Rheopheresis for Digital Ulcers and Raynaud's Phenomenon in Systemic Sclerosis Refractory to Conventional Treatments
Identifieur interne : 000295 ( Pmc/Curation ); précédent : 000294; suivant : 000296Rheopheresis for Digital Ulcers and Raynaud's Phenomenon in Systemic Sclerosis Refractory to Conventional Treatments
Auteurs : Peter Korsten ; Gerhard A. Müller ; Jan-Gerd Rademacher ; Michael Zeisberg ; Björn TampeSource :
- Frontiers in Medicine [ 2296-858X ] ; 2019.
Abstract
Raynaud's phenomenon (RP) is almost universally present in patients with Systemic Sclerosis (SSc). RP represents a generalized vasculopathy and potentially lead to digital ulcers (DU), which may be complicated by superinfection, tissue necrosis, and limb loss. We report the analysis of an extracorporeal procedure in a 36-year-old female patient with diffuse SSc with refractory RP and DU despite treatment with diltiazem, candesartan, sildenafil, and intravenous iloprost. We performed rheopheresis (RheoP), a variant of double-filtration plasmapheresis, as a potential new treatment option for refractory patients despite optimal medical therapy. We performed two RheoP per week every 4 weeks for a total of 3 months. Clinical improvement in DU healing occurred with no adverse events directly related to the treatment. While there was no reduction in the number of Raynaud attacks with RheoP, a significant reduction of the duration of attacks from a median of 15 (5–45, 95% CI 10–15) to 7 (3–30, 95% CI 6–10) minutes with an improvement of the Raynaud Condition Score (RCS) improved from 4 to 2. In conclusion, RheoP is a feasible and potentially beneficial treatment modality in patients with refractory RP and DU. We propose that RheoP should be investigated in a larger number of patients in a clinical trial setting.
Url:
DOI: 10.3389/fmed.2019.00208
PubMed: 31620442
PubMed Central: 6759692
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<front><div type="abstract" xml:lang="en"><p>Raynaud's phenomenon (RP) is almost universally present in patients with Systemic Sclerosis (SSc). RP represents a generalized vasculopathy and potentially lead to digital ulcers (DU), which may be complicated by superinfection, tissue necrosis, and limb loss. We report the analysis of an extracorporeal procedure in a 36-year-old female patient with diffuse SSc with refractory RP and DU despite treatment with diltiazem, candesartan, sildenafil, and intravenous iloprost. We performed rheopheresis (RheoP), a variant of double-filtration plasmapheresis, as a potential new treatment option for refractory patients despite optimal medical therapy. We performed two RheoP per week every 4 weeks for a total of 3 months. Clinical improvement in DU healing occurred with no adverse events directly related to the treatment. While there was no reduction in the number of Raynaud attacks with RheoP, a significant reduction of the duration of attacks from a median of 15 (5–45, 95% CI 10–15) to 7 (3–30, 95% CI 6–10) minutes with an improvement of the Raynaud Condition Score (RCS) improved from 4 to 2. In conclusion, RheoP is a feasible and potentially beneficial treatment modality in patients with refractory RP and DU. We propose that RheoP should be investigated in a larger number of patients in a clinical trial setting.</p>
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<pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Front Med (Lausanne)</journal-id>
<journal-id journal-id-type="iso-abbrev">Front Med (Lausanne)</journal-id>
<journal-id journal-id-type="publisher-id">Front. Med.</journal-id>
<journal-title-group><journal-title>Frontiers in Medicine</journal-title>
</journal-title-group>
<issn pub-type="epub">2296-858X</issn>
<publisher><publisher-name>Frontiers Media S.A.</publisher-name>
</publisher>
</journal-meta>
<article-meta><article-id pub-id-type="pmid">31620442</article-id>
<article-id pub-id-type="pmc">6759692</article-id>
<article-id pub-id-type="doi">10.3389/fmed.2019.00208</article-id>
<article-categories><subj-group subj-group-type="heading"><subject>Medicine</subject>
<subj-group><subject>Case Report</subject>
</subj-group>
</subj-group>
</article-categories>
<title-group><article-title>Rheopheresis for Digital Ulcers and Raynaud's Phenomenon in Systemic Sclerosis Refractory to Conventional Treatments</article-title>
</title-group>
<contrib-group><contrib contrib-type="author"><name><surname>Korsten</surname>
