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Ozcinar B, Ocak Z, Billur D, Ertugrul B, Timirci-Kahraman O. Whole-Exome Sequencing: Discovering Genetic Causes of Granulomatous Mastitis. Int J Mol Sci 2025; 26:425. [PMID: 39796280 PMCID: PMC11721990 DOI: 10.3390/ijms26010425] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/23/2024] [Revised: 01/03/2025] [Accepted: 01/04/2025] [Indexed: 01/30/2025] Open
Abstract
Granulomatous mastitis (GM) is a rare, benign, but chronic and recurrent inflammatory breast disease that significantly impacts physical and psychological well-being. It often presents symptoms such as pain, swelling, and discharge, leading to diagnostic confusion with malignancy. The etiology of GM remains unclear, though autoimmune and multifactorial components are suspected. This study aimed to explore the genetic underpinnings of GM using whole-exome sequencing (WES) on 22 GM patients and 52 healthy controls to identify single nucleotide variants (SNVs) and copy number variations (CNVs) potentially linked to the disease. WES analysis revealed novel SNVs in six genes: BRCA2 (rs169547), CFTR (rs4727853), NCF1 (rs10614), PTPN22 (rs2476601), HLA-DRB1 (seven variants), and C3 (rs406514). Notably, most of these variants are associated with immune regulation and inflammatory pathways, supporting the hypothesis that GM is an autoimmune disease. However, all identified variants were classified as benign according to the American College of Medical Genetics and Genomics (ACMG) guidelines, necessitating further investigation into their potential functional effects. Despite conducting CNV analysis, no significant variations were identified. This study represents a foundational step in linking genetic predisposition to GM and highlights the need for integrating genetic, clinical, and functional data to better understand GM's pathophysiology. Future research should focus on larger cohorts, functional studies, and exploring multifactorial contributors to GM, including hormonal and environmental factors.
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Affiliation(s)
- Beyza Ozcinar
- Department of General Surgery, Istanbul Medical Faculty, Istanbul University, 34093 Istanbul, Türkiye;
| | - Zeynep Ocak
- Department of Medical Genetics, Medical Faculty, Istinye University, 34396 Istanbul, Türkiye;
| | - Deryanaz Billur
- Department of Molecular Medicine, Aziz Sancar Institute of Experimental Medicine, Istanbul University, 34093 Istanbul, Türkiye; (D.B.); (B.E.)
- Department of Molecular Medicine, Institute of Graduate Studies in Health Sciences, Istanbul University, 34093 Istanbul, Türkiye
| | - Baris Ertugrul
- Department of Molecular Medicine, Aziz Sancar Institute of Experimental Medicine, Istanbul University, 34093 Istanbul, Türkiye; (D.B.); (B.E.)
| | - Ozlem Timirci-Kahraman
- Department of Molecular Medicine, Aziz Sancar Institute of Experimental Medicine, Istanbul University, 34093 Istanbul, Türkiye; (D.B.); (B.E.)
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Dogan S, Dal F, Guler M, Sevik H, Oguz Idiz U. Is peripheral blood immunophenotyping useful to understand the etiology of Idiopathic Granulomatous? Hum Immunol 2023:S0198-8859(23)00070-8. [PMID: 37202243 DOI: 10.1016/j.humimm.2023.05.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/29/2022] [Revised: 05/01/2023] [Accepted: 05/01/2023] [Indexed: 05/20/2023]
Abstract
BACKGROUND The etiology of idiopathic granulomatous mastitis (IGM) has not been clearly established. However, autoimmunity has recently become popular in etiopathogenesis. We aimed to investigate the immunophenotyping of immune cells to help clarify the etiopathogenesis of the disease. METHODS Patients with IGM and healthy volunteers were included in the study. Patients were divided into active and remission groups based on their disease status. The ratios of total T cells, helper T cells, cytotoxic T cells, natural killer cells, regulatory T cells, and monocyte subtypes were measured using flow cytometry. In addition, age, complete blood count for leukocyte, lymphocyte, neutrophil, and eosinophil counts, and the smoking status of all volunteers were evaluated. RESULTS A total of 33 volunteers, including 11 patients with active IGM, 10 patients with remission IGM, and 12 healthy volunteers, were included in the study. The neutrophil, eosinophil, neutrophil/lymphocyte, and non-classical monocyte values were significantly higher in IGM patients than in healthy volunteers. Additionally, the CD4+ CD25+ CD127- regulatory T cell was significantly lower in IGM patients than in healthy volunteers. Furthermore, neutrophil, neutrophil/lymphocyte ratio, CD4+ CD25+ CD127- regulatory T cells, and non-classical monocytes showed significant differences when IGM patients were divided into active and remission groups. IGM patients had higher smoking rates, but this was not statistically significant. CONCLUSION The changes in many cell types evaluated in our study were similar to the cell profiles of some autoimmune diseases. This could provide minor evidence to suggest that IGM is an autoimmune granulomatous disease with a local course.
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Affiliation(s)
- Selim Dogan
- Istanbul Teaching and Research Hospital, Department of General Surgery, Turkey
| | - Fatih Dal
- Istanbul Teaching and Research Hospital, Department of General Surgery, Turkey
| | - Mert Guler
- Istanbul Teaching and Research Hospital, Department of General Surgery, Turkey
| | - Husnu Sevik
- Istanbul Teaching and Research Hospital, Department of General Surgery, Turkey.
