Clinical findings in MuSK-antibody positive myasthenia gravis: A U.S. experience
We performed a retrospective chart review on 53 muscle‐specific kinase antibody (MuSK‐Ab)‐positive myasthenia gravis (MG) patients at nine university‐based centers in the U.S. Of these, 66% were Caucasian, 85% were women, and age of onset was 9–79 years. Twenty‐seven patients were nonresponsive to a...
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| Veröffentlicht in: | Muscle & nerve 2010-03, Vol.41 (3), p.370-374 |
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| creator | Pasnoor, Mamatha Wolfe, Gil I. Nations, Sharon Trivedi, Jaya Barohn, Richard J. Herbelin, Laura McVey, April Dimachkie, Mazen Kissel, John Walsh, Ronan Amato, Anthony Mozaffar, Tahseen Hungs, Marcel Chui, Luis Goldstein, Jonathan Novella, Steven Burns, Ted Phillips, Lawrence Claussen, Gwendolyn Young, Angela Bertorini, Tulio Oh, Shin |
| description | We performed a retrospective chart review on 53 muscle‐specific kinase antibody (MuSK‐Ab)‐positive myasthenia gravis (MG) patients at nine university‐based centers in the U.S. Of these, 66% were Caucasian, 85% were women, and age of onset was 9–79 years. Twenty‐seven patients were nonresponsive to anticholinesterase therapy. Myasthenia Gravis Foundation of America improvement status was achieved in 53% patients on corticosteroids, 51% with plasma exchange, and in 20% on intravenous immunoglobulin (IVIG). Thymectomy was beneficial in 7/18 patients at 3 years. Long‐term (≥3 years) outcome was very favorable in 58% of patients who achieved remission and/or minimal manifestation status. Overall, 73% improved. There was one MG‐related death. This survey reinforces several cardinal features of MuSK‐Ab‐positive MG, including prominent bulbar involvement and anticholinesterase nonresponsiveness. Facial or tongue atrophy was rare. Most patients respond favorably to immunotherapy. The best clinical response was to corticosteroids and plasma exchange, and the poorest response was to IVIG. Long‐term outcome is favorable in about 60% of cases. Muscle Nerve, 2009 |
| doi_str_mv | 10.1002/mus.21533 |
| format | Article |
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Twenty‐seven patients were nonresponsive to anticholinesterase therapy. Myasthenia Gravis Foundation of America improvement status was achieved in 53% patients on corticosteroids, 51% with plasma exchange, and in 20% on intravenous immunoglobulin (IVIG). Thymectomy was beneficial in 7/18 patients at 3 years. Long‐term (≥3 years) outcome was very favorable in 58% of patients who achieved remission and/or minimal manifestation status. Overall, 73% improved. There was one MG‐related death. This survey reinforces several cardinal features of MuSK‐Ab‐positive MG, including prominent bulbar involvement and anticholinesterase nonresponsiveness. Facial or tongue atrophy was rare. Most patients respond favorably to immunotherapy. The best clinical response was to corticosteroids and plasma exchange, and the poorest response was to IVIG. Long‐term outcome is favorable in about 60% of cases. Muscle Nerve, 2009</description><identifier>ISSN: 0148-639X</identifier><identifier>EISSN: 1097-4598</identifier><identifier>DOI: 10.1002/mus.21533</identifier><identifier>PMID: 19882635</identifier><identifier>CODEN: MUNEDE</identifier><language>eng</language><publisher>Hoboken: Wiley Subscription Services, Inc., A Wiley Company</publisher><subject>acetylcholine receptor antibody ; Adolescent ; Adult ; Age of Onset ; Aged ; Biological and medical sciences ; Child ; Disease-Free Survival ; Diseases of striated muscles. Neuromuscular diseases ; Electromyography ; Female ; Humans ; Immunoglobulins, Intravenous - therapeutic use ; Immunotherapy ; Male ; Medical Records ; Medical sciences ; Middle Aged ; MuSK ; myasthenia gravis ; Myasthenia Gravis - immunology ; Myasthenia Gravis - therapy ; Neurology ; Plasma Exchange ; Prednisone - therapeutic use ; Receptor Protein-Tyrosine Kinases - immunology ; Receptors, Cholinergic - immunology ; Remission Induction ; Retrospective Studies ; Severity of Illness Index ; Thymectomy ; Treatment Outcome ; United States</subject><ispartof>Muscle & nerve, 2010-03, Vol.41 (3), p.370-374</ispartof><rights>Copyright © 2009 Wiley Periodicals, Inc.