Postinfectious Anti–Myelin Oligodendrocyte Glycoprotein Antibody Positive Optic Neuritis and Myelitis

Abstract

We report the case of a 9-year-old girl admitted with fever, headache, and a cerebrospinal fluid lymphocytic pleocytosis. Polymerase chain reaction was positive for human herpes virus 6. She subsequently developed ataxia and bilateral loss of vision. Magnetic resonance imaging (MRI) showed bilateral optic nerve lesions with extension to optic chiasm and a short-segment myelitis. Serologic studies were positive for Borrelia burgdorferi IgM. Anti-aquaporin 4 antibody was negative and anti-myelin oligodendrocyte glycoprotein antibody (MOG) positive. After intravenous methylprednisolone, ceftriaxone, and intravenous immunoglobulin, her vision slowly recovered. The patient was discharged with only mild visual acuity loss, 1 month after admission. Brain MRI was repeated later and was normal and MOG assay became negative. In our view, this patient suffered from a postinfectious, anti-MOG-mediated, spinal cord and optic nerve demyelination. Borrelia burgdorferi infection is associated with neurologic manifestations in up to 15% of cases.1 Optic neuritis related with Lyme borreliosis has been reported infrequently and there is uncertainty about the validity of the diagnosis in many cases.2 Neurologic manifestations associated with human herpes virus 6 (HHV6) infection are usually febrile seizures in infants and encephalitis in immunosuppressed patients. 3Optic neuritis may have multiple causes, including infection and several autoimmune diseases, such as multiple sclerosis, acute disseminated encephalomyelitis, or neuromyelitis optica spectrum disorders.We describe a case of one female adolescent with antimyelin oligodendrocyte glycoprotein (MOG)-positive optic neuritis and myelitis. She had a positive polymerase chain reaction for HHV6 in cerebrospinal fluid and positive enzyme-linked immunosorbent assay and Western blot for Borrelia burgdorferi in serum. We propose that our case expands the spectrum of the association between infection and immunemediated MOG demyelination. Case ReportA previously healthy 9-year-old girl of African descent living in the Azores Islands was admitted to her local hospital with a 6-day history of a febrile illness, headache, and somnolence, previously treated with amoxicillin-clavulanic acid without resolution of symptoms. She had no history of recent travel. Her white blood cell count was 16340/mL (80% neutrophils) and c-reactive protein 0.88 mg/dL. Cerebrospinal fluid examination revealed a cell count of 95 cells/mL, with lymphocytic predominance and elevated protein (66 mg/dL). Polymerase chain reaction in the cerebrospinal fluid was positive for HHV6 and negative for Borrelia burgdorferi, and no serologic tests were performed regarding HHV6. Four days later, she presented with gait imbalance, abdominal pain, and dysuria. Neurologic examination was positive for gait ataxia with a positive Romberg sign. She underwent treatment with a 21-day course of ganciclovir (10 mg/kg/d) and was discharged on the 26th day after admission with a normal physical examination. Brain

COVID-19, HHV6 and MOG antibody: A perfect storm

“…An increased incidence of HHV6-related diseases such as Kawasaki disease and pityriasis rosea has been reported during the COVID-19 pandemic, suggesting a link between COVID-19 immune dysfunction and reactivation of HHV6 (Dursun, 2020;Drago et al, 2020, Abadías-Granado et al, 2021. Interestingly, the HHV6 and SARS-CoV-2 viruses have each been separately linked to MOG-IgG positive transverse myelitis (Zhou et al, 2020 andVieira et al, 2017). To our knowledge, this is the first report of possible HHV6 reactivation in the central nervous system in the setting of COVID-19 immune dysfunction, and the first with parainfectious MOG-IgG myelitis in the setting of SARS-CoV-2/HHV6 coinfection.…”

Postinfectious Acute Disseminated Encephalomyelitis Associated With Antimyelin Oligodendrocyte Glycoprotein Antibody

“…The clinical characteristics of postinfectious demyelinating diseases with antimyelin oligodendrocyte glycoprotein antibody are reviewed in Table 1. [8][9][10][11][12] All cases had an onset of postinfectious demyelinating disease with antimyelin oligodendrocyte glycoprotein antibody of 3 to 12 days (median 8.5 days) within 2 weeks from the primary infection. However, the interval between the primary infection and onset of demyelinating disease was too short to determine whether or not antimyelin oligodendrocyte glycoprotein antibody was produced by the primary infection because the measured antimyelin oligodendrocyte glycoprotein antibody was suspected to be immunoglobulin G fractionation, not immunoglobulin M. We speculate that antimyelin oligodendrocyte glycoprotein antibody transferred through the blood-brain barrier from the periphery into the central nervous system rather than being produced within the central nervous system.…”

Myelin Oligodendrocyte Glycoprotein Antibody–Associated Optic Neuritis and Myelitis in COVID-19

“…As many as 50% of patients with ADEM have been reported to test positive for serum MOG antibodies, and this proportion may be even higher in ADEM patients with recurrent polyphasic disease (22). Hence, there is a large body of established literature linking viral pathogens and the development of ADEM and MOG antibody-mediated CNS injury (23)(24)(25)(26). Pertinent to the ongoing COVID-19 pandemic, in 2004, Yeh et al (27) described a patient with ADEM associated with a human coronavirus (HCoV-OC43) detected in his serum and CSF samples, and murine hepatitis coronavirus has been implicated in CNS demyelinating disease for over 2 decades (28).…”