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A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab

A 36-year-old woman with a 6-month history of painful unilateral mammary nodules and abscesses was initially treated by gynecologists under the diagnosis of idiopathic granulomatous mastitis (IGM). IGM is an entity that has not been described in classical dermatological textbooks and is considered a...

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Detalles Bibliográficos
Autores principales: Breznik, Vesna, Marko, Pij Bogomir
Formato: Online Artículo Texto
Lenguaje:English
Publicado: S. Karger AG 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9149353/
https://www.ncbi.nlm.nih.gov/pubmed/35702373
http://dx.doi.org/10.1159/000523801
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author Breznik, Vesna
Marko, Pij Bogomir
author_facet Breznik, Vesna
Marko, Pij Bogomir
author_sort Breznik, Vesna
collection PubMed
description A 36-year-old woman with a 6-month history of painful unilateral mammary nodules and abscesses was initially treated by gynecologists under the diagnosis of idiopathic granulomatous mastitis (IGM). IGM is an entity that has not been described in classical dermatological textbooks and is considered a rare inflammatory breast disease assumingly associated with trauma, infection, or autoimmune system manifestations. In this patient, the lesions were refractory to conventional treatment of IGM comprising of surgical incisions, systemic antibiotics, dexamethasone, and methotrexate. At the initial visit to the dermatology department, a working diagnosis of localized hidradenitis suppurativa (HS) of breast was established, and treatment with systemic doxycycline was initiated. After the diagnostic incisional biopsy, the inflamed nodule deteriorated into a painful ulceration, implying a pathergy phenomenon. Histopathological features were consistent with the granulomatous type of pyoderma gangrenosum (PG). Treatment with systemic methylprednisolone and mycophenolate mofetil was unsuccessful. Subsequently, the patient developed nodules in the inguinal and axillary areas, typical for HS. Finally, adalimumab treatment resulted in the complete resolution of all lesions without relapse even after the biologic therapy was discontinued. Although in this case, IGM was not confirmed histopathologically, we noted several etiopathological and therapeutic similarities between IGM, PG, and HS and summarized them in a unique table. Further observations are needed to ascertain the potential associations among the three entities.
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spelling pubmed-91493532022-06-13 A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab Breznik, Vesna Marko, Pij Bogomir Case Rep Dermatol Single Case A 36-year-old woman with a 6-month history of painful unilateral mammary nodules and abscesses was initially treated by gynecologists under the diagnosis of idiopathic granulomatous mastitis (IGM). IGM is an entity that has not been described in classical dermatological textbooks and is considered a rare inflammatory breast disease assumingly associated with trauma, infection, or autoimmune system manifestations. In this patient, the lesions were refractory to conventional treatment of IGM comprising of surgical incisions, systemic antibiotics, dexamethasone, and methotrexate. At the initial visit to the dermatology department, a working diagnosis of localized hidradenitis suppurativa (HS) of breast was established, and treatment with systemic doxycycline was initiated. After the diagnostic incisional biopsy, the inflamed nodule deteriorated into a painful ulceration, implying a pathergy phenomenon. Histopathological features were consistent with the granulomatous type of pyoderma gangrenosum (PG). Treatment with systemic methylprednisolone and mycophenolate mofetil was unsuccessful. Subsequently, the patient developed nodules in the inguinal and axillary areas, typical for HS. Finally, adalimumab treatment resulted in the complete resolution of all lesions without relapse even after the biologic therapy was discontinued. Although in this case, IGM was not confirmed histopathologically, we noted several etiopathological and therapeutic similarities between IGM, PG, and HS and summarized them in a unique table. Further observations are needed to ascertain the potential associations among the three entities. S. Karger AG 2022-05-03 /pmc/articles/PMC9149353/ /pubmed/35702373 http://dx.doi.org/10.1159/000523801 Text en Copyright © 2022 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 Single Case
Breznik, Vesna
Marko, Pij Bogomir
A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab
title A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab
title_full A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab
title_fullStr A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab
title_full_unstemmed A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab
title_short A Case of Overlapping Clinical Features of Idiopathic Granulomatous Mastitis, Hidradenitis Suppurativa, and Pyoderma Gangrenosum Successfully Treated with Adalimumab
title_sort case of overlapping clinical features of idiopathic granulomatous mastitis, hidradenitis suppurativa, and pyoderma gangrenosum successfully treated with adalimumab
topic Single Case
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9149353/
https://www.ncbi.nlm.nih.gov/pubmed/35702373
http://dx.doi.org/10.1159/000523801
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