Eosinophilic fasciitis in a pregnant woman with corticosteroid dependence and good response to infliximab
Eosinophilic fasciitis (EF) is characterized by symmetrical thickening of subcutaneous muscular fascia, causing skin induration with wrinkles and prominent hair follicles: the classic peau d’orange . Eosinophilia is a characteristic—albeit not universal—finding. We present the case of a 43-year-old...
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Veröffentlicht in: | Rheumatology international 2021-08, Vol.41 (8), p.1531-1539 |
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description | Eosinophilic fasciitis (EF) is characterized by symmetrical thickening of subcutaneous muscular fascia, causing skin induration with wrinkles and prominent hair follicles: the classic
peau d’orange
. Eosinophilia is a characteristic—albeit not universal—finding. We present the case of a 43-year-old pregnant woman diagnosed with EF during pregnancy who had extensive cutaneous involvement and severe functional repercussions, including worsening of lung function and intrauterine growth restriction as a possible complication. Treatment with prednisone was initiated during gestation and it was necessary to increase the dose. After delivery, methotrexate treatment was initiated and the corticosteroid dose progressively decreased, with progressive worsening in the torso and abdomen and secondary dyspnea due to thoracic pressure. Treatment with infliximab was then initiated, with favorable progress, though residual ankle and tarsal joint stiffness and significant muscular atrophy in the limbs continued. The triggering factor of EF was not identified. In a systematic search of the medical literature, three cases of EF in pregnant woman without clear triggers were found. Interestingly, all three cases progressed favorably with steroid treatment. Apart from this case, there are only seven published cases of infliximab use in the literature, all with moderate or complete response. Infliximab could be an option for corticosteroid-dependent EF with no response to other options. |
doi_str_mv | 10.1007/s00296-021-04787-6 |
format | Article |
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peau d’orange
. Eosinophilia is a characteristic—albeit not universal—finding. We present the case of a 43-year-old pregnant woman diagnosed with EF during pregnancy who had extensive cutaneous involvement and severe functional repercussions, including worsening of lung function and intrauterine growth restriction as a possible complication. Treatment with prednisone was initiated during gestation and it was necessary to increase the dose. After delivery, methotrexate treatment was initiated and the corticosteroid dose progressively decreased, with progressive worsening in the torso and abdomen and secondary dyspnea due to thoracic pressure. Treatment with infliximab was then initiated, with favorable progress, though residual ankle and tarsal joint stiffness and significant muscular atrophy in the limbs continued. The triggering factor of EF was not identified. In a systematic search of the medical literature, three cases of EF in pregnant woman without clear triggers were found. Interestingly, all three cases progressed favorably with steroid treatment. Apart from this case, there are only seven published cases of infliximab use in the literature, all with moderate or complete response. Infliximab could be an option for corticosteroid-dependent EF with no response to other options.</description><identifier>ISSN: 0172-8172</identifier><identifier>EISSN: 1437-160X</identifier><identifier>DOI: 10.1007/s00296-021-04787-6</identifier><identifier>PMID: 33484332</identifier><language>eng</language><publisher>Berlin/Heidelberg: Springer Berlin Heidelberg</publisher><subject>Adult ; Antirheumatic Agents - therapeutic use ; Biopsy ; Case Based Review ; Drug dosages ; Eosinophilia - drug therapy ; Fasciitis - drug therapy ; Female ; Hepatitis ; Hospitals ; Humans ; Infliximab - therapeutic use ; Internal medicine ; Leukemia ; Light therapy ; Medicine ; Medicine & Public Health ; Monoclonal antibodies ; Pathology ; Prednisolone - administration & dosage ; Pregnancy ; Pregnancy Complications - drug therapy ; Rheumatology ; Scleroderma ; Skin ; Skin - pathology ; Steroids ; TNF inhibitors ; Tumors</subject><ispartof>Rheumatology international, 2021-08, Vol.41 (8), p.1531-1539</ispartof><rights>The Author(s), under exclusive licence to Springer-Verlag GmbH, DE part of Springer Nature 2021</rights><rights>The Author(s), under exclusive licence to Springer-Verlag GmbH, DE part of Springer Nature 2021.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c375t-bd112d6d92cba9985b776c59e258199bb63894f8e111821dc9e389d17a3d97e03</citedby><cites>FETCH-LOGICAL-c375t-bd112d6d92cba9985b776c59e258199bb63894f8e111821dc9e389d17a3d97e03</cites><orcidid>0000-0002-6839-0303 ; 0000-0002-3012-3947</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s00296-021-04787-6$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s00296-021-04787-6$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,780,784,27923,27924,41487,42556,51318</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/33484332$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Jiménez-García, Nicolás</creatorcontrib><creatorcontrib>Aguilar-García, Josefa</creatorcontrib><creatorcontrib>Fernández-Canedo, Inés</creatorcontrib><creatorcontrib>Blázquez-Sánchez, Nuria</creatorcontrib><creatorcontrib>Fúnez-Liébana, Rafael</creatorcontrib><creatorcontrib>Romero-Gómez, Carlos</creatorcontrib><title>Eosinophilic fasciitis in a pregnant woman with corticosteroid dependence and good response to infliximab</title><title>Rheumatology international</title><addtitle>Rheumatol Int</addtitle><addtitle>Rheumatol Int</addtitle><description>Eosinophilic fasciitis (EF) is characterized by symmetrical thickening of subcutaneous muscular fascia, causing skin induration with wrinkles and prominent hair follicles: the classic
