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Journal of Clinical Immunology
Published in: Journal of Clinical Immunology 1/2020

01-01-2020 | Thymoma | Letter to Editor

Successful Treatment of a Patient with Chronic Systemic Capillary Leak Syndrome, Neutropenia and Thymoma

Authors: Rohit R. Ghurye, Asim Khan, Timothy Yung, Sorena Kiani-Alikhan, Debasish Pyne, Sofia Grigoriadou

Published in: Journal of Clinical Immunology | Issue 1/2020

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Excerpt

To the Editor: …
Literature
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Clarkson B, Thompson D, Horwith M, Luckey EH. Cyclical edema and shock due to increased capillary permeability. Am J Med. 1960;29:193–216.CrossRef
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Droder RM, Kyle RA, Greipp PR. Control of systemic capillary leak syndrome withaminophylline and terbutaline. Am J Med. 1992;92(5):523–6.CrossRef
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Airaghi L, Montori D, Santambrogio L, Miadonna A, Tedeschi A. Chronic systemic capillary leak syndrome. Report of a case and review of the literature. J Intern Med. 2000;247(6):731–5.CrossRef
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Druey KM, Greipp PR. Narrative review: the systemic capillary leak syndrome. Ann Intern Med. 2010;153(2):90–8.CrossRef
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Kapoor P, Greipp PT, Schaefer EW, Mandrekar SJ, Kamal AH, Gonzalez-Paz NC, et al. Idiopathic systemic capillary leak syndrome (Clarkson's disease): the Mayo clinic experience. Mayo Clin Proc. 2010;85(10):905–12.CrossRef
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Marra AM, Gigante A, Rosato E. Intravenous immunoglobulin in systemic capillary leak syndrome: a case report and review of literature. Expert Rev Clin Immunol. 2014;10(3):349–52.CrossRef
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Pineton de Chambrun M, Gousseff M, Mauhin W, Lega JC, Lambert M, Rivière S, et al. EurêClark Study Group. Intravenous immunoglobulins improve survival in monoclonal gammopathy-associated systemic capillary-leak syndrome. Am J Med. 2017;130(10):1219.e19–27.CrossRef
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Tahirkheli NK, Greipp PR. Treatment of the systemic capillary leak syndrome with terbutaline and theophylline. A case series. Ann Intern Med. 1999;130(11):905–9.CrossRef
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Guffroy A, Dervieux B, Gravier S, Martinez C, Deibener-Kaminsky J, Hachulla E, et al. Club des Rhumatismes et Inflammations (CRI). Systemic capillary leak syndrome and autoimmune diseases: a case series. Semin Arthritis Rheum. 2017;46(4):509–12.CrossRef
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Browne SK. Anticytokine autoantibody-associated immunodeficiency. Annu Rev Immunol. 2014;32:635–57.CrossRef
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Weksler B, Lu B. Alterations of the immune system in thymic malignancies. J Thorac Oncol. 2014;9(9 Suppl 2):S137–42.CrossRef
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Belizna C, Duijvestijn A, Hamidou M, Tervaert JW. Antiendothelial cell antibodies in vasculitis and connective tissue disease. Ann Rheum Dis. 2006;65(12):1545–50.CrossRef
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Assaly R, Olson D, Hammersley J, Fan PS, Liu J, Shapiro JI, et al. Initial evidence of endothelial cell apoptosis as a mechanism of systemic capillary leak syndrome. Chest. 2001;120(4):1301–8.CrossRef
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Xie Z, Ghosh CC, Patel R, Iwaki S, Gaskins D, Nelson C, et al. Vascular endothelial hyperpermeability induces the clinical symptoms of Clarkson disease (the systemic capillary leak syndrome). Blood. 2012;119(18):4321–32.CrossRef
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Ballow M. The IgG molecule as a biological immune response modifier: mechanisms of action of intravenous immune serum globulin in autoimmune and inflammatory disorders. J Allergy Clin Immunol. 2011;127(2):315–23.CrossRef
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Mullane R, Langewisch E, Florescu M, Plumb T. Chronic systemic capillary leak syndrome treatment with intravenous immune globulin: Case report and review of the literature. Clin Nephrol. 2019;91(1):59–63.CrossRef
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Alkhunaizi AM, Kabbani AH, ElTigani MA. Chronic idiopathic systemic capillary leak syndrome: a case report. Allergy Asthma Clin Immunol. 2019;15:34.CrossRef
Metadata
Title
Successful Treatment of a Patient with Chronic Systemic Capillary Leak Syndrome, Neutropenia and Thymoma
Authors
Rohit R. Ghurye
Asim Khan
Timothy Yung
Sorena Kiani-Alikhan
Debasish Pyne
Sofia Grigoriadou
Publication date
01-01-2020
Publisher
Springer US
Published in
Journal of Clinical Immunology / Issue 1/2020
Print ISSN: 0271-9142
Electronic ISSN: 1573-2592
DOI
https://doi.org/10.1007/s10875-019-00722-4

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Keynote webinar | Spotlight on medication adherence

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WHO estimates that half of all patients worldwide are non-adherent to their prescribed medication. The consequences of poor adherence can be catastrophic, on both the individual and population level.

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