| | - Ufuk Oguz Idiz
- Istanbul Teaching and Research Hospital, Department of General Surgery, Turkey
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Laor L, Ganguli S, Fakioglu E. Granulomatous mastitis, erythema nodosum, and polyarthritis: a case report. J Med Case Rep 2022; 16:146. [PMID: 35382864 PMCID: PMC8985372 DOI: 10.1186/s13256-022-03327-5] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/24/2020] [Accepted: 02/11/2022] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Granulomatous mastitis is a rare inflammatory disease of the breast, typically seen in woman of child-bearing age. No definitive etiology has been described. In rare instances, this condition has been reported to be associated with extramammary manifestations such as erythema nodosum and arthritis. We describe this rare condition in an adolescent female. CASE PRESENTATION A 16-year-old, Hispanic female presented with right-sided painful breast swelling, polyarthritis, and erythema nodosum on bilateral shins and lower thighs. Physical examination was negative for lymphadenopathy and pulmonary, gastrointestinal, and cardiovascular findings. Ophthalmologic examination for uveitis and serologic tests for autoimmune diseases were negative. Diagnosis of idiopathic granulomatous mastitis was made by exclusion of other etiologies and conditions. Confirmation was made by histopathologic examination demonstrating noncaseating granuloma within breast lobules with neutrophils and microabscess formation. After wide local excision and a short course of trimethoprim-sulfamethoxazole, our patient was placed on naproxen and prednisone, the latter being tapered off over 3 months, with steady and complete resolution of all symptoms. CONCLUSION This is the first reported case of idiopathic granulomatous mastitis in a pediatric patient who also had extramammary manifestations, including erythema nodosum and polyarthritis. In this case-based review, we summarize the phenotype, risk factors, prognosis, and treatment options of this rare condition, chiefly to make the readers cognizant of such a diagnostic possibility in similar clinical presentation in the future.
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Affiliation(s)
- Leanna Laor
- Flushing Hospital Medical Center, 4500 Parsons Boulevard, Flushing, NY, 11355, USA.
| | - Suhas Ganguli
- Pediatric Rheumatology Marshfield Clinic, 1000 North Oak Ave, Suite 1A1,, Marshfield, WI, 5449, USA
| | - Esra Fakioglu
- Flushing Hospital Medical Center, 4500 Parsons Boulevard, Flushing, NY, 11355, USA
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Zhu Q, Wang L, Wang P. The Identification of Gene Expression Profiles Associated with Granulomatous Mastitis. Breast Care (Basel) 2021; 16:319-327. [PMID: 34602937 DOI: 10.1159/000507474] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/28/2019] [Accepted: 03/24/2020] [Indexed: 12/13/2022] Open
Abstract
Background Granulomatous mastitis (GM) is a rare chronic inflammatory disease of the breast. The current therapeutic effects of the antibiotic therapy and surgical or immunomodulatory (steroid) treatment are normally poor due to the unclear etiology. Method This study aimed to identify the differentially expressed mRNAs in GM tissues using RNA sequencing and further explored the functions of differentially expressed mRNAs resulting in GM. Moreover, we revealed the relationship between GM and breast cancer by shared highly expressed genes in GM tissues and breast cancer tissues. Results A total of 12,115 mRNAs were analyzed in the whole expression profile, and 207 mRNAs (136 upregulated and 71 downregulated mRNAs) were differently expressed between the GM tissues and normal tissues. The enrichment analysis showed that the differentially expressed mRNAs were enriched in the biological processes and played a significant role in the immune system. Besides, the genes expressed significantly highly in breast cancer tissues are found to be enriched with GM genes, which may explain the similar clinical features between breast cancer and GM. We also found that the HSD11B1 gene which was differentially expressed in GM was used as drug target of prednisone, which is a common treatment for GM. Conclusion This study is the first to use sequencing technology to elucidate the genetic mechanisms of GM. The finding of this study may have potential value in GM diagnosis and also provides potential drug targets for GM treatment.
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Affiliation(s)
- Qiang Zhu
- Department of Breast Surgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Lin Wang
- Department of Breast Surgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
| | - Pilin Wang
- Department of Breast Surgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
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Saydam M, Yilmaz KB, Sahin M, Yanik H, Akinci M, Yilmaz I, Balas S, Azili C, Gulcelik MA. New Findings on Autoimmune Etiology of Idiopathic Granulomatous Mastitis: Serum IL-17, IL-22 and IL-23 Levels of Patients. J INVEST SURG 2021; 34:993-997. [PMID: 32046543 DOI: 10.1080/08941939.2020.1725190] [Citation(s) in RCA: 33] [Impact Index Per Article: 8.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/19/2022]
Abstract
BACKGROUND Idiopathic Granulomatous Mastitis (IGM) is a benign chronic inflammatory breast disease that mimics breast cancer, and the etiopathogenesis has not yet been fully evaluated. Autoimmunity has received the most focus as a possible etiology. Our aim in this prospective clinical study was to investigate the possible association between the cytokines, interleukin IL-17, IL-22, IL-23 and IGM. MATERIALS AND METHODS The current study was conducted in 26 women with histopathologically diagnosed IGM, and 15 control women of reproductive age having no breast disease history. Blood samples were collected, and serum concentrations of IL-17, IL-22, and IL-23 were determined. RESULTS In the analysis of variables, the patients with IGM and the control group had statistically significant differences between serum IL-22 titers (p = 0.0378) and IL-23 titers (p = 0.0469. No statistically significant difference was found between IGM patients and the control group in serum IL-17 titers (p = 0.9724). CONCLUSION The results of the current study, especially pertaining to serum IL-22 and IL-23 levels, support the etiopathogenesis of IGM in favor of the autoinflammatory thesis. Nevertheless, this thesis should be supported by a large case number and prospective clinical studies.