peau d’orange
. Eosinophilia is a characteristic—albeit not universal—finding. We present the case of a 43-year-old pregnant woman diagnosed with EF during pregnancy who had extensive cutaneous involvement and severe functional repercussions, including worsening of lung function and intrauterine growth restriction as a possible complication. Treatment with prednisone was initiated during gestation and it was necessary to increase the dose. After delivery, methotrexate treatment was initiated and the corticosteroid dose progressively decreased, with progressive worsening in the torso and abdomen and secondary dyspnea due to thoracic pressure. Treatment with infliximab was then initiated, with favorable progress, though residual ankle and tarsal joint stiffness and significant muscular atrophy in the limbs continued. The triggering factor of EF was not identified. In a systematic search of the medical literature, three cases of EF in pregnant woman without clear triggers were found. Interestingly, all three cases progressed favorably with steroid treatment. Apart from this case, there are only seven published cases of infliximab use in the literature, all with moderate or complete response. Infliximab could be an option for corticosteroid-dependent EF with no response to other options.</description><subject>Adult</subject><subject>Antirheumatic Agents - therapeutic use</subject><subject>Biopsy</subject><subject>Case Based Review</subject><subject>Drug dosages</subject><subject>Eosinophilia - drug therapy</subject><subject>Fasciitis - drug therapy</subject><subject>Female</subject><subject>Hepatitis</subject><subject>Hospitals</subject><subject>Humans</subject><subject>Infliximab - therapeutic use</subject><subject>Internal medicine</subject><subject>Leukemia</subject><subject>Light therapy</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Monoclonal antibodies</subject><subject>Pathology</subject><subject>Prednisolone - administration & dosage</subject><subject>Pregnancy</subject><subject>Pregnancy Complications - drug therapy</subject><subject>Rheumatology</subject><subject>Scleroderma</subject><subject>Skin</subject><subject>Skin - pathology</subject><subject>Steroids</subject><subject>TNF inhibitors</subject><subject>Tumors</subject><issn>0172-8172</issn><issn>1437-160X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNp9kT1PHDEQhq0IFA6SP5AispQmzYK_1h9lhEhAQkoDEp3ltWcPoz17sfcE-fcxHAkSBc1YM37mnbFfhL5QckwJUSeVEGZkRxjtiFBadfIDWlHBVUcludlDK0IV63QLB-iw1jvScinJR3TAudCCc7ZC8SzXmPJ8G6fo8eiqj3GJFceEHZ4LrJNLC37IG5fwQ1xusc9liT7XBUqOAQeYIQVIHrBLAa9zDrhAnXOqgJfcdMYpPsaNGz6h_dFNFT6_nEfo-ufZ1el5d_n718Xpj8vOc9Uv3RAoZUEGw_zgjNH9oJT0vQHWa2rMMEiujRg1UEo1o8EbaIVAlePBKCD8CH3f6c4l32-hLnYTq4dpcgnytlomNOFMCmYa-u0Nepe3JbXtLOuFVEZIIRvFdpQvudYCo51Le1D5YymxT0bYnRG2GWGfjbBPTV9fpLfDBsL_ln8_3wC-A2q7Smsor7Pfkf0Lrv-UFg</recordid><startdate>20210801</startdate><enddate>20210801</enddate><creator>Jiménez-García, Nicolás</creator><creator>Aguilar-García, Josefa</creator><creator>Fernández-Canedo, Inés</creator><creator>Blázquez-Sánchez, Nuria</creator><creator>Fúnez-Liébana, Rafael</creator><creator>Romero-Gómez, Carlos</creator><general>Springer Berlin Heidelberg</general><general>Springer Nature B.V</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0002-6839-0303</orcidid><orcidid>https://orcid.org/0000-0002-3012-3947</orcidid></search><sort><creationdate>20210801</creationdate><title>Eosinophilic fasciitis in a pregnant woman with corticosteroid dependence and good response to infliximab</title><author>Jiménez-García, Nicolás ; 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peau d’orange
. Eosinophilia is a characteristic—albeit not universal—finding. We present the case of a 43-year-old pregnant woman diagnosed with EF during pregnancy who had extensive cutaneous involvement and severe functional repercussions, including worsening of lung function and intrauterine growth restriction as a possible complication. Treatment with prednisone was initiated during gestation and it was necessary to increase the dose. After delivery, methotrexate treatment was initiated and the corticosteroid dose progressively decreased, with progressive worsening in the torso and abdomen and secondary dyspnea due to thoracic pressure. Treatment with infliximab was then initiated, with favorable progress, though residual ankle and tarsal joint stiffness and significant muscular atrophy in the limbs continued. The triggering factor of EF was not identified. In a systematic search of the medical literature, three cases of EF in pregnant woman without clear triggers were found. Interestingly, all three cases progressed favorably with steroid treatment. Apart from this case, there are only seven published cases of infliximab use in the literature, all with moderate or complete response. Infliximab could be an option for corticosteroid-dependent EF with no response to other options.</abstract><cop>Berlin/Heidelberg</cop><pub>Springer Berlin Heidelberg</pub><pmid>33484332</pmid><doi>10.1007/s00296-021-04787-6</doi><tpages>9</tpages><orcidid>https://orcid.org/0000-0002-6839-0303</orcidid><orcidid>https://orcid.org/0000-0002-3012-3947</orcidid></addata></record> |
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subjects | Adult Antirheumatic Agents - therapeutic use Biopsy Case Based Review Drug dosages Eosinophilia - drug therapy Fasciitis - drug therapy Female Hepatitis Hospitals Humans Infliximab - therapeutic use Internal medicine Leukemia Light therapy Medicine Medicine & Public Health Monoclonal antibodies Pathology Prednisolone - administration & dosage Pregnancy Pregnancy Complications - drug therapy Rheumatology Scleroderma Skin Skin - pathology Steroids TNF inhibitors Tumors |
title | Eosinophilic fasciitis in a pregnant woman with corticosteroid dependence and good response to infliximab |
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