Literature DB >> 33976992

A Rare Case of Idiopathic Capillary Leak Syndrome.

Suman Rao1, Omar Nasser1, Akhila Sunkara1, Rishi Singhal1, Dragos Manta2.   

Abstract

Idiopathic capillary leak syndrome (ICLS) occurs as a result of vascular membrane instability, which results in the leakage of several proteins from the vascular compartment to the interstitial spaces. It is an extremely rare disorder, with around 260 cases documented thus far. We present a case of a 35-year-old male with a past medical history of asthma and gastroesophageal reflux disease who initially presented to our hospital for the treatment of chronic demyelinating inflammatory neuropathy requiring plasmapheresis and steroid therapy. After removal of his vascular catheter, he experienced sudden onset of dyspnea, hypotension, and respiratory distress. His lab work showed a hemoglobin of 21.3 g/dL and a hematocrit of 62.6%. Protein electrophoresis showed a mildly decreased albumin at 3.28 g/dL. These findings were consistent with ICLS. He required management with colloids and systemic steroids. The difficult diagnosis of ICLS is due to its overlap with several medical emergencies, such as sepsis and anaphylaxis. Further studies are required to study the role of steroids and colloids in the management of ICLS.
Copyright © 2021, Rao et al.

Entities:  

Keywords:  idiopathic capillary leak syndrome

Year:  2021        PMID: 33976992      PMCID: PMC8106471          DOI: 10.7759/cureus.14370

Source DB:  PubMed          Journal:  Cureus        ISSN: 2168-8184


  10 in total

1.  The systemic capillary leak syndrome: appearance of interleukin-2-receptor-positive cells during attacks.

Authors:  M Cicardi; M Gardinali; G Bisiani; A Rosti; P Allavena; A Agostoni
Journal:  Ann Intern Med       Date:  1990-09-15       Impact factor: 25.391

2.  Systemic capillary leak syndrome and monoclonal IgG gammopathy; studies in a sixth patient and a review of the literature.

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Journal:  Medicine (Baltimore)       Date:  1977-05       Impact factor: 1.889

Review 3.  Idiopathic systemic capillary leak syndrome (Clarkson disease).

Authors:  Kirk M Druey; Samir M Parikh
Journal:  J Allergy Clin Immunol       Date:  2016-12-22       Impact factor: 10.793

4.  The Clinical Picture of Severe Systemic Capillary-Leak Syndrome Episodes Requiring ICU Admission.

Authors:  Marc Pineton de Chambrun; Charles-Edouard Luyt; François Beloncle; Marie Gousseff; Wladimir Mauhin; Laurent Argaud; Stanislas Ledochowski; Anne-Sophie Moreau; Romain Sonneville; Bruno Verdière; Sybille Merceron; Nathalie Zappella; Mickael Landais; Damien Contou; Alexandre Demoule; Sylvie Paulus; Bertrand Souweine; Bernard Lecomte; Antoine Vieillard-Baron; Nicolas Terzi; Elie Azoulay; Raymond Friolet; Marc Puidupin; Jérôme Devaquet; Jean-Marc Mazou; Yannick Fedun; Jean-Paul Mira; Jean-Herlé Raphalen; Alain Combes; Zahir Amoura
Journal:  Crit Care Med       Date:  2017-07       Impact factor: 7.598

Review 5.  Disorders of capillary permeability.

Authors:  J Marks; S Shuster
Journal:  Br J Dermatol       Date:  1973-06       Impact factor: 9.302

6.  Initial evidence of endothelial cell apoptosis as a mechanism of systemic capillary leak syndrome.

Authors:  R Assaly; D Olson; J Hammersley; P S Fan; J Liu; J I Shapiro; M B Kahaleh
Journal:  Chest       Date:  2001-10       Impact factor: 9.410

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

Authors:  Zhihui Xie; Chandra C Ghosh; Roshni Patel; Shoko Iwaki; Donna Gaskins; Celeste Nelson; Nina Jones; Philip R Greipp; Samir M Parikh; Kirk M Druey
Journal:  Blood       Date:  2012-03-12       Impact factor: 22.113

8.  The systemic capillary leak syndrome: a case series of 28 patients from a European registry.

Authors:  Marie Gousseff; Laurent Arnaud; Marc Lambert; Arnaud Hot; Mohamed Hamidou; Pierre Duhaut; Thomas Papo; Martin Soubrier; Marc Ruivard; Giuseppe Malizia; Nathalie Tieulié; Sophie Rivière; Jacques Ninet; Pierre-Yves Hatron; Zahir Amoura
Journal:  Ann Intern Med       Date:  2011-04-05       Impact factor: 25.391

Review 9.  Idiopathic systemic capillary leak syndrome (SCLS): case report and systematic review of cases reported in the last 16 years.

Authors:  Varun Dhir; Vivek Arya; Ishwar Chandra Malav; B S Suryanarayanan; Rajiva Gupta; A B Dey
Journal:  Intern Med       Date:  2007-06-15       Impact factor: 1.271

10.  Systemic Capillary Leak Syndrome associated with hypovolemic shock and compartment syndrome. Use of transpulmonary thermodilution technique for volume management.

Authors:  Bernd Saugel; Andreas Umgelter; Friedrich Martin; Veit Phillip; Roland M Schmid; Wolfgang Huber
Journal:  Scand J Trauma Resusc Emerg Med       Date:  2010-07-05       Impact factor: 2.953

  10 in total

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