Systemic capillary leak syndrome complicated by lower extremity compartment syndrome: a case report.

IF 0.8 Q3 ANESTHESIOLOGY

JA Clinical Reports Pub Date : 2025-06-09 DOI:10.1186/s40981-025-00795-8

Asahi Ishihara, Katsuyuki Sagishima, Tadashi Ejima, Manami Kuwahara, Naoyuki Hirata

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引用次数: 0

Abstract

Background: Systemic capillary leak syndrome (SCLS) is a rare disorder characterized by hypotension, hypoalbuminemia, and hemoconcentration, typically caused by increased vascular permeability due to endothelial dysfunction. We report a case of SCLS complicated by bilateral lower extremity compartment syndrome.

Case presentation: A 29-year-old man developed fever, cough, and rhinorrhea. He was restless, hypotensive, and had generalized edema with tense extremities. Laboratory findings included a hemoglobin level of 24.9 g/dL, hematocrit of 69.3%, albumin of 1.8 g/dL, and creatinine of 3.27 mg/dL. SCLS-induced shock was diagnosed with detection of monoclonal gammopathy of the IgG-λ type. Treatment consisted of fluid resuscitation, vasopressors, high-dose corticosteroids, and intravenous immunoglobulin. Although hemodynamic status improved, he developed bilateral lower-limb compartment syndrome, necessitating fasciotomy. Although the patient exhibited sensory deficits and impaired dorsiflexion and plantarflexion in both ankles, he was able to ambulate with a cane and was discharged on hospital day 50.

Conclusion: This case highlights the risk of serious complications such as compartment syndrome in patients with SCLS.

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全身毛细血管渗漏综合征合并下肢筋膜室综合征1例。

背景：全身性毛细血管渗漏综合征（SCLS）是一种以低血压、低白蛋白血症和血液浓缩为特征的罕见疾病，通常由内皮功能障碍引起的血管通透性增加引起。我们报告一例scs合并双侧下肢隔室综合征。病例介绍：一名29岁男子出现发热、咳嗽和鼻漏。他坐立不安，低血压，全身水肿，四肢紧张。实验室检查结果包括血红蛋白水平24.9 g/dL，红细胞压积69.3%，白蛋白1.8 g/dL，肌酐3.27 mg/dL。采用IgG-λ型单克隆γ病检测诊断scs性休克。治疗包括液体复苏、血管加压剂、大剂量皮质类固醇和静脉注射免疫球蛋白。虽然血流动力学状况有所改善，但他出现双侧下肢筋膜室综合征，需要行筋膜切开术。尽管患者表现出感觉缺陷和双踝关节背屈和跖屈受损，但他能够用手杖行走，并于住院第50天出院。结论：本病例强调了scs患者发生严重并发症的风险，如筋膜室综合征。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

JA Clinical Reports ANESTHESIOLOGY-

CiteScore

1.30

自引率

0.00%

发文量

审稿时长

9 weeks

期刊介绍： JA Clinical Reports is a companion journal to the Journal of Anesthesia (JA), the official journal of the Japanese Society of Anesthesiologists (JSA). This journal is an open access, peer-reviewed, online journal related to clinical anesthesia practices such as anesthesia management, pain management and intensive care. Case reports are very important articles from the viewpoint of education and the cultivation of scientific thinking in the field of anesthesia. However, submissions of anesthesia research and clinical reports from Japan are notably decreasing in major anesthesia journals. Therefore, the JSA has decided to launch a new journal, JA Clinical Reports, to encourage JSA members, particularly junior Japanese anesthesiologists, to publish papers in English language.