Munasinghe B M, Arambepola A G, Pathirage N, Fernando U P M, Subramaniam N, Nimalan S, Gajanthan T
Department of Anaesthesia and Intensive Care, District General Hospital, Mannar, Sri Lanka.
National Blood Transfusion Service, Colombo, Sri Lanka.
Case Rep Infect Dis. 2021 Jul 8;2021:3451155. doi: 10.1155/2021/3451155. eCollection 2021.
Secondary haemophagocytic lymphohistiocytosis (sHLH), often associated with an array of infections, malignancies, and autoimmune diseases, is rarely seen with leptospirosis, which carries a relatively poor prognosis even with modern state-of-the-art medical care. We describe a patient with leptospirosis complicated by sHLH who succumbed to illness following multiorgan dysfunction. . A 74-year-old farmer presented with high-grade, unsettling fever for a week. Muddy water exposure and suggestive symptoms prompted investigation and management in the line of leptospirosis (IV ceftriaxone was instituted, and later, MAT (microscopic agglutination test) became positive). Subsequently, he developed severe acute hypoxemia requiring mechanical ventilation and acute renal failure requiring renal replacement therapy. Bone marrow biopsy and markedly elevated serum ferritin and triglyceride levels done on day 10 (with unresolving fever, hepatosplenomegaly, and pancytopaenia) confirmed the diagnosis of HLH. The routine cultures, retroviral studies, CMV, dengue, hanta and mycoplasma antibodies, tuberculosis and COVID-19 PCR, and malaria screening were all normal. There was no improvement of hypoxemia following intravenous methylprednisolone. He died on day 15 despite escalating organ support.
Leptospirosis is a common zoonotic disease in the tropics with significant morbidity and mortality. In the case of severe leptospirosis, overlapping clinical features with sHLH make the diagnosis of the latter challenging. No assessment tools are available to date to predict the risk of developing sHLH in a patient having leptospirosis. Outcome following sHLH due to leptospirosis still remains majorly ominous. A high index of suspicion and low threshold for specific investigations could possibly alter the outcome following such an occurrence.
继发性噬血细胞性淋巴组织细胞增生症(sHLH)常与一系列感染、恶性肿瘤和自身免疫性疾病相关,与钩端螺旋体病并发者罕见,即便采用现代先进医疗手段,其预后仍相对较差。我们报告一例钩端螺旋体病并发sHLH的患者,该患者因多器官功能障碍而病故。一名74岁农民,持续高热一周,令人不安。接触过浑水且有相关症状,促使对其进行钩端螺旋体病方面的检查与治疗(开始静脉注射头孢曲松,随后显微镜凝集试验(MAT)呈阳性)。随后,他出现严重急性低氧血症,需要机械通气,还出现急性肾衰竭需要肾脏替代治疗。在第10天进行的骨髓活检以及血清铁蛋白和甘油三酯水平显著升高(伴有持续发热、肝脾肿大和全血细胞减少),确诊为HLH。常规培养、逆转录病毒研究、巨细胞病毒、登革热、汉坦病毒和支原体抗体检测、结核病及新冠病毒PCR检测以及疟疾筛查结果均正常。静脉注射甲泼尼龙后低氧血症无改善。尽管不断加强器官支持,他仍于第15天死亡。
钩端螺旋体病是热带地区常见的人畜共患病,发病率和死亡率都很高。在严重钩端螺旋体病病例中,与sHLH重叠的临床特征使得后者的诊断具有挑战性。目前尚无评估工具可预测钩端螺旋体病患者发生sHLH的风险。钩端螺旋体病所致sHLH的预后仍然大多不容乐观。高度的怀疑指数和较低的特异性检查门槛可能会改变此类情况发生后的结局。