|
1
|
Jeries H, Braun-Moscovici Y, Zohar Y, Balbir-Gurman A. Immunoglobulin G 4-related breast disease: two cases treated successfully with rituximab. Scand J Rheumatol 2024; 53:442-444. [PMID: 39279634 DOI: 10.1080/03009742.2024.2372899] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/02/2024] [Accepted: 06/24/2024] [Indexed: 09/18/2024]
Affiliation(s)
- H Jeries
- Rheumatology Unit, Galilee Medical Center, Nahariya, Israel
- Azrieli Faculty of Medicine, Bar-Ilan University, Safed, Israel
| | - Y Braun-Moscovici
- B Shine Rheumatology Institute, Rambam Health Care Campus, Haifa, Israel
- Rappaport Faculty of Medicine, Technion, Haifa, Israel
| | - Y Zohar
- Rappaport Faculty of Medicine, Technion, Haifa, Israel
- Department of Pathology, Rambam Health Care Campus, Haifa, Israel
| | - A Balbir-Gurman
- B Shine Rheumatology Institute, Rambam Health Care Campus, Haifa, Israel
- Rappaport Faculty of Medicine, Technion, Haifa, Israel
| |
Collapse
|
|
2
|
Jeries H, Braun-Moscovici Y, Balbir-Gurman A. IgG4-related Breast Disease: Review of the Literature. Rambam Maimonides Med J 2024; 15:RMMJ.10532. [PMID: 39503548 PMCID: PMC11524419 DOI: 10.5041/rmmj.10532] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/08/2024] Open
Abstract
IgG4-related disease (IgG4-RD) is a rare illness with inflammatory and fibrotic changes in affected organs such as pancreas, thyroid, salivary or lacrimal glands, and retroperitoneal space; rarely other organs may be involved. IgG4-related breast disease (IgG4-BD) is very rare and generally presents as a lump or mastitis. IgG4-BD as a presenting feature of IgG4-RD is extremely rare. Hence, this paper reviews the known (n=48) IgG-BD cases reported in the literature to date. The majority of cases were diagnosed on routine mammography or during assessment for other clinically significant features. The absence of a lump border, and especially the absence of calcifications on ultrasonography, mammography, or computed tomography, is typical for IgG4-BD. Characteristic IgG4-BD pathological findings were dense lymphoplasmacytic infiltration with stromal fibrosis, and more than 10% IgG4+ plasma cells/high-power field (HPF); the mean percentage of IgG4+/IgG+ plasma cells was 54.2%, and only one-third of the patients had all "classical" signs of IgG4-BD including storiform fibrosis and obliterative phlebitis. Most of the cases had a benign course and responded to surgical excision with or without steroid therapy.
Collapse
Affiliation(s)
- Helana Jeries
- Rheumatology Unit, Galilee Medical Center, Nahariya, Israel
- The Azrieli Faculty of Medicine, Bar-Ilan University, Safed, Israel
| | - Yolanda Braun-Moscovici
- The B. Shine Rheumatology Institute, Rambam Health Care Campus, Haifa, Israel
- Ruth and Bruce Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel
| | - Alexandra Balbir-Gurman
- The B. Shine Rheumatology Institute, Rambam Health Care Campus, Haifa, Israel
- Ruth and Bruce Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel
| |
Collapse
|
|
3
|
Zhou Y, Gong J, Deng X, Shen L, Liu L. Novel insights: crosstalk with non-puerperal mastitis and immunity. Front Immunol 2024; 15:1431681. [PMID: 39148739 PMCID: PMC11324573 DOI: 10.3389/fimmu.2024.1431681] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/12/2024] [Accepted: 07/16/2024] [Indexed: 08/17/2024] Open
Abstract
The two primary types of non-puerperal mastitis (NPM) are granulomatous lobular mastitis (GLM) and plasma cell mastitis (PCM). Existing research indicates that immune inflammatory response is considered to be the core of the pathogenesis of GLM and PCM, and both innate and adaptive immune responses play an important role in the pathophysiology of PCM and GLM. However, the regulatory balance between various immune cells in these diseases is still unclear. Consequently, we present a comprehensive summary of the immune-related variables and recent advances in GLM and PCM.