<given-names>Peter</given-names>
</name>
<xref ref-type="corresp" rid="c001"><sup>*</sup>
</xref>
<uri xlink:type="simple" xlink:href="http://loop.frontiersin.org/people/283116/overview"></uri>
</contrib>
<contrib contrib-type="author"><name><surname>Müller</surname>
<given-names>Gerhard A.</given-names>
</name>
</contrib>
<contrib contrib-type="author"><name><surname>Rademacher</surname>
<given-names>Jan-Gerd</given-names>
</name>
<uri xlink:type="simple" xlink:href="http://loop.frontiersin.org/people/452248/overview"></uri>
</contrib>
<contrib contrib-type="author"><name><surname>Zeisberg</surname>
<given-names>Michael</given-names>
</name>
<uri xlink:type="simple" xlink:href="http://loop.frontiersin.org/people/36207/overview"></uri>
</contrib>
<contrib contrib-type="author"><name><surname>Tampe</surname>
<given-names>Björn</given-names>
</name>
<uri xlink:type="simple" xlink:href="http://loop.frontiersin.org/people/576062/overview"></uri>
</contrib>
</contrib-group>
<aff><institution>Department of Nephrology and Rheumatology, University Medical Center Göttingen</institution>
,<addr-line>Göttingen</addr-line>
,<country>Germany</country>
</aff>
<author-notes><fn fn-type="edited-by"><p>Edited by: Lorenzo Cavagna, San Matteo Hospital Foundation (IRCCS), Italy</p>
</fn>
<fn fn-type="edited-by"><p>Reviewed by: Konstantinos Triantafyllias, ACURA Karl Aschoff Rehabilitation Clinic, Germany; Gianluca Bagnato, University of Messina, Italy; Veronica Codullo, Hôpital Cochin, France</p>
</fn>
<corresp id="c001">*Correspondence: Peter Korsten <email>peter.korsten@med.uni-goettingen.de</email>
</corresp>
<fn fn-type="other" id="fn001"><p>This article was submitted to Rheumatology, a section of the journal Frontiers in Medicine</p>
</fn>
</author-notes>
<pub-date pub-type="epub"><day>18</day>
<month>9</month>
<year>2019</year>
</pub-date>
<pub-date pub-type="collection"><year>2019</year>
</pub-date>
<volume>6</volume>
<elocation-id>208</elocation-id>
<history><date date-type="received"><day>09</day>
<month>7</month>
<year>2019</year>
</date>
<date date-type="accepted"><day>05</day>
<month>9</month>
<year>2019</year>
</date>
</history>
<permissions><copyright-statement>Copyright © 2019 Korsten, Müller, Rademacher, Zeisberg and Tampe.</copyright-statement>
<copyright-year>2019</copyright-year>
<copyright-holder>Korsten, Müller, Rademacher, Zeisberg and Tampe</copyright-holder>
<license xlink:href="http://creativecommons.org/licenses/by/4.0/"><license-p>This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.</license-p>
</license>
</permissions>
<abstract><p>Raynaud's phenomenon (RP) is almost universally present in patients with Systemic Sclerosis (SSc). RP represents a generalized vasculopathy and potentially lead to digital ulcers (DU), which may be complicated by superinfection, tissue necrosis, and limb loss. We report the analysis of an extracorporeal procedure in a 36-year-old female patient with diffuse SSc with refractory RP and DU despite treatment with diltiazem, candesartan, sildenafil, and intravenous iloprost. We performed rheopheresis (RheoP), a variant of double-filtration plasmapheresis, as a potential new treatment option for refractory patients despite optimal medical therapy. We performed two RheoP per week every 4 weeks for a total of 3 months. Clinical improvement in DU healing occurred with no adverse events directly related to the treatment. While there was no reduction in the number of Raynaud attacks with RheoP, a significant reduction of the duration of attacks from a median of 15 (5–45, 95% CI 10–15) to 7 (3–30, 95% CI 6–10) minutes with an improvement of the Raynaud Condition Score (RCS) improved from 4 to 2. In conclusion, RheoP is a feasible and potentially beneficial treatment modality in patients with refractory RP and DU. We propose that RheoP should be investigated in a larger number of patients in a clinical trial setting.</p>
</abstract>
<kwd-group><kwd>systemic sclerosis</kwd>
<kwd>extracorporeal circulation</kwd>
<kwd>raynaud phenomenon</kwd>
<kwd>skin ulcers</kwd>
<kwd>plasmapheresis</kwd>
</kwd-group>
<counts><fig-count count="4"></fig-count>
<table-count count="0"></table-count>
<equation-count count="0"></equation-count>
<ref-count count="19"></ref-count>
<page-count count="5"></page-count>
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</front>
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