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PubMed2004

[Follicular lymphoma complicated with autoimmune hemolytic anemia and pure red cell aplasia].

Malignant lymphomas are often associated with immunological disorders. We describe here a 54-year-old woman with follicular lymphoma, simultaneously complicated with autoimmune hemolytic anemia and pure red cell aplasia. The patient had bilateral cervical, axillar and inguinal lymph node swellings. Peripheral blood analysis revealed severe anemia (Hb 3.4g/dl) and reticulocytopenia (2260/ml), and then the bone marrow showed erythroid hypoplasia. Furthermore, a direct Coombs test was positive and the serum haptoglobin level was undetectable. After treatment with CHOP followed by 1 mg/kg of prednisolone daily, the patient obtained complete remission and her anemia improved to the normal level.

Anemia, Hemolytic, Autoimmune↗
PubMed1992

[Immunoblastic lymphadenopathy-like T cell lymphoma accompanied by autoimmune hemolytic anemia].

A 62-year-old man was admitted to our hospital because of generalized lymphadenopathy, fever and skin eruptions. The histology of the right cervical lymph nodes showed immunoblastic lymphadenopathy (IBL)-like T cell lymphoma. His laboratory data were as follows: hemoglobin concentration 7.1 g/dl, red blood cells 1,850,000/microliters, reticulocytes 4.2%, total bilirubin 2.6mg/dl, direct bilirubin 0.5mg/dl, haptoglobin less than 10mg/dl, positive Coombs test. He was diagnosed as having IBL-like T cell lymphoma accompanied by autoimmune hemolytic anemia. He was successfully treated with combination chemotherapy (Pro-MACE), and lymph node swelling and hemolytic anemia disappeared. He has been in complete remission for more than 1 year.

Anemia, Hemolytic, Autoimmune↗
PubMed1979

Impaired regulation of erythrocyte autoantibody production after splenectomy.

C3H mice were given 4 i.p. injections, eac of 2 X 10(8) WAG rat RBC, at weekly intervals. The production of erythrocyte autoantibodies elicited by the cross-reacting rat RBC was assessed using the average direct Coombs' test (DCT) score. Autoantibody production reached higher levels and persisted significantly longer in mice splenectomized 15 days before the first injection of rat RBC. This increased production of autoantibodies was not prevented by injecting each splenectomized mouse i.v. with 5 X 10(7) syngeneic spleen cells immediately after splenectomy. Similarly, splenectomy of mice already DCT+ significantly prolonged autoantibody production which was not prevented by injections of 10(8) cells prepared from the spleens removed at splenectomy. Transfer of spleen cells from mice already DCT+ to mice before the injections of rat RBC were started in the recipients caused a significant reduction in the amount of RBC autoantibodies produced. This suppression of autoantibody production was greater in unsplenectomized mice than in splenectomized mice. The results show that the spleen is involved in the regulation of these erythrocyte autoantibody responses. It is hypothesized that, in addition to the cellular component of the spleen, the splenic architecture and/or environment contributes to the regulation of autoantibody responses.

Animals↗
PubMedDate not supplied

[Thrombotic thrombocytopenic purpura: report of seven cases].

From May 1999 to January 2002 we observed 7 patients (4 females and 3 males, median age 55 years, range 31-81 years) with thrombotic thrombocytopenic purpura (TTP). Six patients has been previously undiagnosed and 1 patient was at second relapse. Trigger factors of TTP were identified in 6 patients: ticlopidine treatment (2 patients); an acute cutaneous infection episode immediately before the features of TTP (1 patient); presence of devices: orthodontic (1 patient) and intrauterine contraceptive (1 patient), Mycoplasma urealyticum vaginal infection (1 patient). In all the 7 patients the clinical status was mainly related to the hemolytic anemia, thrombocytopenia and neurological events. One of these patients presented with hemolytic-uremic syndrome with acute renal failure and macrohematuria at onset, another one showed a systemic exanthema post-infection-like. Six out of 7 patients presented with different neurological events: headache, confusion, focal neurological failure. All the 7 patients were promptly treated with plasma-exchange and cryosupernatant plasma infusion. In addition they received prednisone 25-50 mg/day. All the 7 patients achieved a complete remission after plasma-exchange, one relapsed 3 months later and was treated with plasma-exchange again. All the patients are in complete remission with a median follow-up of 36.3 months (range 20-62 months). From these cases we suggest: 1) clinicians should take in mind the suspicion of TTP in every patient with hemolytic, negative direct Coombs test, anemia, thrombocytopenia, high level of lactate dehydrogenase; 2) the treatment of choice is plasma-exchange; 3) the response of treatment is good if therapy is promptly and aggressively administered; 4) the possible role of a trigger factor for removing it and to prevent relapses.