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Affiliation(s)
- Mehmet Saydam
- Department of General Surgery, University of Health Sciences, Ankara Diskapi Yildirim Beyazit Training and Research Hospital, Ankara, Turkey
| | - Kerim Bora Yilmaz
- Department of General Surgery, University of Health Sciences, Gulhane Training and Research Hospital, Ankara, Turkey
- Department of Medical and Surgical Research, University of Health Sciences, Hacettepe University, Ankara, Turkey
| | - Mutlu Sahin
- Department of General Surgery, University of Health Sciences, Ankara Kecioren Training and Research Hospital, Ankara, Turkey
| | - Hamdullah Yanik
- Department of Basic Oncology, Cancer Institute, Hacettepe University, Ankara, Turkey
| | - Melih Akinci
- Department of General Surgery, University of Health Sciences, Gulhane Training and Research Hospital, Ankara, Turkey
| | - Ibrahim Yilmaz
- Department of General Surgery, University of Health Sciences, Ankara Diskapi Yildirim Beyazit Training and Research Hospital, Ankara, Turkey
| | - Sener Balas
- Department of General Surgery, University of Health Sciences, Ankara Diskapi Yildirim Beyazit Training and Research Hospital, Ankara, Turkey
| | - Cem Azili
- Department of General Surgery, University of Health Sciences, Ankara Diskapi Yildirim Beyazit Training and Research Hospital, Ankara, Turkey
| | - Mehmet Ali Gulcelik
- Department of General Surgery, University of Health Sciences, Gulhane Training and Research Hospital, Ankara, Turkey
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Dağ A, Edizsoy A, Berkeşoğlu M. Oncoplastic Breast Surgery Techniques When Surgery is Essential for the Management of the Idiopatic Granulomatous Mastitis. J INVEST SURG 2021; 35:653-658. [PMID: 34180771 DOI: 10.1080/08941939.2021.1922552] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/21/2022]
Abstract
BACKGROUND Immunosuppressive agents are the main treatment options for idiopathic granulomatous mastitis (IGM). However, in some patients, the remaining large pouch and seroma may cause recurrence and severe deformity. Oncoplastic breast surgery (OBS) techniques can be used when surgery is required. MATERIAL AND METHODS In this study, surgical results of the patients histopathologically diagnosed with IGM who underwent any of the OBS techniques between 2016 and 2020 were evaluated. Patient characteristics, surgical indications, and technical details of the surgery were recorded. RESULTS Eighteen patients who underwent wide excision combined with the OBS technique were included in the study. Surgery was performed owning to persistent disease (n = 12), recurrence (n = 3), and patient preference (n = 3). No major complications occurred after the OBS technique. None of the patients had recurrence at the surgical site. CONCLUSIONS When surgery is indicated in patients with IGM, successful results can be obtained with acceptable complication rates by using the simplest and easiest applicable OBS techniques, including volume displacement technique.
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Affiliation(s)
- Ahmet Dağ
- Faculty of Medicine, Clinic of General Surgery, Mersin University, Mersin, Turkey
| | - Akay Edizsoy
- Clinic of Surgical Oncology, Isparta City Hospital, Isparta, Turkey
| | - Mustafa Berkeşoğlu
- Faculty of Medicine, Clinic of General Surgery, Mersin University, Mersin, Turkey
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Papila Kundaktepe B, Velidedeoğlu M, Mete B. The effect of methotrexate monotherapy on treatment-resistant idiopathic granulomatous mastitis patients. Surgeon 2021; 20:e13-e19. [PMID: 33836950 DOI: 10.1016/j.surge.2021.03.001] [Citation(s) in RCA: 16] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/13/2020] [Revised: 02/16/2021] [Accepted: 03/09/2021] [Indexed: 11/25/2022]
Abstract
BACKGROUND Idiopathic granulomatous mastitis (IGM) is a disease of unknown etiology, involving a chronic inflammatory process, characterized by noncaseating granuloma formation. IGM can mimic a tumor clinically and radiologically. Since we are a tertiary referral center, most of our patients (n = 56, 87.5%) are secondary admissions who have previously had antibiotics and steroid treatments; therefore, we accept these patients as resistant cases. Here, we aim to present our single-center series of 64 patients with resistant IGM who underwent methotrexate monotherapy. To the best of our knowledge, our study includes the highest number of patients described in the literature with IGM who have undergone this treatment. METHODS This study included 64 patients, 56 of which were resistant cases, diagnosed with IGM between January 2013 and January 2020 at Istanbul University Cerrahpasa, Cerrahpasa Medical Faculty, General Surgery Breast Outpatient Clinic that were followed-up at least once. These patients were administered oral methotrexate monotherapy 15 mg/week for 24 weeks, and in relapsed cases, the treatment was up to 20 mg/week for 1 year. Folic acid 10 mg/week was given as a supplement to all patients. RESULTS Complete recovery was observed in 52 (81.25%) of the 64 patients. Follow-up was discontinued by 4 patients. The dose was increased and the duration of treatment was extended up to 1 year when relapse was observed in 8 patients and complete response was then obtained in these cases. Only 3 patients (4.69%) experienced side effects and were switched to subcutaneous treatment due to nausea. CONCLUSION Considering the high patient compliance, low recurrence, minimal side effects, and overall success of the treatment, we believe that methotrexate monotherapy may be used in treatment-resistant IGM patients and may also be the first choice for first-line treatment in the future.
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Affiliation(s)
- Berrin Papila Kundaktepe
- Istanbul University-Cerrahpasa, Cerrahpaşa Faculty of Medicine, Department of General Surgery, Turkey.
| | - Mehmet Velidedeoğlu
- Istanbul University-Cerrahpasa, Cerrahpaşa Faculty of Medicine, Department of General Surgery, Turkey.
| | - Bilgül Mete
- Istanbul University-Cerrahpasa, Cerrahpaşa Faculty of Medicine, Department of Infectious Diseases and Clinical Microbiology, Turkey.
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Yin Y, Liu X, Meng Q, Han X, Zhang H, Lv Y. Idiopathic Granulomatous Mastitis: Etiology, Clinical Manifestation, Diagnosis and Treatment. J INVEST SURG 2021; 35:709-720. [PMID: 33691563 DOI: 10.1080/08941939.2021.1894516] [Citation(s) in RCA: 34] [Impact Index Per Article: 8.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/20/2022]
Abstract
Idiopathic granulomatous mastitis (IGM) is a rare form of chronic inflammatory breast disease. Although it is a benign breast lesion, it may be sometimes difficult to distinguish from breast cancer. The cause of IGM is unknown, but may be associated with autoimmunity, abnormal hormone levels and infection. While the clinical manifestations of IGM involve various manifestations of inflammation, the diagnosis is principally established by histopathology, characterized by non-caseating granulomas and microabscess formation centered on the breast lobules. Therapeutic options for IGM range from observation to various medical treatments, such as steroids, immunosuppressants, and antibiotics, to surgical intervention, particularly if secondarily infected. Given that the controversy on etiology and treatment choices, we accomplished the present review through reviewing IGM-related literature published in 'Pubmed' and 'Web of science' databases during 1997 to 2020, aiming to provide the basis for rational clinical diagnosis and treatment.
