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Paraneoplastic opsoclonus myoclonus syndrome associated with inflammatory myofibroblastic tumor in a pediatric patient
Opsoclonus myoclonus syndrome (OMS) is a rare neurological syndrome caused by a paraneoplastic autoimmune process that affects children with neuroblastic tumors. Treatment includes corticosteroids, intravenous gamma globulin (IVIG), rituximab, and other immunosuppressive therapies. Here, we describe...
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| Published in: | Pediatric blood & cancer 2020-08, Vol.67 (8), p.e28218-n/a |
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| container_start_page | e28218 |
| container_title | Pediatric blood & cancer |
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| creator | Gerstle, Karyn Siddiqui, Amir Schulte, Jefree J. Cohn, Susan L. |
| description | Opsoclonus myoclonus syndrome (OMS) is a rare neurological syndrome caused by a paraneoplastic autoimmune process that affects children with neuroblastic tumors. Treatment includes corticosteroids, intravenous gamma globulin (IVIG), rituximab, and other immunosuppressive therapies. Here, we describe a patient diagnosed with OMS associated with a localized inflammatory myofibroblastic tumor. The patient has no evidence of tumor recurrence following surgical resection with 8‐month follow‐up. The neurologic symptoms resolved with corticosteroids and IVIG. This case demonstrates that in children, neoplasms other than neuroblastoma may be associated with this paraneoplastic syndrome, and highlights the importance of evaluating patients with OMS for underlying malignancies. |
| doi_str_mv | 10.1002/pbc.28218 |
| format | article |
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Treatment includes corticosteroids, intravenous gamma globulin (IVIG), rituximab, and other immunosuppressive therapies. Here, we describe a patient diagnosed with OMS associated with a localized inflammatory myofibroblastic tumor. The patient has no evidence of tumor recurrence following surgical resection with 8‐month follow‐up. The neurologic symptoms resolved with corticosteroids and IVIG. This case demonstrates that in children, neoplasms other than neuroblastoma may be associated with this paraneoplastic syndrome, and highlights the importance of evaluating patients with OMS for underlying malignancies.</description><identifier>ISSN: 1545-5009</identifier><identifier>EISSN: 1545-5017</identifier><identifier>DOI: 10.1002/pbc.28218</identifier><identifier>PMID: 32472953</identifier><language>eng</language><publisher>United States: Wiley Subscription Services, Inc</publisher><subject>Adrenal Cortex Hormones - administration & dosage ; anaplastic lymphoma kinase ; Antineoplastic Combined Chemotherapy Protocols - administration & dosage ; Autoimmune diseases ; Child, Preschool ; Children ; Corticosteroids ; Follow-Up Studies ; Globulins ; Hematology ; Humans ; Immunoglobulins ; Immunoglobulins, Intravenous - administration & dosage ; Immunosuppressive agents ; Immunosuppressive Agents - administration & dosage ; Inflammation ; inflammatory myofibroblastic tumor ; Intravenous administration ; IVIG ; Male ; Monoclonal antibodies ; Myoclonus ; Neoplasms, Muscle Tissue - pathology ; Neoplasms, Muscle Tissue - therapy ; Neuroblastoma ; Neuromuscular diseases ; Oncology ; opsoclonus myoclonus syndrome ; Opsoclonus-Myoclonus Syndrome - pathology ; Opsoclonus-Myoclonus Syndrome - therapy ; Paraneoplastic syndrome ; Patients ; Pediatrics ; Rituximab ; Rituximab - administration & dosage ; Targeted cancer therapy ; Tumors</subject><ispartof>Pediatric blood & cancer, 2020-08, Vol.67 (8), p.e28218-n/a</ispartof><rights>2020 Wiley Periodicals, Inc.</rights><rights>2020 Wiley Periodicals LLC</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4438-b84dcec2cbc98504a47d35993eef44ab776d059b4a3f0910f0325e3460a17a423</citedby><cites>FETCH-LOGICAL-c4438-b84dcec2cbc98504a47d35993eef44ab776d059b4a3f0910f0325e3460a17a423</cites><orcidid>0000-0001-8759-120X ; 0000-0001-5749-7650</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,780,784,885,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/32472953$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Gerstle, Karyn</creatorcontrib><creatorcontrib>Siddiqui, Amir</creatorcontrib><creatorcontrib>Schulte, Jefree J.</creatorcontrib><creatorcontrib>Cohn, Susan L.</creatorcontrib><title>Paraneoplastic opsoclonus myoclonus syndrome associated with inflammatory myofibroblastic tumor in a pediatric patient</title><title>Pediatric blood & cancer</title><addtitle>Pediatr Blood Cancer</addtitle><description>Opsoclonus myoclonus syndrome (OMS) is a rare neurological syndrome caused by a paraneoplastic autoimmune process that affects children with neuroblastic tumors. Treatment includes corticosteroids, intravenous gamma globulin (IVIG), rituximab, and other immunosuppressive therapies. Here, we describe a patient diagnosed with OMS associated with a localized inflammatory myofibroblastic tumor. The patient has no evidence of tumor recurrence following surgical resection with 8‐month follow‐up. The neurologic symptoms resolved with corticosteroids and IVIG. 