMC Pulmonary Medicine
https://www.readbyqxmd.com/read/29618690/-neutrophil-recovery-by-eltrombopag-treatment-in-a-patient-with-adult-onset-autoimmune-neutropenia-and-immune-thrombocytopenia
#10
Masahiro Tokunaga, Masahide Yamada, Atsushi Kondo, Mai Mishima, Shinichi Yoshikawa, Shinya Inoue, Takako Morita, Nobuhiko Tominaga
Adult-onset autoimmune neutropenia (AIN) is rarely self-limiting, unlike infant-onset AIN. Although several therapeutic agents have been reported, including corticosteroids, more effective treatment options may exist. Here, we describe neutrophil recovery by eltrombopag in a 52-year-old male AIN patient with immune thrombocytopenia (ITP) who was referred to our hospital with severe neutropenia. Within a year of referral, he developed moderate thrombocytopenia. He was diagnosed with AIN and concurrent ITP, based on the detection of antineutrophil antibodies and bone marrow aspiration, respectively...
2018: [Rinshō Ketsueki] the Japanese Journal of Clinical Hematology
https://www.readbyqxmd.com/read/29613972/chronic-neutropenia-in-children-with-abscess-forming-cervical-lymphadenitis-caused-by-staphylococcus-aureus
#11
Mari Asakura, Toshiyuki Tanaka, Kensuke Shoji, Shuhei Karakawa, Akira Ishiguro, Isao Miyairi
BACKGROUND: Suppurative cervical lymphadenitis is rare in children with limited information about the frequency of neutropenia in any series. METHODS: We conducted a retrospective review of pediatric patients in whom suppurative cervical lymphadenitis was diagnosed between April 2011 and March 2016 at a tertiary children's hospital in Tokyo, Japan. Microbiologically confirmed cases of cervical lymphadenitis with abscess formation were included in the analysis. RESULTS: Twenty-five patients (median age: 1 year old, 64% female) with abscess formation due to Staphylococcus aureus were identified...
April 2, 2018: Pediatric Infectious Disease Journal
https://www.readbyqxmd.com/read/29491289/concurrent-autoimmune-neutropenia-and-idiopathic-thrombocytopenic-purpura-associated-with-igg4-related-diease
#12
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...
July 1, 2018: Internal Medicine
https://www.readbyqxmd.com/read/29475723/umbralisib-a-novel-pi3k%C3%AE-and-casein-kinase-1%C3%AE%C2%B5-inhibitor-in-relapsed-or-refractory-chronic-lymphocytic-leukaemia-and-lymphoma-an-open-label-phase-1-dose-escalation-first-in-human-study
#13
Howard A Burris, Ian W Flinn, Manish R Patel, Timothy S Fenske, Changchun Deng, Danielle M Brander, Martin Gutierrez, James H Essell, John G Kuhn, Hari P Miskin, Peter Sportelli, Michael S Weiss, Swaroop Vakkalanka, Michael R Savona, Owen A O'Connor
BACKGROUND: Umbralisib (TGR-1202) is a novel next-generation inhibitor of phosphatidylinositol 3-kinase (PI3K) isoform p110δ (PI3Kδ), which is structurally distinct from other PI3Kδ inhibitors and shows improved isoform selectivity. Umbralisib also uniquely inhibits casein kinase-1ε, a major regulator of protein translation. The aim of this first-in-human phase 1 study was to establish the safety and preliminary activity profile of umbralisib in patients with haematological malignancies...
April 2018: Lancet Oncology
https://www.readbyqxmd.com/read/29405299/adult-onset-primary-cyclic-autoimmune-neutropenia-a-case-report
#14
Tomohiro Yabushita, Nobuhiro Hiramoto, Yuichiro Ono, Satoshi Yoshioka, Shuhei Karakawa, Masao Kobayashi, Takayuki Ishikawa
BACKGROUND: A few cases of primary autoimmune neutropenia (AIN) have been reported in adults, but cyclic primary AIN, which is characterized by the periodic oscillation of neutrophils, is uncommon in adults. STUDY DESIGN AND METHODS: Herein, we report a 70-year-old man referred to our hospital with severe neutropenia and thrombocytopenia. He had experienced intermittent episodes of low-extremity purpura for the past 3 months, with cellulitis on the skin of the scalp 1 month previously...
April 2018: Transfusion
https://www.readbyqxmd.com/read/29333079/type-1-autoimmune-hepatitis-presenting-with-severe-autoimmune-neutropenia
#15
Petros Doumtsis, Theodora Oikonomou, Ioannis Goulis, Kaliopi Zachou, George Dalekos, Evangelos Cholongitas
Autoimmune hepatitis (AIH) is a progressive, chronic liver disease characterized by unresolving hepatocellular inflammation of autoimmune origin. The clinical spectrum may vary from asymptomatic presentation, to non-specific symptoms such as fatigue, arthralgias, nausea and abdominal pain, to acute severe liver disease. AIH is characterized by the presence of interface hepatitis and portal plasma cell infiltration on histological examination, hypergammaglobulinemia, and positive autoantibodies. AIH is associated with other autoimmune diseases and its course is often accompanied by various non-specific hematological disorders...
