Antisynthetase syndrome with refractory lung involvement and myositis successfully treated with double filtration plasmapheresis.
Identifieur interne : 000337 ( PubMed/Checkpoint ); précédent : 000336; suivant : 000338Antisynthetase syndrome with refractory lung involvement and myositis successfully treated with double filtration plasmapheresis.
Auteurs : Duygu Emine Ersozlu Bozkirli [Turquie] ; Ilknur Kozanoglu ; Emre Bozkirli ; Eftal YucelSource :
- Journal of clinical apheresis [ 1098-1101 ] ; 2013.
Descripteurs français
- KwdFr :
- Association thérapeutique, Autoanticorps (immunologie), Azathioprine (usage thérapeutique), Capacité de diffusion pulmonaire, Creatine kinase (sang), Cyclophosphamide (usage thérapeutique), Femelle, Humains, Hydroxychloroquine (usage thérapeutique), Immunosuppresseurs (usage thérapeutique), Induction de rémission, Myosite (), Myosite (traitement médicamenteux), Méthylprednisolone (usage thérapeutique), Plasma sanguin, Plasmaphérèse (), Pneumopathies interstitielles (), Pneumopathies interstitielles (traitement médicamenteux), Pneumopathies interstitielles (étiologie), Sujet âgé, Urgences.
- MESH :
- immunologie : Autoanticorps.
- sang : Creatine kinase.
- traitement médicamenteux : Myosite, Pneumopathies interstitielles.
- usage thérapeutique : Azathioprine, Cyclophosphamide, Hydroxychloroquine, Immunosuppresseurs, Méthylprednisolone.
- étiologie : Pneumopathies interstitielles.
- Association thérapeutique, Capacité de diffusion pulmonaire, Femelle, Humains, Induction de rémission, Myosite, Plasma sanguin, Plasmaphérèse, Pneumopathies interstitielles, Sujet âgé, Urgences.
English descriptors
- KwdEn :
- Aged, Autoantibodies (immunology), Azathioprine (therapeutic use), Combined Modality Therapy, Creatine Kinase (blood), Cyclophosphamide (therapeutic use), Emergencies, Female, Humans, Hydroxychloroquine (therapeutic use), Immunosuppressive Agents (therapeutic use), Lung Diseases, Interstitial (drug therapy), Lung Diseases, Interstitial (etiology), Lung Diseases, Interstitial (therapy), Methylprednisolone (therapeutic use), Myositis (complications), Myositis (drug therapy), Myositis (therapy), Plasma, Plasmapheresis (methods), Pulmonary Diffusing Capacity, Remission Induction.
- MESH :
- chemical , blood : Creatine Kinase.
- chemical , immunology : Autoantibodies.
- chemical , therapeutic use : Azathioprine, Cyclophosphamide, Hydroxychloroquine, Immunosuppressive Agents, Methylprednisolone.
- complications : Myositis.
- drug therapy : Lung Diseases, Interstitial, Myositis.
- etiology : Lung Diseases, Interstitial.
- methods : Plasmapheresis.
- therapy : Lung Diseases, Interstitial, Myositis.
- Aged, Combined Modality Therapy, Emergencies, Female, Humans, Plasma, Pulmonary Diffusing Capacity, Remission Induction.
Abstract
Antisynthetase syndrome (ASS) is characterized by inflammatory muscle disease, pulmonary and joint involvement, and antisynthetase autoantibodies, with anti-Jo-1 antibody being the most common. Despite the use of immunosuppressive drugs, the prognosis of lung involvement seems poor. Herein, we report a case of refractory ASS, which maintained long-term remission by double filtration plasmapheresis (DFPP) combined with immunosuppressive therapy. For a 65-year-old woman, who was diagnosed with ASS, immunosuppressive therapy was initiated and plasmapheresis (PP) was performed five times due to acute interstitial pulmonary disease and inflammatory myopathy. She remained in remission for eight months following PP. Increase in interstitial involvement was identified by lung tomography when the patient presented again with complaint of progressive increase in dyspnea and muscle pain. Although the immunosuppressive therapy was increased for the patient with elevated creatine phosphokinase (CPK) (2776 IU/mL), a rapid decrease in diffusion capacity of the lung for carbon monoxide (DLCO) was observed and the patient underwent PP. After four sessions of therapy, insufficient clinical and laboratory response was obtained (control CPK 1797 IU/mL) and because of that issue DFPP using a 2A filter was performed to the patient. There was a marked improvement in complaints of the patient, DLCO, and laboratory findings (control CPK 508 IU/mL) after three sessions of DFPP. The patient, who continued the immunosuppressive therapy after DFPP procedure, is being followed for 12 months in remission. Although our experience is limited with only one patient, DFPP seems promising as a treatment option for ASS with severe lung involvement.
