Rare Cause Of Anemia Research Articles

Algorithm of differential diagnosis of anemia involving laboratory medicine specialists to advance diagnostic excellence.

Anemia is a severe global public health issue. Testing practices for anemia suggest overuse of screening laboratory tests and misinterpretation of studies even in "easy-to-diagnose" underlying causes, leading to late diagnoses and missed treatment opportunities. We aimed to develop a complete and efficient algorithm for clinical pathologists and laboratory medicine physicians for the differential diagnosis of anemia. Comprehensive literature search encompassing original articles, studies, reviews, gold standard books, and other evidence. We created a complex algorithm, primarily for clinical pathology/laboratory use, that explores all major and several rare causes of anemia in an efficient and evidence-based manner. The algorithm includes gold-standard diagnostic laboratory tests available in most clinical laboratories and indices that can be easily calculated to provide an evidence-based differential diagnosis of anemia. The diagnostic strategy combines previously available diagnostic tests and protocols in an efficient order. Clinical pathologists following the algorithm can independently provide valuable diagnostic support for healthcare providers. Clinical pathologists providing complete differential diagnostic services with the proposed algorithm may create an opportunity for an advanced diagnostic service that supports diagnostic excellence and helps patients receive a timely diagnosis and early treatment opportunities.

A rare cause of anemia and hepatosplenomegaly-marble bone disease

Abstract Marble bone disease (“osteopetrosis”) is a rare, heritable disorder of the skeleton due to a defective function of the osteoclasts and characterized by increased bone density on radiographs. It is an uncommon disease with an unknown etiology. The presentation and severity of osteopetrotic conditions vary greatly, from neonatal onset with potentially fatal complications such as bone marrow failure (e.g., classic or “malignant” or autosomal recessive osteopetrosis) to the incidental discovery of osteopetrosis on radiographs (e.g., osteopoikilosis), susceptibility to infections, growth failure, and generalized osteosclerosis. If left untreated most of the cases of infantile form osteopetrosis die as a result of bone marrow suppression in the first decade of life. Normocytic anemia is caused either by hypoproliferation of hemopoietic tissue or increased destruction of red cells. Osteopetrosis is a rare cause of anemia. We are presenting here an unusual case that was diagnosed clinically and radiologically as malignant infantile osteopetrosis and was referred to us for the evaluation of anemia and hepatosplenomegaly.

A rare cause of anaemia in multiple myeloma

Multiple myeloma is a neoplastic plasma cell dyscrasia. Anaemia in multiple myeloma is usually related to many factors, of which the commonest cause being anaemia of chronic disease. We reported a 67 years old hypertensive male presented with low back pain and loss of appetite. Investigations revealed severe anaemia and elevated total serum protein with albumin/globulin (A/G) reversal (1:4). Bone marrow aspiration revealed plasma cell dyscrasia. Skeletal survey evaluation showed multiple osteolytic lesions. Serum protein electrophoresis revealed M spike in gamma globulin region with immunofixation suggestive of IgG and kappa monoclonal gammopathy. He was diagnosed as a case of multiple myeloma.. On evaluating the cause of severe anaemia, interestingly various findings suggestive of Autoimmune hemolytic anaemia (AIHA) were found. Packed red blood cell transfusion along with steroids was done for correction of severe anaemia. Review of literature showed that only about 4% of AIHA patients had multiple myeloma. We reported a rare case of multiple myeloma who presented with AIHA.

Rare causes of anemia in liver diseases.

Anemia is a common finding among patients with liver diseases. Patients who suffer from anemia are at a higher risk of liver function decompensation and hospitalization. It affects significantly their quality of life and contributes to mortality. Anemia is present in 70% of patients with liver cirrhosis and with varying incidence accompanies other liver disorders. As the etiology of anemia in liver diseases is multifactorial, various cases represent different clinical entities. Anemia accompanying hepatic disorders can be broadly divided into several types, such as anemia associated with blood loss, as well as aplastic, hemolytic and micronutrient deficiency anemia. However, it is sometimes difficult to delineate between those types in the clinical practice, as several pathophysiological causes can be present in one patient. It is reported that the most common cause of anemia in liver disease is blood loss and iron deficiency. Still, the incidence of unclear cases reaching over 50% suggests that other types of anemia can be underdiagnosed. This review comprehensively describes less frequent types of anemia associated with liver disease, namely hemolytic and aplastic anemia (AA). Hemolytic anemia can complicate autoimmune liver diseases or be a manifestation of membranopathy of red blood cells, dependent on severe hepatic function impairment or alcoholic liver disease. Aplastic anemia is best known as a sequela of viral hepatitis, but some degree of bone marrow inhibition can complicate virtually all advanced liver diseases.

