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

**Authors:** Asahi Ishihara, Katsuyuki Sagishima, Tadashi Ejima, Manami Kuwahara, Naoyuki Hirata

PMC · DOI: 10.1186/s40981-025-00795-8 · 2025-06-09

## TL;DR

A 29-year-old man with systemic capillary leak syndrome developed a rare complication of bilateral lower extremity compartment syndrome, requiring surgical intervention.

## Contribution

This case report highlights the previously under-recognized risk of compartment syndrome in patients with systemic capillary leak syndrome.

## Key findings

- A patient with SCLS developed bilateral lower-limb compartment syndrome requiring fasciotomy.
- The patient showed sensory deficits and impaired ankle movement but recovered enough to ambulate with a cane.
- The case underscores the importance of monitoring for musculoskeletal complications in SCLS patients.

## Abstract

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.

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.

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

## Linked entities

- **Diseases:** Systemic capillary leak syndrome (MONDO:0001956), compartment syndrome (MONDO:0004001)

## Full-text entities

- **Genes:** ALB (albumin) [NCBI Gene 213] {aka FDAHT, HSA, PRO0883, PRO0903, PRO1341}
- **Diseases:** fever (MESH:D005334), sensory deficits (MESH:D012678), rhinorrhea (MESH:D012818), monoclonal gammopathy (MESH:D010265), cough (MESH:D003371), hypoalbuminemia (MESH:D034141), SCLS (MESH:D019559), compartment syndrome (MESH:D003161), impaired dorsiflexion and plantarflexion (MESH:D060825), hypotension (MESH:D007022), shock (MESH:D012769), edema (MESH:D004487)
- **Chemicals:** creatinine (MESH:D003404)
- **Species:** Homo sapiens (human, species) [taxon 9606]

## Figures

3 figures with captions in the complete paper: https://tomesphere.com/paper/PMC12149068/full.md

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Source: https://tomesphere.com/paper/PMC12149068