January 2018: Annals of Gastroenterology: Quarterly Publication of the Hellenic Society of Gastroenterology
https://www.readbyqxmd.com/read/29296772/high-grade-neutropenia-in-a-patient-successfully-treated-with-nivolumab-for-refractory-primary-mediastinal-b-cell-lymphoma
#16
Zachary Wright, Alexander Brown
Autoimmune neutropenia is a rare side effect of nivolumab which may respond to antibody-based therapies.Nivolumab can lead to durable complete remission in primary mediastinal B-cell lymphoma refractory to multiple lines of therapy.
July 25, 2017: Blood Advances
https://www.readbyqxmd.com/read/29248492/r-loops-cause-genomic-instability-in-t-helper-lymphocytes-from-patients-with-wiskott-aldrich-syndrome
#17
Koustav Sarkar, Seong-Su Han, Kuo-Kuang Wen, Hans D Ochs, Loïc Dupré, Michael M Seidman, Yatin M Vyas
BACKGROUND: Wiskott-Aldrich syndrome (WAS), X-linked thrombocytopenia (XLT), and X-linked neutropenia, which are caused by WAS mutations affecting Wiskott-Aldrich syndrome protein (WASp) expression or activity, manifest in immunodeficiency, autoimmunity, genomic instability, and lymphoid and other cancers. WASp supports filamentous actin formation in the cytoplasm and gene transcription in the nucleus. Although the genetic basis for XLT/WAS has been clarified, the relationships between mutant forms of WASp and the diverse features of these disorders remain ill-defined...
July 2018: Journal of Allergy and Clinical Immunology
https://www.readbyqxmd.com/read/29247540/efficacy-and-safety-of-rituximab-for-systemic-lupus-erythematosus-associated-immune-cytopenias-a-multicenter-retrospective-cohort-study-of-71-adults
#18
Alexandra Serris, Zahir Amoura, Florence Canouï-Poitrine, Benjamin Terrier, Eric Hachulla, Nathalie Costedoat-Chalumeau, Thomas Papo, Olivier Lambotte, David Saadoun, Miguel Hié, Philippe Blanche, Bertrand Lioger, Jacques-Eric Gottenberg, Bertrand Godeau, Marc Michel
The aim of the study was to assess the efficacy and safety of rituximab (RTX) for treating systemic lupus erythematosus (SLE)-associated immune cytopenias. This multicenter retrospective cohort study of adults from French referral centers and networks for adult immune cytopenias and SLE involved patients ≥18 years old with a definite diagnosis of SLE treated with RTX specifically for SLE-associated immune cytopenia from 2005 to 2015. Response assessment was based on standard definitions. In total, 71 patients, 61 women (85...
March 2018: American Journal of Hematology
https://www.readbyqxmd.com/read/29230955/diagnostic-value-of-cell-bound-and-circulating-neutrophil-antibody-detection-in-pediatric-neutropenia
#19
Laura Porretti, Piero Farruggia, Federico Simone Colombo, Alessandra Cattaneo, Roberta Ghilardi, Nadia Mirra, Lucia Dora Notarangelo, Baldassarre Martire, Elena Trombetta, Silvano Milani, Claudia Vener, Paolo Rebulla
BACKGROUND: Chronic benign neutropenia of infancy includes primary autoimmune neutropenia (pAIN) and chronic idiopathic neutropenia (CIN). A diagnosis of CIN is supported by the absence of free and/or cell-bound neutrophil autoantibodies, which can be detected by flow cytometry with the indirect-granulocyte immunofluorescence test (I-GIFT) and direct-granulocyte immunofluorescence test (D-GIFT), respectively. Conclusive evidence is lacking on the diagnostic value of the D-GIFT, whose performance requires specific laboratory expertise, may be logistically difficult, and hampered by very low neutrophil count in patient samples...
April 2018: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/29177911/mechanisms-of-sustained-neutrophilia-in-patient-whim-09-cured-of-whim-syndrome-by-chromothripsis
#20
Qian Liu, Zhanzhuo Li, Alexander Y Yang, Ji-Liang Gao, Daniel S Velez, Elena J Cho, David H McDermott, Philip M Murphy
WHIM-09 is the first patient described with WHIM syndrome, an autosomal dominant form of neutropenia related to bone marrow retention of neutrophils. Originally diagnosed incorrectly with autoimmune neutropenia, the patient underwent splenectomy at age 9, but the absolute neutrophil count (ANC) did not rise. Subsequently, she was spontaneously cured by chromothripsis (chromosome shattering), which deleted the disease allele CXCR4 R334X , and 163 other genes, on chromosome 2 in a single hematopoietic stem cell (HSC)...
January 2018: Journal of Clinical Immunology
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