Acquired Hyperzincaemia Due to Zinc-Laden Denture Adhesives Leading to Hypocupraemia as a Cause of Neutropenia

IntroductionCopper deficiency or hypocupraemia is a rare cause of anaemia and neutropenia.Case descriptionWe hereby present the case of a 34-year-old female with gastric bypass surgery who presented with neutropenic fever, abdominal pain and diarrhoea, later found to have extended-spectrum beta-lactamase resistant Escherichia coli urinary tract infection and small bowel bacterial overgrowth syndrome, with her anaemia and neutropenia being caused by copper deficiency due to hyperzincaemia induced by using zinc denture adhesive cream.DiscussionVarious causes of copper deficiency have been recognized including, but not limited to, malnutrition, gastrectomy, gastric bypass surgery, protein-losing enteropathies (coeliac disease, tropical sprue), Wilson disease and Menkes syndrome. Copper deficiency caused by zinc overuse is not very prevalent. The haematologic abnormalities associated with copper deficiency are neutropenia, sideroblastic anaemia and/or pancytopenia.ConclusionBecause of its low prevalence and nonspecific haematologic and clinical manifestations, the diagnosis of zinc-induced copper deficiency (ZICD) can be missed.LEARNING POINTSZinc toxicity is a rare cause of hypocupraemia.The bone marrow and haematological manifestations of copper deficiency are not pathognomonic but include sideroblastic anaemia, pancytopenia or neutropenia.Early detection of hypocupraemia as a cause for neutropenia will improve morbidity and mortality by avoiding untoward infections due to neutropenia, neurological decline or unnecessary interventions.

ABCL-226: Triple Whammy Hemolytic Anemia: Malaria, Cold and Warm Antibody

Introduction Autoimmune hemolytic anemias (AIHAs) are a group of disorders with warm-, cold-, or mixed-reactive antibodies against the red blood cell surface. AIHA is a disorder of hyperfunctioning B lymphocytes associated with immune-related disorders, infections, and other lymphoproliferative disorders. We describe a case of both warm and cold hemolytic anemia associated with diffuse large B cell lymphoma (DLBCL). Case Report A 47-year-old gentleman from Liberia initially presented in October 2014 with severe fatigue. Evaluation revealed pelvic lymph node enlargement, hemoglobin 3.5 g/dl, indirect bilirubin 1.6 mg/dl, and haptoglobin Discussion Immune-mediated hemolytic anemia is a rare cause of anemia in DLBCL. More than 80% of cases are due to warm antibodies, and the incidence of cold antibodies is even rarer. In our case, initially, IgG warm antibodies and C3d cold antibodies were present; over time, IgG disappeared, and C3d persisted. He also had hemolysis secondary to Plasmodium reactivation. Although hemolysis was initially responsive to rituximab monotherapy, upon lymphoma recurrence, treatment with BR regimen resulted in simultaneous lymphoma remission and hemolysis control. Non-immune causes of hemolytic anemia and the recurrence of lymphoma should be considered in patients in whom hemolysis persists after treatment.

Multiorgan failure with abnormal receptor metabolism in mice mimicking Samd9/9L syndromes.

Autosomal dominant sterile α motif domain containing 9 (Samd9) and Samd9L (Samd9/9L) syndromes are a large subgroup of currently established inherited bone marrow failure syndromes that includes myelodysplasia, infection, growth restriction, adrenal hypoplasia, genital phenotypes, and enteropathy (MIRAGE), ataxia pancytopenia, and familial monosomy 7 syndromes. Samd9/9L genes are located in tandem on chromosome 7 and have been known to be the genes responsible for myeloid malignancies associated with monosomy 7. Additionally, as IFN-inducible genes, Samd9/9L are crucial for protection against viruses. Samd9/9L syndromes are caused by gain-of-function mutations and develop into infantile myelodysplastic syndromes associated with monosomy 7 (MDS/-7) at extraordinarily high frequencies. We generated mice expressing Samd9LD764N, which mimic MIRAGE syndrome, presenting with growth retardation, a short life, bone marrow failure, and multiorgan degeneration. In hematopoietic cells, Samd9LD764N downregulates the endocytosis of transferrin and c-Kit, resulting in a rare cause of anemia and a low bone marrow reconstitutive potential that ultimately causes MDS/-7. In contrast, in nonhematopoietic cells we tested, Samd9LD764N upregulated the endocytosis of EGFR by Ship2 phosphatase translocation to the cytomembrane and activated lysosomes, resulting in the reduced expression of surface receptors and signaling. Thus, Samd9/9L is a downstream regulator of IFN that controls receptor metabolism, with constitutive activation leading to multiorgan dysfunction.

