Maeba Shinji, Hasegawa Shunji, Shimomura Maiko, Ichimura Takuya, Takahashi Kazumasa, Motoyama Masashi, Fukunaga Shinnosuke, Ito Yoshinori, Ichiyama Takashi, Ohga Shouichi
Department of Pediatrics, Yamaguchi University Graduate School of Medicine, Yamaguchi, Japan.
Department of Pediatrics, Nagoya University Graduate School of Medicine, Aichi, Japan.
AJP Rep. 2015 Oct;5(2):e089-92. doi: 10.1055/s-0034-1398392. Epub 2015 Apr 6.
Background Herpes simplex virus (HSV) infection carries one of the poorest outcomes of neonatal liver failure (NLF). Neonates with disseminated HSV infection can develop hemophagocytic lymphohistiocytosis (HLH), and occasionally need orthotopic liver transplantation. Early interventions may be critical for the cure of NLF. Case Report We describe herewith a 6-day-old neonate with fulminant hepatic failure due to disseminated HSV-1 infection, who successfully responded to high-dose corticosteroid therapy 72 hours after the onset of disease. Preceding acyclovir, gamma globulin, and exchange blood transfusion therapies failed to control the disease. Methylprednisolone pulse therapy led to a drastic improvement of liver function and cytokine storms, and prevented the disease progression to HLH. Sustained levels of plasma and cerebrospinal fluid HSV DNA declined after prolonged acyclovir therapy. Bilateral lesions of the periventricular white matter areas, assessed by magnetic resonance imaging, disappeared at 3 months of age. The infant showed normal growth and development at 4 years of age. Conclusion Early anti-hypercytokinemia therapy using corticosteroid, and prolonged antiviral therapy might only provide the transplantation-free cure of NLF with HSV dissemination.
背景 单纯疱疹病毒(HSV)感染是新生儿肝衰竭(NLF)预后最差的病因之一。患有播散性HSV感染的新生儿可发展为噬血细胞性淋巴组织细胞增生症(HLH),偶尔需要进行原位肝移植。早期干预对于治愈NLF可能至关重要。病例报告 我们在此描述一名6日龄因播散性HSV-1感染导致暴发性肝衰竭的新生儿,该患儿在疾病发作72小时后对大剂量皮质类固醇治疗成功产生反应。在使用阿昔洛韦、丙种球蛋白和换血疗法之前未能控制该疾病。甲泼尼龙冲击疗法使肝功能和细胞因子风暴得到显著改善,并防止疾病进展为HLH。在长时间使用阿昔洛韦治疗后,血浆和脑脊液中HSV DNA的持续水平下降。通过磁共振成像评估,脑室周围白质区域的双侧病变在3个月大时消失。该婴儿在4岁时生长发育正常。结论 早期使用皮质类固醇进行抗高细胞因子血症治疗以及长时间的抗病毒治疗可能仅能使播散性HSV感染的NLF在不进行移植的情况下治愈。