Adult↗
PubMed1979

[Immunomechanisms in drug-induced hemolytic anemias].

Among the acquired hemolytic anemias, drug-induced immune hemolytic anemias (IHA) play a significant role. Many different drugs are capable of causing IHA, alphamethyldopa and penicillins have been responsible for the greatest number of cases. For the majority of the other drugs single cases of IHA are reported. Drug-induced immune hemolysis may be mediated by different immune mechanisms including the immune cytotoxicity type (penicillins and cephalosporins) with predominantly extravascular hemolysis. Immune hemolysis of most other drugs is due to immune complexes formed by the drug and specific antibodies leading to intravascular hemolysis in the presence of complement activation. So far, the autoimmune type of IHA caused by alphamethyldopa is unexplained, serologically this form cannot be distinguished from warm antibody auto-IHA. The clinical importance of drug-induced IHA depends on the severity of anemia and the problem of diagnosis leading to adequate therapeutic measures. Only a small number of cases showing a positive direct antiglobulin (Coombs') test caused by drugs will develop IHA. Therefore this finding does not require absolutely withdrawal of the drug, but careful observation of the patient. The diagnosis of drug-induced IHA is necessary for its distinction from auto-IHA and other secondary IHA. The study of such IHA has great importance for the understanding of other drug-induced cytopenias and may give explanations of the pathogenetic mechanisms of yet unexplained autoimmune phenomena.

Anemia, Hemolytic, Autoimmune↗
PubMed2005

[Basedow disease associated with Evans syndrome].

A 60-year-old woman was admitted to a hospital complaining of dizziness and general fatigue in October, 2004. Because of heart failure and severe anemia, she was referred to our hospital. Based on a positive direct Coombs test and an elevated level of platelet-associated IgG (PAIgG), the patient was diagnosed as having autoimmune hemolytic anemia (AIHA) associated with idiopathic thrombocytopenic purpura (ITP), i.e., Evans syndrome. Basedow disease was also diagnosed due to hyperthyroidism with an elevation of anti-thyroid stimulating hormone (TSH) receptor antibodies. Both the Evans syndrome and Basedow disease were considerably ameliorated with plasma exchange, corticosteroid and thiamazole therapy. Although Basedow disease is known to be associated with hematological disorders such as AIHA or ITP, the combination of Basedow disease and Evans syndrome is rare. We report here a case of Basedow disease associated with Evans syndrome.

Anemia, Hemolytic, Autoimmune↗
PubMed2006

Update on the management of non-anti D antibodies.

With the development of the Rh D immunoglobulin and utilisation in clinical practice, severe rhesus iso-immunisation is rarely seen today. Antibodies against other blood groups are now more common than anti-D and while the majority do not cause significant haemolysis, there are reports of serious morbidity with antibodies other than anti-D. We reviewed retrospectively all cases (excluding anti-D and anti kell antibodies) where antibodies were detected at routine booking visit between 1997-2001 and correlated the type of antibody to clinical outcome as assessed by coombs test, postnatal phototherapy and transfusion. Our approach to these cases does not involve invasive test as amniocentesis and relies on the ultrasound monitoring, level of antibodies and timely delivery with appropriate paediatric follow up. Over five years period 150 women were identified with antibodies other than anti-D or anti-kell. There were no directly related deaths. Many authorities suggest amniocentesis for specific antibodies to assess fetal haemolysis and plan management. This study support a non-invasive approach with mortality and minimal morbidity.

Blood Group Antigens↗
PubMed2004

Serum IL-2 and platelet-associated immunoglobulins are good prognostic markers in immune thrombocytopenic purpura.

