Medical Intensive Care Unit and Division of Respiratory Diseases, Department of Internal Medicine, Third Affiliated Hospital of Sun Yat-sen University, Guangzhou, China.
Department of Gastroenterology, The Third Affiliated Hospital of Sun Yat-sen University, Guangzhou, China.
Front Immunol. 2020 Aug 4;11:1634. doi: 10.3389/fimmu.2020.01634. eCollection 2020.
Previous infectious or inflammatory events may be involved in the pathogenesis of neuromyelitis optica (NMO), potentially by triggering an autoimmune response. Cytomegalovirus (CMV)-related NMO (CMV-NMO) is rarely reported. Acute hemorrhagic rectal ulcer (AHRU) is a rare disease with a largely unknown pathogenesis. Herein, we reported a co-NMO and AHRU case associated with CMV infection. In addition, we review previously reported cases of CMV-NMO and CMV-AHRU. A 40-year-old female diagnosed with aquaporin4 (AQP4)-IgG NMO and a poor response to high-dose intravenous methylprednisolone and immunoglobulin, followed by three rounds of plasma exchange was transferred to Third Affiliated Hospital of Sun Yat-sen University, Guangzhou, China. She developed repeated acute lower gastrointestinal hemorrhage from the third day of admission. Abdominal computed tomography angiography (CTA) and interventional angiography did not detect any bleeding vessel. Bedside colonoscopy revealed a large ulcer-like lesion at 10 cm above the anus. Rectal biopsy pathology confirmed a CMV infection on day 23 post-admission, and cerebrospinal fluid (CSF) pathogen gene sequencing detected CMV gene copies on day 25 post-admission. After 2 weeks of treatment with ganciclovir and sodium phosphinate, the patient's lower gastrointestinal bleeding stopped, and her limb muscle strength and visual acuity gradually improved. After 4 weeks of antiviral therapy, colonoscopy showed that the intestinal wall of the original lesion was smooth. Hematoxylin and eosin (HE) staining and immunohistochemistry (IHC) of a biopsy specimen was negative for CMV, her right eye vision was normal, and limb muscle strength had recovered. Serum AQP4-IgG was negative, and lesions on brain magnetic resonance imaging (MRI) manifested shrinkage. The benefits of antiviral therapy remain unclear; however, clinicians should be aware of the possibility of CMV-related NMO, if NMO was refractory to high-dose intravenous methylprednisolone, immunoglobulin, and plasma exchange. Moreover, clinicians should consider the possibility of CMV-related AHRU when recurrent acute lower gastrointestinal bleeding occurs in a patient.
先前的感染或炎症事件可能参与了视神经脊髓炎（NMO）的发病机制，可能通过触发自身免疫反应。巨细胞病毒（CMV）相关的 NMO（CMV-NMO）很少见。急性出血性直肠溃疡（AHRU）是一种罕见疾病，其发病机制在很大程度上未知。在此，我们报告了一例与 CMV 感染相关的合并 NMO 和 AHRU 病例。此外，我们还回顾了之前报道的 CMV-NMO 和 CMV-AHRU 病例。
一名 40 岁女性，被诊断为水通道蛋白 4（AQP4）-IgG NMO，对大剂量静脉注射甲基强的松龙和免疫球蛋白反应不佳，随后进行了三轮血浆置换，转入中国广州中山大学附属第三医院。她在入院后的第三天开始反复出现急性下消化道出血。腹部 CT 血管造影（CTA）和介入血管造影均未发现出血血管。床边结肠镜检查发现距肛门 10cm 处有一个大的溃疡样病变。直肠活检病理在入院后第 23 天证实为 CMV 感染，入院后第 25 天脑脊液（CSF）病原体基因测序检测到 CMV 基因拷贝。给予更昔洛韦和膦酸钠钠治疗 2 周后，患者的下消化道出血停止，四肢肌力和视力逐渐改善。抗病毒治疗 4 周后，结肠镜检查显示原病变肠壁光滑。活检标本的苏木精和伊红（HE）染色和免疫组化（IHC）均为 CMV 阴性，右眼视力正常，四肢肌力恢复。血清 AQP4-IgG 阴性，脑部磁共振成像（MRI）病变缩小。
抗病毒治疗的益处尚不清楚；但是，如果 NMO 对大剂量静脉注射甲基强的松龙、免疫球蛋白和血浆置换无反应，临床医生应该意识到 CMV 相关 NMO 的可能性。此外，当患者反复发生急性下消化道出血时，临床医生应该考虑 CMV 相关 AHRU 的可能性。