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Affiliation(s)
- Yulong Yin
- Department of Thyroid Breast Surgery, The Affiliated Hospital of Northwest University, Xi'an, Shaanxi Province, China
| | - Xianghua Liu
- Department of Thyroid Breast Surgery, The Affiliated Hospital of Northwest University, Xi'an, Shaanxi Province, China
| | - Qingjie Meng
- Department of Thyroid Breast Surgery, The Affiliated Hospital of Northwest University, Xi'an, Shaanxi Province, China
| | - Xiaogang Han
- Department of Thyroid Breast Surgery, The Affiliated Hospital of Northwest University, Xi'an, Shaanxi Province, China
| | - Haomeng Zhang
- Department of Thyroid Breast Surgery, The Affiliated Hospital of Northwest University, Xi'an, Shaanxi Province, China
| | - Yonggang Lv
- Department of Thyroid Breast Surgery, The Affiliated Hospital of Northwest University, Xi'an, Shaanxi Province, China
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Grover H, Grover SB, Goyal P, Hegde R, Gupta S, Malhotra S, Li S, Gupta N. Clinical and imaging features of idiopathic granulomatous mastitis - The diagnostic challenges and a brief review. Clin Imaging 2021; 69:126-132. [PMID: 32717540 DOI: 10.1016/j.clinimag.2020.06.022] [Citation(s) in RCA: 9] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/31/2020] [Revised: 05/28/2020] [Accepted: 06/12/2020] [Indexed: 02/08/2023]
Abstract
Idiopathic granulomatous mastitis (IGM), or granulomatous lobular mastitis is a rare, benign, inflammatory condition of the breast, without an identifiable underlying etiology. The clinical and imaging diagnosis of this entity is challenging with the presentation frequently mimicking inflammatory breast carcinoma (IBC). Mammography and breast ultrasound (US) have an important role in its detection, however, biopsy is imperative for histopathological confirmation. We present three cases of biopsy proven IGM, highlighting the variability of the clinical and imaging features,brieflyreview the relevant literature and discuss the challenges associated with its diagnosis and management.
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Affiliation(s)
- Hemal Grover
- Department of Radiology, Icahn School of Medicine at Mount Sinai West, New York, NY, USA.
| | - Shabnam Bhandari Grover
- Department of Radiology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi 110029, India
| | - Pradeep Goyal
- Department of Radiology, St. Vincent's Medical Center, Bridgeport, CT, USA
| | - Rahul Hegde
- Department of Radiology, Yale New Haven Health, New Haven, CT, USA
| | - Sonali Gupta
- Department of Medicine, St. Vincent's Medical Center, Bridgeport, CT, USA
| | - Saurabh Malhotra
- Department of Pathology, Charlotte Hungerford Hospital, Torrington, CT, USA
| | - Shuo Li
- Department of Radiology, Yale New Haven Health, New Haven, CT, USA
| | - Nishant Gupta
- Department of Radiology, Columbia University at Bassett Healthcare, Cooperstown, NY, USA
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Ben Abid F, Abdel Rahman S Al Soub H. A case report of TB versus idiopathic granulomatous mastitis with erythema nodosum, reactive arthritis, cough, and headache. Aging Male 2020; 23:411-414. [PMID: 30293476 DOI: 10.1080/13685538.2018.1504915] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/28/2022] Open
Abstract
Tuberculous mastitis (TBM) is relatively rare disease with an incidence ranging between 0.1 and 4%. Most of the cases are culture negative and often mistaken with chronic benign idiopathic granulomatous mastitis (IGM). It is very crucial to distinguish culture negative TBM from other causes of mastitis as the treatment differs tremendously. We describe here in a young woman originally from India and residing in Qatar; a non endemic area of tuberculosis; for more then fifteen years. She presented with 2 months history of right breast mass, followed by low grade fever, dry cough, headache, erythema nodosum, arthritis, and arthralgia. In view of the origin of the patient, positive family history for tuberculosis and positive quantiferon, the patient was started empirically on anti-tuberculous treatment (ATT). One week later she developed paradoxical reaction to ATT. This case illustrates unusual and rare manifestations of primary TBM and highlights the importance of differentiating and treating culture negative TBM from IGM.
