Efficacy of immunotherapy in seropositive and seronegative putative autoimmune autonomic ganglionopathy
To evaluate the efficacy of immunotherapy in the treatment of patients with seropositive and seronegative putative autoimmune autonomic ganglionopathy (AAG) using validated autonomic function tests and instruments. AAG is an immune-mediated disorder characterized by prominent and selective involveme...
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| Veröffentlicht in: | Neurology 2009-06, Vol.72 (23), p.2002-2008 |
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| creator | IODICE, V KIMPINSKI, K VERNINO, S SANDRONI, P FEALEY, R. D LOW, P. A |
| description | To evaluate the efficacy of immunotherapy in the treatment of patients with seropositive and seronegative putative autoimmune autonomic ganglionopathy (AAG) using validated autonomic function tests and instruments.
AAG is an immune-mediated disorder characterized by prominent and selective involvement of autonomic nerve fibers or ganglia. Treatment with i.v. immunoglobulin (IVIg) or plasma exchange (PE) has been reported to be effective in single case reports.
We studied six patients, four with seropositive and two with seronegative putative AAG, who underwent autonomic function tests and completed two validated questionnaires, to assess autonomic symptoms before and after immunomodulatory treatment. Patients were treated with standard doses of IVIg, PE, or immunosuppressants in a specific sequential therapy protocol depending on clinical response.
Of the six patients (all women, mean ages 49.3 +/- 10.6 years), four patients were ganglionic (alpha3) AChR autoantibody positive and two were autoantibody negative. All patients showed clinical improvement after treatment. Sudomotor function assessed by quantitative sudomotor axon reflex test and thermoregulatory sweat test improved in four patients after treatment.
Immunomodulatory treatment can be effective in both seropositive and seronegative putative autoimmune autonomic ganglionopathy. Plasma exchange or combined therapy with immunosuppressive agents should be considered in patients who do not benefit from i.v. immunoglobulin alone. |
| doi_str_mv | 10.1212/wnl.0b013e3181a92b52 |
| format | Article |
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AAG is an immune-mediated disorder characterized by prominent and selective involvement of autonomic nerve fibers or ganglia. Treatment with i.v. immunoglobulin (IVIg) or plasma exchange (PE) has been reported to be effective in single case reports.
We studied six patients, four with seropositive and two with seronegative putative AAG, who underwent autonomic function tests and completed two validated questionnaires, to assess autonomic symptoms before and after immunomodulatory treatment. Patients were treated with standard doses of IVIg, PE, or immunosuppressants in a specific sequential therapy protocol depending on clinical response.
Of the six patients (all women, mean ages 49.3 +/- 10.6 years), four patients were ganglionic (alpha3) AChR autoantibody positive and two were autoantibody negative. All patients showed clinical improvement after treatment. Sudomotor function assessed by quantitative sudomotor axon reflex test and thermoregulatory sweat test improved in four patients after treatment.