Collapse
Affiliation(s)
- Yao Zhou
- Department of Galactophore, The First Hospital of Hunan University of Chinese Medicine, Changsha, Hunan, China
- Hunan University of Chinese Medicine, Changsha, Hunan, China
| | - Jie Gong
- Department of Galactophore, The First Hospital of Hunan University of Chinese Medicine, Changsha, Hunan, China
- Hunan University of Chinese Medicine, Changsha, Hunan, China
| | - Xianguang Deng
- Department of Galactophore, The First Hospital of Hunan University of Chinese Medicine, Changsha, Hunan, China
- Hunan University of Chinese Medicine, Changsha, Hunan, China
| | - Lele Shen
- Hunan University of Chinese Medicine, Changsha, Hunan, China
| | - Lifang Liu
- Department of Galactophore, The First Hospital of Hunan University of Chinese Medicine, Changsha, Hunan, China
| |
Collapse
|
|
4
|
Matsushima H, Kikuchi M, Miyabe R, Yamaguchi S, Kita H, Kobayashi J, Ando T, Atsuta K, Soga T, Shintani T. IgG4-related mastitis managed without excision or steroid therapy. Surg Case Rep 2024; 10:32. [PMID: 38302639 PMCID: PMC10834898 DOI: 10.1186/s40792-024-01826-9] [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: 12/01/2023] [Accepted: 01/19/2024] [Indexed: 02/03/2024] Open
Abstract
IgG4-related mastitis is an extremely rare IgG4-related sclerosing disease involving the breast that must be differentiated from breast cancer. There is currently no consensus regarding the optimal treatment strategies. Here, we report a case of IgG4-related mastitis followed up without excision or steroid therapy. Although the association between IgG4-related mastitis and breast cancer remains unclear, regular follow-up imaging and measurement of serum concentrations of disease activity markers may allow for follow-up without excision or steroid therapy.
Collapse
Affiliation(s)
- Hirokazu Matsushima
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| | - Masayuki Kikuchi
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan.
| | - Rika Miyabe
- Tosen Clinic, 1-20, Gohukucho, Aoi-Ku, Shizuoka, Shizuoka, 420-0031, Japan
| | - Sota Yamaguchi
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| | - Hidenori Kita
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| | - Junko Kobayashi
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| | - Takashi Ando
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| | - Koji Atsuta
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| | - Takayoshi Soga
- Department of Rheumatology, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| | - Tsunehiro Shintani
- Department of Surgery, Japanese Red Cross Shizuoka Hospital, 8-2, Ottemachi, Aoi-Ku, Shizuoka, Shizuoka, 420-0853, Japan
| |
Collapse
|
|
5
|
Cui L, Sun C, Guo J, Zhang X, Liu S. Pathological manifestations of granulomatous lobular mastitis. Front Med (Lausanne) 2024; 11:1326587. [PMID: 38371511 PMCID: PMC10869469 DOI: 10.3389/fmed.2024.1326587] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/25/2023] [Accepted: 01/16/2024] [Indexed: 02/20/2024] Open
Abstract
Granulomatous lobular mastitis (GLM) is a rare inflammatory breast disease with unknown etiology, characterized by non-caseous granulomatous inflammation of the lobules, which infiltrate lymphocytes, neutrophils, plasma cells, monocytes, and eosinophils may accompany. GLM is often misdiagnosed as breast cancer due to the lack of specificity in clinical and imaging examinations, and therefore histopathology is the main basis for confirming the diagnosis. This review provides an overview of the pathological features of granulomatous lobular mastitis and cystic neutrophil granulomatous mastitis (CNGM, a pathologic subtype of GLM). As well as pathologic manifestations of other breast diseases that need to be differentiated from granulomatous lobular mastitis such as breast tuberculosis, lymphocytic mastopathy/diabetic mastopathy, IgG4-related sclerosing mastitis (IgG4-RSM), nodular disease, Wegener's granulomatosis, and plasma cell mastitis. Besides, discusses GLM and CNGM, GLM and breast cancer, emphasizing that their relationship deserves further in-depth exploration. The pathogenesis of GLM has not yet been clearly articulated and needs to be further explored, pathology enables direct observation of the microscopic manifestations of the disease and contributes to further investigation of the pathogenesis.