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Affiliation(s)
- Fatma Ben Abid
- Medicine, Infectious Disease Department, Hamad General Hospital-HMC, Hamad Medical Corp, Doha, Qatar
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Choi EC, Wong SBJ, Ho SAJ. Idiopathic Granulomatous Mastitis and Erythema nodosum - A Unifying Pathophysiology? Australas J Dermatol 2020; 62:e149-e153. [PMID: 32935853 DOI: 10.1111/ajd.13463] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/08/2020] [Revised: 07/29/2020] [Accepted: 08/07/2020] [Indexed: 12/30/2022]
Affiliation(s)
- Ellie Ce Choi
- Division of Dermatology, Department of Medicine, National University Healthcare System, Singapore, Singapore
| | | | - Sue-Ann Je Ho
- Division of Dermatology, Department of Medicine, National University Healthcare System, Singapore, Singapore
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Wu JM, Turashvili G. Cystic neutrophilic granulomatous mastitis: an update. J Clin Pathol 2020; 73:445-453. [PMID: 32094275 DOI: 10.1136/jclinpath-2019-206180] [Citation(s) in RCA: 49] [Impact Index Per Article: 9.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/22/2019] [Revised: 01/22/2020] [Accepted: 01/23/2020] [Indexed: 02/07/2023]
Abstract
Cystic neutrophilic granulomatous mastitis (CNGM) is a rare subtype of granulomatous mastitis with a highly distinct histological pattern often associated with Corynebacterium species. CNGM is characterised by suppurative lipogranulomas that are composed of central lipid vacuoles rimmed by neutrophils and an outer cuff of epithelioid histiocytes. Some of the lipid vacuoles may contain sparse, rod-shaped, gram-positive bacilli that can be easily missed or dismissed. The surrounding mixed inflammatory infiltrate contains Langhans-type giant cells, lymphocytes and neutrophils. CNGM occurs in reproductive age women with a history of pregnancy and typically presents as a palpable mass that can be painful. CNGM has many mimickers, most significantly breast carcinoma. In many cases, CNGM has significant pathological and clinical overlap with other forms of granulomatous mastitis. Given the association with Corynebacterium species, early diagnosis of CNGM is essential in offering patients the most appropriate treatment. Prolonged antibiotic therapy specifically directed to corynebacteria is required, sometimes even beyond resolution of clinical symptoms. This comprehensive review of the existing literature on CNGM describes clinical-pathological features, microbiological findings, challenges associated with the microscopic differential diagnosis, clinical implications of this diagnosis and emerging treatment options. Morphological criteria and suggested comments to convey the degree of diagnostic certainty are also proposed for standard pathology reporting.
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Affiliation(s)
- Jessie M Wu
- Department of Pathology and Laboratory Medicine, Mount Sinai Hospital, University of Toronto, Toronto, Ontario, Canada
| | - Gulisa Turashvili
- Department of Pathology and Laboratory Medicine, Mount Sinai Hospital, University of Toronto, Toronto, Ontario, Canada
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[Idiopathic granulomatous mastitis associated with erythema nodosum]. Ann Dermatol Venereol 2020; 146:571-576. [PMID: 31151772 DOI: 10.1016/j.annder.2019.04.023] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/03/2018] [Revised: 12/24/2018] [Accepted: 04/08/2019] [Indexed: 01/15/2023]
Abstract
BACKGROUND Idiopathic granulomatous mastitis (IGM) is a rare, chronic, granulomatous, inflammatory disorder with potentially misleading clinical and radiological features. IGM is diagnosed after exclusion of infectious or tumoral diseases. Herein we report a case of erythema nodosum associated with IGM, which serves as a reminder that erythema nodosum may constitute an extra-mammary sign of IGM. PATIENTS AND METHODS A 36-year-old nulliparous woman presented with fever and skin rash at our dermatology clinic at the North Franche-Comté hospital in July 2017. Symptoms had occurred 2 days previously. The patient had a fever of 38.8°C and typical erythema nodosum on the limbs. Physical examination showed an irregular, inflammatory, abscessed mass measuring 20cm with multiple shrinkage zones that had been developing for 4 weeks and for which breast examinations were being performed. Laboratory tests showed an inflammatory state (CRP 155mg/mL, WBC 14.6×109/L), other tests (serum electrolytes and calcium, hepatic and renal tests, Streptotest, T-spot, HIV, HBV, HCV serology, anti-streptolysin, anti-streptodornase, local microbiology samples, antinuclear antibodies, soluble antigens antibodies, hemocultures, angiotensin-converting-enzyme and chest x-rays) were normal. Microbiology investigations were negative. Mammography revealed invasive mastitis. Breast sample biopsies showed giant cell granulomas without caseous necrosis or tumor cells, and histochemical staining (PAS, Ziehl, Grocott, Gram) was negative. The final diagnosis was of IGM associated with erythema nodosum. Symptoms rapidly improved with oral steroids. DISCUSSION As reported herein, erythema nodosum may be associated with IGM and support the diagnosis thereof. This association is rare, with fewer than 30 case reports described in the literature. IGM is an uncommon benign disorder of the breast that can mimic two frequent breast disorders: breast carcinoma and breast abscess. It usually occurs in young sexually active women. It appears as a tumor with an inflammatory solid painful mass, with nipple and skin retraction, occasionally with abscesses and accompanied by homolateral axillary lymphadenopathies. Radiologic findings are usually not specific for or suggestive of cancer. The histopathological picture of IGM is characterized by the presence of multinucleated giant cells and epithelioid histiocytes forming non-caseating granulomas around lobules. Minor ductal and periductal inflammation is usually present. IGM is rarely associated with autoimmune system manifestations, especially erythema nodosum, arthritis, episcleritis or hidradenitis suppurativa. Although the physiopathology of IGM remains unclear, this case serves as additional evidence that the etiology of IGM is of autoimmune origin. While there is no recommendation for the treatment of IGM, oral steroids remain the cornerstone of therapy. CONCLUSION We report a case of IGM associated with erythema nodosum. Dermatologists must be aware of this association.
