Another promising treatment option for neuroblastoma-associated opsoclonus–myoclonus syndrome by oral high-dose dexamethasone pulse: Lymphocyte markers as disease activity
Abstract A one-year-old boy with neuroblastoma (NBoma)-associated opsoclonus–myoclonus syndrome (OMS) was treated by oral high-dose dexamethasone (DEX) pulses (20 mg/m2 /day of DEX for three consecutive days) every 28 days for 6 months after resection of the tumor. All OMS symptoms improved after th...
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Veröffentlicht in: | Brain & development (Tokyo. 1979) 2012-03, Vol.34 (3), p.251-254 |
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creator | Oguma, Makiko Morimoto, Akira Takada, Akiko Kashii, Yoshifumi Fukuda, Tokiko Mori, Masato Yamagata, Takanori Sugie, Hideo Momoi, Mariko Y |
description | Abstract A one-year-old boy with neuroblastoma (NBoma)-associated opsoclonus–myoclonus syndrome (OMS) was treated by oral high-dose dexamethasone (DEX) pulses (20 mg/m2 /day of DEX for three consecutive days) every 28 days for 6 months after resection of the tumor. All OMS symptoms improved after the first course of DEX pulse therapy and disappeared after the last course. No adverse effects were observed. Minor deterioration of his developmental quotient was noted 33 months after the onset of the disease. NBoma remission has been maintained since treatment. Before DEX pulse therapy, frequency of T lymphocyte, in particular CD4-positive cell decreased markedly resulted in low CD4/8 ratio in the peripheral blood (PB). The frequency of B lymphocyte increased, especially in cerebrospinal fluid. These aberrant values in PB were reversed by DEX pulse therapy and correlated well with the neurological symptoms. A prospective study that assesses the efficacy of this promising and inexpensive treatment for OMS is warranted. |
doi_str_mv | 10.1016/j.braindev.2011.04.005 |
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All OMS symptoms improved after the first course of DEX pulse therapy and disappeared after the last course. No adverse effects were observed. Minor deterioration of his developmental quotient was noted 33 months after the onset of the disease. NBoma remission has been maintained since treatment. Before DEX pulse therapy, frequency of T lymphocyte, in particular CD4-positive cell decreased markedly resulted in low CD4/8 ratio in the peripheral blood (PB). The frequency of B lymphocyte increased, especially in cerebrospinal fluid. These aberrant values in PB were reversed by DEX pulse therapy and correlated well with the neurological symptoms. A prospective study that assesses the efficacy of this promising and inexpensive treatment for OMS is warranted.</description><identifier>ISSN: 0387-7604</identifier><identifier>EISSN: 1872-7131</identifier><identifier>DOI: 10.1016/j.braindev.2011.04.005</identifier><identifier>PMID: 21531096</identifier><language>eng</language><publisher>Netherlands: Elsevier B.V</publisher><subject>Anti-Inflammatory Agents - administration & dosage ; B-Lymphocytes - immunology ; Biomarkers - blood ; CD4-Positive T-Lymphocytes - immunology ; CD4/CD8 ratio ; CD8-Positive T-Lymphocytes - immunology ; Child, Preschool ; Dexamethasone ; Dexamethasone - administration & dosage ; Humans ; Kidney Neoplasms - complications ; Kidney Neoplasms - immunology ; Male ; Neuroblastoma ; Neuroblastoma - complications ; Neuroblastoma - immunology ; Neurology ; Opsoclonus-Myoclonus Syndrome - drug therapy ; Opsoclonus-Myoclonus Syndrome - etiology ; Opsoclonus-Myoclonus Syndrome - immunology ; Opsoclonus–myoclonus syndrome</subject><ispartof>Brain & development (Tokyo. 1979), 2012-03, Vol.34 (3), p.251-254</ispartof><rights>The Japanese Society of Child Neurology</rights><rights>2011 The Japanese Society of Child Neurology</rights><rights>Copyright © 2011 The Japanese Society of Child Neurology. 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All rights reserved.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c507t-3cea19307587175ba88200cabd9a977e061c91c9e26018a779aad867020435673</citedby><cites>FETCH-LOGICAL-c507t-3cea19307587175ba88200cabd9a977e061c91c9e26018a779aad867020435673</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S0387760411000945$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3536,27903,27904,65309</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/21531096$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Oguma, Makiko</creatorcontrib><creatorcontrib>Morimoto, Akira</creatorcontrib><creatorcontrib>Takada, Akiko</creatorcontrib><creatorcontrib>Kashii, Yoshifumi</creatorcontrib><creatorcontrib>Fukuda, Tokiko</creatorcontrib><creatorcontrib>Mori, Masato</creatorcontrib><creatorcontrib>Yamagata, Takanori</creatorcontrib><creatorcontrib>Sugie, Hideo</creatorcontrib><creatorcontrib>Momoi, Mariko Y</creatorcontrib><title>Another promising treatment option for neuroblastoma-associated opsoclonus–myoclonus syndrome by oral high-dose dexamethasone pulse: Lymphocyte markers as disease activity</title><title>Brain & development (Tokyo. 