Zhao Jing, Chen Xilong, Yue Shixia
Gansu Provincial Maternity and Child-care Hospital (Gansu Provincial Central Hospital), Lanzhou, China.
Front Pediatr. 2025 Aug 14;13:1635534. doi: 10.3389/fped.2025.1635534. eCollection 2025.
Hemophagocytic syndrome (HPS), also known as hemophagocytic lymphohistiocytosis (HLH), is a life-threatening disorder that can occur in both children and adults, closely associated with genetic factors and acquired immune dysregulation. This article presents a case report of HLH with NBAS gene mutation and chronic active Epstein-Barr virus (EBV) infection. Despite undergoing a series of aggressive treatments, the patient failed to achieve a favorable clinical response. The clinical course was complicated by hepatic injury, coagulopathy, progressive cytopenia in all three blood cell lineages, and significant elevation of serum ferritin, ultimately resulting in fatal outcome. Current research on genetic predisposing factors has identified 17 causative genes for HLH, including PRF1 and UNC13D. However, NBAS-related cases have been rarely reported. The discovery of additional potential pathogenic genes holds significant value for advancing diagnostic and therapeutic approaches in HLH management.
噬血细胞综合征(HPS),也称为噬血细胞性淋巴组织细胞增生症(HLH),是一种危及生命的疾病,可发生于儿童和成人,与遗传因素和获得性免疫失调密切相关。本文报告一例伴有NBAS基因突变和慢性活动性EB病毒(EBV)感染的HLH病例。尽管接受了一系列积极治疗,患者仍未获得良好的临床反应。临床过程中出现肝损伤、凝血功能障碍、全血细胞进行性减少以及血清铁蛋白显著升高,最终导致死亡。目前关于遗传易感因素的研究已确定了17个HLH致病基因,包括PRF1和UNC13D。然而,与NBAS相关的病例报道很少。发现更多潜在致病基因对推进HLH治疗中的诊断和治疗方法具有重要价值。