Tetsuka Syuichi, Suzuki Tomohiro, Ogawa Tomoko, Hashimoto Ritsuo, Kato Hiroyuki
Department of Neurology, International University of Health and Welfare Hospital, 537-3 Iguchi, Nasushiobara, Tochigi 329-2763, Japan.
Case Rep Infect Dis. 2021 Jun 4;2021:5589739. doi: 10.1155/2021/5589739. eCollection 2021.
Primary cytomegalovirus (CMV) infection in healthy young adults is usually an asymptomatic or mononucleosis-like syndrome, whereas in immunocompromised patients, CMV can cause significant disease. In this study, we report an unusual case of primary CMV infection wherein the patient, an immunocompetent 21-year-old woman, presented severe encephalopathy, acute hepatitis, retinitis, and reactivation of latent Epstein-Barr virus. She developed confusion, high fever, headache, and tonic-clonic seizures. Brain magnetic resonance imaging showed high-intensity lesions in the medial temporal lobe and basal ganglia. Liver dysfunction was observed, and abdominal computed tomography revealed splenohepatomegaly. After fundus findings, the patient was diagnosed with CMV retinitis. Upon admission, she was treated with intravenous acyclovir and steroid pulse therapy. Considering both her serious clinical condition and elevated serum levels of interleukin-6, we speculated that her condition was similar to cytokine-storm-induced encephalopathy. On day 2 after admission, she showed prompt recovery from these clinical manifestations. Since blood CMV pp65 antigenemia was found to be positive, we administered ganciclovir for 2 weeks. On the basis of her clinical manifestations and the presence of blood CMV DNA and CMV pp65 antigenemia along with IgM kinetics, we finally diagnosed this patient with severe primary CMV infection. She left our hospital without sequelae 20 days after admission. The incidence of severe CMV disease in immunocompetent young adults might be higher than previously recognized. Noninvasive testing for CMV (such as CMV pp65 antigenemia and CMV DNAemia) is widely available and can help early diagnosis. Short-term glucocorticoid therapy might be beneficial in the treatment of encephalopathy in the early stages of primary CMV infection. Considering such a background, clinicians should keep severe primary CMV infection in mind as a differential diagnosis in the clinical setting.
健康年轻成年人的原发性巨细胞病毒（CMV）感染通常为无症状或类似单核细胞增多症的综合征，而在免疫功能低下的患者中，CMV可导致严重疾病。在本研究中，我们报告了一例原发性CMV感染的罕见病例，患者为一名21岁免疫功能正常的女性，出现了严重脑病、急性肝炎、视网膜炎以及潜伏性EB病毒的再激活。她出现了意识模糊、高热、头痛和强直阵挛性发作。脑部磁共振成像显示颞叶内侧和基底神经节有高强度病变。观察到肝功能障碍，腹部计算机断层扫描显示脾肝肿大。眼底检查后，患者被诊断为CMV视网膜炎。入院时，她接受了静脉注射阿昔洛韦和类固醇脉冲疗法。考虑到她严重的临床状况以及血清白细胞介素-6水平升高，我们推测她的病情类似于细胞因子风暴诱发的脑病。入院后第2天，她的这些临床表现迅速恢复。由于发现血液CMV pp65抗原血症呈阳性，我们给予更昔洛韦治疗2周。根据她的临床表现、血液中CMV DNA和CMV pp65抗原血症的存在以及IgM动力学，我们最终诊断该患者为严重原发性CMV感染。入院20天后，她未留后遗症出院。免疫功能正常的年轻成年人中严重CMV疾病的发生率可能高于先前的认识。CMV的非侵入性检测（如CMV pp65抗原血症和CMV DNA血症）广泛可用，有助于早期诊断。短期糖皮质激素治疗可能有助于原发性CMV感染早期脑病的治疗。考虑到这样的背景，临床医生在临床环境中应将严重原发性CMV感染作为鉴别诊断予以考虑。
