Case report: The histopathological analyses of two myelin oligodendrocyte glycoprotein antibody-associated diseases with a distinctive linear radiating gadolinium enhancement on MRI

Front Immunol. 2024 Aug 29:15:1426236. doi: 10.3389/fimmu.2024.1426236. eCollection 2024.

Abstract

Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) has highly heterogeneous clinical presentations, in which encephalitis is an important phenotype. Moreover, MOGAD has been reported to exhibit diverse imaging findings. However, there have been no previous reports of cases with perivascular radial gadolinium enhancement in periventricular regions, commonly reported in autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy. In this paper, we present two cases of MOGAD with this MRI feature, both of which underwent brain biopsy for the lesions. Brain biopsies revealed perivenous demyelination and inflammation consistent with acute disseminated encephalomyelitis (ADEM), with pronounced axonal damage in Case 1 and minimal axonal involvement in Case 2. Case 1 exhibited more severe cerebral atrophy than Case 2, correlating with the extent of axonal damage. Through these cases, we highlight the heterogeneity of radiological manifestations of MOGAD, expanding the spectrum beyond previously defined MRI patterns. Furthermore, histopathological analysis revealed distinct axonal involvement as a potential prognostic marker of brain atrophy. These observations emphasize the importance of considering MOGAD in the differential diagnosis, even in cases with atypical imaging findings, and highlight the significance of brain biopsy in guiding both diagnosis and prognosis.

Keywords: MRI; axonal damage; brain atrophy; histopathology; myelin oligodendrocyte glycoprotein antibody-associated disease; the radial linear periventricular enhancement pattern.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Autoantibodies* / immunology
  • Biopsy
  • Brain / diagnostic imaging
  • Brain / pathology
  • Encephalomyelitis, Acute Disseminated / diagnostic imaging
  • Encephalomyelitis, Acute Disseminated / immunology
  • Encephalomyelitis, Acute Disseminated / pathology
  • Female
  • Gadolinium*
  • Humans
  • Magnetic Resonance Imaging*
  • Male
  • Middle Aged
  • Myelin-Oligodendrocyte Glycoprotein* / immunology

Substances

  • Myelin-Oligodendrocyte Glycoprotein
  • Gadolinium
  • Autoantibodies
  • MOG protein, human

Grants and funding

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This work was supported by JSPS KAKENHI Grant Number JP 22H04923(CoBiA) (to SM) and AMED under Grant Numbers JP21wm0425018 (to GB), JP20dm017106 (to SM), and JP21wm0425019 (to SM).