Intravenous cyclophosphamide and immunoglobulin ameliorated visual field defects in a patient with eosinophilic granulomatosis with polyangiitis

Yuto Motobayashi; Chiyako Oshikata; Yuka Kodama; Kosuke Terada; Yuga Yamashita; Ryo Nakadegawa; Hinako Masumitsu; Reeko Osada; Hirokazu Takayasu; Nami Masumoto; Saki Manabe; Takeshi Kaneko; Akira Shiraishi; Naomi Tsurikisawa

doi:10.1016/j.rmcr.2024.101980

Intravenous cyclophosphamide and immunoglobulin ameliorated visual field defects in a patient with eosinophilic granulomatosis with polyangiitis

Respir Med Case Rep. 2024 Jan 9:47:101980. doi: 10.1016/j.rmcr.2024.101980. eCollection 2024.

Authors

Yuto Motobayashi¹, Chiyako Oshikata^{1

2

3}, Yuka Kodama¹, Kosuke Terada¹, Yuga Yamashita¹, Ryo Nakadegawa¹, Hinako Masumitsu¹, Reeko Osada¹, Hirokazu Takayasu¹, Nami Masumoto^{1

3}, Saki Manabe¹, Takeshi Kaneko³, Akira Shiraishi⁴, Naomi Tsurikisawa^{1

2

3}

Affiliations

¹ Department of Respirology, National Hospital Organization Yokohama Medical Center, 3-60-2 Harajuku, Totsuka-ku, Yokohama, 245-8575, Japan.
² Department of Allergy and Respirology, Hiratsuka City Hospital, 1-19-1 Minamihara, Hiratsuka, Kanagawa, 254-0065, Japan.
³ Department of Pulmonology, Yokohama City University Graduate School of Medicine, 3-9 Fukuura, Kanazawa-ku, Yokohama, Kanagawa, 236-0004, Japan.
⁴ Department of Ophthalmology, Hiratsuka City Hospital, 1-19-1 Minamihara, Hiratsuka, Kanagawa, 254-0065, Japan.

Abstract

Treating ocular involvement in eosinophilic granulomatosis with polyangiitis (EGPA) can be challenging. We present the case of a 37-year-old woman with EGPA who had severe bilateral visual field defects. Laboratory results showed leukocytosis (17,500 WBC/μL, 25.8 % eosinophils), negative MPO-ANCA titer, and elevated PR3-ANCA level (33.2 IU/mL). Diffusion-weighted MRI revealed bilateral hyperintense occipital lesions, which were more prominent on the left. Her therapy initially included a steroid pulse, followed by daily prednisolone, but her visual field defects remained refractory. The addition of intravenous cyclophosphamide (5 courses) and intravenous immunoglobulin decreased her optic neuropathy and resolved her visual field defects.

Keywords: ANCA-Associated vasculitis; Cyclophosphamide; Eosinophilic granulomatosis with polyangiitis; IVIG; Optic neuritis; Visual field defects.

Publication types

Case Reports