Collapse
Affiliation(s)
- Leyin Cui
- Department of Breast Surgery, Longhua Hospital Shanghai University of Traditional Chinese Medicine, Shanghai, China
| | - Chenping Sun
- Department of Breast Surgery, Longhua Hospital Shanghai University of Traditional Chinese Medicine, Shanghai, China
| | - Jierong Guo
- Department of Breast Surgery, Longhua Hospital Shanghai University of Traditional Chinese Medicine, Shanghai, China
| | - Xuliu Zhang
- Department of Breast Surgery, Longhua Hospital Shanghai University of Traditional Chinese Medicine, Shanghai, China
| | - Sheng Liu
- Department of Breast Surgery, Longhua Hospital Shanghai University of Traditional Chinese Medicine, Shanghai, China
- Graduate School, Shanghai University of Traditional Chinese Medicine, Shanghai, China
| |
Collapse
|
|
6
|
Itakura M, Horimoto Y, Ishizuka Y, Onagi H, Hayashi T, Kawamoto T, Watanabe J, Iijima K. IgG4-related mastitis characterized by skin thickening of the breast: a case report. Surg Case Rep 2023; 9:192. [PMID: 37907692 PMCID: PMC10618130 DOI: 10.1186/s40792-023-01770-0] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/30/2023] [Accepted: 10/20/2023] [Indexed: 11/02/2023] Open
Abstract
BACKGROUND IgG4-related diseases are characterized by marked infiltration and IgG4-positive plasma cells and fibrosis, and involve multiple organs. However, IgG4-related mastitis is rare. We report a case of mastitis associated with IgG4-related disease. CASE PRESENTATION A 78-year-old woman visited our hospital with a complaint of multiple subcutaneous nodules. A biopsy of a dorsal subcutaneous mass was performed but did not yield a definitive diagnosis. However, blood tests showed a high level of IgG4, thus she was referred to the department of collagen disease for further examination. Computed tomography (CT) showed a thickening of the skin of the right breast, and the patient was referred to our department. On physical examination, a large area of thickened skin was observed in the right breast without inflammatory breast cancer-like redness, and no mass was palpable. A needle biopsy was performed on an indistinct hypoechoic area in the breast, and she was diagnosed with mastitis associated with IgG4-related disease. Systemic steroid therapy was then administered and the symptoms of multiple skin nodules and mastitis improved. CONCLUSIONS We reached the diagnosis based on a biopsy of the mammary gland enabling the patient to begin treatment for IgG4-related disease. This case was characterized by breast skin thickening, which is different from inflammatory breast cancer.
Collapse
Affiliation(s)
- Moe Itakura
- Department of Breast Oncology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan
| | - Yoshiya Horimoto
- Department of Breast Oncology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan.
- Department of Human Pathology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan.
| | - Yumiko Ishizuka
- Department of Breast Oncology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan
| | - Hiroko Onagi
- Department of Human Pathology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan
| | - Takuo Hayashi
- Department of Human Pathology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan
| | - Toshio Kawamoto
- Department of Internal Medicine and Rheumatology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan
| | - Junichiro Watanabe
- Department of Breast Oncology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan
| | - Kotaro Iijima
- Department of Breast Oncology, Juntendo University Faculty of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 1113-0033, Japan
| |
Collapse
|
|
7
|
Wang Q, Bradley K, Zhang M, Li S, Li X. Rosai-Dorfman disease of the breast: a clinicoradiologic and pathologic study. Hum Pathol 2023; 141:30-42. [PMID: 37673345 DOI: 10.1016/j.humpath.2023.08.009] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/03/2023] [Revised: 08/26/2023] [Accepted: 08/31/2023] [Indexed: 09/08/2023]
Abstract
Rosai-Dorfman disease (RDD) is an uncommon histiocytic disorder typically involving lymph nodes and less frequently extranodal tissues. RDD involving the breast is rare and may clinically and radiologically mimic neoplastic and non-neoplastic disorders. We report seven patients with breast RDD, describe their clinicoradiologic and pathologic features, and discuss the differential diagnosis. Patients, ranging from 15 to 74 years of age, presented with unilateral and unifocal (5/7) or bilateral and multifocal (2/7) masses. RDD was either confined to the breast (6/7) or concurrently involved a lymph node (1/7). Masses ranged from 8 to 31 mm, categorized as Breast Imaging-Reporting and Data System (BI-RADS) 4 (6/7) or 5 (1/7). All cases showed similar morphology with many large histiocytes displaying emperipolesis with associated fibrosis and dense lymphoplasmacytic infiltrate. The abnormal histiocytes co-expressed CD68/CD163, S100, OCT2, and Cyclin D1 (7/7), and were negative for CK AE1/AE3 (7/7), CD1a (7/7), and BRAF V600E (6/6). Flow cytometry (n = 3), kappa/lambda in situ hybridization (n = 5), and IgG4/IgG immunohistochemistry (n = 1) did not reveal lymphoma or IgG4-related disease. No mycobacterial or fungal organisms were identified on acid-fast bacillus (AFB) and Grocott methenamine silver (GMS) stains (n = 5). Three patients underwent complete excision and none recurred or progressed to systemic disease during follow-up (88-151 months). In summary, breast RDD should be included in the differential diagnosis of a mass-forming breast lesion. Histopathology with ancillary studies and clinicoradiologic correlation is essential for accurate diagnosis and optimal clinical management. Patients with RDD of the breast have an excellent prognosis after complete excision.
