scholarly journals Plasma filtration with dialysis for the treatment of capillary leak syndrome occurring secondary to surgery for colon cancer‐related perforating peritonitis

2021 ◽  
Author(s):  
Ginga Suzuki ◽  
Ryo Ichibayashi ◽  
Saki Yamamoto ◽  
Hibiki Serizawa ◽  
Yoshimi Nakamichi ◽  
...  
Keyword(s):  
Colon Cancer ◽  
Capillary Leak Syndrome ◽  
Capillary Leak ◽  
Plasma Filtration

Coupled Plasma Filtration Adsorption for Treatment of Capillary Leak Syndrome Superimposed to Acute Generalized Exanthematous Pustolosis: A Case Report

2020 ◽  
Vol 49 (3) ◽  
pp. 372-378 ◽  
Author(s):  
Vittorio Di Maso ◽  
Martina Cozzi ◽  
Ugo Gerini ◽  
Elisa Bedina ◽  
Elisa Olivo ◽  
...  
Keyword(s):  
Hypovolemic Shock ◽  
Supportive Therapy ◽  
Intravenous Immunoglobulins ◽  
Abrupt Onset ◽  
Capillary Leak Syndrome ◽  
Capillary Leak ◽  
Plasma Filtration ◽  
Threatening Condition ◽  
Life Threatening ◽  
Exanthematous Pustulosis

Coupled plasma filtration adsorption (CPFA) is an extracorporeal supportive therapy based on nonspecific adsorption of pro- and anti-inflammatory mediators combined with continuous renal replacement therapy. The main field of CPFA application is septic shock, and there are limited data about its efficacy in the treatment of other acute conditions characterized by a dysregulation in immune homeostasis. Capillary leak syndrome (CLS) defines a life-threatening condition sustained by hypercytokinemia and characterized by abrupt onset of increased capillary permeability leading to severe generalized edema and hypovolemic shock refractory to fluid administration. Therapy for CLS is not specific and, at present time, it consists in the use of steroids or intravenous immunoglobulins. We present the case of a 34-year-old woman who developed CLS superimposed to acute generalized exanthematous pustulosis after initiating therapy with hydroxychloroquine for undifferentiated connective tissue disease. CLS did not respond to steroids and intravenous immunoglobulins, while it was successfully treated with CPFA. This observation supports the possible role of CPFA in restoring a proper immunologic homeostasis not only in sepsis but also in other devastating conditions sustained by hypercytokinemia.

scholarly journals Continuous renal replacement therapy rescued life-threatening capillary leak syndrome in an extremely-low-birth-weight premature: a case report

2021 ◽  
Vol 47 (1) ◽  
Author(s):  
Li-Fen Yang ◽  
Jia-Chang Ding ◽  
Ling-Ping Zhu ◽  
Li-Xia Li ◽  
Meng-Qi Duan ◽  
...  
Keyword(s):  
Birth Weight ◽  
Renal Replacement Therapy ◽  
Low Birth Weight ◽  
Continuous Renal Replacement Therapy ◽  
Replacement Therapy ◽  
Therapeutic Option ◽  
Capillary Leak Syndrome ◽  
Extremely Low Birth Weight ◽  
Capillary Leak ◽  
Life Threatening

Abstract Background Capillary leak syndrome (CLS) is a rare disease characterized by profound vascular leakage and presents as a classic triad of hypotension, hypoalbuminemia and hemoconcentration. Severe CLS is mostly induced by sepsis and generally life-threatening in newborns, especially in premature infants. Continuous renal replacement therapy (CRRT) plays an important role of supportive treatment for severe CLS. Unfortunately, CRRT in preterm infants has rarely been well defined. Case presentation We report the case of a 11-day-old girl with CLS caused by sepsis, who was delivered by spontaneous vaginal delivery (SVD) at gestational age of 25 weeks and 4 days, and a birth weight of 0.89 Kilograms(kg). The infant received powerful management consisting of united antibiotics, mechanical ventilation, intravenous albumin and hydroxyethyl starch infusion, vasoactive agents, small doses of glucocorticoids and other supportive treatments. However, the condition rapidly worsened with systemic edema, hypotension, pulmonary exudation, hypoxemia and anuria in about 40 h. Finally, we made great efforts to perform CRRT for her. Fortunately, the condition improved after 82 h’ CRRT, and the newborn was rescued and gradually recovered. Conclusion CRRT is an effective rescue therapeutic option for severe CLS and can be successfully applied even in extremely-low-birth-weight premature.