</rights><rights>2015 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4903-ed8e64de6231d09701afc266e2e7813a3be2720c0929c53ffbbbac21c6e7dcd83</citedby><cites>FETCH-LOGICAL-c4903-ed8e64de6231d09701afc266e2e7813a3be2720c0929c53ffbbbac21c6e7dcd83</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fmus.21533$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fmus.21533$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,780,784,1417,27924,27925,45574,45575</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=22484182$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/19882635$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Pasnoor, Mamatha</creatorcontrib><creatorcontrib>Wolfe, Gil I.</creatorcontrib><creatorcontrib>Nations, Sharon</creatorcontrib><creatorcontrib>Trivedi, Jaya</creatorcontrib><creatorcontrib>Barohn, Richard J.</creatorcontrib><creatorcontrib>Herbelin, Laura</creatorcontrib><creatorcontrib>McVey, April</creatorcontrib><creatorcontrib>Dimachkie, Mazen</creatorcontrib><creatorcontrib>Kissel, John</creatorcontrib><creatorcontrib>Walsh, Ronan</creatorcontrib><creatorcontrib>Amato, Anthony</creatorcontrib><creatorcontrib>Mozaffar, Tahseen</creatorcontrib><creatorcontrib>Hungs, Marcel</creatorcontrib><creatorcontrib>Chui, Luis</creatorcontrib><creatorcontrib>Goldstein, Jonathan</creatorcontrib><creatorcontrib>Novella, Steven</creatorcontrib><creatorcontrib>Burns, Ted</creatorcontrib><creatorcontrib>Phillips, Lawrence</creatorcontrib><creatorcontrib>Claussen, Gwendolyn</creatorcontrib><creatorcontrib>Young, Angela</creatorcontrib><creatorcontrib>Bertorini, Tulio</creatorcontrib><creatorcontrib>Oh, Shin</creatorcontrib><title>Clinical findings in MuSK-antibody positive myasthenia gravis: A U.S. experience</title><title>Muscle & nerve</title><addtitle>Muscle Nerve</addtitle><description>We performed a retrospective chart review on 53 muscle‐specific kinase antibody (MuSK‐Ab)‐positive myasthenia gravis (MG) patients at nine university‐based centers in the U.S. Of these, 66% were Caucasian, 85% were women, and age of onset was 9–79 years. Twenty‐seven patients were nonresponsive to anticholinesterase therapy. Myasthenia Gravis Foundation of America improvement status was achieved in 53% patients on corticosteroids, 51% with plasma exchange, and in 20% on intravenous immunoglobulin (IVIG). Thymectomy was beneficial in 7/18 patients at 3 years. Long‐term (≥3 years) outcome was very favorable in 58% of patients who achieved remission and/or minimal manifestation status. Overall, 73% improved. There was one MG‐related death. This survey reinforces several cardinal features of MuSK‐Ab‐positive MG, including prominent bulbar involvement and anticholinesterase nonresponsiveness. Facial or tongue atrophy was rare. Most patients respond favorably to immunotherapy. The best clinical response was to corticosteroids and plasma exchange, and the poorest response was to IVIG. Long‐term outcome is favorable in about 60% of cases. Muscle Nerve, 2009</description><subject>acetylcholine receptor antibody</subject><subject>Adolescent</subject><subject>Adult</subject><subject>Age of Onset</subject><subject>Aged</subject><subject>Biological and medical sciences</subject><subject>Child</subject><subject>Disease-Free Survival</subject><subject>Diseases of striated muscles. Neuromuscular diseases</subject><subject>Electromyography</subject><subject>Female</subject><subject>Humans</subject><subject>Immunoglobulins, Intravenous - therapeutic use</subject><subject>Immunotherapy</subject><subject>Male</subject><subject>Medical Records</subject><subject>Medical sciences</subject><subject>Middle Aged</subject><subject>MuSK</subject><subject>myasthenia gravis</subject><subject>Myasthenia Gravis - immunology</subject><subject>Myasthenia Gravis - therapy</subject><subject>Neurology</subject><subject>Plasma Exchange</subject><subject>Prednisone - therapeutic use</subject><subject>Receptor Protein-Tyrosine Kinases - immunology</subject><subject>Receptors, Cholinergic - immunology</subject><subject>Remission Induction</subject><subject>Retrospective Studies</subject><subject>Severity of Illness Index</subject><subject>Thymectomy</subject><subject>Treatment Outcome</subject><subject>United States</subject><issn>0148-639X</issn><issn>1097-4598</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2010</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqF0E1P2zAYwHFr2jQK48AXmHyZJg4pfolfshuqRkEDNqlU9GY5zhPmLXGKnQD99mRrx07TTr78nuex_ggdUTKlhLCTdkhTRgXnr9CEkkJluSj0azQhNNeZ5MVqD-2n9IMQQrVUb9EeLbRmkosJ-jZrfPDONrj2ofLhLmEf8NWw-JLZ0PuyqzZ43SXf-wfA7cam_jsEb_FdtA8-fcKneDldTDE8rSF6CA7eoTe1bRIc7t4DtDz7fDM7zy6_zi9mp5eZywvCM6g0yLwCyTitxi8TamvHpAQGSlNueQlMMeJIwQoneF2XZWkdo06Cqlyl-QH6uN27jt39AKk3rU8OmsYG6IZklMiFUJzL_0vOFZFCq1Eeb6WLXUoRarOOvrVxYygxv0qbsbT5XXq073dbh7KF6q_cpR3Bhx2waexbRxucTy-OsVznVLPRnWzdo29g8--L5mq5-HM620741MPTy4SNP41UXAlzez03K1rMV9ezM8P4Mz6-o2s</recordid><startdate>201003</startdate><enddate>201003</enddate><creator>Pasnoor, Mamatha</creator><creator>Wolfe, Gil I.