Collapse
Affiliation(s)
- Qun Wang
- Department of Pathology, Emory University School of Medicine, Atlanta, GA, 30322, USA.
| | - Kyle Bradley
- Department of Pathology, Emory University School of Medicine, Atlanta, GA, 30322, USA
| | - Meng Zhang
- Department of Radiology and Imaging Sciences, Emory University School of Medicine, Atlanta, GA, 30322, USA
| | - Shiyong Li
- Department of Pathology, Emory University School of Medicine, Atlanta, GA, 30322, USA
| | - Xiaoxian Li
- Department of Pathology, Emory University School of Medicine, Atlanta, GA, 30322, USA.
| |
Collapse
|
|
8
|
Yan M, Wang J, Gadde R, Bomeisl P, Gilmore H, Harbhajanka A. Breast MALT Lymphoma: A Clinical, Histomorphologic, and Immunophenotypic Evaluation. Int J Surg Pathol 2023; 31:1283-1293. [PMID: 36802856 DOI: 10.1177/10668969231152585] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/22/2023]
Abstract
Introduction. Primary breast extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue (MALT lymphoma) is rare and understudied. Embryonically, mammary glands are developed as specialized skin appendages. It is possible that overlapping features exist between breast MALT lymphoma and primary cutaneous marginal zone lymphoma. Methods. We studied 5 primary and 6 secondary breast MALT lymphomas diagnosed in our institution during a 20-year period. Clinical and pathologic features of these lymphomas were analyzed and compared. Results. Most primary and secondary breast MALT lymphomas had similar clinical presentations as unilateral breast lesions without axillary lymphadenopathy. However, primary lymphomas tended to be diagnosed in older patients (median: 77 years old) than secondary lymphomas (median: 60 years old). Thyroid abnormality was a common finding in both primary (3/5) and secondary (5/6) lymphomas. Hashimoto's thyroiditis was diagnosed in one primary lymphoma. No distinct histopathologic findings were found in primary lymphomas. Features for primary cutaneous marginal zone lymphoma, including overexpression of IgG and IgG4 and high IgG4/IgG ratio, were absent in all primary but present in one secondary lymphoma with cutaneous origin. This secondary lymphoma also had expansion of CD30-positive cells. Conclusion. Primary breast MALT lymphoma does not share the distinctive features of primary cutaneous marginal zone lymphoma that set it apart from other extranodal marginal zone lymphomas. Having increased IgG- and IgG4-positive cells with a high IgG/IgG4 ratio in breast MALT lymphoma may indicate cutaneous origin. CD30 overexpression may be a feature seen in marginal zone lymphoma of cutaneous origin, which needs further studies to prove.
