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小儿肉芽肿性多血管炎的临床表现:双病例研究

Pediatric Presentations of Granulomatosis With Polyangiitis: A Double Case Study.

作者信息

Handal Marina, Sharma Amit, Ernst Mara, Khalil Krystina, Weiss Eduardo

机构信息

Department of Dermatology, Nova Southeastern University KP-COM, Fort Lauderdale, Florida, USA.

Department of Dermatology, Larkin Community Hospital Palm Springs Campus, Hialeah, Florida, USA.

出版信息

Case Rep Dermatol Med. 2025 Mar 14;2025:6052518. doi: 10.1155/crdm/6052518. eCollection 2025.

Abstract

Granulomatosis with polyangiitis (GPA) is an ANCA-associated necrotizing vasculitis that causes granulomatous inflammation of small vessels in the respiratory tract and mucosa; GPA in childhood is a rare occurrence that presents distinctly as either a chronic, granulomatous disease that is clinically localized or as an acute vasculitis with rapidly progressive pulmonary or renal hemorrhage. We present two distinct cases of GPA in pediatric patients whose diagnoses were confirmed according to clinical presentation and scoring guidelines offered by the ACR/EULAR GPA Criteria. Despite a negative cANCA result, Patient 1 demonstrated a score of 9 based on the ACR/EULAR criteria for GPA diagnosis. This was based on the patient's physical examination, which revealed tender nodules and plaques along the face as well as a crusted ulceration in the left concha. A punch biopsy of the left lateral forehead revealed necrotizing angiitis with neutrophil-predominant inflammatory infiltrate and giant cells on pathological analysis. In contrast, Patient 2 displayed a score of 13 as reflected in the extent of systemic disease involvement, with ulcerations and nodules scattered along the torso, extremities, and genitalia. Laboratory workup revealed ANCA positivity. Additionally, this patient experienced granuloma formation of the right optic nerve, ethmoid sinus infiltration with damage to the nasal septum, and bilateral cavitary masses on CXR. There is a paucity of data in characterizing GPA in childhood, as evidence is based on small cohort studies and case reports in this unique demographic. The clinical presentations in our report underscore the need for early disease detection and comprehensive workup, as timely diagnosis and optimal treatment regimens may improve the prognoses of pediatric patients with GPA.

摘要

肉芽肿性多血管炎(GPA)是一种与抗中性粒细胞胞浆抗体(ANCA)相关的坏死性血管炎,可导致呼吸道和黏膜小血管的肉芽肿性炎症;儿童期GPA较为罕见,其表现明显不同,可为临床局限性的慢性肉芽肿性疾病,或为伴有快速进展性肺出血或肾出血的急性血管炎。我们报告了两例儿科患者的不同GPA病例,其诊断根据美国风湿病学会(ACR)/欧洲抗风湿病联盟(EULAR)GPA标准提供的临床表现和评分指南得以确诊。尽管患者1的胞浆型ANCA(cANCA)结果为阴性,但根据ACR/EULAR GPA诊断标准,其评分为9分。这是基于患者的体格检查,检查发现面部有压痛性结节和斑块,左侧鼻甲有结痂性溃疡。左侧前额的钻孔活检显示病理分析有坏死性血管炎,炎症浸润以中性粒细胞为主,并可见巨细胞。相比之下,患者2的评分为13分,反映出全身疾病受累程度,躯干、四肢和生殖器有散在的溃疡和结节。实验室检查显示ANCA阳性。此外,该患者右侧视神经形成肉芽肿,筛窦浸润伴鼻中隔损伤,胸部X线检查显示双侧空洞性肿块。关于儿童期GPA特征的数据较少,因为证据基于这一独特人群的小型队列研究和病例报告。我们报告中的临床表现强调了早期疾病检测和全面检查的必要性,因为及时诊断和最佳治疗方案可能改善儿科GPA患者的预后。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ae95/11928219/592e171b2afe/CRIDM2025-6052518.001.jpg

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