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


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          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.

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          Author and article information

          Blood Purif
          Blood Purification
          S. Karger AG
          May 2020
          18 February 2020
          : 49
          : 3
          : 372-378
          [_a] aNephrology and Dialysis Unit, Department of Internal Medicine, ASUITS, Trieste, Italy
          [_b] bDepartment of Anatomical Pathology, ASUITS, Trieste, Italy
          [_c] cDepartment of Perioperative Medicine, Intensive Care and Emergency, ASUITS, Trieste, Italy
          Author notes
          *Vittorio Di Maso, MD, PhD, Nephrology and Dialysis Unit, Department of Internal Medicine, ASUITS, Trieste Hospital, Strada di Fiume, 447, IT–34149 Trieste (Italy), E-Mail vittorio.dimaso@asuits.sanita.fvg.it
          503770 Blood Purif 2020;49:372–378
          © 2020 S. Karger AG, Basel

          Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

          : 26 February 2019
          : 28 September 2019
          Page count
          Figures: 2, Tables: 1, Pages: 7
          Case Report

          Cardiovascular Medicine,Nephrology
          Capillary leak syndrome,Coupled plasma filtration adsorption,Cytokines,Acute generalized exanthematous pustulosis,Hydroxychloroquine


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