Osteopetrosis as a rare cause of anaemia in paediatric patients: a case report

Osteopetrosis as a rare cause of anaemia in paediatric patients: a case report

Autoimmune Hemolytic Anemia in Chronic Myeloid Leukemia from Busulfan to the Era of Tyrosine Kinase Inhibitors

Introduction Autoimmune hemolytic anemia is idiopathic in around half of the patients. However, it is known to be linked in the rest with autoimmune disorders, viral infections, drugs or cancers. The most common associated hematological malignancies are lymphoproliferative disorders, especially chronic lymphocytic leukemia (CLL). The combination of CML and AIHA, on the contrary to CLL, is extremely unusual. In this review, we are going to shed the light on what is known so far about this association, patients' characteristics, how AIHA was treated and to analyze the possible underlying etiology . Results 54 CML patients developed AIHA between 1952 and 2018 and summarized in Table 1. All of them were in the chronic phase of CML except 3 patients: one was in accelerated phase and two were in blast crisis. Different lines of therapy were used for CML: Among the 54 patients, one was treated with focused radiotherapy to the spleen, 3 were treated with Busulfan, 14 with IFNα, 4 were managed with Imatinib and 31 underwent allogenic hematopoietic stem cell transplantation (HSCT). Warm AIHA was confirmed in 40 patients whereas cold agglutinin disease was confirmed in 7 patients and both types were diagnosed in 4 patients. The onset of AIHA was variable with median of 19 months after CML diagnosis in the non-transplant group. The onset of AIHA in the transplant group was earlier with median of 6.5 months. The median of Hemoglobin level at the time of diagnosis of AIHA was 5.9 g/dL (The lowest reported value was 3.4 g/dL and the highest reading was 8 g/dL). It was noteworthy that almost half of the transplant patients had developed viral infections prior to the diagnosis of AIHA (12 due to CMV, 2 due to VZV, 1 due to Parvovirus and 1 due to Influenza virus). From the bone marrow transplanted group, 9 out of 18 cases were diagnosed with AIHA simultaneously with CML relapse. AIHA was treated in 35 cases with Steroids. However, some required second- and third-line treatment with splenectomy (N = 8), IVIg (N = 16), Rituximab (N = 4), Bortezomib (N = 1) and Plasmapheresis (N = 2). More than half of all cases showed improvement with treatment. 14 patients passed away due to different reasons. Discussion The incidence of AIHA in patients with CML is extremely low, but when occurs, it almost always develops after the diagnosis of CML in the chronic phase. The rare occurrence and the variable onset of AIHA in those patients makes it difficult to claim that CML per say is the main culprit. In terms of the possible underlying etiology, patients might be classified into two categories: The hematopoietic stem cell transplant (HSCT) group, which constitutes more than half of the reported cases in this review, and the non-transplant group, which was treated with Busulfan, Interferon or Imatinib. Hemolysis post HCST is rare but well-known complication and is categorized into either alloimmune or autoimmune. The etiology of AIHA in HSCT group is thought to be related either to the donor cell immune reconstitution, concomitant viral infection, or CML relapse. The cause of AIHA in the non-transplant group is thought to be linked to drugs used specifically for CML treatment, despite that the mechanism behind it is poorly understood. Interferon was more frequently reported than both Busulfan and Imatinib combined. It was suggested that hemolysis, in case interferon was used, is mediated either by the formation of immune complexes or through the possible modification of red blood cell surface antigens and production of autoantibodies. New onset severe anemia due to Imatinib is very unusual. It was hypothesized that the mechanism behind it is either because of imatinib effect on hematopoietic stem cells or through an interaction with iron absorption or metabolism, despite that iron replacement didn't improve the outcome. In terms of prognosis, many patients responded well to steroids as first line, but physician should keep in mind that second- and third-line treatment might be required in transplanted patients. Clinician should be aware that despite AIHA is uncommon in CML patients, it should be in the differential diagnosis list for those who develop a sudden drop of hemoglobin without source of bleeding. Conclusion AIHA is a rare cause of anemia in CMl , but when occurs, it develops after the diagnosis of CML in the chronic phase which usually respond to steroids . AIHA that develop in CML patients after transplantation usually required more than steroid therapy to control it. Disclosures No relevant conflicts of interest to declare.

