Bourgarit Anne, Toumelin Philippe Le, Pagnoux Christian, Cohen Pascal, Mahr Alfred, Guern Véronique Le, Mouthon Luc, Guillevin Loïc
From the Department of Internal Medicine (AB, CP, PC, AM, VLG, LM, LG), Hôpital Avicenne, UPRES EA 3409 Université Paris XIII, and Hôpital Cochin, Assistance Publique-Hôpitaux de Paris, Université Paris V, Paris; and Department of Biostatistics (PLT), Hôpital Avicenne, Bobigny, France.
Medicine (Baltimore). 2005 Sep;84(5):323-330. doi: 10.1097/01.md.0000180793.80212.17.
Although combining corticosteroids and cyclophosphamide has greatly improved the prognoses of severe necrotizing vasculitides, some patients continue to have fulminating disease and die within the first year of diagnosis. To evaluate the characteristics of these patients, we retrospectively studied the files of 60 patients who died within the first year (20 patients with hepatitis B virus-associated polyarteritis nodosa [HBV-PAN], 18 with non-HBV PAN, 13 with microscopic polyangiitis [MPA], and 9 with Churg-Strauss syndrome [CSS]) and 535 first-year survivors (89 patients with HBV-PAN, 182 with non-HBV PAN, 140 with MPA, and 124 with CSS), 85 of whom died during a mean follow-up of 6.4 years. The 2 groups were compared for prognostic factors defined by the five-factor score (FFS) and Birmingham Vasculitis Activity Score at baseline, clinical signs, treatment, outcome, and causes of death. For first-year nonsurvivors, the clinical signs predictive of death were as follows: renal involvement (hazard ratio [HR], 1.6; 95% confidence intervals [CI], 1.09-2.3) or central nervous system involvement (HR, 2.3; 95% CI, 1.5-3.7), and a trend toward cardiomyopathy (HR, 1.4; 95% CI, 1.000-2.115). Older patients died earlier (HR, 1.04; 95% CI, 1.023-1.051). Gastrointestinal symptoms were most frequently associated with early death from HBV-PAN, while 83% of CSS patients died of cardiac involvement. Treatment had no significant impact on early death, except for patients with FFS > or = 2, for whom steroids alone were associated (p < 0.05). The major cause of early death was uncontrolled vasculitis (58%), followed by infection (26%). Cyclophosphamide-induced cytopenia and infection were responsible for 2 deaths. Despite these iatrogenic complications, early deaths were more frequently the consequence of insufficient or inappropriate therapy.
尽管联合使用皮质类固醇和环磷酰胺已极大地改善了严重坏死性血管炎的预后,但仍有一些患者病情急剧恶化,在诊断后的第一年内死亡。为了评估这些患者的特征,我们回顾性研究了60例在第一年内死亡的患者(20例乙型肝炎病毒相关性结节性多动脉炎[HBV-PAN]、18例非HBV-PAN、13例显微镜下多血管炎[MPA]和9例变应性肉芽肿性血管炎[CSS])以及535例第一年存活者(89例HBV-PAN、182例非HBV-PAN、140例MPA和124例CSS)的病历,其中85例在平均6.4年的随访期间死亡。比较两组患者的预后因素,包括基线时的五因素评分(FFS)和伯明翰血管炎活动评分、临床体征、治疗、结局及死亡原因。对于第一年的非存活者,预测死亡的临床体征如下:肾脏受累(风险比[HR],1.6;95%置信区间[CI],1.09 - 2.3)或中枢神经系统受累(HR,2.3;95%CI,1.5 - 3.7),以及存在心肌病的趋势(HR,1.4;95%CI,1.000 - 2.115)。年龄较大的患者死亡更早(HR,1.04;95%CI,1.023 - 1.051)。胃肠道症状最常与HBV-PAN早期死亡相关,而83%的CSS患者死于心脏受累。除FFS≥2的患者单独使用类固醇与之相关(p<0.05)外,治疗对早期死亡无显著影响。早期死亡的主要原因是未控制的血管炎(58%),其次是感染(26%)。环磷酰胺诱导的血细胞减少和感染导致2例死亡。尽管存在这些医源性并发症,但早期死亡更常见的原因是治疗不足或不当。