tetano
Editor, Senior Moderator
Front Neurol
. 2023 Mar 10;14:1149612.
doi: 10.3389/fneur.2023.1149612. eCollection 2023.
Recurrent Acute Disseminated Encephalomyelitis (ADEM) after COVID-19-vaccination and after subsequent COVID-19-infection: A case report (part II)
Khouloud Poli[SUP] 1 [/SUP], Markus Kowarik[SUP] 1 2 [/SUP], Klaus Hamprecht[SUP] 3 [/SUP], Thomas Iftner[SUP] 3 [/SUP], Ulrike Ernemann[SUP] 4 [/SUP], Ulf Ziemann[SUP] 1 2 [/SUP], Sven Poli[SUP] 1 2 [/SUP]
Affiliations
Abstract
Acute disseminated encephalomyelitis (ADEM) is an autoimmune disorder of the central nervous system (CNS), which is commonly associated to previous viral infection or immunization. Cases of ADEM with a potential relationship to both severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection and vaccination have been reported. We recently published a rare case of a 65-year-old patient who suffered from a corticosteroid- and immunoglobulin-refractory multiple autoimmune syndrome including ADEM following Pfizer-BioNTech coronavirus disease (COVID)-19 vaccination, and whose symptoms largely resolved after repeated plasma exchange (PE). Four months later, the patient was diagnosed with SARS-CoV-2 omicron variant infection after experiencing mild upper respiratory tract symptoms. Few days later, the patient developed severe tetraparesis with magnetic resonance imaging (MRI) showing multiple new inflammatory contrast-enhancing lesions in the left middle cerebellar peduncle, cervical spinal cord, and ventral conus medullaris. Repeated cerebrospinal fluid (CSF) analyses indicated blood-brain barrier damage (increased albumin ratio) without signs of SARS-CoV-2 invasion (mild pleocytosis, no intrathecal antibody production). SARS-CoV-2 specific immunoglobulin G (IgG) were detected in serum and to a much lower degree in CSF with close correlation between both concentrations over time, reflecting antibody dynamics of vaccine- and infection-induced immune response, and blood-brain barrier patency. Daily PE therapy was initiated. Given the patient's lack of improvement after seven PE, treatment with rituximab was considered. After a first dose, however, the patient suffered epididymo-orchitis leading to sepsis, and declined rituximab continuation. At 3-months follow-up, clinical symptoms had dramatically improved. The patient regained walking ability without assistance. This case of recurrent ADEM after COVID-19-vaccination and after subsequent COVID-19-infection strongly supports the hypotheses of neuroimmunological complications in these conditions being promoted by a systemic immune response and mediated by molecular mimicry of, both, viral and vaccine SARS-CoV-2 antigens and CNS self-antigens.
Keywords: ADEM; COVID-19; CSF; SARS-CoV-2 antigen-specific IgG; biomarkers; recurrent.
. 2023 Mar 10;14:1149612.
doi: 10.3389/fneur.2023.1149612. eCollection 2023.
Recurrent Acute Disseminated Encephalomyelitis (ADEM) after COVID-19-vaccination and after subsequent COVID-19-infection: A case report (part II)
Khouloud Poli[SUP] 1 [/SUP], Markus Kowarik[SUP] 1 2 [/SUP], Klaus Hamprecht[SUP] 3 [/SUP], Thomas Iftner[SUP] 3 [/SUP], Ulrike Ernemann[SUP] 4 [/SUP], Ulf Ziemann[SUP] 1 2 [/SUP], Sven Poli[SUP] 1 2 [/SUP]
Affiliations
- PMID: 36970530
- PMCID: PMC10036343
- DOI: 10.3389/fneur.2023.1149612
Abstract
Acute disseminated encephalomyelitis (ADEM) is an autoimmune disorder of the central nervous system (CNS), which is commonly associated to previous viral infection or immunization. Cases of ADEM with a potential relationship to both severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection and vaccination have been reported. We recently published a rare case of a 65-year-old patient who suffered from a corticosteroid- and immunoglobulin-refractory multiple autoimmune syndrome including ADEM following Pfizer-BioNTech coronavirus disease (COVID)-19 vaccination, and whose symptoms largely resolved after repeated plasma exchange (PE). Four months later, the patient was diagnosed with SARS-CoV-2 omicron variant infection after experiencing mild upper respiratory tract symptoms. Few days later, the patient developed severe tetraparesis with magnetic resonance imaging (MRI) showing multiple new inflammatory contrast-enhancing lesions in the left middle cerebellar peduncle, cervical spinal cord, and ventral conus medullaris. Repeated cerebrospinal fluid (CSF) analyses indicated blood-brain barrier damage (increased albumin ratio) without signs of SARS-CoV-2 invasion (mild pleocytosis, no intrathecal antibody production). SARS-CoV-2 specific immunoglobulin G (IgG) were detected in serum and to a much lower degree in CSF with close correlation between both concentrations over time, reflecting antibody dynamics of vaccine- and infection-induced immune response, and blood-brain barrier patency. Daily PE therapy was initiated. Given the patient's lack of improvement after seven PE, treatment with rituximab was considered. After a first dose, however, the patient suffered epididymo-orchitis leading to sepsis, and declined rituximab continuation. At 3-months follow-up, clinical symptoms had dramatically improved. The patient regained walking ability without assistance. This case of recurrent ADEM after COVID-19-vaccination and after subsequent COVID-19-infection strongly supports the hypotheses of neuroimmunological complications in these conditions being promoted by a systemic immune response and mediated by molecular mimicry of, both, viral and vaccine SARS-CoV-2 antigens and CNS self-antigens.
Keywords: ADEM; COVID-19; CSF; SARS-CoV-2 antigen-specific IgG; biomarkers; recurrent.