Megaloblastic anemia with pancytopenia in infancy:a rare entity

Megaloblastic anemia in infants present with generalized weakness, failure to thrive, or irritability. Diagnosis is usually centered on complete blood count and peripheral smear, which may show macrocytes, hyper segmented neutrophils, reticulocytopenia and a raised mean corpuscular volume (MCV >100 fl). Pancytopenia has also been noted.We report an exclusively breast fed six months old female child who presented with irritability, poor feeding and developmental delay. Her initial blood picture revealed pancytopenia, with normal MCV. Vitamin B12 levels were found to be reduced. Maternal levels of Vitamin B12 were also found to be borderline low. The child was treated as per protocols and improvement was evidenced with return of hematological parameters to normal and gradual advancement of milestones. The authors wish to underscore the importance ofMegaloblastic anemia as an important and rare cause of anemia in infancy. Early recognition and treatment are imperative to prevent catastrophic sequelae.

Evaluation of musculoskeletal manifestations of hemoglobino-pathies by conventional imaging

Introduction: The hemoglobinopathies are a group of genetic disorders in which there is alteration of hemoglobin structure. These hematologic disorders of the skeleton produce radiologic appearances related to marrow infiltration and/or replacement. Common findings are in place of such as osteopenia with trabecular prominence, growth disturbances, and extramedullary hematopoiesis. Conventional Radiography is required for knowing the imaging appearances of sickle cell disease, thalassemia, and other rare causes of anemia. Objectives: To study the role of conventional imaging in detecting various radiological manifestations of hemaglobinopathies. Materials & Methods: An observational descriptive study of 21 cases with hemaglobinopathies was done by conventional radiography & its varied musculoskeletal manifestations were studied. Result: Most common site of involvement was head of femur which was seen in 16 cases out of which isolated involvement was seen in 10 cases. Rest of the cases was associated with spine, skull and upper limb manifestations. Conclusion: Knowledge of the distribution and site of involvement and its musculoskeletal features is of considerable diagnostic importance in the evaluation and treatment of hemoglobinopathies. Conventional Radiography is required and its primary investigation modality It is important to know the extent of the range of radiologic appearances in order to accurately diagnose complications and to provide appropriate treatment.

A Case Report on Acquired Pure Red Cell Aplasia in a Patient of Rheumatoid Arthritis

Pure red cell aplasia (PRCA) is a clinical syndrome defined by the absence of mature erythroid precursors in another wise normocellular bone marrow. PRCA is considered to be an extra articular manifestation of rheumatoid arthritis and is very rare cause of anemia in Rheumatoid Arthritis with only very few cases reported till now. Herein we report a case of 52 year old female with long standing untreated seropositive arthritis who presented to us with severe symptomatic anemia. After proper work up including bone marrow examination, her findings were consistent with pure red cell aplasia.

P-215: Evans syndrome: a rare cause of anemia in the elderly

P-215: Evans syndrome: a rare cause of anemia in the elderly

Idiopathic pulmonary hemosiderosis - a diagnostic challenge

Idiopathic pulmonary hemosiderosis is a rare disorder that can occur at any age and is characterized by the triad of hemoptysis, iron deficiency anemia and diffuse pulmonary infiltrates. The clinical course is exceedingly variable especially in children and a substantial proportion of this age group is undiagnosed. It is probably due to the fact that iron deficiency anemia may be the first and the only manifestation of IPH, preceding other symptoms and signs by several months and IPH is not considered as a rare cause of anemia, unless the typical triad is present. We present a case of IPH in a 13-year-old girl, treated for several months with persistent iron deficiency anemia, without response to therapy.

Watermelon stomach: a rare cause of anemia in patients with end-stage renal disease

Watermelon Stomach or gastric antral vascular ectasia (GAVE) is a rare cause of chronic gastrointestinal blood loss. It has been commonly associated with autoimmune connective tissue and chronic liver disease. It is characterized by endoscopic appearances of erythematous patches secondary to dilated vessels that can be diffusely located or in linear stripes. Histology is characterized by dilated mucosa capillaries with fibrin thrombi and fibromuscular hyperplasia. Unless recognized, the endoscopic findings can be misdiagnosed as severe gastritis. GAVE can be treated using various modalities with variable success. We report four cases of GAVE as the cause of chronic gastrointestinal blood loss in patients with end stage renal disease. A literature review of this rare and interesting entity is presented.

