小儿嗜酸性肉芽肿合并多血管炎和心内血栓1例。

IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL SAGE Open Medical Case Reports Pub Date : 2024-12-19 eCollection Date: 2024-01-01 DOI:10.1177/2050313X241309966
Katharine V Jensen, Nicholas Brochez, Christopher Spence, Joel Livingston, Michael Khoury, Jeanine McColl
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引用次数: 0

摘要

嗜酸性肉芽肿病合并多血管炎(EGPA)是一种罕见的系统性坏死性血管炎,以嗜酸性粒细胞增多和血管外肉芽肿为特征,主要影响呼吸道。本报告详细介绍了一例独特的EGPA病例,该病例发生在一名6岁女孩,心脏广泛受损伤,表现为非典型心内肿块,提示心内膜纤维化并伴有血栓。临床过程分为三个阶段:最初的前驱症状伴哮喘;随后的外周嗜酸性粒细胞增多;最后是系统性血管炎。心脏受累,特别是右心室心内肿块延伸至室间隔,强调了EGPA的多样性。该患者符合美国风湿病学会和欧洲风湿病协会联盟对EGPA诊断的充分标准,表现为嗜酸性粒细胞增多、气道阻塞性疾病和活检证实的炎症,主要以血管外嗜酸性粒细胞为特征。治疗包括大剂量甲基强的松龙和环磷酰胺,导致临床改善和炎症标志物正常化。为了阻止右心室血栓的进展,开始使用无分离肝素治疗,并改用华法林治疗,这导致心脏肿块完全消失。本病例强调了多学科方法治疗复杂EGPA表现的必要性,特别是在儿科,并强调了及时干预减轻EGPA相关心脏并发症影响的重要性。
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Pediatric eosinophilic granulomatosis with polyangiitis and intracardiac thrombus: A case report.

Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare systemic necrotizing vasculitis marked by eosinophilia and extravascular granulomas, predominantly affecting the respiratory tract. This report details a unique EGPA case in a 6-year-old girl with extensive cardiac involvement, featuring an atypical intracardiac mass suggestive of endomyocardial fibrosis and a concomitant thrombus. The clinical course unfolded in three phases: an initial prodrome with asthma; subsequent peripheral hypereosinophilia; and ultimately systemic vasculitis. Cardiac involvement, notably an intracardiac mass in the right ventricular apex extending into the interventricular septum, underscored the diverse nature of EGPA. The patient fulfilled sufficient criteria outlined by the American College of Rheumatology and the European Alliance of Associations for Rheumatology for an EGPA diagnosis, displaying hypereosinophilia, obstructive airway disease, and biopsy-confirmed inflammation predominantly characterized by extravascular eosinophils. Treatment included high-dose methylprednisolone and cyclophosphamide, which resulted in clinical improvement and inflammatory marker normalization. To halt right ventricular thrombus progression, therapeutic unfractionated heparin was initiated, and she was transitioned to warfarin, which resulted in complete resolution of the cardiac mass. This case highlights the necessity of a multidisciplinary approach for managing complex EGPA manifestations, particularly in pediatrics, and emphasizes the importance of timely intervention in mitigating the impact of cardiac complications associated with EGPA.

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来源期刊
SAGE Open Medical Case Reports
SAGE Open Medical Case Reports MEDICINE, GENERAL & INTERNAL-
CiteScore
0.60
自引率
0.00%
发文量
320
审稿时长
8 weeks
期刊介绍: SAGE Open Medical Case Reports (indexed in PubMed Central) is a peer reviewed, open access journal. It aims to provide a publication home for short case reports and case series, which often do not find a place in traditional primary research journals, but provide key insights into real medical cases that are essential for physicians, and may ultimately help to improve patient outcomes. SAGE Open Medical Case Reports does not limit content due to page budgets or thematic significance. Papers are subject to rigorous peer review and are selected on the basis of whether the research is sound and deserves publication. By virtue of not restricting papers to a narrow discipline, SAGE Open Medical Case Reports facilitates the discovery of the connections between papers, whether within or between disciplines. Case reports can span the full spectrum of medicine across the health sciences in the broadest sense, including: Allergy/Immunology Anaesthesia/Pain Cardiovascular Critical Care/ Emergency Medicine Dentistry Dermatology Diabetes/Endocrinology Epidemiology/Public Health Gastroenterology/Hepatology Geriatrics/Gerontology Haematology Infectious Diseases Mental Health/Psychiatry Nephrology Neurology Nursing Obstetrics/Gynaecology Oncology Ophthalmology Orthopaedics/Rehabilitation/Occupational Therapy Otolaryngology Palliative Medicine Pathology Pharmacoeconomics/health economics Pharmacoepidemiology/Drug safety Psychopharmacology Radiology Respiratory Medicine Rheumatology/ Clinical Immunology Sports Medicine Surgery Toxicology Urology Women''s Health.
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