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Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy

Meningoencephalitis is a rare complication of relapsing polychondritis. We report a case of a 25-year-old male who presented with visual hallucinations and symptoms of depression and anxiety, white matter changes on MRI, and CSF lymphocytosis, along with inflammatory chondritis seen in his auricle c...

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Autores principales: Al-Tabbaa, Mohammad Mousbah, Habal, Hani
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6192138/
https://www.ncbi.nlm.nih.gov/pubmed/30402308
http://dx.doi.org/10.1155/2018/1873582
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author Al-Tabbaa, Mohammad Mousbah
Habal, Hani
author_facet Al-Tabbaa, Mohammad Mousbah
Habal, Hani
author_sort Al-Tabbaa, Mohammad Mousbah
collection PubMed
description Meningoencephalitis is a rare complication of relapsing polychondritis. We report a case of a 25-year-old male who presented with visual hallucinations and symptoms of depression and anxiety, white matter changes on MRI, and CSF lymphocytosis, along with inflammatory chondritis seen in his auricle cartilage biopsy. Eventually he was given the diagnosis of RP presenting with meningoencephalitis based on CSF analysis, brain MRI findings, negative serologies, and neurologic exam findings. The patient's clinical state did not improve despite being on IV methylprednisolone for a period of 7 days; afterwards he was switched to oral prednisone with no clinical improvement. As a result, he was given cyclophosphamide and rituximab, respectively, without benefit. He also underwent craniectomy with VP shunt due to worsening hydrocephalus and a brain biopsy was done to confirm the diagnosis. He is currently on methotrexate and steroid dependent with a goal to taper down. Even though all 19 reported cases of meningoencephalitis with RP in the literature did respond to immunosuppressive therapy, in our case, however the patient did not respond to immunosuppressive treatment and currently is in mute dementia status after three years of treatment.
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spelling pubmed-61921382018-11-06 Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy Al-Tabbaa, Mohammad Mousbah Habal, Hani Case Rep Neurol Med Case Report Meningoencephalitis is a rare complication of relapsing polychondritis. We report a case of a 25-year-old male who presented with visual hallucinations and symptoms of depression and anxiety, white matter changes on MRI, and CSF lymphocytosis, along with inflammatory chondritis seen in his auricle cartilage biopsy. Eventually he was given the diagnosis of RP presenting with meningoencephalitis based on CSF analysis, brain MRI findings, negative serologies, and neurologic exam findings. The patient's clinical state did not improve despite being on IV methylprednisolone for a period of 7 days; afterwards he was switched to oral prednisone with no clinical improvement. As a result, he was given cyclophosphamide and rituximab, respectively, without benefit. He also underwent craniectomy with VP shunt due to worsening hydrocephalus and a brain biopsy was done to confirm the diagnosis. He is currently on methotrexate and steroid dependent with a goal to taper down. Even though all 19 reported cases of meningoencephalitis with RP in the literature did respond to immunosuppressive therapy, in our case, however the patient did not respond to immunosuppressive treatment and currently is in mute dementia status after three years of treatment. Hindawi 2018-10-03 /pmc/articles/PMC6192138/ /pubmed/30402308 http://dx.doi.org/10.1155/2018/1873582 Text en Copyright © 2018 Mohammad Mousbah Al-Tabbaa and Hani Habal. https://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Al-Tabbaa, Mohammad Mousbah
Habal, Hani
Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy
title Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy
title_full Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy
title_fullStr Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy
title_full_unstemmed Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy
title_short Relapsing Polychondritis with Meningoencephalitis Refractory to Immunosuppressant Therapy
title_sort relapsing polychondritis with meningoencephalitis refractory to immunosuppressant therapy
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6192138/
https://www.ncbi.nlm.nih.gov/pubmed/30402308
http://dx.doi.org/10.1155/2018/1873582
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