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일차성쇼그렌증후군 환자에서 폐결핵 치료 중 발생한 Rifampin 유발 면역용혈성빈혈
석진우,김양기,장계일,정현석,어수택,김기업,구소미,이보영,노현진,신우용,신정원,진소영 대한수혈학회 2019 大韓輸血學會誌 Vol.30 No.3
Drug-induced immune hemolytic anemia is a rare disease that occurs in 1 in 1 million individuals of the general population. Rifampin-induced immune hemolytic anemia is caused by drug-dependent antibodies and this can be treated without complication by drug cessation. Herein, we present a case of rifampin-induced immune hemolytic anemia in a patient with primary Sjogren’s syndrome (pSS) which occurred during treatment of pulmonary tuberculosis. At admission, the patient’s laboratory tests revealed hemolytic anemia and positive direct antiglobulin test result. Since the incidence of autoimmune hemolytic anemia (AIHA) in pSS is reported to be 3 percent, which is higher than that of the general population, differential diagnosis between AIHA and rifampin-induced immune hemolytic anemia was required for planning future anti-tuberculous treatment. We identified rifampin-dependent antibody by drug-induced immune complex test and diagnosed rifampin-induced immune hemolytic anemia. Based on this experience, if rifampin administration is considered in patients with systemic autoimmune disease such as pSS, which has a high incidence of AIHA, we suggest evaluating the presence and the cause of hemolytic anemia at baseline by testing serum lactate dehydrogenase, haptoglobin, and direct and indirect antiglobulin tests before drug administration to promptly identify the cause of hemolysis if hemolytic anemia develops. (Korean J Blood Transfus 2019;30:246-252)
Quetiapine Induced Autoimmune Hemolytic Anemia in a Child Patient: A Case Report
Asiye Arıcı,Hatice Altun,Can Acıpayam 대한정신약물학회 2018 CLINICAL PSYCHOPHARMACOLOGY AND NEUROSCIENCE Vol.16 No.4
Autoimmune hemolytic anemia is a disease characterized with destruction of erythrocytes as a result of antibody produce against patient’s own erythrocytes and anemia. Autoimmune hemolytic anemia can be roughly stratified into two groups according to serological features and secondary causes including drugs induced hemolytic anemia. Drugs induced autoimmune hemolytic anemia is very rare in pediatric patients. Even though hematological side effects such as leucopenia, agranulocytosis, eosinophilia, thrombocytopenic purpura and aplastic anemia might occur due to psychotropic drug use; to the best of our knowledge there is no autoimmune hemolytic anemia case due to quetiapine, an atypical antipsychotics, in literature. We hereby describe the first child case of autoimmune hemolytic anemia during quetiapine treatment.We also are pointing out that one should keep in mind serious hematological side effects with atypical antipsychotic drug use with this case report.
Raghuveer Prabhu,Renjitha Bhaskaran,Veena Shenoy,Rema G,Neeraj Sidharthan 대한혈액학회 2016 Blood Research Vol.51 No.2
BackgroundAutoimmune hemolytic anemia (AIHA) is a less recognized, potentially fatal condition. There is a scarcity of data on clinicoserological characteristics and response to therapy concerning this disease from South India.MethodsData for 33 patients with primary AIHA recorded from July 2009 to June 2015 were retro-spectively analyzed for clinical presentation, response to frontline therapy, durability of response, time to next treatment (TTNT), and response to second-line agents.ResultsThe median follow-up period was 50 months. Among 33 patients, 48% of the cases were warm autoimmune hemolytic anemia (WAIHA), 46% were cold agglutinin disease (CAD), and 6% were atypical. Three-fourth of patients had severe anemia (<8 g/dL hemoglobin [Hb]) at onset; younger patients (age <40 yr) had more severe anemia. All of the patients who required treatment received oral prednisolone at 1.5 mg/kg/d as a frontline therapy, and the response rate was 90% (62% complete response [CR] and 28% partial response [PR]). The overall response to corticosteroids in WAIHA and CAD was 87% and 92%, respectively. The median corticosteroid duration was 14 months, and 50% of the patients required second-line agents. Fourteen patients received azathioprine as a second-line agent, and 11 of these patients responded well, with half of them not requiring a third agent. Four patients developed severe infections (pneumonia, sepsis, and soft tissue ab-scess) and two had life-threatening venous thrombosis. One case of death was recorded.ConclusionAIHA is a heterogeneous disease that requires care by physicians experienced in treating these patients.
Atypical Hemolytic Uremic Syndrome after Traumatic Rectal Injury: A Case Report
( Ji-hyoun Kang ),( Donghyun Lee ),( Yunchul Park ) 대한외상학회 2021 大韓外傷學會誌 Vol.34 No.4
Atypical hemolytic uremic syndrome (aHUS) is a rare, progressive, life-threatening condition of thrombotic microangiopathy characterized by thrombocytopenia, microangiopathic hemolytic anemia, and renal impairment. The mechanisms underlying aHUS remain unclear. Herein, we present the first case in the literature of aHUS after a traumatic injury. A 55-year-old male visited the emergency department after a traumatic injury caused by a tree limb. Abdominal computed tomography revealed a rectal wall defect with significant air density in the perirectal space and preperitoneum, implying rectal perforation. Due to the absence of intraperitoneal intestinal perforation, we performed diverting sigmoid loop colostomy. An additional intermittent simple repair was performed due to perianal and anal injuries. One day postoperatively, his urine output abruptly decreased and serum creatinine level increased. His platelet level decreased, and a spiking fever occurred after 2 days. The patient was diagnosed with acute renal failure secondary to aHUS and was treated with fresh frozen plasma replacement. Continuous renal replacement therapy (CRRT) was also started for oliguria and uremic symptoms. The patient received CRRT for 3 days and intermittent hemodialysis thereafter. After hemodialysis and subsequent supportive treatment, his urine output and renal function improved. The hemolytic anemia and thrombocytopenia also gradually improved. Dialysis was terminated on day 22 of admission and the patient was discharged after recovery. This case suggests that that a traumatic event can trigger aHUS, which should be considered in patients who have thrombocytopenia and acute renal failure with microangiopathic hemolytic anemia. Early diagnosis and appropriate management are critical for favorable outcomes.
