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PubMed2003

Pure red cell aplasia in a patient with trisomy X chromosome abnormality and reactivated Epstein-Barr virus infection.

We describe a woman with a congenital chromosome anomaly, 47,XXX, who developed chronic pure red cell aplasia (PRCA). The patient had serologic reactivity consistent with that of reactivated Epstein-Barr virus (EBV) infection, as judged by high titers for anti-EBV viral capsid antigen (VCA) immunoglobulin G (IgG) and anti-early antigen (EA) IgG. Detection of EBV genome in peripheral blood cells and cell-free serum also supported the diagnosis. Although EBV infection has been implicated in the pathogenesis of acute PRCA, the viral infection rarely results in a chronic disease state. So far, only 1 case of EBV-associated chronic PRCA has been reported, to the best of our knowledge. Chronic PRCA also is known to occur on an autoimmune basis. Individuals carrying an extra X chromosome, such as XXY and XXX, are prone to development of immune abnormalities. Our patient had an anti-DNA autoantibody and a positive result of the direct Coombs test. The pathogenesis of PRCA in this case seemed to involve multiple factors. In addition to the infectious agent, host factors may have played a role. Although the etiologic link between chronic PRCA and trisomy X remains to be elucidated, our findings suggest the importance of karyotype analysis as well as search for infectious agents in patients with chronic PRCA.

Chromosomes, Human, X↗
PubMed2003

Angioimmunoblastic T-cell lymphoma associated with an antibody to human immunodeficiency virus protein.

A 58-year-old man was admitted with generalized lymphadenopathy. On admission, the patient showed polyclonal hyper-gammopathy in a blood examination, positive results in the direct/indirect Coombs test, and an elevated cold agglutinin titer. Autoimmune thrombocytopenia with a high level of platelet-associated immunoglobulin G complicated the patient's condition. An enzyme immunoassay kit for human immunodeficiency virus (HIV) recombinant proteins p24, gp41, and gp36 showed positive results. Western blot analysis showed the presence of antibodies cross-reacting with HIV p24 gag protein. HIV RNA was not detected by means of a reverse transcriptase-polymerase chain reaction assay, so the patient was not an HIV carrier. Angioimmunoblastic T-cell lymphoma (AILT) was diagnosed on the basis of lymph node biopsy specimens. We speculated that in this case some of the numerous subtypes of polyclonal gamma globulin had coincidentally cross-reacted with HIV p24. Cross-reactive phenomena with HIV in patients with systemic lupus erythematosus have been well investigated, but to our knowledge our patient is the first case of such cross-reactivity involving AILT. Physicians should pay close attention to serologic tests to determine whether the patient truly is a viral carrier.

Antibodies, Viral↗
PubMed2005

Prospective surveillance of Canadian children with the haemolytic uraemic syndrome.

We determined prospectively the incidence of childhood diarrhoea associated haemolytic uraemic syndrome (D+ HUS) or HUS due to Streptococcus pneumoniae (SP+ HUS) in Canada through the Canadian Paediatric Surveillance Program (CPSP) network. From April 2000, to March 2002, 82% (1,886/2,300) of all Canadian paediatricians reported possible new cases of D+ HUS or SP+ HUS. Diagnosis was validated with a second questionnaire. There were a total of 136 children with D+ HUS (epidemic, n =15; endemic, n =121), corresponding to an annual incidence rate of 1.11 cases per 100,000 children under 16 years of age. Excluding incomplete forms, the annual incidence rate among endemic cases was 0.74 cases per 100,000. The provinces of Ontario (40%), Quebec (31%), and Alberta (18%) accounted for 89% of cases. Evidence of E. coli O157 or O157:H7 were found in 94% (79/84) of children who had positive stool cultures. The mortality rate was 4% (n =5) and 34% (n =41) of children underwent dialysis for a median of 12 days (2-60 days). SP-HUS was diagnosed in four children with pneumonia and pleural effusion (n =2) or meningitis (n =2) who survived. One child with positive direct Coombs testing had definitive evidence of SP+ HUS. The remaining three were considered possible cases. We conclude that SP+ HUS is rare in Canada. Over the last 15 years, the incidence of childhood D+ HUS in Canadian children may have decreased.

Adolescent↗
PubMed2003

Visceral leishmaniasis resembling systemic lupus erythematosus.

