Literature DB >> 29619573

[Idiopathic systemic capillary leak syndrome (Clarkson disease) : A rare cause of recurrent life-threatening edema].

S Dasdelen1, S-O Grebe2.   

Abstract

This case report describes the episodic occurrence of severe generalized edema in a young female patient, who developed hypertension with a massive hemoconcentration (hematocrit >0.5, hemoglobin >20g/dl) and hypoalbuminemia during the course of these acute disease phases. After the first two disease exacerbations were overcome, there was a complete regression of symptoms. After a long symptom-free interval, a new exacerbation occurred as a result of which critical organ ischemia occurred due to the severe hypotension and massive edema. Despite all treatment measures a severe compartment syndrome of the lower extemities with subsequent rhabdomyelosis developed. The patient ultimately died as a result of treatment-refractory cardiovascular failure. The idiopathic systemic capillary leak syndrome (SCLS, also known as Clarkson disease) is a rare and potentially life-threatening disease with a high mortality. Since the first description of the disease only approximately 500 cases have been published worldwide. The pathophysiology of this disease remains unclear despite all previous attempts at clarification. Regulation processes of endothelial permeability seem to be essentially disturbed. Affected patients have a monoclonal gammopathy of undetermined signficance conspicuously often; however, the knowledge of the limited treatment options is of fundamental importance for the prognosis and overall survival of patients.

Entities:  

Keywords:  Edema, generalized; Hyperpermeability, vascular, endothelial; Hypotension; Hypovolemic shock; Monoclonal gammopathy of undetermined significance

Mesh:

Year:  2018        PMID: 29619573     DOI: 10.1007/s00108-018-0409-2

Source DB:  PubMed          Journal:  Internist (Berl)        ISSN: 0020-9554            Impact factor:   0.743


  51 in total

1.  [Shock, hemoconcentration, and generalized edema in a 47-year-old man].

Authors:  T Rahn; A Plehn; H Lemm; S Abromeit; S Dambeck; F Busanny; L Neumann; K Werdan; H Ebelt
Journal:  Internist (Berl)       Date:  2012-03       Impact factor: 0.743

2.  Vascular endothelial growth factor in systemic capillary leak syndrome.

Authors:  Willem Joost Lesterhuis; Alexander J Rennings; William P Leenders; Arjan Nooteboom; Cornelis J Punt; Fred C Sweep; Peter Pickkers; Anneke Geurts-Moespot; Hanneke W Van Laarhoven; Johan Van der Vlag; Jo H Berden; Cor T Postma; Jos W Van der Meer
Journal:  Am J Med       Date:  2009-06       Impact factor: 4.965

3.  Mimicker of hereditary angioedema: Idiopathic systemic capillary leak syndrome successfully treated with intravenous immunoglobulin.

Authors:  Xiao Chloe Wan; Andrew Lai; Tejaswi Kompala; Rosa Ten
Journal:  Ann Allergy Asthma Immunol       Date:  2017-04-01       Impact factor: 6.347

Review 4.  Capillary leak syndrome: etiologies, pathophysiology, and management.

Authors:  Eric Siddall; Minesh Khatri; Jai Radhakrishnan
Journal:  Kidney Int       Date:  2017-03-17       Impact factor: 10.612

5.  Systemic capillary leak syndrome.

Authors:  N L Kao; G W Richmond; A T Luskin
Journal:  Chest       Date:  1993-11       Impact factor: 9.410

6.  A case of brucellosis complicated with fatal capillary leak syndrome.

Authors:  H Karsen; S T Koruk; C Calisur; L Karagac; F Duygu
Journal:  Bratisl Lek Listy       Date:  2012       Impact factor: 1.278

7.  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

8.  Intravenous Immunoglobulins Improve Survival in Monoclonal Gammopathy-Associated Systemic Capillary-Leak Syndrome.

Authors:  Marc Pineton de Chambrun; Marie Gousseff; Wladimir Mauhin; Jean-Christophe Lega; Marc Lambert; Sophie Rivière; Antoine Dossier; Marc Ruivard; François Lhote; Gilles Blaison; Laurent Alric; Christian Agard; David Saadoun; Julie Graveleau; Martin Soubrier; Marie-Josée Lucchini-Lecomte; Christine Christides; Annick Bosseray; Hervé Levesque; Jean-François Viallard; Nathalie Tieulie; Pierre-Yves Lovey; Sylvie Le Moal; Béatrice Bibes; Giuseppe Malizia; Pierre Abgueguen; François Lifermann; Jacques Ninet; Pierre-Yves Hatron; Zahir Amoura
Journal:  Am J Med       Date:  2017-06-09       Impact factor: 4.965

9.  Idiopathic systemic capillary leak syndrome in children.

Authors:  Peter Hsu; Zhihui Xie; Katie Frith; Melanie Wong; Alyson Kakakios; Kelly D Stone; Kirk M Druey
Journal:  Pediatrics       Date:  2015-03       Impact factor: 7.124

Review 10.  Interendothelial junctions and their role in the control of angiogenesis, vascular permeability and leukocyte transmigration.

Authors:  E Dejana; R Spagnuolo; G Bazzoni
Journal:  Thromb Haemost       Date:  2001-07       Impact factor: 5.249

View more
  1 in total

1.  [Necrotizing myositis of the thigh : Cause or result of a systemic capillary leak syndrome?]

Authors:  Paul Schmitz; Nadine Hausmann
Journal:  Unfallchirurgie (Heidelb)       Date:  2021-11-30
  1 in total

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