Immunomodulatory treatment can be effective in both seropositive and seronegative putative autoimmune autonomic ganglionopathy. Plasma exchange or combined therapy with immunosuppressive agents should be considered in patients who do not benefit from i.v. immunoglobulin alone.</description><identifier>ISSN: 0028-3878</identifier><identifier>EISSN: 1526-632X</identifier><identifier>DOI: 10.1212/wnl.0b013e3181a92b52</identifier><identifier>PMID: 19506222</identifier><identifier>CODEN: NEURAI</identifier><language>eng</language><publisher>Hagerstown, MD: Lippincott Williams & Wilkins</publisher><subject>Adult ; Aged ; Autoantibodies - analysis ; Autoantibodies - blood ; Autonomic Nervous System Diseases - blood ; Autonomic Nervous System Diseases - drug therapy ; Autonomic Nervous System Diseases - immunology ; Biological and medical sciences ; Body Temperature Regulation - drug effects ; Body Temperature Regulation - immunology ; Female ; Ganglia, Autonomic - drug effects ; Ganglia, Autonomic - immunology ; Ganglia, Autonomic - physiopathology ; Humans ; Immunoglobulins, Intravenous - administration & dosage ; Immunotherapy - methods ; Immunotherapy - statistics & numerical data ; Medical sciences ; Middle Aged ; Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis ; Neurology ; Plasmapheresis - statistics & numerical data ; Polyradiculoneuropathy - blood ; Polyradiculoneuropathy - drug therapy ; Polyradiculoneuropathy - immunology ; Receptors, Nicotinic - immunology ; Surveys and Questionnaires ; Sweat Glands - innervation ; Sweat Glands - physiopathology ; Treatment Outcome</subject><ispartof>Neurology, 2009-06, Vol.72 (23), p.2002-2008</ispartof><rights>2009 INIST-CNRS</rights><rights>Copyright © 2009 by AAN Enterprises, Inc.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c533t-5b6ebf0145b7b50e94302e68ca64dff35dafec8a219ca3e53cac2f58183d99043</citedby><cites>FETCH-LOGICAL-c533t-5b6ebf0145b7b50e94302e68ca64dff35dafec8a219ca3e53cac2f58183d99043</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,776,780,881,27901,27902</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=21669660$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/19506222$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>IODICE, V</creatorcontrib><creatorcontrib>KIMPINSKI, K</creatorcontrib><creatorcontrib>VERNINO, S</creatorcontrib><creatorcontrib>SANDRONI, P</creatorcontrib><creatorcontrib>FEALEY, R. D</creatorcontrib><creatorcontrib>LOW, P. A</creatorcontrib><title>Efficacy of immunotherapy in seropositive and seronegative putative autoimmune autonomic ganglionopathy</title><title>Neurology</title><addtitle>Neurology</addtitle><description>To evaluate the efficacy of immunotherapy in the treatment of patients with seropositive and seronegative putative autoimmune autonomic ganglionopathy (AAG) using validated autonomic function tests and instruments.
AAG is an immune-mediated disorder characterized by prominent and selective involvement of autonomic nerve fibers or ganglia. Treatment with i.v. immunoglobulin (IVIg) or plasma exchange (PE) has been reported to be effective in single case reports.
We studied six patients, four with seropositive and two with seronegative putative AAG, who underwent autonomic function tests and completed two validated questionnaires, to assess autonomic symptoms before and after immunomodulatory treatment. Patients were treated with standard doses of IVIg, PE, or immunosuppressants in a specific sequential therapy protocol depending on clinical response.
Of the six patients (all women, mean ages 49.3 +/- 10.6 years), four patients were ganglionic (alpha3) AChR autoantibody positive and two were autoantibody negative. All patients showed clinical improvement after treatment. Sudomotor function assessed by quantitative sudomotor axon reflex test and thermoregulatory sweat test improved in four patients after treatment.
