Vanthuyne Aurelie, Riemann Sebastian, Brusselle Guy
Department of Respiratory Medicine Ghent University Hospital Ghent Belgium.
Respirol Case Rep. 2025 May 7;13(5):e70186. doi: 10.1002/rcr2.70186. eCollection 2025 May.
We present the case of an ANCA+ EGPA patient with a long disease history and remission for over 8 years (between 2015 and 2023), who developed a life-threatening relapse marked by diffuse alveolar haemorrhage and mononeuritis multiplex despite low dose mepolizumab (100 mg q4w) maintenance therapy. She was treated with pulse dose corticosteroids, rituximab, and mepolizumab at an increased dose of 300 mg q4w. This treatment regimen led to clinical and serological improvement, but she has persistent neuropathic pain and reduced handgrip strength. Use of low dose mepolizumab in high risk EGPA patients (i.e., life-threatening disease, prior organ involvement) might be insufficient to prevent relapses causing permanent organ damage such as sensory or motor paralysis. It is important to take into account the EGPA phenotype based upon ANCA serology as well as type and severity of organ involvement, as these characteristics might be associated with different treatment requirements and responses.
我们报告了一例抗中性粒细胞胞浆抗体(ANCA)阳性的嗜酸性肉芽肿性多血管炎(EGPA)患者,该患者病程较长,已缓解8年以上(2015年至2023年),尽管接受了低剂量美泊利珠单抗(100mg,每4周一次)维持治疗,但仍出现了以弥漫性肺泡出血和多发性单神经炎为特征的危及生命的复发。她接受了大剂量脉冲式糖皮质激素、利妥昔单抗治疗,美泊利珠单抗剂量增加至300mg,每4周一次。这种治疗方案使临床症状和血清学指标得到改善,但她仍有持续性神经病理性疼痛,握力下降。在高危EGPA患者(即危及生命的疾病、既往有器官受累)中使用低剂量美泊利珠单抗可能不足以预防导致永久性器官损害(如感觉或运动麻痹)的复发。根据ANCA血清学以及器官受累类型和严重程度来考虑EGPA表型很重要,因为这些特征可能与不同的治疗需求和反应相关。