Takehara Runa, Ebihara Itaru, Honda Yoshifumi, Ooba Norimasa, Kurosawa Hiromi, Sato Chihiro, Ohtani Haruo, Tsutsumi Yutaka, Nose Masato, Kobayashi Masaki
Department of Nephrology, Mito Saiseikai General Hospital, 3-3-10 Futabadai, Mito, Ibaraki, 311-4198, Japan.
Ooba Internal Medicine Clinic, Ibaraki, Japan.
CEN Case Rep. 2025 Jun;14(3):477-485. doi: 10.1007/s13730-024-00950-7. Epub 2024 Dec 12.
Visceral disseminated varicella zoster virus (VZV) infection is a severe complication, characterized by a notably high mortality rate. Herein, we present a case of a 36-year-old-man involving visceral disseminated VZV infection that emerged during remission induction therapy involving high-dose prednisolone (PSL), mycophenolate mofetil (MMF), and hydroxychloroquine for lupus nephritis. Two months after starting the immunosuppressive therapy, he experienced a rapid onset of severe upper abdominal pain. The following day, clinical manifestations and laboratory abnormalities rapidly deteriorated. Hyperferritinemia and hypertriglyceridemia, indicative of hemophagocytic lymphohistiocytosis (HLH), emerged, along with escalating liver and renal impairment and newly appeared disseminated intravascular coagulation, and multiple organ failure is suggested. The patient developed widespread blistering predominantly on the trunk and face, patient's condition failed to ameliorate, ultimately culminating in his demise a few hours later. At autopsy, the cutaneous lesions with blisters revealed positive immunostaining with anti-VZV antibody, and similar findings were detected in multiple organs. HLH was confirmed in lymph nodes. It is crucial to emphasize the awareness of visceral disseminated VZV, particularly in cases patients are undergoing concurrent PSL therapy alongside MMF for SLE. The progression of this fatal condition usually begins with abdominal pain, followed by a skin rash a few days later. The present case is the first to show evidence of HLH occurring as a result of visceral disseminated VZV infection. This disease is extremely rare but extremely serious, therefore, VZV-DNA should be measured in cases where you suspect this disease for early diagnosis and treatment.
内脏播散性水痘带状疱疹病毒(VZV)感染是一种严重并发症,死亡率显著较高。在此,我们报告一例36岁男性内脏播散性VZV感染病例,该感染发生在狼疮性肾炎缓解诱导治疗期间,使用了大剂量泼尼松龙(PSL)、霉酚酸酯(MMF)和羟氯喹。开始免疫抑制治疗两个月后,他迅速出现严重上腹部疼痛。次日,临床表现和实验室异常迅速恶化。出现了提示噬血细胞性淋巴组织细胞增生症(HLH)的高铁蛋白血症和高甘油三酯血症,同时肝肾功能损害不断加重,新出现了弥散性血管内凝血,提示多器官功能衰竭。患者躯干和面部出现广泛水疱,病情未能改善,最终数小时后死亡。尸检时,水疱性皮肤病变抗VZV抗体免疫染色呈阳性,多个器官也发现类似结果。淋巴结确诊为HLH。必须强调对内脏播散性VZV的认识,特别是在系统性红斑狼疮患者同时接受PSL和MMF治疗的情况下。这种致命疾病的进展通常始于腹痛,几天后出现皮疹。本病例是首例显示因内脏播散性VZV感染导致HLH的证据。这种疾病极其罕见但极其严重,因此,在怀疑该病时应检测VZV-DNA以进行早期诊断和治疗。