Immune thrombocytopenic purpura (ITP) is an acquired disease in which autoantibodies to platelets cause their sequestration and destruction by mononuclear macrophages, principally in the spleen. While most children with the disease experience a relatively short and benign clinical course, ITP in adults often lasts more than 6 months (chronic ITP) and is resistant to conventional treatment (corticosteroids, intravenous immunoglobulin, or splenectomy). This work was done to study the immunological difference between acute and chronic ITP, the effect of treatment on the studied immunological parameters, and to evaluate the role of prednisone therapy in chronic ITP. The study included 49 patients, twenty-three children with acute ITP, and twenty-six with chronic ITP. After taking the history, clinical examination was performed for all patients and control subjects. Laboratory investigations included complete blood count, bone marrow aspirate examination (patients), direct and indirect Coombs' test, antinuclear antibodies, lymphocyte phenotyping, cytokine (IL-2, IFN-gamma, and IL-6) measurement, and platelet antibodies by immunofluorescence. Results showed that acute ITP is more prevalent in preschool children and its relapse is lower when steroids are used for treatment. Platelet counts were significantly elevated in both acute and chronic ITP, especially with good response to steroids. Also, CD4 and CD4/CD8 were significantly reduced in chronic ITP with good response to therapy. Both IL-2 and IFN-gamma were significantly increased in chronic ITP when compared to acute ITP or control. Platelet associated IgM was detected more in acute than in chronic ITP, while IgG was equally detectable in both cases. This work shows that IL-2 is a good prognostic factor in chronic ITP and steroids are important for its treatment. It also shows that platelet associated IgG is a good monitoring parameter for response to treatment.

Acute Disease↗
PubMed1991

[Severe immunoallergic hemolytic anemia caused by a glafenine metabolite].

Three weeks after a surgical operation, a 74-year old woman was admitted to hospital for severe haemolytic anaemia. A strongly positive IgG type direct Coombs test pointed to a iatrogenic origin, but a search for anti-molecule antibodies directed against all medicines taken by the patient after surgery was negative. We extended our immunological investigations and were able to demonstrate the presence of IgG type antibodies directed against an ex vivo metabolite of glafenine.

Aged↗
PubMed2006

[Plasmapheresis for fulminant autoimmune hemolytic anemia with rapidly progressing disturbance of consciousness].

A 64-year-old male was diagnosed as having autoimmune hemolytic anemia (AIHA) in December 2001. The diagnosis of AIHA was established based on the presence of anemia, hyperbilirubinemia, reticulocytosis, positive Coombs test and serum LDH elevation. Although he received steroid pulse therapy, disturbance of consciousness appeared and the anemia and icterus did not improve. Plasma exchange was carried out to eliminate antibodies as an emergency measure. The patient's symptoms were ameliorated following four courses of plasma exchange. This case represents a case of aggressive AIHA with disturbance of consciousness and refractory to conventional corticosteroid therapy. Plasma exchange rapidly led to complete remission.

Anemia, Hemolytic, Autoimmune↗
PubMed2006

Intravenous immunoglobulin in ABO and Rh hemolytic diseases of newborn.

OBJECTIVE: To evaluate whether the use of intravenous immunoglobulin in newborn infants with isoimmune hemolytic jaundice due to Rh and ABO incompatibility is an effective treatment in reducing the need for exchange transfusion. METHODS: This study included all direct Coombs' test positive Rh and ABO isoimmunized babies, who admitted in the Neonatal Intensive Care Unit of Ghaem Hospital of Mashhad University of Medical Sciences, Iran, from October 2003 to October 2004. Significant hyperbilirubinemia was defined as rising by >or=0.5 mg/dl per hour. Babies were randomly assigned to received phototherapy with intravenous immunoglobulin (IVIg) 0.5 g/kg over 4 hours, every 12 hours for 3 doses (study group) or phototherapy alone (control group). Exchange transfusion was performed in any group if serum bilirubin exceeded >or=20mg/dl or rose by >or=1mg/dl/h. RESULTS: A total of 34 babies were eligible for this study (17 babies in each group). The number of exchange transfusion, duration of phototherapy and hospitalization days, were significant shorter in the study group versus control group. When we analyzed the outcome results in ABO and Rh hemolytic disease separately, the efficacy of IVIg was significantly better in Rh versus ABO isoimmunization. Late anemia was more common in the IVIg group 11.8% versus 0%, p=0.48. Adverse effects were not observed during IVIg administration. CONCLUSION: Administration of IVIg to newborns with significant hyperbilirubinemia due to Rh hemolytic disease reduced the need for exchange transfusion but in ABO hemolytic disease there was no significant difference between IVIg and double surface blue light phototherapy.

