Ohashi Keiji, Morishita Michiko, Watanabe Haruki, Sada Ken-Ei, Katsuyama Takayuki, Miyawaki Yoshia, Katsuyama Eri, Narazaki Mariko, Tatebe Noriko, Watanabe Katsue, Kawabata Tomoko, Wada Jun
Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Japan.
Intern Med. 2017 Nov 1;56(21):2943-2948. doi: 10.2169/internalmedicine.8683-16. Epub 2017 Sep 25.
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.
我们在此描述两例难治性抗中性粒细胞胞浆抗体相关性血管炎(AAV)合并可能与肥厚性硬脑膜炎(HP)相关的尿崩症(DI)的病例。一名患者患有显微镜下多血管炎和HP,对环磷酰胺、硫唑嘌呤、利妥昔单抗、霉酚酸酯(MMF)和咪唑立宾治疗无效。最终使用依那西普实现缓解,但5年后发生了DI。另一名患者患有肉芽肿性多血管炎,对环磷酰胺、甲氨蝶呤、MMF和利妥昔单抗治疗无效。随后发生了DI,但用依那西普成功治疗。在后一例中肉眼观察到硬脑膜肥厚。