We describe three cases with visceral leishmaniasis (VL) associated with autoimmune manifestations. The patients presented with anemia, leukopenia, thrombocytopenia, renal involvement and low complement levels. Autoimmune features were present (antinuclear and anticardiolipin antibodies, VDRL, RF, positive direct Coombs' test), which subsided after therapy. Leishmaniasis should be taken into consideration in the differential diagnosis of autoimmune disorders.

Adult↗
PubMed2001

Induction of remission with intravenous immunoglobulin and cyclophosphamide in steroid-resistant Evans' syndrome associated with dermatomyositis.

Evans' syndrome is characterised by the simultaneous or sequential occurrence of Coombs'-positive haemolytic anaemia (AIHA) and immune thrombocytopenia without underlying aetiology. It has been found to be associated with collagen vascular diseases, especially systemic lupus erythematosus (SLE) and scleroderma. However, Evans' syndrome with dermatomyositis is very rare. A 59-year-old woman, who had been taking high-dose prednisolone for a month and cyclosporin for 10 days for dermatomyositis, developed purpura on the left popliteal fossa. The platelet and haemoglobin levels decreased to 77,000/mm3 and 9.8 g/dl, respectively. Antiplatelet antibody was positive. Thrombocytopenia responded to intravenous immunoglobulin (IVIG) for a short time, but further decreased in a week. Her blood film showed features of haemolytic anaemia. Laboratory findings showed reticulocytosis and a positive direct Coombs' test. Bone marrow examination showed a mild hyperplasia of erythroid precursors and megakaryocytes. The patient was successfully treated with cyclophosphamide in addition to oral prednisolone. AIHA in connective tissue disease may develop gradually and show a benign clinical course in most patients. Therefore, we suggest that patients with dermatomyositis and anaemia should always be checked for haemolysis if there is no other explanation.

Anemia, Hemolytic, Autoimmune↗
PubMed1975

Angio-immunoblastic lymphadenopathy. Diagnosis and clinical course.

The clinical and pathologic findings in 24 patients with "angio-immunoblastic lymphadenopathy with dysproteinemia" (AILD) are presented. The patients' ages ranged from 44 to 80 years, with a median age of 68 years. The disease has an acute onset. In many respects, the clinical presentation is suggestive of malignant lymphoma. Generalized lymphadenopathy was always present. Hepatomegaly was found in 20 patients, splenomegaly in 17, constitutional symptoms in 20 and skin rashes in nine. Twenty patients had anemia, with positive Coombs' test in eight of 14 tested. Polyclonal hypergammaglobulinemia was found in 17 of 22 patients. Two patterns of evolution were recognizable: (1) long survival (24 to 67 months) without treatment or after the administration of intensive combination chemotherapy; and (2) rapid progression (one to 19 months) regardless of the treatment given. Sixteen patients died; postmortem examination in 10 cases showed the cause of death to be attributable to severe infection in eight patients, to renal disease in one and to cardiovascular disease in one. No evidence of malignant lymphoma was seen in any of these autopsies. Histologically, the disease is systemic, with specific lesions in the lymph nodes. The spleen, liver, bone marrow, skin and lung are also involved, but the changes are less characteristic than in the lymph nodes. In the patients in whom sequential biopsies were performed, a trend toward restoration of the nodal architecture was observed. AILD is a clinical-pathologic entity in a spectrum of yet to be defined immune reactions. The clinical, laboratory and pathologic manifestations of AILD are consistent with an autoimmune disorder, in which a deficiency of the T-cell regulatory functions probably predisposes to an abnormal proliferative and autoaggressive reaction of the B-cell system. Surgical staging procedures do not appear to be indicated. Intensive cytotoxic treatment may be hazardous in some patients, precipitating their death, but long survival after such therapy has been observed in others. Supportive therapy and small doses of steroids appear to be a safer therapeutic approach.

Adult↗
PubMed1975

Drug-induced immune hemolytic anemia.

