Division of Hematology, Tokyo Metropolitan Cancer and Infectious Diseases Center Komagome Hospital, 3-18-22 Hon-komagome, Tokyo, 1138677, Bunkyo-ku, Japan.
Department of Infection Prevention and Control, Department of Clinical Laboratory, Tokyo Metropolitan Cancer and Infectious Diseases Center Komagome Hospital, 3-18-22 Hon-komagome, Tokyo, 1138677, Bunkyo-ku, Japan.
BMC Infect Dis. 2019 Jan 17;19(1):65. doi: 10.1186/s12879-019-3721-0.
Herpes simplex virus (HSV) has various presentations, depending on the patient's immune status, age, and the route of transmission. In adults, HSV type 1 is found predominantly in the oral area, and HSV type 2 (HSV-2) is commonly found in the genital area. HSV-2 infection without genital lesions is uncommon. Herein we report a unique case of pharyngotonsillitis as an initial manifestation of disseminated HSV-2 infection without genital involvement.
A 46-year-old male was admitted to our hospital with a 1-week history of fever and sore throat. His past medical history included hypereosinophilic syndrome diagnosed at age 45 years. Physical examination revealed throat congestion, bilaterally enlarged tonsils with exudates, tender cervical lymphadenopathy in the left posterior triangle, and mild epigastric tenderness. The laboratory data demonstrated bicytopenia, elevated liver enzyme levels, and hyperferritinemia. A bone marrow smear showed hypocellular marrow with histiocytes and hemophagocytosis. The diagnosis of HLH was confirmed, and the patient was treated with methylprednisolone pulse therapy on days 1-3. On day 5, despite initial improvement of the fever and sore throat, multiple, new, small bullae developed on the patient's face, trunk, and extremities. Additional testing showed that he was positive for HSV-specific immunoglobulin M and immunoglobulin G. Disseminated HSV infection was suspected, and intravenous acyclovir (10 mg/kg every 8 h) was begun. A subsequent direct antigen test of a bulla sample was positive for HSV-2. Moreover, tonsillar and esophageal biopsies revealed viral inclusion bodies. Immunohistochemical staining and a quantitative real-time polymerase chain reaction (PCR) assay confirmed the presence of HSV-2. Disseminated HSV-2 infection with multiple bullae, tonsillitis, esophagitis, and suspected hepatic involvement was diagnosed. After a 2-week course of intravenous acyclovir, his hematological status and liver function normalized, and his cutaneous skin lesions resolved. He was discharged on day 22 in good general health and continued taking oral valacyclovir for viral suppression due to his immunosuppressed status.
Disseminated HSV-2 infection should be considered as one of the differential diagnoses in patients with pharyngotonsillitis and impaired liver function of unknown etiology even if there are no genital lesions.
单纯疱疹病毒（HSV）的表现形式多种多样，取决于患者的免疫状态、年龄和传播途径。在成人中，HSV-1 主要出现在口腔区域，HSV-2（HSV-2）通常出现在生殖器区域。无生殖器病变的 HSV-2 感染并不常见。在此，我们报告了一例独特的咽扁桃体炎病例，这是播散性 HSV-2 感染无生殖器受累的初始表现。
一名 46 岁男性因发热和咽痛 1 周入院。他的既往病史包括 45 岁时诊断的嗜酸性粒细胞增多综合征。体格检查显示喉咙充血，双侧扁桃体肿大并有渗出物，左颈后三角区有压痛的颈淋巴结肿大，上腹部轻度压痛。实验室数据显示两系血细胞减少、肝酶水平升高和铁蛋白血症。骨髓涂片显示细胞减少的骨髓中有组织细胞和噬血细胞。诊断为 HLH，并在第 1-3 天接受甲基强的松龙脉冲治疗。第 5 天，尽管发热和咽痛最初有所改善，但患者面部、躯干和四肢出现多个新的小水疱。进一步检查显示他的 HSV 特异性免疫球蛋白 M 和免疫球蛋白 G 阳性。怀疑播散性 HSV 感染，并开始静脉用阿昔洛韦（10mg/kg，每 8 小时 1 次）。随后对水疱样本进行直接抗原检测，结果为 HSV-2 阳性。此外，扁桃体和食管活检显示病毒包涵体。免疫组化染色和实时定量 PCR 检测证实存在 HSV-2。诊断为播散性 HSV-2 感染，伴有多个水疱、扁桃体炎、食管炎和疑似肝受累。静脉用阿昔洛韦治疗 2 周后，患者的血液学状态和肝功能正常，皮肤病变消退。他在一般状况良好的情况下于第 22 天出院，并由于免疫抑制状态继续口服伐昔洛韦进行病毒抑制。
即使没有生殖器病变，咽扁桃体炎和不明原因肝损伤的患者也应考虑播散性 HSV-2 感染作为鉴别诊断之一。
