Effectiveness of Tocilizumab in the Treatment of Fasciitis with Eosinophilia: Two Case Reports.
Identifieur interne : 000169 ( PubMed/Checkpoint ); précédent : 000168; suivant : 000170Effectiveness of Tocilizumab in the Treatment of Fasciitis with Eosinophilia: Two Case Reports.
Auteurs : Guiomar Pinheiro [Portugal] ; Ana Rita Costa [Portugal] ; Ana Campar [Portugal] ; Teresa Mendonça [Portugal]Source :
- European journal of case reports in internal medicine [ 2284-2594 ] ; 2020.
Abstract
Fasciitis with eosinophilia (FE) is a rare connective tissue disease. Due to its rarity, large-scale studies are lacking, which makes its treatment challenging. Systemic corticosteroids (SCSs) are the cornerstone of treatment; however, additional immunosuppressive drugs (ISDs) are frequently necessary (usually methotrexate). We report 2 patients, for whom an SCS and methotrexate were not a viable long-term option. In the first case, we were unable to taper the SCS dose without symptom relapse, the patient showed only a partial response to methotrexate and presented side effects. The second case never fully responded to the SCS and methotrexate and demonstrated serious SCS adverse effects. Both patients were started on tocilizumab with extremely favourable results, making this drug a potential therapeutic weapon for these patients.
DOI: 10.12890/2020_001485
PubMed: 32309257
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the Treatment of Fasciitis with Eosinophilia: Two Case Reports.</title><author><name sortKey="Pinheiro, Guiomar" sort="Pinheiro, Guiomar" uniqKey="Pinheiro G" first="Guiomar" last="Pinheiro">Guiomar Pinheiro</name><affiliation wicri:level="1"><nlm:affiliation>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto, Portugal.</nlm:affiliation><country xml:lang="fr">Portugal</country><wicri:regionArea>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto</wicri:regionArea><wicri:noRegion>Porto</wicri:noRegion></affiliation></author><author><name sortKey="Costa, Ana Rita" sort="Costa, Ana Rita" uniqKey="Costa A" first="Ana Rita" last="Costa">Ana Rita Costa</name><affiliation wicri:level="1"><nlm:affiliation>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto, Portugal.</nlm:affiliation><country xml:lang="fr">Portugal</country><wicri:regionArea>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto</wicri:regionArea><wicri:noRegion>Porto</wicri:noRegion></affiliation></author><author><name sortKey="Campar, Ana" sort="Campar, Ana" uniqKey="Campar A" first="Ana" last="Campar">Ana Campar</name><affiliation wicri:level="1"><nlm:affiliation>Department of Clinical Immunology, Centro Hospitalar Universitário do Porto, Porto, Portugal.</nlm:affiliation><country xml:lang="fr">Portugal</country><wicri:regionArea>Department of Clinical Immunology, Centro Hospitalar Universitário do Porto, Porto</wicri:regionArea><wicri:noRegion>Porto</wicri:noRegion></affiliation></author><author><name sortKey="Mendonca, Teresa" sort="Mendonca, Teresa" uniqKey="Mendonca T" first="Teresa" last="Mendonça">Teresa Mendonça</name><affiliation wicri:level="1"><nlm:affiliation>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto, Portugal.</nlm:affiliation><country xml:lang="fr">Portugal</country><wicri:regionArea>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto</wicri:regionArea><wicri:noRegion>Porto</wicri:noRegion></affiliation></author></analytic><series><title level="j">European journal of case reports in internal medicine</title><idno type="eISSN">2284-2594</idno><imprint><date when="2020" type="published">2020</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Fasciitis with eosinophilia (FE) is a rare connective tissue disease. Due to its rarity, large-scale studies are lacking, which makes its treatment challenging. Systemic corticosteroids (SCSs) are the cornerstone of treatment; however, additional immunosuppressive drugs (ISDs) are frequently necessary (usually methotrexate). We report 2 patients, for whom an SCS and methotrexate were not a viable long-term option. In the first case, we were unable to taper the SCS dose without symptom relapse, the patient showed only a partial response to methotrexate and presented side effects. The second case never fully responded to the SCS and methotrexate and demonstrated serious SCS adverse effects. Both patients were started on tocilizumab with extremely favourable results, making this drug a potential therapeutic weapon for these patients.