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Fazzio RT, Shah SS, Sandhu NP, Glazebrook KN. Idiopathic granulomatous mastitis: imaging update and review. Insights Imaging 2016; 7:531-9. [PMID: 27221974 PMCID: PMC4956627 DOI: 10.1007/s13244-016-0499-0] [Citation(s) in RCA: 74] [Impact Index Per Article: 8.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/01/2016] [Revised: 04/02/2016] [Accepted: 05/03/2016] [Indexed: 12/15/2022] Open
Abstract
Objectives The purpose of this study was to review the imaging features of idiopathic granulomatous mastitis (IGM) with clinical and pathology correlation. Methods With institutional review board (IRB) approval, a retrospective search of the surgical pathology database from January 2000 to July 2015 was performed. Clinical, imaging and histology findings were reviewed. Cases of granulomatous mastitis without a known source, diagnosed with percutaneous or surgical biopsy, were included in our analysis. Results Seventeen cases of IGM were identified with imaging available for review. The majority of patients presented with a palpable abnormality, whereas a minority were asymptomatic with an abnormal screening mammogram. At imaging, IGM most often demonstrated a focal asymmetry at mammography, a hypoechoic mass with irregular or angular margins at ultrasound, and robust enhancement with mixed progressive and plateau kinetics at magnetic resonance imaging (MRI). Axillary lymph nodes were reactive in appearance at ultrasound. Molecular breast imaging performed in one case showed mild focal asymmetric radiotracer uptake. Conclusion IGM is a rapidly progressive rare inflammatory condition of the breast resulting in non-necrotizing granuloma formation. Imaging features mimic breast carcinoma and diagnosis can be difficult. Radiologists’ awareness of this condition is essential to prevent delayed or unnecessary treatment. Teaching points • Idiopathic granulomatous mastitis is rapidly progressive inflammatory condition. • Imaging features may mimic breast carcinoma or infection. • Ultrasound shows irregular hypoechoic masses with increased vascularity and sinus tracts. • MRI shows irregular, enhancing masses or non-mass enhancement with microabscesses. • MRI is useful for assessment of breast involvement and response to treatment.
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Affiliation(s)
- Robert T Fazzio
- Department of Radiology, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA.
| | - Sejal S Shah
- Department of Laboratory Medicine and Pathology, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA
| | - Nicole P Sandhu
- Department of Medicine, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA
| | - Katrina N Glazebrook
- Department of Radiology, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA
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15
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Benson JR, Dumitru D. Idiopathic granulomatous mastitis: presentation, investigation and management. Future Oncol 2016; 12:1381-94. [PMID: 27067146 DOI: 10.2217/fon-2015-0038] [Citation(s) in RCA: 83] [Impact Index Per Article: 9.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/21/2022] Open
Abstract
Idiopathic granulomatous mastitis (IGM) is a rare chronic inflammatory condition of the breast which although benign can mimic carcinoma. Establishing a diagnosis can be challenging and requires a high index of suspicion with exclusion of infective and autoimmune breast diseases. IGM is characterized histologically by noncaseating granulomas which are of a lobulo-centric pattern and often associated with microabscess formation. Management of confirmed cases remains controversial with proponents of initial surgical or medical therapies - each has its associated problems which can be worse than the original symptoms of IGM. However, many patients require more than one modality of treatment to completely resolve IGM lesions and careful judgment is necessary to ensure optimal type and sequencing of treatments.
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Affiliation(s)
- John R Benson
- Cambridge Breast Unit, Addenbrooke's Hospital & University of Cambridge, Cambridge, CB2 0QQ, UK.,University of Cambridge, Cambridge, UK
| | - Dorin Dumitru
- Cambridge Breast Unit, Addenbrooke's Hospital & University of Cambridge, Cambridge, CB2 0QQ, UK
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16
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Zabetian S, Friedman BJ, McHargue C. A case of idiopathic granulomatous mastitis associated with erythema nodosum, arthritis, and reactive cough. JAAD Case Rep 2016; 2:125-7. [PMID: 27051851 PMCID: PMC4810290 DOI: 10.1016/j.jdcr.2016.01.011] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023] Open
Affiliation(s)
- Saba Zabetian
- Correspondence to: Saba Zabetian, MD, 3031 West Grand Blvd, Suite 800, Detroit, MI 48202.3031 West Grand Blvd, Suite 800DetroitMI48202
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17
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Sheybani F, Naderi HR, Gharib M, Sarvghad M, Mirfeizi Z. Idiopathic granulomatous mastitis: Long-discussed but yet-to-be-known. Autoimmunity 2016; 49:236-9. [PMID: 26829298 DOI: 10.3109/08916934.2016.1138221] [Citation(s) in RCA: 41] [Impact Index Per Article: 4.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/18/2022]
Abstract
Idiopathic granulomatous mastitis (IGM) is a chronic benign inflammatory disease of the breast with unknown etiology. It is an important diagnostic and therapeutic challenge, as most patients were initially misdiagnosed by their primary care physicians, leading to diagnostic confusion and heightened anxiety. Although several triggers have been proposed for development of IGM, the etiologic association of neither of them has been documented. Three main hypotheses about the possible causes of IGM have been suggested, including autoimmune response, infectious disease, and hormonal disruption. Here, we discuss a hypothetical perspective of IGM to explain the possible role of autoinflammation in the pathogenesis of the disease. We also reviewed the previously published literature on pathogenesis of IGM.
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Affiliation(s)
| | | | | | | | - Zahra Mirfeizi
- c Rheumatic Diseases Research Center, School of Medicine, Mashhad University of Medical Sciences , Mashhad , Iran
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18
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Abstract
OBJECTIVE To describe the clinical and paraclinical characteristics of idiopathic granulomatous mastitis, the management of our patients, and review the literature on this topic. METHODS Patients referred to our clinic with a presumptive diagnosis of infectious mastitis were only included in the study if they had histopathologic confirmation of granulomatous mastitis. A systemic or infectious etiology was excluded by the testing of tissues and fluids by staining, culture, and polymerase chain reaction methods. Those patients with no reported etiology were categorized as idiopathic granulomatous mastitis and treated with antiinflammatory drugs. All were monitored for signs of recurrence. RESULTS All 22 patients with a final diagnosis of idiopathic granulomatous mastitis were women with a mean age (± standard deviation) of 32.8 ± 6.2 years. Initial treatment with a prednisone regimen was given in 15 (68.2%) patients, prednisone plus methotrexate (MTX) in six (27.3%), and MTX alone in one (4.5%). Among the total of 22 patients with idiopathic granulomatous mastitis, 12 (54.5%) received MTX either as an initial treatment or as an alternative regimen. Of the 22 patients, three (13.6%) had disease recurrence and four (18.2%) experienced adverse drug reactions. Sixteen (72.7%) patients discontinued the initial treatment regimen with acceptable control of disease activity and without recurrence or adverse drug reactions during the follow-up period. CONCLUSION Idiopathic granulomatous mastitis is a distinct benign breast condition of unknown etiology but several triggers are suspected, including inflammatory, infectious, and hormonal factors. Corticosteroids and MTX, with or without surgery, are the treatment of choice in these patients.
