Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report 

BACKGROUND: Melkersson–Rosenthal syndrome is a rare disease characterized by the triad of recurrent orofacial swelling with facial paralysis and fissured dorsal tongue. Histologically, noncaseating granulomatous inflammation occurs that confirms the diagnosis. Overlaps between granulomatous diseases...

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Autores principales: Casper, J., Mohammad-Khani, S., Schmidt, J. J., Kielstein, J. T., Lenarz, T., Haller, H., Wagner, Annette D.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2021
Materias:
Case Report
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8489098/
https://www.ncbi.nlm.nih.gov/pubmed/34602094
http://dx.doi.org/10.1186/s13256-021-03044-5
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author Casper, J.
Mohammad-Khani, S.
Schmidt, J. J.
Kielstein, J. T.
Lenarz, T.
Haller, H.
Wagner, Annette D.
author_facet Casper, J.
Mohammad-Khani, S.
Schmidt, J. J.
Kielstein, J. T.
Lenarz, T.
Haller, H.
Wagner, Annette D.
author_sort Casper, J.
collection PubMed
description BACKGROUND: Melkersson–Rosenthal syndrome is a rare disease characterized by the triad of recurrent orofacial swelling with facial paralysis and fissured dorsal tongue. Histologically, noncaseating granulomatous inflammation occurs that confirms the diagnosis. Overlaps between granulomatous diseases such as sarcoidosis and Crohn’s disease are described. Systemic corticosteroid therapy is the treatment of choice for acute attacks. CASE PRESENTATION: We here present a case of a 59-year-old White woman suffering from Melkersson–Rosenthal syndrome with a past history of sarcoidosis on therapy with leflunomide in combination with low-dose tacrolimus successfully treated with the anti-leprosy drug clofazimine after failure of systemic steroid therapy. CONCLUSIONS: We propose clofazimine as an alternative treatment in steroid-refractory cases.
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spelling pubmed-84890982021-10-05 Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report  Casper, J. Mohammad-Khani, S. Schmidt, J. J. Kielstein, J. T. Lenarz, T. Haller, H. Wagner, Annette D. J Med Case Rep Case Report BACKGROUND: Melkersson–Rosenthal syndrome is a rare disease characterized by the triad of recurrent orofacial swelling with facial paralysis and fissured dorsal tongue. Histologically, noncaseating granulomatous inflammation occurs that confirms the diagnosis. Overlaps between granulomatous diseases such as sarcoidosis and Crohn’s disease are described. Systemic corticosteroid therapy is the treatment of choice for acute attacks. CASE PRESENTATION: We here present a case of a 59-year-old White woman suffering from Melkersson–Rosenthal syndrome with a past history of sarcoidosis on therapy with leflunomide in combination with low-dose tacrolimus successfully treated with the anti-leprosy drug clofazimine after failure of systemic steroid therapy. CONCLUSIONS: We propose clofazimine as an alternative treatment in steroid-refractory cases. BioMed Central 2021-10-04 /pmc/articles/PMC8489098/ /pubmed/34602094 http://dx.doi.org/10.1186/s13256-021-03044-5 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
Casper, J.
Mohammad-Khani, S.
Schmidt, J. J.
Kielstein, J. T.
Lenarz, T.
Haller, H.
Wagner, Annette D.
Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report 
title Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report 
title_full Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report 
title_fullStr Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report 
title_full_unstemmed Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report 
title_short Melkersson–Rosenthal syndrome in the context of sarcoidosis: a case report 
title_sort melkersson–rosenthal syndrome in the context of sarcoidosis: a case report 
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8489098/
https://www.ncbi.nlm.nih.gov/pubmed/34602094
http://dx.doi.org/10.1186/s13256-021-03044-5
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