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Case Reports
. 2017 Nov 1;56(21):2943-2948.
doi: 10.2169/internalmedicine.8683-16. Epub 2017 Sep 25.

Central Diabetes Insipidus in Refractory Antineutrophil Cytoplasmic Antibody-associated Vasculitis

Keiji Ohashi  1 , Michiko Morishita  1 , Haruki Watanabe  1 , Ken-Ei Sada  1 , Takayuki Katsuyama  1 , Yoshia Miyawaki  1 , Eri Katsuyama  1 , Mariko Narazaki  1 , Noriko Tatebe  1 , Katsue Watanabe  1 , Tomoko Kawabata  1 , Jun Wada  1
Affiliations
Case Reports

Central Diabetes Insipidus in Refractory Antineutrophil Cytoplasmic Antibody-associated Vasculitis

Keiji Ohashi et al. Intern Med. .

Abstract

We herein describe two cases of refractory antineutrophil cytoplasmic antibody-associated vasculitis (AAV) complicated with diabetes insipidus (DI) possibly related to hypertrophic pachymeningitis (HP). One patient had microscopic polyangiitis and HP, which were refractory to cyclophosphamide, azathioprine, rituximab, mycophenolate mofetil (MMF), and mizoribine. Remission was finally achieved with the use of etanercept, but DI occurred 5 years later. The other patient had granulomatosis with polyangiitis, which that was refractory to cyclophosphamide, methotrexate, MMF, and rituximab. DI subsequently developed, but was successfully treated with etanercept. Dura mater hypertrophy was macroscopically observed in the latter case.

Keywords: antineutrophil cytoplasmic antibody-associated vasculitis; diabetes insipidus; etanercept; hypertrophic pachymeningitis; rituximab.

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Figures

Figure 1.
Figure 1.
Magnetic resonance imaging of the head (Case 1). The white arrowheads show gadolinium enhancement of the hypertrophic dura mater.
Figure 2.
Figure 2.
The clinical course in Case 1. IVCY: intravenous cyclophosphamide, RTX: rituximab, PSL: prednisolone, mPSL: methylprednisolone, AZA: azathioprine, MZB: mizoribine, MMF: mycophenolate mofetil, ETN: etanercept, HP: hypertrophic pachymeningitis
Figure 3.
Figure 3.
The clinical course in Case 1 during the development of diabetes insipidus. PSL: prednisolone, mPSL: methylprednisolone, DDAVP: 1-desamino-8-D-arginine vasopressin, DI: diabetes insipidus
Figure 4.
Figure 4.
Magnetic resonance imaging of the pituitary gland (Case 2). A: Before relapse. B: At relapse. The white arrowheads show the excessively enlarged pituitary gland.
Figure 5.
Figure 5.
The clinical course in Case 2. RTX: rituximab, PSL: prednisolone, MMF: mycophenolate mofetil, ETN: etanercept, DDAVP: 1-desamino-8-D-arginine vasopressin, DI: diabetes insipidus
See this image and copyright information in PMC

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