Cooper deficiency anemia: A complication after bariatric surgery

Copper is a trace mineral essential to hematopoiesis and to the structure and function of the nervous system. Copper acts as a ligand to ferroxidase II, which oxidizes iron, allowing it to be mobilized and transported from the hepatic stores to the bone marrow to be used in erythropoiesis..Copper deficiency is a rare cause of anemia, leukopenia, and myeloneuropathy. In this report we describe a case of a 49 year-old overweight Caucasian woman who was referred June 2008 to the Obesity Outpatient Clinic at UNIFESP for evaluation of muscular pain and weakness in both legs, fatigue and chronic anemia. The past medical history included Scopinaro Surgery for severe obesity in November 2004 when her weight was approximately 180 Kg (BMI 68.6 kg/m2). She lost considerable weight after surgery, with her weight being stable around 80 kg (BMI 30). Because of clinical manifestations of sub nutrition, protein and vitamin deficiencies and severe anemia, which lead to several hospital admissions, in February 2008 she was submitted to another surgery from conversion of the gastrointestinal surgical Scopinaro procedure to that performed by Capella. She also had a history of hypothyroidism, hyperuricemia and panic syndrome, requiring specific medications, beyond parenteral iron and B12 vitamin therapies for anemia. Physical examination revealed a bilateral edema and no other abnormality. Laboratory tests revealed hypoalbuminemia and hypochromic megaloblastic anemia with normal serum levels of B12 vitamin, iron and ferritin. Due to the persistence of the neurologic symptoms and anemia in spite of adequate iron and B12 vitamin therapies, the hypothesis of copper deficiency was considered and a low serum level of 40μg/dl was found. An oral supplementation with high doses of copper resulted in evident clinical and laboratorial improvements after eight weeks of therapy. The diagnosis of copper deficiency has to be part of the differential diagnosis in patients with unexplained neurologic symptoms, anemia, and leukopenia, especially in those who underwent to previous gastrointestinal surgery.

Re: case report “Idiopathic pulmonary haemosiderosis: A rare cause of anaemia”

The Journal is the primary organ of Continuing Paediatric Medical Education in Sri Lanka. The journal also has a website. Free full text access is available for all readers.The Sri Lanka Journal of Child Health is now indexed in SciVerse Scopus (Source Record ID 19900193609), Index Medicus for South-East Asia Region (IMSEAR), CABI (Centre for Agriculture and Bioscience International Global Health Database), DOAJ and is available in Google, as well as Google Scholar.The policies of the journal are modelled on the Committee on Publication Ethics (COPE) Guidelines on Principles of Transparency and Best Practice in Scholarly Publishing. Sri Lanka Journal of Child Health is recognised by the International Committee of Medical Journal Editors (ICMJE) as a publication following the ICMJE Recommendations.

Copper deficiency anemia after bariatric surgery

ABSTRACTCopper is a trace mineral essential to hematopoiesis and to the structure and function of the nervous system. Copper acts as a ligand to ferroxidase II, which oxidizes iron, allowing it to be mobilized and transported from the hepatic stores to the bone marrow to be used in erythropoiesis..Copper deficiency is a rare cause of anemia, leukopenia, and myeloneuropathy. In this report we describe a case of a 49 year-old overweight Caucasian woman who was referred June 2008 to the Obesity Outpatient Clinic at UNIFESP for evaluation of muscular pain and weakness in both legs, fatigue and chronic anemia. The past medical history included Scopinaro Surgery for severe obesity in November 2004 when her weight was approximately 180 Kg (BMI 68.6 kg/m2). She lost considerable weight after surgery, with her weight being stable around 80 kg (BMI 30). Because of clinical manifestations of sub nutrition, protein and vitamin deficiencies and severe anemia, which lead to several hospital admissions, in February 2008 she was submitted to another surgery from conversion of the gastrointestinal surgical Scopinaro procedure to that performed by Capella. She also had a history of hypothyroidism, hyperuricemia and panic syndrome, requiring specific medications, beyond parenteral iron and B12 vitamin therapies for anemia. Physical examination revealed a bilateral edema and no other abnormality. Laboratory tests revealed hypoalbuminemia and hypochromic megaloblastic anemia with normal serum levels of B12 vitamin, iron and ferritin. Due to the persistence of the neurologic symptoms and anemia in spite of adequate iron and B12 vitamin therapies, the hypothesis of copper deficiency was considered and a low serum level of 40μg/dl was found. An oral supplementation with high doses of copper resulted in evident clinical and laboratorial improvements after eight weeks of therapy. The diagnosis of copper deficiency has to be part of the differential diagnosis in patients with unexplained neurologic symptoms, anemia, and leukopenia, especially in those who underwent to previous gastrointestinal surgery.

