University of Health Sciences, Diskapi Yildirim Beyazit Training and Research Hospital, Department of Hematology, Ankara, Turkey.
University of Health Sciences, Diskapi Yildirim Beyazit Training and Research Hospital, Department of Hematology, Ankara, Turkey.
Transfus Apher Sci. 2020 Aug;59(4):102747. doi: 10.1016/j.transci.2020.102747. Epub 2020 Feb 28.
Hemophagocytic Lymphohistiocytosis (HLH) is a reactive disorder of the mononuclear phagocytic system characterized by increased histiocytic proliferation, activation and hemaphagocytosis. The underlying etiology may be genetic (primary) or acquired (secondary). Secondary causes include drugs, autoimmune diseases, malignancies and infections of which EBV is the most common. A 28-year old male patient who was a shepherd with no known concomitant comorbid disease was admitted to the Emergency Department with the complaints of abdominal pain, fever, severe fatigue. Physical examination revealed high fever, hepatosplenomegaly and laboratory examination revealed pancytopenia, hyperferritinemia and hypertriglyceridemia. Hemophagocytes were observed in the bone marrow biopsy and the patient was diagnosed as HLH. The patient was treated with cyclosporine A, dexamethasone, intravenous immunoglobulin (IvIg) and etoposide according to the HLH 2004 protocol. Coxiella burnetii was detected in the serological evaluation of the etiology and doxycycline was added to the current treatment. Fever was controlled in the second week of the treatment and the patient was discharged after complete recovery of the cytopenia in the fourth week. In the outpatient setting, treatment was completed in 8 weeks and follow-up of the patient is still ongoing without medication. To the best of our knowledge, this is the first case from Turkey of HLH secondary to Q-fever which was treated and managed successfully. Since the mortality of HLH is quite high, the etiology should be determined as soon as possible to be able to provide appropriate treatment.
噬血细胞性淋巴组织细胞增生症(HLH)是一种单核吞噬细胞系统的反应性疾病,其特征为组织细胞增生、活化和噬血作用增强。其潜在病因可能是遗传的(原发性)或获得性的(继发性)。继发性病因包括药物、自身免疫性疾病、恶性肿瘤和感染,其中 EBV 最为常见。一名 28 岁男性患者,作为一名牧羊人,无已知并存的合并症,因腹痛、发热、严重疲劳而到急诊就诊。体格检查发现高热、肝脾肿大,实验室检查发现全血细胞减少、铁蛋白血症和高甘油三酯血症。骨髓活检中观察到噬血细胞,患者被诊断为 HLH。根据 HLH 2004 方案,患者接受环孢素 A、地塞米松、静脉注射免疫球蛋白(IvIg)和依托泊苷治疗。病因学的血清学评估中检测到柯克斯体,在当前治疗中加入多西环素。治疗的第二周,发热得到控制,第四周全血细胞减少完全恢复后,患者出院。在门诊环境中,8 周完成治疗,患者仍在继续随访,无需用药。据我们所知,这是土耳其首例 Q 热引起的成功治疗和管理的 HLH 病例。由于 HLH 的死亡率相当高,应尽快确定病因,以便提供适当的治疗。