Drug administration causes from 16 to 18 per cent of cases of acquired immune hemolytic anemia. The pathogenesis of erythrocyte sensitization by drug-related antibody with or without fixation of complement is variable, and there is a relationship between the responsible drug, the mechanism of red cell sensitization, clinical manifestations and laboratory methods of diagnosis. Drugs such as phenacetin and quinidine form a complex with the antidrug antibody, and the immune complex attaches to red cells usually fixing complement and causing acute intravascular hemolysis. Other drugs (e.g., penicillins), when given in large doses, coat normal red cells in vivo and in some patients a high titer IgG anti-drug antibody develops which reacts with the coated cells. Hemolytic anemia may develop with red cell destruction being primarily extravascular. Cephalosporins cause positive direct antiglobulin tests in a small percentage of patients either by the same mechanism as penicillins or by modification of the red cell membrane leading to nonimmunologic absorption of serum proteins. Hemolytic anemia has been reported only rarely. A few drugs (notably alpha methyldopa) cause the development of autoimmune hemolytic anemia. Knowledge of clinical manifestations and laboratory aids to diagnosis is necessary to distinguish immunohematologic abnormalities caused by drugs from other causes.

Adsorption↗
PubMed1982

B-cell neoplasms with homogeneous cold-reacting antibodies (cold agglutinins).

Among 78 patients with persistent cold agglutinins, 31 had lymphoma, 13 had macroglobulinemia of Waldenstrom, six had chronic lymphocytic leukemia and 28 had chronic cold agglutinin disease. The average age was over 60 years. Patients wit chronic cold agglutinin disease had more hemolytic crises, bleeding and Raynaud's phenomena, and less frequently lymphadenopathy or hepatosplenomegaly. The frequency of anemia, positive Coombs test results, cryoglobulinemia and Bence Jones proteinuria was similar in the various groups. Survival time from diagnosis was on average two years in lymphoma, two and a half years in Waldenstrom's macroglobulinemia, more than six years in chronic lymphocytic leukemia and more than five years in chronic cold agglutinin disease. Anti-I were common in chronic cold agglutinin disease (74 percent) and rare in other groups (32 to 33 percent). Anti-I and other cold agglutinins were rare in chronic cold agglutinin disease and common in lymphoma and Waldenstrom's macroglobulinemia. In chronic cold agglutinin disease, and in Waldenstrom's macroglobulinemia, cold agglutinins usually had K light chains--92 percent and 71 percent, respectively--whereas in lymphoma, 71 percent of cold agglutinins had lambda light chains. The type of light chains related to the specifically of cold agglutinins: 58 percent of IgM/K were anti-I, 75 percent of IgM/lambda had other specificities. Cold agglutinins were cytotoxic to autologous and allogeneic cells were killed implying that the former may be precoated in vivo with the antibodies. In conclusion, conditions with persistent cold agglutinins are a spectrum that varies from "benign" autoimmune-like chronic cold agglutinin disease to malignant lymphoma. Marked differences in the light chain type of cold agglutinins, specificity toward membranous antigens and severity of clinical manifestations were noted in benign and malignant varieties.

Adult↗
PubMed1987

Hemolytic anemia following intravenous gamma globulin administration.

A young homosexual man with immune thrombocytopenia recently had transient intravascular hemolysis during intravenous gamma globulin treatment. The hemolysis, manifested by a sharp decrease in hemoglobin and by a direct Coombs' test with a positive result, was mediated by anti-A antibody present in the gamma globulin preparation. In view of the increasing use of intravenous gamma globulin in the treatment of patients with immune cytopenia, this problem may result in crossmatching difficulties and should be recognized as a potential complication of therapy.

Adult↗
PubMed1979

Is the d an allelic gene of D in the Rh blood group system? A quantitative study using 99mTechnetium-pyrophosphate-labelled antibody (TcLA) method.

This article reports on studies of Rh antigens, such as D, Du and d, using uranyl-labelled antibody (ULA) and TcLA (99mTc-pyrophosphate-labelled antibody) methods for the first time for this purpose. TcLA method proved to be simple in labelling and very sensitive (20--100 times more so than the indirect Coombs test) in the detection of Rh antigen-antibody binding. Results of this quantitative study demonstrate convincingly that the d is not an allelic gene of D but rather the weakest of the series D less than Du less than d. Although the evidence from this study demonstrates clearly that differences between D and d are only quantitative, the authors do not think that the Rh nomenclature should be changed but they do think that the present evidence should be used in regard to the understanding of the allelism in the Rh blood group system. The c is an allelic gene of C as the e is an allelic gene of E; specific test sera detecting every one of these antigens exist and the family studies verify these statements. However, the d is not a distinct antigen as c and e are, even if the pattern of inheritance from family studies, using the existent anti-D serum, would suggest the allelism as probability. That is why in the past the anti-Du and anti-d specific test sera never incidentally found or artificially produced.