DOI: 10.1002/jca.21285
PubMed: 23908096
Affiliations:
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Despite the use of immunosuppressive drugs, the prognosis of lung involvement seems poor. Herein, we report a case of refractory ASS, which maintained long-term remission by double filtration plasmapheresis (DFPP) combined with immunosuppressive therapy. For a 65-year-old woman, who was diagnosed with ASS, immunosuppressive therapy was initiated and plasmapheresis (PP) was performed five times due to acute interstitial pulmonary disease and inflammatory myopathy. She remained in remission for eight months following PP. Increase in interstitial involvement was identified by lung tomography when the patient presented again with complaint of progressive increase in dyspnea and muscle pain. Although the immunosuppressive therapy was increased for the patient with elevated creatine phosphokinase (CPK) (2776 IU/mL), a rapid decrease in diffusion capacity of the lung for carbon monoxide (DLCO) was observed and the patient underwent PP. After four sessions of therapy, insufficient clinical and laboratory response was obtained (control CPK 1797 IU/mL) and because of that issue DFPP using a 2A filter was performed to the patient. There was a marked improvement in complaints of the patient, DLCO, and laboratory findings (control CPK 508 IU/mL) after three sessions of DFPP. The patient, who continued the immunosuppressive therapy after DFPP procedure, is being followed for 12 months in remission. Although our experience is limited with only one patient, DFPP seems promising as a treatment option for ASS with severe lung involvement.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">23908096</PMID><DateCompleted><Year>2014</Year><Month>09</Month><Day>22</Day></DateCompleted><DateRevised><Year>2015</Year><Month>10</Month><Day>23</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1098-1101</ISSN><JournalIssue CitedMedium="Internet"><Volume>28</Volume><Issue>6</Issue><PubDate><Year>2013</Year><Month>Dec</Month></PubDate></JournalIssue><Title>Journal of clinical apheresis</Title><ISOAbbreviation>J Clin Apher</ISOAbbreviation></Journal><ArticleTitle>Antisynthetase syndrome with refractory lung involvement and myositis successfully treated with double filtration plasmapheresis.</ArticleTitle><Pagination><MedlinePgn>422-5</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1002/jca.21285</ELocationID><Abstract><AbstractText>Antisynthetase syndrome (ASS) is characterized by inflammatory muscle disease, pulmonary and joint involvement, and antisynthetase autoantibodies, with anti-Jo-1 antibody being the most common. Despite the use of immunosuppressive drugs, the prognosis of lung involvement seems poor. Herein, we report a case of refractory ASS, which maintained long-term remission by double filtration plasmapheresis (DFPP) combined with immunosuppressive therapy. For a 65-year-old woman, who was diagnosed with ASS, immunosuppressive therapy was initiated and plasmapheresis (PP) was performed five times due to acute interstitial pulmonary disease and inflammatory myopathy. She remained in remission for eight months following PP. Increase in interstitial involvement was identified by lung tomography when the patient presented again with complaint of progressive increase in dyspnea and muscle pain. Although the immunosuppressive therapy was increased for the patient with elevated creatine phosphokinase (CPK) (2776 IU/mL), a rapid decrease in diffusion capacity of the lung for carbon monoxide (DLCO) was observed and the patient underwent PP. After four sessions of therapy, insufficient clinical and laboratory response was obtained (control CPK 1797 IU/mL) and because of that issue DFPP using a 2A filter was performed to the patient. There was a marked improvement in complaints of the patient, DLCO, and laboratory findings (control CPK 508 IU/mL) after three sessions of DFPP. The patient, who continued the immunosuppressive therapy after DFPP procedure, is being followed for 12 months in remission. Although our experience is limited with only one patient, DFPP seems promising as a treatment option for ASS with severe lung involvement.</AbstractText><CopyrightInformation>Copyright © 2013 Wiley Periodicals, Inc.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Bozkirli</LastName><ForeName>Duygu Emine Ersozlu</ForeName><Initials>DE</Initials><AffiliationInfo><Affiliation>Department of Rheumatology, Baskent University School of Medicine, Adana, Turkey.