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19
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Altintoprak F, Kivilcim T, Ozkan OV. Aetiology of idiopathic granulomatous mastitis. World J Clin Cases 2014; 2:852-858. [PMID: 25516860 PMCID: PMC4266833 DOI: 10.12998/wjcc.v2.i12.852] [Citation(s) in RCA: 142] [Impact Index Per Article: 12.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/01/2014] [Revised: 07/07/2014] [Accepted: 10/10/2014] [Indexed: 02/05/2023] Open
Abstract
Idiopathic granulomatous mastitis is a rare chronic inflammatory lesion of the breast that can clinically and radiographically mimic breast carcinoma. The most common clinical presentation is an unilateral, discrete breast mass, nipple retraction and even a sinus formation often associated with an inflammation of the overlying skin. The etiology of idiopathic granulomatous mastitis is still obscure. Its treatment remains controversial. The cause may be the autoimmune process, infection, a chemical reaction associated with oral contraceptive pills, or even lactation. Various factors, including hormonal imbalance, autoimmunity, unknown microbiological agents, smoking and α 1-antitrypsin deficiency have been suggested to play a role in disease aetiology. In this review, causing factors in the aetiology of idiopathic granulomatous mastitis are reviewed in detail.
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20
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Hida T, Minami M, Kawaguchi H, Oshiro Y, Kubo Y. Case of erythema nodosum associated with granulomatous mastitis probably due toCorynebacteriuminfection. J Dermatol 2014; 41:821-3. [DOI: 10.1111/1346-8138.12604] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/25/2014] [Accepted: 07/18/2014] [Indexed: 11/29/2022]
Affiliation(s)
- Tetsuya Hida
- Divisions of Dermatology; Matsuyama Red Cross Hospital; Matsuyama Japan
| | - Mitsuyoshi Minami
- Divisions of Dermatology; Matsuyama Red Cross Hospital; Matsuyama Japan
| | | | - Yumi Oshiro
- Divisions of Diagnostic Pathology; Matsuyama Red Cross Hospital; Matsuyama Japan
| | - Yoshiaki Kubo
- Department of Dermatology; Institute of Health Biosciences; The University of Tokushima Graduate School; Tokushima Japan
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21
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Granulomatous mastitis: changing clinical and imaging features with image-guided biopsy correlation. Eur Radiol 2014; 24:2404-11. [PMID: 24962828 DOI: 10.1007/s00330-014-3273-z] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/06/2014] [Revised: 04/25/2014] [Accepted: 06/03/2014] [Indexed: 01/16/2023]
Abstract
OBJECTIVES To review clinical presentation, revisit patient demographics and imaging findings in granulomatous mastitis and determine the optimal biopsy method for diagnosis. METHODS A retrospective study was performed to review the clinical presentation, imaging findings and biopsy methods in patients with granulomatous mastitis. Twenty-seven patients with pathology-proven granulomatous mastitis were included. RESULTS The average age at presentation was 38.0 years (range, 21-73 years). Seven patients were between 48 and 73 years old. Twenty-four patients presented with symptoms and three patients were asymptomatic. Nineteen patients were imaged with mammography demonstrating mammographically occult lesions as the predominant finding. Twenty-six patients were imaged with ultrasound and the most common finding was a mass lesion. Pathological diagnosis was made by image-guided biopsy in 44 % of patients. The imaging features of granulomatous mastitis on mammography are infrequently described. CONCLUSIONS Our study demonstrates that granulomatous mastitis can occur in postmenopausal or asymptomatic patients, although previously reported exclusively in young women with palpable findings. Presentation on mammography as calcifications requiring mammographically guided vacuum-assisted biopsy has not been previously described. The diagnosis of granulomatous mastitis can easily be made by image-guided biopsy and surgical excision should be reserved for definitive treatment. KEY POINTS • Characterizes radiographic appearance of granulomatous mastitis in postmenopausal or asymptomatic patients. • Granulomatous mastitis can present exclusively as calcifications on mammography. • The diagnosis of granulomatous mastitis is made by image-guided biopsy techniques.
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22
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Nakamura Y, Yamaguchi M, Nakamura A, Muto M. Tender, red nodules and arthralgia in a young woman with mastitis. Clin Exp Dermatol 2014; 39:410-2. [PMID: 24635090 DOI: 10.1111/ced.12276] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 10/13/2013] [Indexed: 11/30/2022]
Affiliation(s)
- Y Nakamura
- Department of Dermatology, Yamaguchi University Graduate School of Medicine, Yamaguchi, Japan
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23
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Binesh F, Shiryazdi M, Bagher Owlia M, Azimi S. Idiopathic granulomatous mastitis, erythema nodosum and bilateral ankle arthritis in an Iranian woman. BMJ Case Rep 2013; 2013:bcr2012007636. [PMID: 23355571 PMCID: PMC3604348 DOI: 10.1136/bcr-2012-007636] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Here we report a case of idiopathic granulomatous mastitis (IGM) associated with erythema nodosum (EN) and ankle arthritis. The skin, joint and mammary symptoms improved with corticosteroid. Coincidence of granulomatous mastitis, EN and arthritis is a rare feature.
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Affiliation(s)
- Fariba Binesh
- Shahid Sadoughi University of Medical Sciences, Yazd, Iran.
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24
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Binesh F, Shiryazdi M, Bagher Owlia M, Azimi S. Idiopathic granulomatous mastitis, erythema nodosum and bilateral ankle arthritis in an Iranian woman. BMJ Case Rep 2013; 2013:bcr2012007636. [PMID: 23355571 PMCID: PMC3777118 DOI: 10.1155/2013/148727] [Citation(s) in RCA: 59] [Impact Index Per Article: 4.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/03/2013] [Accepted: 08/03/2013] [Indexed: 02/06/2023] Open
Abstract
Here we report a case of idiopathic granulomatous mastitis (IGM) associated with erythema nodosum (EN) and ankle arthritis. The skin, joint and mammary symptoms improved with corticosteroid. Coincidence of granulomatous mastitis, EN and arthritis is a rare feature.