</creator><creator>Nations, Sharon</creator><creator>Trivedi, Jaya</creator><creator>Barohn, Richard J.</creator><creator>Herbelin, Laura</creator><creator>McVey, April</creator><creator>Dimachkie, Mazen</creator><creator>Kissel, John</creator><creator>Walsh, Ronan</creator><creator>Amato, Anthony</creator><creator>Mozaffar, Tahseen</creator><creator>Hungs, Marcel</creator><creator>Chui, Luis</creator><creator>Goldstein, Jonathan</creator><creator>Novella, Steven</creator><creator>Burns, Ted</creator><creator>Phillips, Lawrence</creator><creator>Claussen, Gwendolyn</creator><creator>Young, Angela</creator><creator>Bertorini, Tulio</creator><creator>Oh, Shin</creator><general>Wiley Subscription Services, Inc., A Wiley Company</general><general>Wiley</general><scope>BSCLL</scope><scope>IQODW</scope><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>7X8</scope><scope>7T5</scope><scope>7TK</scope><scope>H94</scope></search><sort><creationdate>201003</creationdate><title>Clinical findings in MuSK-antibody positive myasthenia gravis: A U.S. experience</title><author>Pasnoor, Mamatha ; Wolfe, Gil I. ; Nations, Sharon ; Trivedi, Jaya ; Barohn, Richard J. ; Herbelin, Laura ; McVey, April ; Dimachkie, Mazen ; Kissel, John ; Walsh, Ronan ; Amato, Anthony ; Mozaffar, Tahseen ; Hungs, Marcel ; Chui, Luis ; Goldstein, Jonathan ; Novella, Steven ; Burns, Ted ; Phillips, Lawrence ; Claussen, Gwendolyn ; Young, Angela ; Bertorini, Tulio ; Oh, Shin</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4903-ed8e64de6231d09701afc266e2e7813a3be2720c0929c53ffbbbac21c6e7dcd83</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2010</creationdate><topic>acetylcholine receptor antibody</topic><topic>Adolescent</topic><topic>Adult</topic><topic>Age of Onset</topic><topic>Aged</topic><topic>Biological and medical sciences</topic><topic>Child</topic><topic>Disease-Free Survival</topic><topic>Diseases of striated muscles. 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Twenty‐seven patients were nonresponsive to anticholinesterase therapy. Myasthenia Gravis Foundation of America improvement status was achieved in 53% patients on corticosteroids, 51% with plasma exchange, and in 20% on intravenous immunoglobulin (IVIG). Thymectomy was beneficial in 7/18 patients at 3 years. Long‐term (≥3 years) outcome was very favorable in 58% of patients who achieved remission and/or minimal manifestation status. Overall, 73% improved. There was one MG‐related death. This survey reinforces several cardinal features of MuSK‐Ab‐positive MG, including prominent bulbar involvement and anticholinesterase nonresponsiveness. Facial or tongue atrophy was rare. Most patients respond favorably to immunotherapy. The best clinical response was to corticosteroids and plasma exchange, and the poorest response was to IVIG. Long‐term outcome is favorable in about 60% of cases. Muscle Nerve, 2009</abstract><cop>Hoboken</cop><pub>Wiley Subscription Services, Inc., A Wiley Company</pub><pmid>19882635</pmid><doi>10.1002/mus.21533</doi><tpages>5</tpages></addata></record> |
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| subjects | acetylcholine receptor antibody Adolescent Adult Age of Onset Aged Biological and medical sciences Child Disease-Free Survival Diseases of striated muscles. Neuromuscular diseases Electromyography Female Humans Immunoglobulins, Intravenous - therapeutic use Immunotherapy Male Medical Records Medical sciences Middle Aged MuSK myasthenia gravis Myasthenia Gravis - immunology Myasthenia Gravis - therapy Neurology Plasma Exchange Prednisone - therapeutic use Receptor Protein-Tyrosine Kinases - immunology Receptors, Cholinergic - immunology Remission Induction Retrospective Studies Severity of Illness Index Thymectomy Treatment Outcome United States |
| title | Clinical findings in MuSK-antibody positive myasthenia gravis: A U.S. experience |
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