Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case Report
Acute hemorrhagic encephalomyelitis (AHEM) is a rare hyperacute form of acute disseminated encephalomyelitis (ADEM). The disease is characterized by fulminant inflammation and demyelination in the brain and spinal cord and is often preceded by an infection or vaccination. This case report presents a...
| Main Authors: | , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
MDPI AG
2022-11-01
|
| Series: | Vaccines |
| Subjects: | |
| Online Access: | https://www.mdpi.com/2076-393X/10/12/2046 |
| _version_ | 1827636653671317504 |
|---|---|
| author | Annika Kits Mattia Russel Pantalone Christopher Illies Aleksandra Antovic Anne-Marie Landtblom Ellen Iacobaeus |
| author_facet | Annika Kits Mattia Russel Pantalone Christopher Illies Aleksandra Antovic Anne-Marie Landtblom Ellen Iacobaeus |
| author_sort | Annika Kits |
| collection | DOAJ |
| description | Acute hemorrhagic encephalomyelitis (AHEM) is a rare hyperacute form of acute disseminated encephalomyelitis (ADEM). The disease is characterized by fulminant inflammation and demyelination in the brain and spinal cord and is often preceded by an infection or vaccination. This case report presents a 53-year-old male with rheumatoid arthritis and ongoing treatment with methotrexate and etanercept who developed fatal AHEM following the second dose of the COVID-19 vaccine. The disease course was complicated by multiorgan thromboembolic disease and the presence of high/moderate levels of cardiolipin IgG antibodies and anti-beta-2 glycoprotein 1 IgG antibodies suggesting a possible antiphospholipid syndrome. Treatment with immunosuppressive therapies failed to improve the course. The report comprises comprehensive clinical, neuroimaging, and neuropathological findings. The case highlights diagnostic challenges in a patient with several preceding risk factors, including autoimmune disease, immunotherapy, and vaccination, with possible pathophysiological implications. The temporal association with the COVID-19 vaccination may suggest possible causality although evidence cannot be ascertained. Reporting possible adverse events following COVID-19 vaccination is important to identify at-risk populations and to accomplish control of the current pandemic. |
| first_indexed | 2024-03-09T15:45:47Z |
| format | Article |
| id | doaj.art-6698244dc35f4623b81af4eea0a867d8 |
| institution | Directory Open Access Journal |
| issn | 2076-393X |
| language | English |
| last_indexed | 2024-03-09T15:45:47Z |
| publishDate | 2022-11-01 |
| publisher | MDPI AG |
| record_format | Article |
| series | Vaccines |
| spelling | doaj.art-6698244dc35f4623b81af4eea0a867d82023-11-24T18:31:45ZengMDPI AGVaccines2076-393X2022-11-011012204610.3390/vaccines10122046Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case ReportAnnika Kits0Mattia Russel Pantalone1Christopher Illies2Aleksandra Antovic3Anne-Marie Landtblom4Ellen Iacobaeus5Department of Neuroradiology, Karolinska University Hospital, 171 76 Stockholm, SwedenDepartment of Clinical Neuroscience, Division of Neurology, Karolinska Institute and Neurology, Karolinska University Hospital, 171 76 Stockholm, SwedenKarolinska University Laboratory, Department of Clinical Pathology and Cancerdiagnostics, Karolinska University Hospital, 171 76 Stockholm, SwedenDepartment of Medicine, Division of Rheumatology, Karolinska Institute and Rheumatology, Karolinska University Hospital, 171 76 Stockholm, SwedenDepartment of Medical Sciences, Division of Neurology, Uppsala University, 751 85 Uppsala, SwedenDepartment of Clinical Neuroscience, Division of Neurology, Karolinska Institute and Neurology, Karolinska University Hospital, 171 76 Stockholm, SwedenAcute hemorrhagic encephalomyelitis (AHEM) is a rare hyperacute form of acute disseminated encephalomyelitis (ADEM). The disease is characterized by fulminant inflammation and demyelination in the brain and spinal cord and is often preceded by an infection or vaccination. This case report presents a 53-year-old male with rheumatoid arthritis and ongoing treatment with methotrexate and etanercept who developed fatal AHEM following the second dose of the COVID-19 vaccine. The disease course was complicated by multiorgan thromboembolic disease and the presence of high/moderate levels of cardiolipin IgG antibodies and anti-beta-2 glycoprotein 1 IgG antibodies suggesting a possible antiphospholipid syndrome. Treatment with immunosuppressive therapies failed to improve the course. The report comprises comprehensive clinical, neuroimaging, and neuropathological findings. The case highlights diagnostic challenges in a patient with several preceding risk factors, including autoimmune disease, immunotherapy, and vaccination, with possible pathophysiological implications. The temporal association with the COVID-19 vaccination may suggest possible causality although evidence cannot be ascertained. Reporting possible adverse events following COVID-19 vaccination is important to identify at-risk populations and to accomplish control of the current pandemic.https://www.mdpi.com/2076-393X/10/12/2046COVID-19vaccinationacute hemorrhagic encephalomyelitisantiphospholipid syndromeantiphospholipid antibodiesadverse event |
| spellingShingle | Annika Kits Mattia Russel Pantalone Christopher Illies Aleksandra Antovic Anne-Marie Landtblom Ellen Iacobaeus Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case Report Vaccines COVID-19 vaccination acute hemorrhagic encephalomyelitis antiphospholipid syndrome antiphospholipid antibodies adverse event |
| title | Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case Report |
| title_full | Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case Report |
| title_fullStr | Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case Report |
| title_full_unstemmed | Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case Report |
| title_short | Fatal Acute Hemorrhagic Encephalomyelitis and Antiphospholipid Antibodies following SARS-CoV-2 Vaccination: A Case Report |
| title_sort | fatal acute hemorrhagic encephalomyelitis and antiphospholipid antibodies following sars cov 2 vaccination a case report |
| topic | COVID-19 vaccination acute hemorrhagic encephalomyelitis antiphospholipid syndrome antiphospholipid antibodies adverse event |
| url | https://www.mdpi.com/2076-393X/10/12/2046 |
| work_keys_str_mv | AT annikakits fatalacutehemorrhagicencephalomyelitisandantiphospholipidantibodiesfollowingsarscov2vaccinationacasereport AT mattiarusselpantalone fatalacutehemorrhagicencephalomyelitisandantiphospholipidantibodiesfollowingsarscov2vaccinationacasereport AT christopherillies fatalacutehemorrhagicencephalomyelitisandantiphospholipidantibodiesfollowingsarscov2vaccinationacasereport AT aleksandraantovic fatalacutehemorrhagicencephalomyelitisandantiphospholipidantibodiesfollowingsarscov2vaccinationacasereport AT annemarielandtblom fatalacutehemorrhagicencephalomyelitisandantiphospholipidantibodiesfollowingsarscov2vaccinationacasereport AT elleniacobaeus fatalacutehemorrhagicencephalomyelitisandantiphospholipidantibodiesfollowingsarscov2vaccinationacasereport |