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A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate

Multicentric reticulohistiocytosis (MRH) is categorized as a rare non-Langerhans cell histiocytosis most commonly seen in women in the fourth to fifth decade of life. This systemic inflammatory condition affects multiple organ systems and can result in severe joint destruction which can progress to...

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Autores principales: Patel, Suraj, Alhajj, Mandy, Brimhall, Conrad
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Author(s). Published by S. Karger AG 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9827444/
https://www.ncbi.nlm.nih.gov/pubmed/36632441
http://dx.doi.org/10.1159/000528254
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author Patel, Suraj
Alhajj, Mandy
Brimhall, Conrad
author_facet Patel, Suraj
Alhajj, Mandy
Brimhall, Conrad
author_sort Patel, Suraj
collection PubMed
description Multicentric reticulohistiocytosis (MRH) is categorized as a rare non-Langerhans cell histiocytosis most commonly seen in women in the fourth to fifth decade of life. This systemic inflammatory condition affects multiple organ systems and can result in severe joint destruction which can progress to arthritis mutilans. To date, various underlying malignancies have been discovered in patients with MRH including breast, gastric, thymic, hepatic, and melanoma. There has been 1 case of underlying renal cell carcinoma reported in a patient diagnosed with MRH. Additionally, there is no consistently recognized treatment for MRH described in the literature. The rarity of the disease contributes to the difficulty in defining a standardized treatment. We present the case of a patient with extensive joint and skin involvement who was successfully treated with infliximab and methotrexate, experienced clinical improvement, and was later diagnosed with clear cell renal cell carcinoma. The synergistic effects of infliximab and methotrexate, in combination with the low side-effect profile, appear to be promising in the setting of MRH and in our patient resulted in the resolution of symptoms and cutaneous manifestations. We suggest this regimen as an effective combination therapy. We emphasize thorough and continuous screening for underlying malignancy associated with MRH, despite clinical improvement or negative malignancy work-up upon initial diagnosis.
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spelling pubmed-98274442023-01-10 A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate Patel, Suraj Alhajj, Mandy Brimhall, Conrad Case Rep Dermatol Case Report Multicentric reticulohistiocytosis (MRH) is categorized as a rare non-Langerhans cell histiocytosis most commonly seen in women in the fourth to fifth decade of life. This systemic inflammatory condition affects multiple organ systems and can result in severe joint destruction which can progress to arthritis mutilans. To date, various underlying malignancies have been discovered in patients with MRH including breast, gastric, thymic, hepatic, and melanoma. There has been 1 case of underlying renal cell carcinoma reported in a patient diagnosed with MRH. Additionally, there is no consistently recognized treatment for MRH described in the literature. The rarity of the disease contributes to the difficulty in defining a standardized treatment. We present the case of a patient with extensive joint and skin involvement who was successfully treated with infliximab and methotrexate, experienced clinical improvement, and was later diagnosed with clear cell renal cell carcinoma. The synergistic effects of infliximab and methotrexate, in combination with the low side-effect profile, appear to be promising in the setting of MRH and in our patient resulted in the resolution of symptoms and cutaneous manifestations. We suggest this regimen as an effective combination therapy. We emphasize thorough and continuous screening for underlying malignancy associated with MRH, despite clinical improvement or negative malignancy work-up upon initial diagnosis. The Author(s). Published by S. Karger AG 2023-01-09 /pmc/articles/PMC9827444/ /pubmed/36632441 http://dx.doi.org/10.1159/000528254 Text en © 2023 The Author(s). Published by S. Karger AG, Basel https://creativecommons.org/licenses/by-nc/4.0/This article is licensed under the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC) (http://www.karger.com/Services/OpenAccessLicense). Usage and distribution for commercial purposes requires written permission.
spellingShingle Case Report
Patel, Suraj
Alhajj, Mandy
Brimhall, Conrad
A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate
title A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate
title_full A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate
title_fullStr A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate
title_full_unstemmed A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate
title_short A Novel Case of Multicentric Reticulohistiocytosis Associated with Renal Cell Carcinoma Successfully Treated with Infliximab and Methotrexate
title_sort novel case of multicentric reticulohistiocytosis associated with renal cell carcinoma successfully treated with infliximab and methotrexate
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9827444/
https://www.ncbi.nlm.nih.gov/pubmed/36632441
http://dx.doi.org/10.1159/000528254
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