Isolated CNS involvement in eosinophilic granulomatosis with polyangiitis treated with mepolizumab: A case report

Hyoshin Son; Woo-Jin Lee; Jangsup Moon; Sungjoon Yoon; Keun-Hwa Jung; Kyung-Il Park; Sang Kun Lee; Kon Chu

doi:10.1016/j.jneuroim.2021.577551

Isolated CNS involvement in eosinophilic granulomatosis with polyangiitis treated with mepolizumab: A case report

J Neuroimmunol. 2021 Jun 15:355:577551. doi: 10.1016/j.jneuroim.2021.577551. Epub 2021 Mar 19.

Authors

Hyoshin Son¹, Woo-Jin Lee¹, Jangsup Moon², Sungjoon Yoon¹, Keun-Hwa Jung¹, Kyung-Il Park³, Sang Kun Lee¹, Kon Chu⁴

Affiliations

¹ Department of Neurology, Seoul National University Hospital, Seoul, South Korea.
² Department of Neurology, Seoul National University Hospital, Seoul, South Korea; Laboratory for Neurotherapeutics, Center for Medical Innovations, Biomedical Research Institute, Seoul National University Hospital, Seoul, South Korea.
³ Department of Neurology, Seoul National University Hospital Healthcare System Gangnam Center, Seoul, South Korea.
⁴ Department of Neurology, Seoul National University Hospital, Seoul, South Korea; Laboratory for Neurotherapeutics, Center for Medical Innovations, Biomedical Research Institute, Seoul National University Hospital, Seoul, South Korea. Electronic address: stemcell.snu@gmail.com.

PMID: 33773378
DOI: 10.1016/j.jneuroim.2021.577551

Abstract

Eosinophilic granulomatosis with polyangiitis (EGPA) is a systemic vasculitis disease involving small-sized vessels. The literature has reported involvement of the central nervous system (CNS) in 5% cases, and isolated CNS involvement is extremely rare. Due to its rarity and scarcity of clinical data, standardized treatment of isolated CNS involvement in EGPA is unclear. Mepolizumab, an anti-interleukin-5 monoclonal antibody, was previously introduced to treat EGPA with longer remission period, more patients showing remission, and reduction in prednisolone dose of those who already taking prednisolone compared to placebo. We describe a case of isolated CNS involvement in EGPA, confirmed by brain biopsy and treated with mepolizumab, which was refractory to conventional immunotherapy.

Keywords: Anti-interleukin-5 antibody; Brain biopsy; Central nervous system (CNS); Eosinophilic granulomatosis with polyangiitis (EGPA); Mepolizumab.

Publication types

Case Reports

MeSH terms

Antibodies, Monoclonal, Humanized / pharmacology
Antibodies, Monoclonal, Humanized / therapeutic use*
Brain / diagnostic imaging*
Brain / drug effects
Brain / immunology
Eosinophils* / drug effects
Eosinophils* / immunology
Granulomatosis with Polyangiitis / diagnostic imaging*
Granulomatosis with Polyangiitis / drug therapy*
Granulomatosis with Polyangiitis / immunology
Humans
Male
Middle Aged
Treatment Outcome

Substances

Antibodies, Monoclonal, Humanized
mepolizumab