Seror Raphaèle, Mahr Alfred, Ramanoelina Jacky, Pagnoux Christian, Cohen Pascal, Guillevin Loïc
From Department of Internal Medicine, Hôpital Cochin, Université René-Descartes Paris 5, Assistance Publique-Hôpitaux de Paris, Paris, France.
Medicine (Baltimore). 2006 Jan;85(1):53-65. doi: 10.1097/01.md.0000200166.90373.41.
Wegener granulomatosis (WG) is an antineutrophil cytoplasmic antibody (ANCA)-associated granulomatous vasculitis of small and medium-sized vessels. This vasculitis involves mainly the upper and lower respiratory tracts and kidneys, although WG may affect any organ. Central nervous system (CNS) involvement is an uncommon manifestation of WG, reported in 7%-11% of patients. Three major mechanisms have been incriminated as causing CNS disease in WG: contiguous invasion of granuloma from extracranial sites, remote intracranial granuloma, and CNS vasculitis. Herein we describe 6 patients with WG-related CNS involvement, 2 of whom had chronic hypertrophic pachymeningitis, 3 with pituitary involvement, and 1 with cerebral vasculitis. CNS involvement was present at disease onset in 2 patients and occurred 5-18 years after WG diagnosis in the remaining 4. Based on these observations and a review of the literature, we discuss the pathogenic mechanisms, clinical features, imaging findings, treatment, and outcome of meningeal, pituitary, and vascular involvement, with an emphasis on differential diagnoses, prognosis, and therapeutic management of WG-related CNS involvement.
韦格纳肉芽肿病(WG)是一种与抗中性粒细胞胞浆抗体(ANCA)相关的中小血管肉芽肿性血管炎。这种血管炎主要累及上、下呼吸道和肾脏,不过WG也可能影响任何器官。中枢神经系统(CNS)受累是WG的一种不常见表现,在7% - 11%的患者中有所报道。有三种主要机制被认为可导致WG患者出现中枢神经系统疾病:肉芽肿从颅外部位的连续性侵袭、远处颅内肉芽肿以及中枢神经系统血管炎。在此,我们描述6例与WG相关的中枢神经系统受累患者,其中2例患有慢性肥厚性硬脑膜炎,3例累及垂体,1例患有脑血管炎。2例患者在疾病发作时即出现中枢神经系统受累,其余4例在WG诊断后5 - 18年出现。基于这些观察结果并结合文献复习,我们讨论了脑膜、垂体和血管受累的致病机制、临床特征、影像学表现、治疗及预后,重点关注WG相关中枢神经系统受累的鉴别诊断、预后及治疗管理。