Alleles↗
PubMed1993

Stress and dexfenfluramine: effects on the immune response and energy balance in the rat.

The effects of stress, dexfenfluramine (d-Fen), and a combination of both were investigated on ingestive behavior, body weight, and the humoral immune response in the rat. Three-hundred and 84 male Sprague-Dawley rats were split into four groups of 96 animals. In a balanced design, each group was submitted or not to repeated intense stress for 20 consecutive days. Animals were also treated with 5 mg/kg/day d-Fen (IP, 1 ml/kg) or an equal volume of placebo (saline) for 28 days. The humoral immune response of rats to sheep red blood cells (50% solution, 1 ml IP at day 0) was assessed from the antibody titer on days 4, 8, 12, 16, 20, and 28. Antibodies were assayed by direct hemagglutination and by the Coombs' test. Plasma corticosterone was also measured on days 0 and 12. The effects of stress and d-Fen on ingestive behavior and body weight were consistent with previously published results. In addition, rats treated with d-Fen had a significantly reduced body weight (-20 g) 5 weeks after the end of the treatment, whereas the loss in body weight induced by stress had totally disappeared. Stress did not decrease animals' immune response despite a massive corticosterone secretion on day 0, with a marked response lasting for at least 12 days. d-Fen reduced the corticosterone levels determined on day 12. Antibody production was slightly but significantly reduced in rats receiving d-Fen.(ABSTRACT TRUNCATED AT 250 WORDS)

Animals↗
PubMed1989

In vitro determination of phagocytosis and intracellular killing of Brucella melitensis by goat's polymorphonuclear phagocytes.

One hundred and forty samples of goats serum taken from animals suspected of having brucellosis, have been studied by trying to assess the phagocytosis in vitro of Brucella melitensis Rev-1 by polymorphonuclear neutrophil phagocytes opsonized with antibrucella serum. The serum agglutination test and the Coombs antiglobulin test were carried out with the serum samples. The opsonization ingestion, intracellular killing and germ killing power of the serum were also assessed. The results demonstrate that the phagocytic activity of the polymorphonuclear neutrophils opsonized by a particular serum can be of use to determine its titre; its opsonic activity varies according to whether the serums are inactivated or not. Likewise it shows the resistance of Brucella meilitensis Rev-1 to digestion by caprine polymorphonuclear neutrophils, this resistance being independent of the animal's degree of infection.

Agglutination Tests↗
PubMed1982

A laser nephelometric method for measuring alternative pathway complement activity of human and murine serum.

A kinetic method for the evaluation of alternative pathway complement activity in man and mice is presented. A laser nephelometer was employed for detection of non-sensitized rabbit erythrocyte lysis based on the observation that the intensity of laser scatter (LS) is proportional to the number of erythrocytes in suspension. During erythrocyte lysis a continuous decline in LS is observed since lytic products do not evoke LS. Utilizing the indirect Coombs test and cross-electrophoresis it was shown that rabbit erythrocyte lysis causes activation of the alternative complement pathway. This method is modified for room temperature, is independent of sample hemoglobin content and, in its micro version, it can be done with 10 microliter of human serum, i.e. 50 microliters of murine serum.

Animals↗
PubMed1982

Serological response of cattle after vaccination and challenge with Brucella abortus.

New and currently used serological procedures were evaluated using sera from cattle that were challenged with B. abortus S544 (S544) after vaccination with either B. abortus S19 (S19) or B. abortus 45/20 (S45/20) as calves or adults. In animals vaccinated with S19, titres to the indirect haemolysis test (IHLT) rose more slowly, declined more rapidly and involved fewer animals than did titres to the complement fixation test (CFT). In animals vaccinated with S45/20 the rough antigen complement fixation test (RCFT) showed persistent titres. At slaughter the IHLT and CFT were found to be more specific and more sensitive than the Rose Bengal Plate Test (RBPT) and Serum Agglutination Test (SAT) in the detection of cattle infected with B. abortus.

Agglutination Tests↗