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Kozanoglu</LastName><ForeName>Ilknur</ForeName><Initials>I</Initials></Author><Author ValidYN="Y"><LastName>Bozkirli</LastName><ForeName>Emre</ForeName><Initials>E</Initials></Author><Author ValidYN="Y"><LastName>Yucel</LastName><ForeName>Eftal</ForeName><Initials>E</Initials></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2013</Year><Month>07</Month><Day>03</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>J Clin Apher</MedlineTA><NlmUniqueID>8216305</NlmUniqueID><ISSNLinking>0733-2459</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D001323">Autoantibodies</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D007166">Immunosuppressive Agents</NameOfSubstance></Chemical><Chemical><RegistryNumber>4QWG6N8QKH</RegistryNumber><NameOfSubstance UI="D006886">Hydroxychloroquine</NameOfSubstance></Chemical><Chemical><RegistryNumber>8N3DW7272P</RegistryNumber><NameOfSubstance UI="D003520">Cyclophosphamide</NameOfSubstance></Chemical><Chemical><RegistryNumber>EC 2.7.3.2</RegistryNumber><NameOfSubstance UI="D003402">Creatine Kinase</NameOfSubstance></Chemical><Chemical><RegistryNumber>MRK240IY2L</RegistryNumber><NameOfSubstance UI="D001379">Azathioprine</NameOfSubstance></Chemical><Chemical><RegistryNumber>X4W7ZR7023</RegistryNumber><NameOfSubstance UI="D008775">Methylprednisolone</NameOfSubstance></Chemical></ChemicalList><SupplMeshList><SupplMeshName Type="Disease" UI="C537778">Antisynthetase syndrome</SupplMeshName></SupplMeshList><CitationSubset>IM</CitationSubset><CommentsCorrectionsList><CommentsCorrections RefType="CommentIn"><RefSource>J Clin Apher. 2015 Aug;30(4):257-8</RefSource><PMID Version="1">25346339</PMID></CommentsCorrections></CommentsCorrectionsList><MeshHeadingList><MeshHeading><DescriptorName UI="D000368" MajorTopicYN="N">Aged</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D001323" MajorTopicYN="N">Autoantibodies</DescriptorName><QualifierName UI="Q000276" MajorTopicYN="N">immunology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D001379" MajorTopicYN="N">Azathioprine</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D003131" MajorTopicYN="N">Combined Modality Therapy</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D003402" MajorTopicYN="N">Creatine Kinase</DescriptorName><QualifierName UI="Q000097" MajorTopicYN="N">blood</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D003520" MajorTopicYN="N">Cyclophosphamide</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D004630" MajorTopicYN="N">Emergencies</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006886" MajorTopicYN="N">Hydroxychloroquine</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D007166" MajorTopicYN="N">Immunosuppressive Agents</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D017563" MajorTopicYN="N">Lung Diseases, Interstitial</DescriptorName><QualifierName UI="Q000188" MajorTopicYN="N">drug therapy</QualifierName><QualifierName UI="Q000209" MajorTopicYN="N">etiology</QualifierName><QualifierName UI="Q000628" MajorTopicYN="Y">therapy</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D008775" MajorTopicYN="N">Methylprednisolone</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D009220" MajorTopicYN="N">Myositis</DescriptorName><QualifierName UI="Q000150" MajorTopicYN="N">complications</QualifierName><QualifierName UI="Q000188" MajorTopicYN="N">drug therapy</QualifierName><QualifierName UI="Q000628" MajorTopicYN="Y">therapy</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D010949" MajorTopicYN="N">Plasma</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D010956" MajorTopicYN="N">Plasmapheresis</DescriptorName><QualifierName UI="Q000379" MajorTopicYN="Y">methods</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D011653" MajorTopicYN="N">Pulmonary Diffusing Capacity</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D012074" MajorTopicYN="N">Remission Induction</DescriptorName></MeshHeading></MeshHeadingList><KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">antisynthetase syndrome</Keyword><Keyword MajorTopicYN="N">double filtration plasmapheresis</Keyword><Keyword MajorTopicYN="N">plasmapheresis</Keyword></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2013</Year><Month>03</Month><Day>10</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2013</Year><Month>05</Month><Day>30</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2013</Year><Month>06</Month><Day>03</Day></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2013</Year><Month>8</Month><Day>3</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2013</Year><Month>8</Month><Day>3</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2014</Year><Month>9</Month><Day>23</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="pubmed">23908096</ArticleId><ArticleId IdType="doi">10.1002/jca.21285</ArticleId></ArticleIdList></PubmedData></pubmed><affiliations><list><country><li>Turquie</li></country></list><tree><noCountry><name sortKey="Bozkirli, Emre" sort="Bozkirli, Emre" uniqKey="Bozkirli E" first="Emre" last="Bozkirli">Emre Bozkirli</name><name sortKey="Kozanoglu, Ilknur" sort="Kozanoglu, Ilknur" uniqKey="Kozanoglu I" first="Ilknur" last="Kozanoglu">Ilknur Kozanoglu</name><name sortKey="Yucel, Eftal" sort="Yucel, Eftal" uniqKey="Yucel E" first="Eftal" last="Yucel">Eftal Yucel</name></noCountry><country name="Turquie"><noRegion><name sortKey="Bozkirli, Duygu Emine Ersozlu" sort="Bozkirli, Duygu Emine Ersozlu" uniqKey="Bozkirli D" first="Duygu Emine Ersozlu" last="Bozkirli">Duygu Emine Ersozlu Bozkirli</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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