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蛋白丢失性肠病作为小儿系统性红斑狼疮的初始表现:越南的一例罕见病例报告及文献综述

Protein-losing enteropathy as initial presentation of pediatric systemic lupus erythematosus: A rare case report from Vietnam and literature review.

作者信息

Mai Thanh Cong, Nguyen Trong Phuoc, Nguyen Thanh Nam

机构信息

Department of Pediatrics, Hanoi Medical University, Hanoi, Vietnam.

Pediatric Center, Bach Mai Hospital, Hanoi, Vietnam.

出版信息

Int J Immunopathol Pharmacol. 2025 Jan-Dec;39:3946320251358304. doi: 10.1177/03946320251358304. Epub 2025 Jul 20.

Abstract

Protein-losing enteropathy (PLE) is a rare but recognized manifestation of systemic lupus erythematosus (SLE). As an initial presentation of SLE, PLE is exceptionally uncommon, particularly in pediatric patients. We report the case of a 15-year-old Vietnamese girl with no significant past medical or family history, who presented with PLE as the initial manifestation of SLE. Clinical features included bilateral eyelid and lower extremity edema, ascites, and hypoalbuminemia, in the absence of nephrotic-range proteinuria, hepatic dysfunction, or malnutrition. Stool α1-antitrypsin concentration was markedly elevated at >231 mg/dL (normal <26.8 mg/dL), supporting the diagnosis of PLE in conjunction with clinical features and therapeutic response. Immunological evaluation revealed positive antinuclear antibody (ANA), anti-double-stranded DNA (anti-dsDNA) antibody, and lupus anticoagulant, hypocomplementemia, along with proteinuria equivalent to >0.5 g/24 h, fulfilling the 2019 EULAR/ACR classification criteria for SLE. The patient also developed cerebral venous sinus thrombosis. Treatment with corticosteroids, hydroxychloroquine, and warfarin resulted in significant clinical improvement. At 7 months of follow-up, she remained clinically stable, with normalized serum albumin levels and resolution of thrombosis. This case highlights the challenges of diagnosing PLE as an initial symptom of SLE in resource-limited settings. Heightened awareness of this rare presentation can facilitate early diagnosis and optimal management, improving patient outcomes.

摘要

蛋白丢失性肠病(PLE)是系统性红斑狼疮(SLE)一种罕见但已被认识到的表现。作为SLE的初始表现,PLE极为罕见,尤其是在儿科患者中。我们报告了一名15岁越南女孩的病例,她既往无重大病史或家族史,以PLE作为SLE的初始表现就诊。临床特征包括双侧眼睑和下肢水肿、腹水以及低白蛋白血症,且不存在肾病范围的蛋白尿、肝功能障碍或营养不良。粪便α1-抗胰蛋白酶浓度显著升高,>231mg/dL(正常<26.8mg/dL),结合临床特征和治疗反应支持PLE的诊断。免疫评估显示抗核抗体(ANA)、抗双链DNA(抗dsDNA)抗体和狼疮抗凝物阳性,补体降低,同时蛋白尿>0.5g/24小时,符合2019年欧洲抗风湿病联盟(EULAR)/美国风湿病学会(ACR)SLE分类标准。该患者还发生了脑静脉窦血栓形成。使用皮质类固醇、羟氯喹和华法林治疗后临床有显著改善。在7个月的随访中,她临床保持稳定,血清白蛋白水平恢复正常,血栓溶解。该病例突出了在资源有限的环境中将PLE诊断为SLE初始症状的挑战。提高对这种罕见表现的认识有助于早期诊断和优化管理,改善患者预后。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9e1a/12277556/fc6f7149903f/10.1177_03946320251358304-fig1.jpg

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