Immunomodulatory treatment can be effective in both seropositive and seronegative putative autoimmune autonomic ganglionopathy. Plasma exchange or combined therapy with immunosuppressive agents should be considered in patients who do not benefit from i.v. immunoglobulin alone.</description><subject>Adult</subject><subject>Aged</subject><subject>Autoantibodies - analysis</subject><subject>Autoantibodies - blood</subject><subject>Autonomic Nervous System Diseases - blood</subject><subject>Autonomic Nervous System Diseases - drug therapy</subject><subject>Autonomic Nervous System Diseases - immunology</subject><subject>Biological and medical sciences</subject><subject>Body Temperature Regulation - drug effects</subject><subject>Body Temperature Regulation - immunology</subject><subject>Female</subject><subject>Ganglia, Autonomic - drug effects</subject><subject>Ganglia, Autonomic - immunology</subject><subject>Ganglia, Autonomic - physiopathology</subject><subject>Humans</subject><subject>Immunoglobulins, Intravenous - administration & dosage</subject><subject>Immunotherapy - methods</subject><subject>Immunotherapy - statistics & numerical data</subject><subject>Medical sciences</subject><subject>Middle Aged</subject><subject>Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis</subject><subject>Neurology</subject><subject>Plasmapheresis - statistics & numerical data</subject><subject>Polyradiculoneuropathy - blood</subject><subject>Polyradiculoneuropathy - drug therapy</subject><subject>Polyradiculoneuropathy - immunology</subject><subject>Receptors, Nicotinic - immunology</subject><subject>Surveys and Questionnaires</subject><subject>Sweat Glands - innervation</subject><subject>Sweat Glands - physiopathology</subject><subject>Treatment Outcome</subject><issn>0028-3878</issn><issn>1526-632X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2009</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFUU1v1DAUtBCILoV_gKpc6C3FH7FjXyqhqqWVVnChKjfrxbGzRomd2kmr_feku6tCuXB6XzOjeRqEPhJ8Riihnx9Df4YbTJhlRBJQtOH0FVoRTkUpGP35Gq0wprJkspZH6F3OvzBejrV6i46I4lhQSleou3TOGzDbIrrCD8Mc4rSxCcZt4UORbYpjzH7yD7aA0O4WwXawW4zztG9gnuKOu29DHLwpOghd75dhhGmzfY_eOOiz_XCox-j26vLHxXW5_v715uLLujScsankjbCNw6TiTd1wbFXFMLVCGhBV6xzjLThrJFCiDDDL2WKdOi6JZK1SuGLH6HyvO87NYFtjw5Sg12PyA6StjuD1y0vwG93FB00lq7kii8DpQSDF-9nmSQ8-G9v3EGycsxY1qzBl4r9ASjCTsnqyVO2BJsWck3XPbgjWT1Hqu29r_W-UC-3k70_-kA7ZLYBPBwBkA71LEIzPzzhKhFBCYPYbxcmtPA</recordid><startdate>20090609</startdate><enddate>20090609</enddate><creator>IODICE, V</creator><creator>KIMPINSKI, K</creator><creator>VERNINO, S</creator><creator>SANDRONI, P</creator><creator>FEALEY, R. D</creator><creator>LOW, P. A</creator><general>Lippincott Williams & Wilkins</general><general>American Academy of Neurology</general><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>7T5</scope><scope>7TK</scope><scope>H94</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20090609</creationdate><title>Efficacy of immunotherapy in seropositive and seronegative putative autoimmune autonomic ganglionopathy</title><author>IODICE, V ; KIMPINSKI, K ; VERNINO, S ; SANDRONI, P ; FEALEY, R. D ; LOW, P. A</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c533t-5b6ebf0145b7b50e94302e68ca64dff35dafec8a219ca3e53cac2f58183d99043</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2009</creationdate><topic>Adult</topic><topic>Aged</topic><topic>Autoantibodies - analysis</topic><topic>Autoantibodies - blood</topic><topic>Autonomic Nervous System Diseases - blood</topic><topic>Autonomic Nervous System Diseases - drug therapy</topic><topic>Autonomic Nervous System Diseases - immunology</topic><topic>Biological and medical sciences</topic><topic>Body Temperature Regulation - drug effects</topic><topic>Body Temperature Regulation - immunology</topic><topic>Female</topic><topic>Ganglia, Autonomic - drug effects</topic><topic>Ganglia, Autonomic - immunology</topic><topic>Ganglia, Autonomic - physiopathology</topic><topic>Humans</topic><topic>Immunoglobulins, Intravenous - administration & dosage</topic><topic>Immunotherapy - methods</topic><topic>Immunotherapy - statistics & numerical data</topic><topic>Medical sciences</topic><topic>Middle Aged</topic><topic>Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis</topic><topic>Neurology</topic><topic>Plasmapheresis - statistics & numerical data</topic><topic>Polyradiculoneuropathy - blood</topic><topic>Polyradiculoneuropathy - drug therapy</topic><topic>Polyradiculoneuropathy - immunology</topic><topic>Receptors, Nicotinic - immunology</topic><topic>Surveys and Questionnaires</topic><topic>Sweat Glands - innervation</topic><topic>Sweat Glands - physiopathology</topic><topic>Treatment Outcome</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>IODICE, V</creatorcontrib><creatorcontrib>KIMPINSKI, K</creatorcontrib><creatorcontrib>VERNINO, S</creatorcontrib><creatorcontrib>SANDRONI, P</creatorcontrib><creatorcontrib>FEALEY, R. D</creatorcontrib><creatorcontrib>LOW, P. A</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Neurosciences Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Neurology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>IODICE, V</au><au>KIMPINSKI, K</au><au>VERNINO, S</au><au>SANDRONI, P</au><au>FEALEY, R. D</au><au>LOW, P. A</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Efficacy of immunotherapy in seropositive and seronegative putative autoimmune autonomic ganglionopathy</atitle><jtitle>Neurology</jtitle><addtitle>Neurology</addtitle><date>2009-06-09</date><risdate>2009</risdate><volume>72</volume><issue>23</issue><spage>2002</spage><epage>2008</epage><pages>2002-2008</pages><issn>0028-3878</issn><eissn>1526-632X</eissn><coden>NEURAI</coden><abstract>To evaluate the efficacy of immunotherapy in the treatment of patients with seropositive and seronegative putative autoimmune autonomic ganglionopathy (AAG) using validated autonomic function tests and instruments.
AAG is an immune-mediated disorder characterized by prominent and selective involvement of autonomic nerve fibers or ganglia. Treatment with i.v. immunoglobulin (IVIg) or plasma exchange (PE) has been reported to be effective in single case reports.
We studied six patients, four with seropositive and two with seronegative putative AAG, who underwent autonomic function tests and completed two validated questionnaires, to assess autonomic symptoms before and after immunomodulatory treatment. Patients were treated with standard doses of IVIg, PE, or immunosuppressants in a specific sequential therapy protocol depending on clinical response.
Of the six patients (all women, mean ages 49.3 +/- 10.6 years), four patients were ganglionic (alpha3) AChR autoantibody positive and two were autoantibody negative. All patients showed clinical improvement after treatment. Sudomotor function assessed by quantitative sudomotor axon reflex test and thermoregulatory sweat test improved in four patients after treatment.
Immunomodulatory treatment can be effective in both seropositive and seronegative putative autoimmune autonomic ganglionopathy. Plasma exchange or combined therapy with immunosuppressive agents should be considered in patients who do not benefit from i.v. immunoglobulin alone.</abstract><cop>Hagerstown, MD</cop><pub>Lippincott Williams & Wilkins</pub><pmid>19506222</pmid><doi>10.1212/wnl.0b013e3181a92b52</doi><tpages>7</tpages><oa>free_for_read</oa></addata></record> |
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| ispartof | Neurology, 2009-06, Vol.72 (23), p.2002-2008 |
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| subjects | Adult Aged Autoantibodies - analysis Autoantibodies - blood Autonomic Nervous System Diseases - blood Autonomic Nervous System Diseases - drug therapy Autonomic Nervous System Diseases - immunology Biological and medical sciences Body Temperature Regulation - drug effects Body Temperature Regulation - immunology Female Ganglia, Autonomic - drug effects Ganglia, Autonomic - immunology Ganglia, Autonomic - physiopathology Humans Immunoglobulins, Intravenous - administration & dosage Immunotherapy - methods Immunotherapy - statistics & numerical data Medical sciences Middle Aged Multiple sclerosis and variants. Guillain barré syndrome and other inflammatory polyneuropathies. Leukoencephalitis Neurology Plasmapheresis - statistics & numerical data Polyradiculoneuropathy - blood Polyradiculoneuropathy - drug therapy Polyradiculoneuropathy - immunology Receptors, Nicotinic - immunology Surveys and Questionnaires Sweat Glands - innervation Sweat Glands - physiopathology Treatment Outcome |
| title | Efficacy of immunotherapy in seropositive and seronegative putative autoimmune autonomic ganglionopathy |
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