Bruns Lina, Völker Linus, Klamroth Robert, Kuhlmann Martin K, Jabs Wolfram J
Department of Nephrology, Vivantes Clinic in Friedrichshain, Berlin.
Department II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University Hospital Cologne, Cologne, and.
Clin Nephrol Case Stud. 2023 Dec 12;11:147-153. doi: 10.5414/CNCS111193. eCollection 2023.
Thrombocytopenia is always of concern when encountered in emergency settings. We report a case of a 29-year-old women in whom a unique constellation of hematological disorders occurred. The patient had been diagnosed with idiopathic immune thrombocytopenia (ITP) in 2007, with a history of several thrombocytopenic flares. She now presented with homonymous hemianopia accompanied by thrombocytopenia and microangiopathic hemolytic anemia (MAHA) and was soon after diagnosed with a posterior stroke. Symptoms were more reminiscent of acquired thrombotic thrombocytopenic purpura (aTTP) rather than ITP. Immediate treatment with plasma exchange and caplacizumab curtailed MAHA, and progressive ischemic disease was averted. ADAMTS-13 testing confirmed the diagnosis of immune-mediated aTTP. Repeated testing for ITP, however, also showed IgG-loaded thrombocytes with the former known anti-GPIIb/IIIa specificity. Furthermore, autoimmune hemolytic anemia (AIHA) could be detected by direct antiglobulin test showing IgG and complement loading of the patient's erythrocytes. The autoimmune background of all three entities suggested an underlying systemic disease. Indeed, systemic lupus erythematosus (SLE) serology was strongly positive allowing for the diagnosis of SLE. ITP and AIHA as well as aTTP can be secondary to SLE, but emergence of all three disorders has not been reported at the same time.
在急诊情况下遇到血小板减少症时总是令人担忧。我们报告一例29岁女性病例,该患者出现了一系列独特的血液系统疾病。该患者于2007年被诊断为特发性免疫性血小板减少症(ITP),有多次血小板减少发作史。她现在出现同向性偏盲,伴有血小板减少和微血管病性溶血性贫血(MAHA),不久后被诊断为后循环卒中。症状更符合获得性血栓性血小板减少性紫癜(aTTP)而非ITP。立即进行血浆置换和卡泊单抗治疗控制了MAHA,避免了进行性缺血性疾病。ADAMTS-13检测确诊为免疫介导的aTTP。然而,对ITP的反复检测也显示血小板上有IgG负载,具有先前已知的抗GPIIb/IIIa特异性。此外,直接抗球蛋白试验可检测到自身免疫性溶血性贫血(AIHA),显示患者红细胞上有IgG和补体负载。所有这三种疾病的自身免疫背景提示存在潜在的系统性疾病。事实上,系统性红斑狼疮(SLE)血清学呈强阳性,从而确诊为SLE。ITP、AIHA以及aTTP都可能继发于SLE,但同时出现这三种疾病的情况尚未见报道。