scholarly journals Vascular endothelial hyperpermeability induces the clinical symptoms of Clarkson disease (the systemic capillary leak syndrome)

Blood ◽  
2012 ◽  
Vol 119 (18) ◽  
pp. 4321-4332 ◽  
Author(s):  
Zhihui Xie ◽  
Chandra C. Ghosh ◽  
Roshni Patel ◽  
Shoko Iwaki ◽  
Donna Gaskins ◽  
...  
Keyword(s):  
Clinical Symptoms ◽  
Case Series ◽  
Fiber Formation ◽  
Capillary Leak Syndrome ◽  
Actin Stress Fiber ◽  
Vascular Endothelial ◽  
Capillary Leak ◽  
Systemic Capillary Leak Syndrome ◽  
Humoral Factors ◽  
Clarkson Disease

Abstract The systemic capillary leak syndrome (SCLS) is a rare disorder characterized by transient episodes of hypotensive shock and anasarca thought to arise from reversible microvascular barrier dysfunction. Although the high prevalence of a monoclonal gammopathy of unknown significance in SCLS suggests a pathogenic contribution of endogenous immunoglobulins, the mechanisms of vascular hyperpermeability remain obscure. Herein, we report clinical and molecular findings on 23 patients, the largest SCLS case series to date. Application of episodic SCLS sera, but neither the purified immunoglobulin fraction nor sera obtained from patients during remission, to human microvascular endothelial cells caused vascular endothelial cadherin internalization, disruption of interendothelial junctions, actin stress fiber formation, and increased permeability in complementary functional assays without inducing endothelial apoptosis. Intravenous immunoglobulin, one promising therapy for SCLS, mitigated the permeability effects of episodic sera. Consistent with the presence of endogenous, nonimmunoglobulin, circulating permeability factor(s) constrained to SCLS episodes, we found that vascular endothelial growth factor (VEGF) and angiopoietin 2 (Ang2), were elevated in episodic SCLS sera but not in remission sera. Ab-based inhibition of Ang2 counteracted permeability induced by episodic SCLS sera. Comparable experiments with anti-VEGF Ab (bevacizumab) yielded less interpretable results, probably because of endothelial toxicity of VEGF withdrawal. Our results support a model of SCLS pathogenesis in which nonimmunoglobulin humoral factors such as VEGF and Ang2 contribute to transient endothelial contraction, suggesting a molecular mechanism for this highly lethal disorder.

Capillary-leak syndrome: an unrecognized early immune adverse effect of checkpoint-inhibitors treatment

Immunotherapy ◽  
2021 ◽  
Author(s):  
Ruth Percik ◽  
Asher Nethanel ◽  
Yair Liel
Keyword(s):  
Adverse Effect ◽  
Immune Response ◽  
Immune Checkpoint Inhibitors ◽  
Checkpoint Inhibitors ◽  
Endothelial Damage ◽  
Capillary Leak Syndrome ◽  
Capillary Leak ◽  
Cytokine Activation ◽  
Complex Interactions ◽  
Cellular Immune

Capillary-leak syndrome is strongly associated with cytokine activity states. It is an ill-recognized adverse effect of checkpoint inhibitors treatment, which are typically associated with cellular immune response. We describe two patients with capillary leak syndrome following immune checkpoint inhibitors treatment. We present linking mechanisms between checkpoint inhibitors, cellular immunity, cytokine action and endothelial damage. We suggest that capillary-leak syndrome is a unique adverse effect of immunotherapy, resulting from complex interactions between cellular and cytokine activation and that its expression is probably depending on inherent host immune variabilities.

Encephalopathy Due to Capillary Leak Syndrome

1997 ◽  
Vol 90 (10) ◽  
pp. 1060-1062 ◽  
Author(s):  
TULIO E. BERTORINI ◽  
MICHAEL S. GELFAND ◽  
THOMAS F. OʼBRIEN
Keyword(s):  
Capillary Leak Syndrome ◽  
Capillary Leak
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