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Recurrent autoimmune hypophysitis treated with rituximab: a case report

BACKGROUND: Autoimmune hypophysitis is a rare condition that often results in enlargement of the pituitary gland and hypopituitarism due to inflammatory infiltration. Management of autoimmune hypophysitis can include long-term hormonal replacement and close control of the inflammatory pituitary mass...

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Autores principales: Kruse, Maria, Olesen, Thomas Bastholm, Markovic, Ljubo, Glintborg, Dorte, Andersen, Marianne Skovsager
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8672614/
https://www.ncbi.nlm.nih.gov/pubmed/34906226
http://dx.doi.org/10.1186/s13256-021-03146-0
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author Kruse, Maria
Olesen, Thomas Bastholm
Markovic, Ljubo
Glintborg, Dorte
Andersen, Marianne Skovsager
author_facet Kruse, Maria
Olesen, Thomas Bastholm
Markovic, Ljubo
Glintborg, Dorte
Andersen, Marianne Skovsager
author_sort Kruse, Maria
collection PubMed
description BACKGROUND: Autoimmune hypophysitis is a rare condition that often results in enlargement of the pituitary gland and hypopituitarism due to inflammatory infiltration. Management of autoimmune hypophysitis can include long-term hormonal replacement and close control of the inflammatory pituitary mass. Mass-related symptoms in patients with autoimmune hypophysitis are treated with anti-inflammatory therapy, surgery, and/or radiotherapy. CASE PRESENTATION: We present a 25-year-old White man with visual field defects of the right eye, headache, and weight loss. Magnetic resonance imaging showed a sellar mass, and the patient underwent transcranial surgery. Histopathology revealed autoimmune hypophysitis with predominantly CD20 positive B-cell infiltration. Progression of visual field defects necessitated postoperatively anti-inflammatory treatment with prednisolone. Azathioprine was initiated under gradual tapering of prednisolone with stable conditions at first, but relapse followed after dose reduction. Therefore, rituximab treatment was initiated, which resulted in regression of the pituitary mass. Rituximab treatment was discontinued after 25 months. The patient has continuously been in remission for 4 years after rituximab treatment was stopped. CONCLUSION: This case illustrates that rituximab might be an effective alternative treatment in B-cell predominant autoimmune hypophysitis.
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spelling pubmed-86726142021-12-17 Recurrent autoimmune hypophysitis treated with rituximab: a case report Kruse, Maria Olesen, Thomas Bastholm Markovic, Ljubo Glintborg, Dorte Andersen, Marianne Skovsager J Med Case Rep Case Report BACKGROUND: Autoimmune hypophysitis is a rare condition that often results in enlargement of the pituitary gland and hypopituitarism due to inflammatory infiltration. Management of autoimmune hypophysitis can include long-term hormonal replacement and close control of the inflammatory pituitary mass. Mass-related symptoms in patients with autoimmune hypophysitis are treated with anti-inflammatory therapy, surgery, and/or radiotherapy. CASE PRESENTATION: We present a 25-year-old White man with visual field defects of the right eye, headache, and weight loss. Magnetic resonance imaging showed a sellar mass, and the patient underwent transcranial surgery. Histopathology revealed autoimmune hypophysitis with predominantly CD20 positive B-cell infiltration. Progression of visual field defects necessitated postoperatively anti-inflammatory treatment with prednisolone. Azathioprine was initiated under gradual tapering of prednisolone with stable conditions at first, but relapse followed after dose reduction. Therefore, rituximab treatment was initiated, which resulted in regression of the pituitary mass. Rituximab treatment was discontinued after 25 months. The patient has continuously been in remission for 4 years after rituximab treatment was stopped. CONCLUSION: This case illustrates that rituximab might be an effective alternative treatment in B-cell predominant autoimmune hypophysitis. BioMed Central 2021-12-15 /pmc/articles/PMC8672614/ /pubmed/34906226 http://dx.doi.org/10.1186/s13256-021-03146-0 Text en © The Author(s) 2021 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
Kruse, Maria
Olesen, Thomas Bastholm
Markovic, Ljubo
Glintborg, Dorte
Andersen, Marianne Skovsager
Recurrent autoimmune hypophysitis treated with rituximab: a case report
title Recurrent autoimmune hypophysitis treated with rituximab: a case report
title_full Recurrent autoimmune hypophysitis treated with rituximab: a case report
title_fullStr Recurrent autoimmune hypophysitis treated with rituximab: a case report
title_full_unstemmed Recurrent autoimmune hypophysitis treated with rituximab: a case report
title_short Recurrent autoimmune hypophysitis treated with rituximab: a case report
title_sort recurrent autoimmune hypophysitis treated with rituximab: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8672614/
https://www.ncbi.nlm.nih.gov/pubmed/34906226
http://dx.doi.org/10.1186/s13256-021-03146-0
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