A case of pure red cell aplasia complicated by Evans syndrome

Toyokawa, Yasuhiko; Kingetsu, Isamu; Yasuda, Chiho; Yasuda, Jun; Yoshida, Ken; Kurosaka, Daitaro; Yamada, Akio

doi:10.1007/s10165-007-0584-9

Cited by 5 publications

(4 citation statements)

References 16 publications

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“…Furthermore, B19V‐infection is known to cause thrombocytopenia and to trigger autoimmune disorders [5,20–24]. We observed markers for acute or recent B19V‐infection in five patients presenting with autoimmune disease: ITP was diagnosed in patients AI and AII, Kawasaki‐disease in patient AIII, thrombocytopenia in patient AIV and Evan’s syndrome in patient A1, even though, contraversially, Kawasaki‐disease and Evan’s syndrome have been associated with B19V‐infection [25–27].…”

Section: Discussionmentioning

confidence: 99%

Impact of parvovirus B19 infection on paediatric patients with haematological and/or oncological disorders

Jitschin

Peters

Plentz

et al. 2011

Clinical Microbiology and Infection

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To determine the frequency and the impact of parvovirus B19 (B19V) infection and its influence on the course of haematological and/or oncological diseases in paediatric patients, consecutive serum and bone marrow samples from 110 were analyzed for markers of acute, past and persistent B19V-infection using qPCR, ELISA and WesternLine. Twenty-seven out of 110 (24.5%) children suffered from non-malignant diseases (anaemia, pancytopenia, autoimmune disorders); 68/110 (61.8%) patients had developed leukaemia, malignant lymphoma or solid malignant tumours; 15/110 patients (13.6%) presented with other symptoms. At admission, B19V-specific IgM and IgG indicating acute or previous B19V-infection were observed in 5 (4.5%) and 48 patients (43.6%), respectively. B19V-DNA (10(3) -10(9) geq/mL) was detectable in serum and/or bone marrow of 22 patients (20.0%). These suffered from leukaemia (5), non-Hodgkin lymphoma (2), solid tumours (6), autoimmune (4) and haematological (4) disease and fever (1). During clinical observation four further leukaemia patients developed viraemia and persistent B19V-infection was observed in 13/22 DNA-positive patients. Treatment of B19V-DNA-positive cancer patients was associated with more supportive therapy involving erythrocyte and thrombocyte transfusion and/or antibiotic therapy. Acute B19V-infection has been frequently observed in paediatric patients with haematological and/or oncological disease. In patients with non-malignant diseases anaemia or autoimmune disorders were diagnosed in association with B19V-infection. Furthermore, a significant number of cancer patients displayed markers for acute, recent or persistent B19V-infection. This association may be strengthened by frequent treatment with blood products combined with therapeutic immune suppression. In B19V-infected cancer patients supportive therapy was more complex.

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Section: Discussionmentioning

confidence: 99%

Impact of parvovirus B19 infection on paediatric patients with haematological and/or oncological disorders

Jitschin

Peters

Plentz

et al. 2011

Clinical Microbiology and Infection

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show abstract

“…As B19 virus targets the erythroblast, an infection which results in apoptosis, the mechanism of B19-associated aplastic anaemia has been presumed to be on the basis of direct infection. However, B19 infection has also been associated with Evans syndrome in two cases 172 173. Evans syndrome is the presence of direct Coombs positive autoimmune haemolytic anaemia (AIHA) in conjunction with immune-mediated thrombocytopenia.…”

Section: Autoimmune Diseases Associated With Parvovirus B19 Infectionmentioning

confidence: 99%

The role of parvovirus B19 in the pathogenesis of autoimmunity and autoimmune disease

Kerr

2015

J Clin Pathol

109

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Human parvovirus B19 is a single-stranded DNA virus which preferentially targets the erythroblasts in the bone marrow. B19 infection commonly causes erythema infectiosum, arthralgia, fetal death, transient aplastic crisis in patients with shortened red cell survival, and persistent infection in people who are immunocompromised. Less common clinical manifestations include atypical skin rashes, neurological syndromes, cardiac syndromes, and various cytopenias. B19 infection has also been associated with development of a variety of different autoimmune diseases, including rheumatological, neurological, neuromuscular, cardiovascular, haematological, nephrological and metabolic. Production of a variety of autoantibodies has been demonstrated to occur during B19 infection and these have been shown to be key to the pathogenesis of the particular disease process in a significant number of cases, for example, production of rheumatoid factor in cases of B19-associated rheumatoid arthritis and production of anti-glutamic acid decarboxylase (GAD) in patients with B19-associated type 1 diabetes mellitus. B19 infection has also been associated with the development of multiple autoimmune diseases in 12 individuals. Documented mechanisms in B19-associated autoimmunity include molecular mimicry (IgG antibody to B19 proteins has been shown to cross react with a variety of recognised human autoantigens, including collagen II, keratin, angiotensin II type 1 receptor, myelin basic protein, cardiolipin, and platelet membrane glycoprotein IIb/IIIa), B19-induced apoptosis with presentation of self-antigens to T lymphocytes, and the phospholipase activity of the B19 unique VP1 protein.

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“…The risk in CLL is reported to be higher in patients with adverse cytogenetics, unmutated IGHV of the malignant CLL clone and specific stereotyped B cell receptor configurations (5–10). Classical AIC cases rarely represent a diagnostic challenge for the hematologist, but the diagnosis may also be more intricate e.g., in cases showing overlapping patterns of immune reactivity with precursor and mature blood cells (11–16). When diagnosed, most cases of AIC—whether primary or secondary—are primarily treated with steroids (17).…”

Section: Introductionmentioning

confidence: 99%

High-Throughput Immunogenetics Reveals a Lack of Physiological T Cell Clusters in Patients With Autoimmune Cytopenias

et al. 2019

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Autoimmune cytopenias (AIC) such as immune thrombocytopenia or autoimmune hemolytic anemia are claimed to be essentially driven by a dysregulated immune system. Using next-generation immunosequencing we profiled 59 T and B cell repertoires ( TRB and IGH ) of 25 newly diagnosed patients with primary or secondary (lymphoma-associated) AIC to test the hypothesis if these patients present a disease-specific immunological signature that could reveal pathophysiological clues and eventually be exploited as blood-based biomarker. Global TRB and IGH repertoire metrics as well as VJ gene usage distribution showed uniform characteristics for all lymphoma patients (high clonality and preferential usage of specific TRBV - and TRBJ genes), but no AIC-specific signature. Since T cell immune reactions toward antigens are unique and polyclonal, we clustered TCRβ clones in-silico based on target recognition using the GLIPH (grouping of lymphocyte interactions by paratope hotspots) algorithm. This analysis revealed a considerable lack of physiological T cell clusters in patients with primary AIC. Interestingly, this signature did not discriminate between the different subentities of AIC and was also found in an independent cohort of 23 patients with active autoimmune hepatitis. Taken together, our data suggests that the identified T cell cluster signature could represent a blood biomarker of autoimmune conditions in general and should be functionally validated in future studies.

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A case of pure red cell aplasia complicated by Evans syndrome

Cited by 5 publications

References 16 publications

Impact of parvovirus B19 infection on paediatric patients with haematological and/or oncological disorders

Impact of parvovirus B19 infection on paediatric patients with haematological and/or oncological disorders

The role of parvovirus B19 in the pathogenesis of autoimmunity and autoimmune disease

High-Throughput Immunogenetics Reveals a Lack of Physiological T Cell Clusters in Patients With Autoimmune Cytopenias

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