Cystic Neutrophilic Granulomatous Mastitis Regression with the Tumor Necrosis Factor-α Inhibitor, Adalimumab

Chiu, Le Wen; Goodwin, Denise; Vohra, Poonam; Amerson, Erin

doi:10.4274/ejbh.galenos.2021.2021-7-2

Cited by 12 publications

(8 citation statements)

References 15 publications

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“…Given that erythema nodosum is mostly secondary to various autoimmune diseases [43] , erythema nodosum and cough may be the systemic signs that indicate CNGM is an autoimmune disease. Studies have reported that patients with CNGM have achieved better therapeutic effects after taking steroids or immunosuppressive drugs such as methotrexate, confirming that CNGM is an autoimmune disease [44, 45] .…”

Section: Discussionmentioning

confidence: 98%

Clinical features of cystic neutrophil granulomatous mastitis in 62 cases

Wang,

Zhang,

et al. 2023

Preprint

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BackgroundCystic Neutrophilic Granulomatous Mastitis (CNGM) is a rare inflammatory condition affecting the breast. Despite its rarity, understanding its pathogenesis and clinical features is crucial for accurate diagnosis and effective management. This study delves into the nuanced aspects of CNGM, shedding light on its unique characteristics and potential underlying mechanisms. Methods: In this meticulous investigation, we meticulously examined and analyzed the biological data, clinical features, ultrasound imaging findings, and histopathological morphological information of 62 patients diagnosed with CNGM after thorough pathological examination. The study cohort was sourced from the Galactophore department of Beijing Hospital of Traditional Chinese Medicine, spanning the period from September 2019 to September 2022. Results: Sixty-two patients, with an average age of 33.30 years, were predominantly female. Among the 52 patients with detailed documentation of onset following the final delivery, various factors were identified, including hyperprolactinemia, pituitary tumors, psychiatric medication history, granulomatous mastitis history, breast trauma history, and a family history of breast cancer. The primary clinical manifestations were characterized by pain and palpable masses, accompanied by localized symptoms such as redness, ulceration, nipple discharge, and nipple retraction. Additionally, systemic symptoms, such as fever, headache, erythema nodosum, and cough, were observed. Ultrasound examinations revealed predominantly hypoechoic masses with heterogeneous echogenicity. Axillary lymphadenopathy, dilated ducts, and thickening of breast tissue were also noted in some cases. Histopathological analyses demonstrated lobular structural destruction, acute and chronic inflammatory cell infiltration, multinucleated giant cell reactions, granulomas, and cyst formation. Gram staining revealed detection rates of 41.94% (26/62) for gram-positive bacteria and 11.29% (7/62) for gram-negative bacteria. Conclusion: This study highlights the occurrence of Chronic Nonspecific Granulomatous Mastitis (CNGM) in women of childbearing age. Factors such as milk stasis, mammary duct secretion overcharge, exogenous trauma, hormonal influences, and bacterial colonization are implicated in the initiation and recurrence of CNGM. Notably, nipple retraction emerged not only as a clinical symptom but also as a potential risk factor for CNGM. The prevalence of multiple hypoechoic regions in CNGM surpassed that observed in breast cancer cases. The detection of gram-positive bacteria underscores the pivotal role of bacterial infections in the development of CNGM.

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Section: Discussionmentioning

confidence: 98%

Clinical features of cystic neutrophil granulomatous mastitis in 62 cases

Wang,

Zhang,

et al. 2023

Preprint

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“…GLM is considered to be an autoimmune inflammatory disease due to the marked clinical treatment effect of immunosuppressants. Chiu et al ( 29 ) reported that the breast symptoms of patients with GLM resolved following treatment with the tumor necrosis factor (TNF)-α inhibitor adalimumab. TNF-α is involved in systemic inflammation and is one of the cytokines that stimulate the acute-phase response.…”

Section: Discussionmentioning

confidence: 99%

Differential expression profiles of miRNA in granulomatous lobular mastitis and identification of possible biomarkers

et al. 2022

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The etiology and pathogenesis of granulomatous lobular mastitis (GLM) remain largely elusive and the expression levels and regulatory roles of microRNAs (miRNAs or miRs) in GLM have remained mostly undetermined. In the present study, the miRNAs that were differentially expressed in breast biopsy samples from patients with GLM and normal tissue adjacent to fibroadenoma were analyzed, a comprehensive differential expression profile of miRNAs was provided and potential biomarkers were screened out. The expression profile of miRNAs was determined by high-throughput sequencing in the tissues of patients with GLM and healthy controls. Significantly differentially expressed miRNAs were screened by threshold setting and cluster analysis and their target genes were analyzed by Gene Ontology (GO) and Kyoto Encyclopedia of Genes and Genomes (KEGG) enrichment. Finally, circulating differentially expressed miRNAs between the GLM and control groups were further analyzed by reverse transcription-quantitative PCR (RT-qPCR). A total of 31,077 miRNAs were detected by high-throughput sequencing. By using the cutoff criteria of |log2 fold change|>2.5 and q<0.001, 13 miRNAs that were indicated to be GLM biomarkers were screened out. The expression levels of these 13 miRNAs in the GLM group were higher than those in the control group. GO and KEGG enrichment analyses suggested that the occurrence and development of GLM may be associated with autoimmune inflammation, metabolism and pathogenic organisms. miR-451a and miR-5571-3p were confirmed to be significantly increased in the serum of patients with GLM compared with their levels in the serum of healthy volunteers, which suggests that they may be used as biomarkers of GLM. To the best of our knowledge, the present study was the first report detailing genome-wide miRNA profiling of patients with GLM compared with controls. The possible targets and pathways of GLM were evaluated by bioinformatics analysis. The present study identified 13 differentially expressed miRNAs with important theoretical significance and potential application. Furthermore, miR-451a and miR-5571-3p were verified by RT-qPCR as possible biomarkers of GLM.

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“…CNGM typically occurs in parous women during the post-lactational period and is more prevalent among Māori, Pacific Islanders, Hispanic, Chilean, and Southeast Asian populations [ 6 ]. While CNGM and IGM share an identical clinical presentation, they differ in histological presentation [ 7 ]. Therefore, clinical and histologic evaluation are required to confirm the CNGM diagnosis.…”

Section: Discussionmentioning

confidence: 99%

Cystic Neutrophilic Granulomatous Mastitis Treatment with Consecutive Dapsone and Adalimumab

2022

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Cystic neutrophilic granulomatous mastitis is a rarely reported, benign, mastitis that is not associated with lactation. The pathophysiology is still unknown; however, there is often a relationship with Corynebacterium kroppenstedtii. Cystic neutrophilic mastitis can have deep seated tender nodules and cutaneous ulceration which can clinically resemble pyoderma gangrenous. It can be treatment refractory and can progress to a point where mastectomy is required. This case series reports two patients treated with adalimumab with remission of disease. One patient first received dapsone with remission of symptoms, but incompatible side effects resulted in discontinuation. Both dapsone and adalimumab appear to provide disease remission in patients with cystic neutrophilic granulomatous mastitis.

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Cystic Neutrophilic Granulomatous Mastitis Regression with the Tumor Necrosis Factor-α Inhibitor, Adalimumab

Cited by 12 publications

References 15 publications

Clinical features of cystic neutrophil granulomatous mastitis in 62 cases

Clinical features of cystic neutrophil granulomatous mastitis in 62 cases

Differential expression profiles of miRNA in granulomatous lobular mastitis and identification of possible biomarkers

Cystic Neutrophilic Granulomatous Mastitis Treatment with Consecutive Dapsone and Adalimumab

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