Cytomegalovirus Infection in an Adult Patient With Neuromyelitis Optica and Acute Hemorrhagic Rectal Ulcer: Case Report and Literature Review
Authors: Jinmei Luo, Xiaowei Shi, Ying Lin, Na Cheng, Yunfeng Shi, Yanhong Wang, Ben-Quan Wu
Journal: Front Immunol (2020)
Keywords: acute hemorrhagic rectal ulcer, case report, cytomegalovirus, immunocompetent, neuromyelitis optica
MeSH terms: Cytomegalovirus Infections / complications, Cytomegalovirus Infections / virology, Gastrointestinal Hemorrhage / etiology, Neuromyelitis Optica / etiology, Ulcer / etiology, Case Reports, Research Support, Non-U.S. Gov't, Review, Adult, Antiviral Agents / therapeutic use, Biomarkers, Colonoscopy, Cytomegalovirus Infections / drug therapy, Cytomegalovirus Infections / immunology, Disease Management, Disease Susceptibility, Female, Gastrointestinal Hemorrhage / diagnosis, Humans, Immunocompromised Host, Immunohistochemistry, Magnetic Resonance Imaging / methods, Neuromyelitis Optica / diagnosis, Symptom Assessment, Tomography, X-Ray Computed, Treatment Outcome, Ulcer / diagnosis, Antiviral Agents, Biomarkers

Background: 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. Case presentation: 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. Conclusions: 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.