스테로이드에 반응을 보이지 않은 자가면역 용혈 빈혈을 동반한 궤양성 대장염 1예
최현종,홍수진,김영지,고봉민,이문성,심찬섭,김현정,홍대식 대한소화기학회 2008 대한소화기학회지 Vol.51 No.2
Autoimmunity is thought to play a central role in the pathogenesis of inflammatory bowel disease and associated extraintestinal manifestations. Autoimmune hemolytic anemia associated with ulcerative colitis is a rare occurrence. No more than 50 cases have been described in the international literatures, and only 2 cases reported in Korea. A 29-year-old woman who was diagnosed as ulcerative colitis two years ago was complicated with autoimmune hemolytic anemia, and did not respond to steroid therapy. Ultimately, total colectomy and splenectomy were carried out for the treatment of ulcerative colitis and hemolytic anemia. After the operation, anemia was resolved. We present the case with a review of literature. (Korean J Gastroenterol 2008;51:137-141)
( Hee Sup Kim ),( Sook Hyang Jeong ),( Je Hyuck Jang ),( Hyung Joon Myung ),( Jin Wook Kim ),( Soo Mee Bang ),( Sang Hoon Song ),( Haer Young Kim ),( Hae Sun Yun ) 대한간학회 2011 Clinical and Molecular Hepatology(대한간학회지) Vol.17 No.4
A 37-year-old male presented with fever and jaundice was diagnosed as hepatitis A complicated with progressive cholestasis and severe autoimmune hemolytic anemia. He was treated with high-dose prednisolone (1.5 mg/kg), and eventually recovered. His initial serum contained genotype IA hepatitis A virus (HAV), which was subsequently replaced by genotype IIIA HAV. Moreover, at the time of development of hemolytic anemia, he became positive for immunoglobulin M (IgM) anti-hepatitis E virus (HEV). We detected HAV antigens in the liver biopsy specimen, while we detected neither HEV antigen in the liver nor HEV RNA in his serum. This is the first report of hepatitis A coinfected with two different genotypes manifesting with autoimmune hemolytic anemia, prolonged cholestasis, and false-positive IgM anti-HEV. (Korean J Hepatol 2011;17:323-327)
Cytomegalovirus-Associated Severe Direct Antiglobulin Test Negative Hemolytic Anemia: A Case Report
홍현정,조영혜,임재영,염정숙,박지숙,박은실,서지현,우향옥,윤희상 대한소아혈액종양학회 2018 Clinical Pediatric Hematology-Oncology Vol.25 No.2
Cytomegalovirus is a common virus that is mostly asymptomatic when infected, but rarely causes life-threatening hemolysis especially in immunocompromised children. We report a case of antiglobulin test negative severe hemolytic anemia caused by cytomegalovirus infection developed in an immune competent 9-year-old girl. The patient’s hemoglobin level was 4.8 g/dL on the day of admission. The diagnosis was achieved by exclusion of other causes of hemolytic anemia and serological evidence of recent CMV infection. The patient was successfully treated with anti-viral agents and steroids resulting in recovery from anemia. Clinicians should consider cytomegalovirus infection in the differential diagnosis of hemolytic anemia in pediatric patients.
Park, Chul Min,Lee, Kyunghoon,Jun, Sun-Hee,Song, Sang Hoon,Song, Junghan Elsevier 2017 Journal of chromatography. B, Analytical technolog Vol.1060 No.-
<P><B>Abstract</B></P> <P>Deficiencies in erythrocyte metabolic enzymes are associated with hereditary hemolytic anemia. Here, we report the development of a novel multiplex enzyme assay for six major enzymes, namely glucose-6-phosphate dehydrogenase, pyruvate kinase, pyrimidine 5′-nucleotidase, hexokinase, triosephosphate isomerase, and adenosine deaminase, deficiencies in which are implicated in erythrocyte enzymopathies. To overcome the drawbacks of traditional spectrophotometric enzyme assays, the present assay was based on ultra-performance liquid chromatography-tandem mass spectrometry (UPLC–MS/MS). The products of the six enzymes were directly measured by using ion pairing UPLC–MS/MS, and the precision, linearity, ion suppression, optimal sample amounts, and incubation times were evaluated. Eighty-three normal individuals and 13 patients with suspected enzymopathy were analyzed. The UPLC running time was within 5min. No ion suppression was observed at the retention time for the products or internal standards. We selected an optimal dilution factor and incubation time for each enzyme system. The intra- and inter-assay imprecision values (CVs) were 2.5–12.1% and 2.9–14.3%, respectively. The linearity of each system was good, with R<SUP>2</SUP> values >0.97. Patient samples showed consistently lower enzyme activities than those from normal individuals. The present ion paring UPLC–MS/MS assay enables facile and reproducible multiplex evaluation of the activity of enzymes implicated in enzymopathy-associated hemolytic anemia.</P>