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Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker

INTRODUCTION: Autoantibody-mediated autoimmunity directed against targets within the voltage-gated potassium channel complex (VGKCC autoantibodies) has been implicated in causing neuropathic pain. METHODS: We report the case of a 76-year-old, United Kingdom male who was diagnosed with contactin-asso...

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Autores principales: Goebel, Andreas, Moore, Austen Peter, Jacob, Anu
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6181468/
https://www.ncbi.nlm.nih.gov/pubmed/30534626
http://dx.doi.org/10.1097/PR9.0000000000000675
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author Goebel, Andreas
Moore, Austen Peter
Jacob, Anu
author_facet Goebel, Andreas
Moore, Austen Peter
Jacob, Anu
author_sort Goebel, Andreas
collection PubMed
description INTRODUCTION: Autoantibody-mediated autoimmunity directed against targets within the voltage-gated potassium channel complex (VGKCC autoantibodies) has been implicated in causing neuropathic pain. METHODS: We report the case of a 76-year-old, United Kingdom male who was diagnosed with contactin-associated protein 2 (CASPR2) autoantibody–associated Morvan syndrome, a rare neurological condition. RESULTS: He had previously worked in a swine abattoir; exposure to aerosol within swine abattoirs has been reported to elicit an immune response resulting in the production of these autoantibodies; however, unusually, his manifestations emerged with several years' latency. Although this patient's Morvan syndrome–associated seizures were well-controlled with antiepileptic drugs, his neuropathic pain and painful muscle fasciculations did not respond to pharmacological interventions. He refused pain management program treatment, but high-dose immunoglobulin treatment or treatment with rituximab, reported to be sometimes effective in this group, was not initiated because of concerns regarding his general frailty. DISCUSSION AND CONCLUSION: This case highlights issues around the identification and treatment of rare patients with chronic pain who have voltage-gated potassium channel complex autoantibodies; it also emphasizes the possibility that former swine abattoir workers might be at risk of developing neuropathic pain even years after their vocational exposure.
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spelling pubmed-61814682018-12-10 Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker Goebel, Andreas Moore, Austen Peter Jacob, Anu Pain Rep Neuropathic INTRODUCTION: Autoantibody-mediated autoimmunity directed against targets within the voltage-gated potassium channel complex (VGKCC autoantibodies) has been implicated in causing neuropathic pain. METHODS: We report the case of a 76-year-old, United Kingdom male who was diagnosed with contactin-associated protein 2 (CASPR2) autoantibody–associated Morvan syndrome, a rare neurological condition. RESULTS: He had previously worked in a swine abattoir; exposure to aerosol within swine abattoirs has been reported to elicit an immune response resulting in the production of these autoantibodies; however, unusually, his manifestations emerged with several years' latency. Although this patient's Morvan syndrome–associated seizures were well-controlled with antiepileptic drugs, his neuropathic pain and painful muscle fasciculations did not respond to pharmacological interventions. He refused pain management program treatment, but high-dose immunoglobulin treatment or treatment with rituximab, reported to be sometimes effective in this group, was not initiated because of concerns regarding his general frailty. DISCUSSION AND CONCLUSION: This case highlights issues around the identification and treatment of rare patients with chronic pain who have voltage-gated potassium channel complex autoantibodies; it also emphasizes the possibility that former swine abattoir workers might be at risk of developing neuropathic pain even years after their vocational exposure. Wolters Kluwer 2018-09-17 /pmc/articles/PMC6181468/ /pubmed/30534626 http://dx.doi.org/10.1097/PR9.0000000000000675 Text en Copyright © 2018 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of The International Association for the Study of Pain. This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY) (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Neuropathic
Goebel, Andreas
Moore, Austen Peter
Jacob, Anu
Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker
title Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker
title_full Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker
title_fullStr Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker
title_full_unstemmed Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker
title_short Delayed onset of severe chronic pain in CASPR2 autoantibody–associated Morvan syndrome in a former UK swine abattoir worker
title_sort delayed onset of severe chronic pain in caspr2 autoantibody–associated morvan syndrome in a former uk swine abattoir worker
topic Neuropathic
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6181468/
https://www.ncbi.nlm.nih.gov/pubmed/30534626
http://dx.doi.org/10.1097/PR9.0000000000000675
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