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Siddiqui, Amir ; Schulte, Jefree J. ; Cohn, Susan L.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4438-b84dcec2cbc98504a47d35993eef44ab776d059b4a3f0910f0325e3460a17a423</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Adrenal Cortex Hormones - administration & dosage</topic><topic>anaplastic lymphoma kinase</topic><topic>Antineoplastic Combined Chemotherapy Protocols - administration & dosage</topic><topic>Autoimmune diseases</topic><topic>Child, Preschool</topic><topic>Children</topic><topic>Corticosteroids</topic><topic>Follow-Up Studies</topic><topic>Globulins</topic><topic>Hematology</topic><topic>Humans</topic><topic>Immunoglobulins</topic><topic>Immunoglobulins, Intravenous - administration & dosage</topic><topic>Immunosuppressive agents</topic><topic>Immunosuppressive Agents - administration & dosage</topic><topic>Inflammation</topic><topic>inflammatory myofibroblastic tumor</topic><topic>Intravenous administration</topic><topic>IVIG</topic><topic>Male</topic><topic>Monoclonal antibodies</topic><topic>Myoclonus</topic><topic>Neoplasms, Muscle Tissue - pathology</topic><topic>Neoplasms, Muscle Tissue - therapy</topic><topic>Neuroblastoma</topic><topic>Neuromuscular diseases</topic><topic>Oncology</topic><topic>opsoclonus myoclonus syndrome</topic><topic>Opsoclonus-Myoclonus Syndrome - pathology</topic><topic>Opsoclonus-Myoclonus Syndrome - therapy</topic><topic>Paraneoplastic syndrome</topic><topic>Patients</topic><topic>Pediatrics</topic><topic>Rituximab</topic><topic>Rituximab - administration & dosage</topic><topic>Targeted cancer therapy</topic><topic>Tumors</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Gerstle, Karyn</creatorcontrib><creatorcontrib>Siddiqui, Amir</creatorcontrib><creatorcontrib>Schulte, Jefree J.</creatorcontrib><creatorcontrib>Cohn, Susan L.</creatorcontrib><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>Oncogenes and Growth Factors Abstracts</collection><collection>Technology Research Database</collection><collection>Engineering Research Database</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Pediatric blood & cancer</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Gerstle, Karyn</au><au>Siddiqui, Amir</au><au>Schulte, Jefree J.</au><au>Cohn, Susan L.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Paraneoplastic opsoclonus myoclonus syndrome associated with inflammatory myofibroblastic tumor in a pediatric patient</atitle><jtitle>Pediatric blood & cancer</jtitle><addtitle>Pediatr Blood Cancer</addtitle><date>2020-08</date><risdate>2020</risdate><volume>67</volume><issue>8</issue><spage>e28218</spage><epage>n/a</epage><pages>e28218-n/a</pages><issn>1545-5009</issn><eissn>1545-5017</eissn><abstract>Opsoclonus myoclonus syndrome (OMS) is a rare neurological syndrome caused by a paraneoplastic autoimmune process that affects children with neuroblastic tumors. Treatment includes corticosteroids, intravenous gamma globulin (IVIG), rituximab, and other immunosuppressive therapies. Here, we describe a patient diagnosed with OMS associated with a localized inflammatory myofibroblastic tumor. The patient has no evidence of tumor recurrence following surgical resection with 8‐month follow‐up. The neurologic symptoms resolved with corticosteroids and IVIG. This case demonstrates that in children, neoplasms other than neuroblastoma may be associated with this paraneoplastic syndrome, and highlights the importance of evaluating patients with OMS for underlying malignancies.</abstract><cop>United States</cop><pub>Wiley Subscription Services, Inc</pub><pmid>32472953</pmid><doi>10.1002/pbc.28218</doi><tpages>3</tpages><orcidid>https://orcid.org/0000-0001-8759-120X</orcidid><orcidid>https://orcid.org/0000-0001-5749-7650</orcidid><oa>free_for_read</oa></addata></record> |
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| subjects | Adrenal Cortex Hormones - administration & dosage anaplastic lymphoma kinase Antineoplastic Combined Chemotherapy Protocols - administration & dosage Autoimmune diseases Child, Preschool Children Corticosteroids Follow-Up Studies Globulins Hematology Humans Immunoglobulins Immunoglobulins, Intravenous - administration & dosage Immunosuppressive agents Immunosuppressive Agents - administration & dosage Inflammation inflammatory myofibroblastic tumor Intravenous administration IVIG Male Monoclonal antibodies Myoclonus Neoplasms, Muscle Tissue - pathology Neoplasms, Muscle Tissue - therapy Neuroblastoma Neuromuscular diseases Oncology opsoclonus myoclonus syndrome Opsoclonus-Myoclonus Syndrome - pathology Opsoclonus-Myoclonus Syndrome - therapy Paraneoplastic syndrome Patients Pediatrics Rituximab Rituximab - administration & dosage Targeted cancer therapy Tumors |
| title | Paraneoplastic opsoclonus myoclonus syndrome associated with inflammatory myofibroblastic tumor in a pediatric patient |
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