</div></front></TEI><pubmed><MedlineCitation Status="PubMed-not-MEDLINE" Owner="NLM"><PMID Version="1">32309257</PMID><DateRevised><Year>2020</Year><Month>04</Month><Day>22</Day></DateRevised><Article PubModel="Electronic-eCollection"><Journal><ISSN IssnType="Electronic">2284-2594</ISSN><JournalIssue CitedMedium="Internet"><Volume>7</Volume><Issue>4</Issue><PubDate><Year>2020</Year></PubDate></JournalIssue><Title>European journal of case reports in internal medicine</Title><ISOAbbreviation>Eur J Case Rep Intern Med</ISOAbbreviation></Journal><ArticleTitle>Effectiveness of Tocilizumab in the Treatment of Fasciitis with Eosinophilia: Two Case Reports.</ArticleTitle><Pagination><MedlinePgn>001485</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.12890/2020_001485</ELocationID><Abstract><AbstractText>Fasciitis with eosinophilia (FE) is a rare connective tissue disease. Due to its rarity, large-scale studies are lacking, which makes its treatment challenging. Systemic corticosteroids (SCSs) are the cornerstone of treatment; however, additional immunosuppressive drugs (ISDs) are frequently necessary (usually methotrexate). We report 2 patients, for whom an SCS and methotrexate were not a viable long-term option. In the first case, we were unable to taper the SCS dose without symptom relapse, the patient showed only a partial response to methotrexate and presented side effects. The second case never fully responded to the SCS and methotrexate and demonstrated serious SCS adverse effects. Both patients were started on tocilizumab with extremely favourable results, making this drug a potential therapeutic weapon for these patients.</AbstractText><AbstractText Label="LEARNING POINTS" NlmCategory="UNASSIGNED">The treatment of FE is challenging and mainly based on retrospective reviews, open-label trials and case reports, all of which included a small number of patients.Currently, systemic corticosteroids are the mainstay of treatment; however, other ISDs are frequently necessary.Cases showing a favourable clinical response to tocilizumab have recently been described in patients with corticosteroid-refractory disease, suggesting that this drug may potentially become a therapeutic weapon for these patients.</AbstractText><CopyrightInformation>© EFIM 2020.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Pinheiro</LastName><ForeName>Guiomar</ForeName><Initials>G</Initials><AffiliationInfo><Affiliation>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Costa</LastName><ForeName>Ana Rita</ForeName><Initials>AR</Initials><AffiliationInfo><Affiliation>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Campar</LastName><ForeName>Ana</ForeName><Initials>A</Initials><AffiliationInfo><Affiliation>Department of Clinical Immunology, Centro Hospitalar Universitário do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Mendonça</LastName><ForeName>Teresa</ForeName><Initials>T</Initials><AffiliationInfo><Affiliation>Internal Medicine Department, Centro Hospitalar Universitário do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2020</Year><Month>03</Month><Day>02</Day></ArticleDate></Article><MedlineJournalInfo><Country>Italy</Country><MedlineTA>Eur J Case Rep Intern Med</MedlineTA><NlmUniqueID>101648453</NlmUniqueID><ISSNLinking>2284-2594</ISSNLinking></MedlineJournalInfo><KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">Eosinophilic fasciitis</Keyword><Keyword MajorTopicYN="N">Shulman’s disease</Keyword><Keyword MajorTopicYN="N">tocilizumab</Keyword></KeywordList><CoiStatement>Conflicts of Interests: The Authors declare that there are no competing interests.</CoiStatement></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2020</Year><Month>01</Month><Day>14</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2020</Year><Month>01</Month><Day>21</Day></PubMedPubDate><PubMedPubDate 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