ABO Blood-Group System↗
PubMed1991

The diagnosis and treatment of the nonimmune hydrops fetalis.

Fetal hydrops is associated with two distinct pathophysiologic situations. The isoimmune hydrops fetalis is a well understood disorder, and as the result of medical advances and prophylactic therapy its frequency is diminishing. The nonimmune hydrops fetalis is a poorly understood disease with a bad prognosis. The two disorders can be differentiated with the indirect Coombs test. In both cases the ultrasound examination plays an important role in the diagnosis, prognosis and management. Examination of the fetal blood sample gives recently a possibility to approach the disease. In NIHF the examination of fetal blood sample would give a relatively quick and effective diagnosis but its value for the treatment is limited. Although with the present technology it is impossible to diagnose all cases of NIHF, the early recognizing, the careful and step by step investigation, the active perinatologic management mostly can show the etiology and can help the perinatal team at the treatment of the disease.

Humans↗
PubMed1991

Systemic lupus erythematosus in a black South African child. First documented case report.

Systemic lupus erythematosus (SLE) is poorly described among black children in Africa despite being more frequent among some black adult populations than their white counterparts. The first black South African child with SLE is documented. The patient was a 10-year-old girl who had fever, facial rash (with complement (C4) deposited at the dermo-epidermal junction of normal skin), weight loss, central nervous system involvement (depression, withdrawal, retinal exudates), renal involvement (glomerular filtration rate 54 ml/min/1.73 m2; membranous nephropathy with mild mesangial proliferation; World Health Organisation classification Vb), alopecia, lymphadenopathy, hepatomegaly, positive Coombs test, hypeocomplementaemia, anti-DNA antibodies and positive anti-nuclear factor.

Black People↗
PubMedDate not supplied

[Fetal hemolytic disease in a patient immunized by six antibodies: diagnosis and treatment].

A case of severe haemolytic disease of the fetus due to six rare alloantibodies is described. In a pregnant women at 23 weeks gestation after the finding of a positive indirect Coombs test maternal antibodies have been precisely identified and titrated: anti-I: 1/23, anti-c: 1/64, anti-S: 1/16, anti-Fya: 1/128, anti-M: 1/64, anti-Jka: 1/32000. Fetal blood group, free and red blood cell adherent antibodies have been investigated on fetal blood samples obtained by means of cord centesis. A compatible donor has been found. Severe fetal anemia has been corrected by ultrasound guided intrauterine transfusions (one abdominal and two intravascular transfusions) with positive outcome of the pregnancy.

Adult↗
PubMed1990

[Evaluation of the bone marrow in patients with brucellosis. Clinico-pathological correlation].

In the present study the 60 patients with brucellosis and evaluation of bone marrow aspirate seen at the Hospital Base Cayetano Heredia from 1980 to 1986 were included. Iron deficiency was found in the bone marrow in 34.5% of patients, 31% in males and 36% in females. No correlation was found between iron deficiency and severity of the hematological or non-hematological clinical features. Bone marrow cytophagocytosis was found in 28.3% of patients. All had moderate to severe clinical features, and it is postulated that this finding may be helpful as a severity marker in patients with brucellosis. Bone marrow cytophagocytosis was significantly associated with the presence of hematologic abnormalities in general; anemia was the most common of these, followed by thrombocytopenia. This finding suggests that cytophagocytosis is an important mechanism in the pathogenesis of these abnormalities in brucellosis. Bone marrow hypercellularity was present in 70% with normocellularity in 28.3% and one case of pure megakaryocytic aplasia. In thirty-five patients pathological study of bone marrow was carried out 10 of these (28.5%) had granulomas. Their presence was not correlated with the clinical severity. Peripheral blood finding were: anemia in 83.3%, with two cases of hemolytic anemia and positive direct Coombs test, one of them associated with thrombocytopenia (Evans syndrome); leukopenia in 21%, basically due to neutropenia; thrombocytopenia in 33.3%, in one case associated with positive antiplatelet antibodies and with pure megakaryocytic aplasia in others; pancytopenia in 13.5% of cases (8 patients) associated to bone marrow cytophagocytosis in 5 cases (64.5%) and thus suggesting that this might be the major underlying pathogenetic mechanism.

Acute Disease↗