Successful treatment with rituximab in unilateral relapsing primary CNS vasculitis: a case report

Ryo Morikawa; Katsuhiko Kunitake; Junichiro Suzuki; Noriyoshi Nakai; Mari Yoshida; Yasuhiro Ito

doi:10.5692/clinicalneurol.cn-002262

Successful treatment with rituximab in unilateral relapsing primary CNS vasculitis: a case report

Ryo Morikawa
, Katsuhiko Kunitake
, Junichiro Suzuki
, Noriyoshi Nakai
, Mari Yoshida
, Yasuhiro Ito

Neurology & Neuroscience

Research output: Contribution to journal › Article › peer-review

Abstract

We report a case of unilateral relapsing primary CNS vasculitis (PCNSV) treated with Rituximab. A 52-year old man initially diagnosed as PCNSV by brain biopsy. He was treated with corticosteroids and no recurrence had been recognized for 4 years. However, after the reduction of corticosteroids to 9 mg, PCNSV recurred many times only in the right hemisphere. Mycophenolate mofetil and cyclophosphamide were not effective. We diagnosed this patient as unilateral relapsing PCNSV (UR-PCNSV), which is resistant to treatment and repeatedly relapses unilaterally. After rituximab was introduced, there was no clinical recurrence and abnormal lesions on head MRI improved. We emphasize that rituximab is a good candidate drug for treating the UR-PCNSV.

Translated title of the contribution	Rituximab で寛解維持に至ったunilateral relapsing primary CNS vasculitis の 1 例
Original language	English
Pages (from-to)	553-558
Number of pages	6
Journal	Clinical Neurology
Volume	66
Issue number	8
DOIs	https://doi.org/10.5692/clinicalneurol.cn-002262
Publication status	Published - 2026

All Science Journal Classification (ASJC) codes

Clinical Neurology

Access to Document

10.5692/clinicalneurol.cn-002262

Cite this

@article{c23eebf35cf140d091cd567fea6c4fa7,

title = "Successful treatment with rituximab in unilateral relapsing primary CNS vasculitis: a case report",

abstract = "We report a case of unilateral relapsing primary CNS vasculitis (PCNSV) treated with Rituximab. A 52-year old man initially diagnosed as PCNSV by brain biopsy. He was treated with corticosteroids and no recurrence had been recognized for 4 years. However, after the reduction of corticosteroids to 9 mg, PCNSV recurred many times only in the right hemisphere. Mycophenolate mofetil and cyclophosphamide were not effective. We diagnosed this patient as unilateral relapsing PCNSV (UR-PCNSV), which is resistant to treatment and repeatedly relapses unilaterally. After rituximab was introduced, there was no clinical recurrence and abnormal lesions on head MRI improved. We emphasize that rituximab is a good candidate drug for treating the UR-PCNSV.",

keywords = "primary cns vasculitis, rituximab, unilateral relapsing",

author = "Ryo Morikawa and Katsuhiko Kunitake and Junichiro Suzuki and Noriyoshi Nakai and Mari Yoshida and Yasuhiro Ito",

note = "Publisher Copyright: {\textcopyright} 2026 Japanese Society of Neurology. This article is available in Japanese with an abstract in English at www.jstage.jst.go.jp/browse/clinicalneurol.",

year = "2026",

doi = "10.5692/clinicalneurol.cn-002262",

language = "English",

volume = "66",

pages = "553--558",

journal = "Clinical Neurology",

issn = "0009-918X",

publisher = "Societas Neurologica Japonica",

number = "8",

}

TY - JOUR

T1 - Successful treatment with rituximab in unilateral relapsing primary CNS vasculitis

T2 - a case report

AU - Morikawa, Ryo

AU - Kunitake, Katsuhiko

AU - Suzuki, Junichiro

AU - Nakai, Noriyoshi

AU - Yoshida, Mari

AU - Ito, Yasuhiro

PY - 2026

Y1 - 2026

N2 - We report a case of unilateral relapsing primary CNS vasculitis (PCNSV) treated with Rituximab. A 52-year old man initially diagnosed as PCNSV by brain biopsy. He was treated with corticosteroids and no recurrence had been recognized for 4 years. However, after the reduction of corticosteroids to 9 mg, PCNSV recurred many times only in the right hemisphere. Mycophenolate mofetil and cyclophosphamide were not effective. We diagnosed this patient as unilateral relapsing PCNSV (UR-PCNSV), which is resistant to treatment and repeatedly relapses unilaterally. After rituximab was introduced, there was no clinical recurrence and abnormal lesions on head MRI improved. We emphasize that rituximab is a good candidate drug for treating the UR-PCNSV.

AB - We report a case of unilateral relapsing primary CNS vasculitis (PCNSV) treated with Rituximab. A 52-year old man initially diagnosed as PCNSV by brain biopsy. He was treated with corticosteroids and no recurrence had been recognized for 4 years. However, after the reduction of corticosteroids to 9 mg, PCNSV recurred many times only in the right hemisphere. Mycophenolate mofetil and cyclophosphamide were not effective. We diagnosed this patient as unilateral relapsing PCNSV (UR-PCNSV), which is resistant to treatment and repeatedly relapses unilaterally. After rituximab was introduced, there was no clinical recurrence and abnormal lesions on head MRI improved. We emphasize that rituximab is a good candidate drug for treating the UR-PCNSV.

KW - primary cns vasculitis

KW - rituximab

KW - unilateral relapsing

UR - https://www.scopus.com/pages/publications/105048538678

UR - https://www.scopus.com/pages/publications/105048538678#tab=citedBy

U2 - 10.5692/clinicalneurol.cn-002262

DO - 10.5692/clinicalneurol.cn-002262

M3 - Article

C2 - 42402381

AN - SCOPUS:105048538678

SN - 0009-918X

VL - 66

SP - 553

EP - 558

JO - Clinical Neurology

JF - Clinical Neurology

IS - 8

ER -

Successful treatment with rituximab in unilateral relapsing primary CNS vasculitis: a case report

Abstract

All Science Journal Classification (ASJC) codes

Access to Document

Other files and links

Fingerprint

Cite this