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Affiliation(s)
- Fariba Binesh
- Shahid Sadoughi University of Medical Sciences, Yazd, Iran.
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25
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Gautier N, Lalonde L, Tran-Thanh D, El Khoury M, David J, Labelle M, Patocskai E, Trop I. Chronic granulomatous mastitis: Imaging, pathology and management. Eur J Radiol 2012. [PMID: 23200627 DOI: 10.1016/j.ejrad.2012.11.010] [Citation(s) in RCA: 74] [Impact Index Per Article: 5.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
PURPOSE The aim of this study is to describe the clinical and radiological presentation of chronic granulomatous mastitis. MATERIAL AND METHODS We retrospectively reviewed the clinical and radiological data of 11 women with histologically proven chronic granulomatous mastitis (CGM) diagnosed between March 2008 and September 2011. RESULTS The diagnosis of CGM is often a challenging one that can mimic infectious and malignant breast conditions. Clinically, CGM most commonly presents as a mass, occasionally with associated erythema. The most frequent mammographic presentation is an asymmetric density, while ultrasound most commonly reveals a hypoechoic mass with tubular extensions and a striated echotexture. On MRI, the most specific finding is peripherally enhancing fluid or solid masses with fistulous tract to the skin, although the latter is not commonly encountered. Diagnosis can be reliably obtained by needle core or vacuum-assisted biopsy, and is established pathologically by the identification of granulomatous inflammation without caseous necrosis. CGM is a diagnosis of exclusion after infectious and foreign body causes are ruled out. Treatment options include oral steroids or surgery; both options are associated with similar recurrence rates. The disease tends to burn itself out and the option of conservative management with observation is a valid one. CONCLUSION CGM is a rare benign disease with no specific features clinically or at imaging. There are no radiologic findings that are specific of CGM, but in the appropriate clinical setting, the diagnosis can be suggested by the radiologist.
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Affiliation(s)
- Nicolas Gautier
- Centre Régional de Lutte Contre le Cancer Eugène Marquis, avenue Bataille Flandres Dunkerque, Rennes, France.
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Abstract
Idiopathic granulomatous lobular mastitis (IGLM) is a rare breast condition with prominent skin findings. It is typically seen in young parous women. Painful breast masses, draining sinuses, scarring, and breast atrophy are the main clinical manifestations. IGLM can resemble a variety of other inflammatory and neoplastic processes of the breast. It is thought to result from obstruction and rupture of breast lobules. Extravasated breast secretions then induce an inflammatory reaction. Corynebacteria have also been implicated in the pathogenesis. Treatment is surgical, but systemic corticosteroids, methotrexate, and antibiotics also play a role.
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27
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Akbulut S, Yilmaz D, Bakir S. Methotrexate in the management of idiopathic granulomatous mastitis: review of 108 published cases and report of four cases. Breast J 2011; 17:661-668. [PMID: 21951547 DOI: 10.1111/j.1524-4741.2011.01162.x] [Citation(s) in RCA: 98] [Impact Index Per Article: 7.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
This study aimed to discuss the role of agents, such as steroids and methotrexate (MTX), in the treatment of patients with idiopathic granulomatous mastitis (IGM). Using Pubmed and Google Scholar data bases, a retrospective study was carried out on IGM cases treated with steroids and/or MTX between 1972 and 2010. Four IGM cases treated with MTX at our clinic were also summarized in this study. A total of 541 IGM cases since 1972, including ours, were retrospectively analyzed. Steroid treatment 5-85 mg was administered over 5 days-22 months to 112 patients aged 21-48 years. Recurrence occurred in 22 patients, steroid-induced diabetes mellitus in 5 patients, no response to treatment was observed in 4 patients, in 2 patients, the mass decreased in size, and static disease was observed in one. The steroid treatment was changed to MTX treatment in 4 patients who had recurrence, 5 with steroid-induced DM and in 4 who were nonrespondents. Three patients were started on steroid together with MTX as a primary treatment. Of the patients treated with MTX, a satisfactory result was obtained in 14 cases and in 2, mastectomy was performed because of recurrence despite the treatment with MTX. IGM is a troublesome condition that presents management problems due to the side effects of steroids. Our study demonstrates that the use of MTX in IGM cases has been effective in preventing complications, in resolving the inflammatory process, and in limiting side effects of corticosteroids.
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Affiliation(s)
- Sami Akbulut
- Department of Surgery Department of Pathology, Diyarbakir Education and Research Hospital, Dagkapi, Diyarbakir, Turkey.
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28
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Ruiter AM, Vegting IL, Nanayakkara PWB. Idiopathic granulomatous mastitis: a great imitator? BMJ Case Rep 2010; 2010:2010/nov17_1/bcr0320102844. [PMID: 22798482 DOI: 10.1136/bcr.03.2010.2844] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
The patient in this case was seen by numerous doctors for a long time before the diagnosis was made because, right from the start, an abscess or a cancer of the breast was suspected. She was first seen by her general practitioner (GP); thereafter, she went to two consultant physicians and a surgeon in a regional hospital and eventually an idiopathic granulomatous mastitis (IGM) diagnosis was made by a specialist in internal medicine in a university hospital. When the diagnosis IGM was made, the patient was treated with steroids and made a complete recovery. Although the incidence of IGM is not high, the GPs are likely to see these patients initially and should be aware of the existence of this disease, which may spare the patient unnecessary consultations, diagnostics tests or even mastectomy.
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Affiliation(s)
- Annabel M Ruiter
- Department of Internal Medicine, VU University Medical Center, Amsterdam, The Netherlands
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