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Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report

BACKGROUND: Autoantibodies targeting node of Ranvier proteins are rarely reported in China. CASE PRESENTATION: We present the case of a 66-year-old Chinese man who concomitantly developed chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duc...

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Autores principales: Hu, Shurong, Hu, Yin, Du, Qiang
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8830009/
https://www.ncbi.nlm.nih.gov/pubmed/35139901
http://dx.doi.org/10.1186/s13256-022-03277-y
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author Hu, Shurong
Hu, Yin
Du, Qiang
author_facet Hu, Shurong
Hu, Yin
Du, Qiang
author_sort Hu, Shurong
collection PubMed
description BACKGROUND: Autoantibodies targeting node of Ranvier proteins are rarely reported in China. CASE PRESENTATION: We present the case of a 66-year-old Chinese man who concomitantly developed chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in liver, which are rarely reported in China. The man presented with chronic progressive sensory and motor symptoms, bilateral periphery facial paralysis, and protein–cell dissociation of cerebrospinal fluid. Nerve conduction study indicated demyelinating neuropathy. Enhanced magnetic resonance imaging of the liver showed diffuse intrahepatic lesions, which were considered as bile duct hamartomas in the liver. He was suspected as having chronic inflammatory demyelinating polyneuropathy and treated with intravenous immunoglobulin and prednisone. However, his condition got worse. One month later, he was diagnosed with chronic inflammatory demyelinating polyneuropathy associated with anti-contactin-associated protein 1 antibody. He received high-dose methylprednisolone, followed by standard plasma exchange and rituximab therapy. His sensory and motor manifestations were significantly improved at 1 year of follow-up. CONCLUSIONS: This case reminds clinicians to be aware of antiparanodal antibodies, which are associated with specific phenotypes and therapeutic response.
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spelling pubmed-88300092022-02-11 Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report Hu, Shurong Hu, Yin Du, Qiang J Med Case Rep Case Report BACKGROUND: Autoantibodies targeting node of Ranvier proteins are rarely reported in China. CASE PRESENTATION: We present the case of a 66-year-old Chinese man who concomitantly developed chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in liver, which are rarely reported in China. The man presented with chronic progressive sensory and motor symptoms, bilateral periphery facial paralysis, and protein–cell dissociation of cerebrospinal fluid. Nerve conduction study indicated demyelinating neuropathy. Enhanced magnetic resonance imaging of the liver showed diffuse intrahepatic lesions, which were considered as bile duct hamartomas in the liver. He was suspected as having chronic inflammatory demyelinating polyneuropathy and treated with intravenous immunoglobulin and prednisone. However, his condition got worse. One month later, he was diagnosed with chronic inflammatory demyelinating polyneuropathy associated with anti-contactin-associated protein 1 antibody. He received high-dose methylprednisolone, followed by standard plasma exchange and rituximab therapy. His sensory and motor manifestations were significantly improved at 1 year of follow-up. CONCLUSIONS: This case reminds clinicians to be aware of antiparanodal antibodies, which are associated with specific phenotypes and therapeutic response. BioMed Central 2022-02-10 /pmc/articles/PMC8830009/ /pubmed/35139901 http://dx.doi.org/10.1186/s13256-022-03277-y Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Hu, Shurong
Hu, Yin
Du, Qiang
Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report
title Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report
title_full Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report
title_fullStr Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report
title_full_unstemmed Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report
title_short Chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report
title_sort chronic inflammatory demyelinating polyneuropathy with anti-contactin-associated protein 1 antibody and bile duct hamartomas in the liver: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8830009/
https://www.ncbi.nlm.nih.gov/pubmed/35139901
http://dx.doi.org/10.1186/s13256-022-03277-y
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AT duqiang chronicinflammatorydemyelinatingpolyneuropathywithanticontactinassociatedprotein1antibodyandbileducthamartomasintheliveracasereport