1979)</title><addtitle>Brain Dev</addtitle><description>Abstract A one-year-old boy with neuroblastoma (NBoma)-associated opsoclonus–myoclonus syndrome (OMS) was treated by oral high-dose dexamethasone (DEX) pulses (20 mg/m2 /day of DEX for three consecutive days) every 28 days for 6 months after resection of the tumor. All OMS symptoms improved after the first course of DEX pulse therapy and disappeared after the last course. No adverse effects were observed. Minor deterioration of his developmental quotient was noted 33 months after the onset of the disease. NBoma remission has been maintained since treatment. Before DEX pulse therapy, frequency of T lymphocyte, in particular CD4-positive cell decreased markedly resulted in low CD4/8 ratio in the peripheral blood (PB). The frequency of B lymphocyte increased, especially in cerebrospinal fluid. These aberrant values in PB were reversed by DEX pulse therapy and correlated well with the neurological symptoms. A prospective study that assesses the efficacy of this promising and inexpensive treatment for OMS is warranted.</description><subject>Anti-Inflammatory Agents - administration & dosage</subject><subject>B-Lymphocytes - immunology</subject><subject>Biomarkers - blood</subject><subject>CD4-Positive T-Lymphocytes - immunology</subject><subject>CD4/CD8 ratio</subject><subject>CD8-Positive T-Lymphocytes - immunology</subject><subject>Child, Preschool</subject><subject>Dexamethasone</subject><subject>Dexamethasone - administration & dosage</subject><subject>Humans</subject><subject>Kidney Neoplasms - complications</subject><subject>Kidney Neoplasms - immunology</subject><subject>Male</subject><subject>Neuroblastoma</subject><subject>Neuroblastoma - complications</subject><subject>Neuroblastoma - immunology</subject><subject>Neurology</subject><subject>Opsoclonus-Myoclonus Syndrome - drug therapy</subject><subject>Opsoclonus-Myoclonus Syndrome - etiology</subject><subject>Opsoclonus-Myoclonus Syndrome - immunology</subject><subject>Opsoclonus–myoclonus syndrome</subject><issn>0387-7604</issn><issn>1872-7131</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2012</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqNks-O0zAQxiMEYsvCK6x845QykzRxwgGxWvFPqsQBOFsTZ7p1N7GL7VTkxjvwHLwUT4KrdjlwAcmS5_CbGc33fVl2hbBEwPrFbtl5Mrbnw7IAxCWslgDVg2yBjSxyiSU-zBZQNjKXNawusich7AAAC4TH2UWBVYnQ1ovs57V1ccte7L0bTTD2VkTPFEe2Ubh9NM6KjfPC8uRdN1CIbqScQnDaUOQ-MakcnJ3Cr-8_xvlcizDbPk1k0c3CeRrE1txu894FFj1_o5HjloKzLPbTEPilWM_jfuv0HFmM5O_YB0FB9CYwpRbS0RxMnJ9mjzaU-Gfn_zL78vbN55v3-frjuw831-tcVyBjXmombEuQVSNRVh01TQGgqetbaqVkqFG36XFRAzYkZUvUN7WEAlZlVcvyMnt-mptU-TpxiCppo3kYyLKbgmqLugWJTfUfJMqilGWdyPpEau9C8LxRe2_SrbNCUEdP1U7de6qOnipYqeRparw6r5i6kfs_bfcmJuD1CeAkycGwV0Ebtpp741lH1Tvz7x2v_hqhB2ONpuGOZw47N3mbBFeoQqFAfTom6xgsxBSqdlWVvwHmVNC0</recordid><startdate>20120301</startdate><enddate>20120301</enddate><creator>Oguma, Makiko</creator><creator>Morimoto, Akira</creator><creator>Takada, Akiko</creator><creator>Kashii, Yoshifumi</creator><creator>Fukuda, Tokiko</creator><creator>Mori, Masato</creator><creator>Yamagata, Takanori</creator><creator>Sugie, Hideo</creator><creator>Momoi, Mariko Y</creator><general>Elsevier 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>7X8</scope><scope>7T5</scope><scope>7TK</scope><scope>H94</scope></search><sort><creationdate>20120301</creationdate><title>Another promising treatment option for neuroblastoma-associated opsoclonus–myoclonus syndrome by oral high-dose dexamethasone pulse: Lymphocyte markers as disease activity</title><author>Oguma, Makiko ; 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All OMS symptoms improved after the first course of DEX pulse therapy and disappeared after the last course. No adverse effects were observed. Minor deterioration of his developmental quotient was noted 33 months after the onset of the disease. NBoma remission has been maintained since treatment. Before DEX pulse therapy, frequency of T lymphocyte, in particular CD4-positive cell decreased markedly resulted in low CD4/8 ratio in the peripheral blood (PB). The frequency of B lymphocyte increased, especially in cerebrospinal fluid. These aberrant values in PB were reversed by DEX pulse therapy and correlated well with the neurological symptoms. A prospective study that assesses the efficacy of this promising and inexpensive treatment for OMS is warranted.</abstract><cop>Netherlands</cop><pub>Elsevier B.V</pub><pmid>21531096</pmid><doi>10.1016/j.braindev.2011.04.005</doi><tpages>4</tpages></addata></record> |
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subjects | Anti-Inflammatory Agents - administration & dosage B-Lymphocytes - immunology Biomarkers - blood CD4-Positive T-Lymphocytes - immunology CD4/CD8 ratio CD8-Positive T-Lymphocytes - immunology Child, Preschool Dexamethasone Dexamethasone - administration & dosage Humans Kidney Neoplasms - complications Kidney Neoplasms - immunology Male Neuroblastoma Neuroblastoma - complications Neuroblastoma - immunology Neurology Opsoclonus-Myoclonus Syndrome - drug therapy Opsoclonus-Myoclonus Syndrome - etiology Opsoclonus-Myoclonus Syndrome - immunology Opsoclonus–myoclonus syndrome |
title | Another promising treatment option for neuroblastoma-associated opsoclonus–myoclonus syndrome by oral high-dose dexamethasone pulse: Lymphocyte markers as disease activity |
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