Preemptive intravenous immunoglobulin allows safe and timely administration of antineoplastic therapies in patients with multiple myeloma and parvovirus B19 disease

Parvovirus B19 (B19) disease is a rare cause of anemia in cancer patients and often goes unrecognized, causing delays in anticancer therapy. A retrospective review was carried out of the records of patients with multiple myeloma who underwent melphalan-based autologous stem cell transplantation (MEL-ASCT) and developed B19 infection (January 2009-December 2011). Cases were defined by the presence of clinical and laboratory findings consistent with B19 disease in patients with repeatedly positive plasma quantitative polymerase chain reaction for parvovirus. Six patients qualified as cases; 5 presented with trilineage cytopenias (chronic in 1) and 1 with anemia later progressing to pancytopenia. Transfusion-dependent thrombocytopenia led to testing in 5 patients. Two of these patients also had manifestations of autoimmune disease. Therapy with intravenous immunoglobulin (IVIG) resulted in clinical and hematologic response in all; however, 1 patient, whose white blood cell counts and serum hemoglobin levels improved, required splenectomy for persistent thrombocytopenia. All patients required additional IVIG for recurrent B19 disease. Although viral load at diagnosis did not correlate with the severity of cytopenia, its decrease was associated with response during 17 of 20 evaluable episodes (P=0.02). Preemptive IVIG allowed the safe administration of chemotherapy in 3 patients, including MEL-ASCT in 1. Parvovirus B19 can cause severe disease in myeloma patients including ASCT recipients. Thrombocytopenia - not anemia - was the leading presentation and may be associated with autoimmune conditions. Patients with unexplained cytopenias, particularly when prolonged, should undergo testing for circulating parvovirus. A reduction in viral load was associated with response to IVIG, although additional therapy was needed for recurrent disease. Most importantly, preemptive IVIG allowed for safe and timely administration of antineoplastic therapy in patients with ongoing B19 disease.

Autoimmune hemolytic anemia in a patient with Malaria

Autoimmune Hemolytic Anemia (AIHA), a very infrequent condition which represents a group of disorders in which presence of autoantibodies directed against self-antigens leads to shortened red cell survival. Till date, a very few cases of AIHA in Malaria patients are reported worldwide but still AIHA should be considered a relatively rare cause of anemia in malaria. A 20 year male presented with intermittent fever since seven days and yellowish discoloration of urine and sclera since 5 days. He was transfused three units of blood at a private clinic before one month. On examination, pallor, icterus and spelnomegaly were present. Hemoglobin (Hb) was 3.2 gm% and peripheral smear revealed ring forms of both Plasmodium vivax and Plasmodium falciparum. Serum LDH and Serum billirubin (Indirect and Direct) were high. This patient’s blood group was B +ve with positive autocontrol. Indirect Antiglobulin Test (IAT), antibody screening and antibody identification were pan-positive with reaction strength of +4 against each cell. Direct Antiglobulin Test was +4 positive anti IgG and negative with anti C3. He was treated with Artesunate and methylprednisone. Least incompatible, saline washed O Neg and B neg red cells were transfused on the 2nd day of starting treatment. Hb was raised to 6.1 gm% on 4th day. Patient was discharged on 9th day with Hb 7.0 gm% with oral tapering dose of steroids. In the above case, patient was suffering from high grade malarial parasitemia with co-existing autoimmune RBC destruction by IgG auto-antibodies which led to sudden drop in Hb and rise in serum LDH and indirect billirubin. Least incompatible packed red cells along with antimalarials and steroids led to clinical improvement. So far, one case report each from India, Korea, Canada and Germany and one case series report of three cases from India have been reported. Under-reporting or rarity of this phenomenon may be accountable for this.