Collapse
Affiliation(s)
- Mingfei Yan
- Department of Pathology, University Hospitals Cleveland Medical Center, Cleveland, OH, USA
| | - Jing Wang
- Department of Pathology, University Hospitals Cleveland Medical Center, Cleveland, OH, USA
| | - Ramya Gadde
- Department of Pathology, University Hospitals Cleveland Medical Center, Cleveland, OH, USA
| | - Philip Bomeisl
- Department of Pathology, University Hospitals Cleveland Medical Center, Cleveland, OH, USA
| | - Hannah Gilmore
- Department of Pathology, University Hospitals Cleveland Medical Center, Cleveland, OH, USA
| | - Aparna Harbhajanka
- Department of Pathology, University Hospitals Cleveland Medical Center, Cleveland, OH, USA
| |
Collapse
|
|
9
|
Turashvili G, Li X. Inflammatory Lesions of the Breast. Arch Pathol Lab Med 2023; 147:1133-1147. [PMID: 37196345 DOI: 10.5858/arpa.2022-0477-ra] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/17/2023] [Indexed: 05/19/2023]
Abstract
CONTEXT.— Inflammatory lesions of the breast are rare but not infrequently pose problems both clinically and morphologically, particularly on needle core biopsies. These lesions range from acute inflammatory conditions to chronic lymphoplasmacytic and lymphohistiocytic to granulomatous inflammatory diseases. OBJECTIVE.— To provide a comprehensive overview of inflammatory lesions of the breast, with etiopathogenesis and clinical, radiologic, and pathologic features as well as differential diagnostic considerations, clinical management, and prognosis. DATA SOURCES.— The existing literature in the English language, including original research articles and review articles describing inflammatory lesions of the breast. CONCLUSIONS.— Inflammatory lesions of the breast are characterized by a wide variety of clinical, radiologic, and morphologic features. The histopathologic differential diagnosis often includes a neoplastic process requiring ancillary studies and correlation with clinical and radiologic findings. Although most specimens display nonspecific findings precluding a definitive pathologic diagnosis, pathologists have a unique opportunity to play a crucial role in identifying key histologic features suggestive of certain entities, such as cystic neutrophilic granulomatous mastitis, immunoglobulin (Ig) G4 mastitis, or squamous metaplasia of lactiferous ducts, in the right clinical and radiologic context, and thereby guiding optimal and timely clinical management. The information presented herein will be helpful to practicing anatomic pathologists and pathology trainees in becoming more familiar with specific morphologic features and overcoming differential diagnostic challenges related to pathology reporting of inflammatory lesions of the breast.
Collapse
Affiliation(s)
- Gulisa Turashvili
- From the Department of Pathology and Laboratory Medicine, Emory University Hospital, Atlanta, Georgia
| | - Xiaoxian Li
- From the Department of Pathology and Laboratory Medicine, Emory University Hospital, Atlanta, Georgia
| |
Collapse
|
|
10
|
Grabenstetter A, D’Alfonso TM. Inflammatory, Reactive, and Infectious Conditions of the Breast. A COMPREHENSIVE GUIDE TO CORE NEEDLE BIOPSIES OF THE BREAST 2022:131-164. [DOI: 10.1007/978-3-031-05532-4_4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
|
|
11
|
Asano Y, Kashiwagi S, Kawano Y, Tanaka S, Kuwae Y, Takashima T, Ohsawa M, Hirakawa K, Ohira M. IgG4-related mastitis requiring differentiation from breast cancer: a case report. J Surg Case Rep 2021; 2021:rjaa240. [PMID: 34858570 PMCID: PMC8634083 DOI: 10.1093/jscr/rjaa240] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/12/2020] [Accepted: 06/15/2020] [Indexed: 01/13/2023] Open
Abstract
Immunoglobulin (Ig) G4-related disease (IgG4-RD) is a group of chronic relapsing inflammatory conditions. Although IgG4-RD can occur in various organs, it is rarely observed in mammary glands. Here, we report a case of IgG4-related mastitis (IgG4-RM) that needed to be differentiated from breast cancer. A 54-year-old woman was examined for a tumor in her left breast. Mammary ultrasonography revealed an irregular hypoechoic tumor measuring 45.0 × 43.0 × 32.0 mm in size. A core-needle biopsy of the left breast tissue revealed a high degree of mixed T and B lymphocytic and plasma cell infiltration, as well as interstitial fibrosis. IgG4-RD was diagnosed based on hematological examination that revealed an abnormal IgG4 value of 332 mg/dl. All the clinical diagnostic criteria for IgG4 were met, resulting in a definitive diagnosis of IgG4-RM.
Collapse
Affiliation(s)
- Yuka Asano
- Department of Breast and Endocrine Surgery, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Shinichiro Kashiwagi
- Department of Breast and Endocrine Surgery, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Yuko Kawano
- Department of Breast and Endocrine Surgery, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Sayaka Tanaka
- Department of Diagnostic Pathology, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Yuko Kuwae
- Department of Diagnostic Pathology, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Tsutomu Takashima
- Department of Breast and Endocrine Surgery, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Masahiko Ohsawa
- Department of Diagnostic Pathology, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Kosei Hirakawa
- Department of Breast and Endocrine Surgery, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| | - Masaichi Ohira
- Department of Breast and Endocrine Surgery, Osaka City University Graduate School of Medicine, Osaka 545-8585, Japan
| |
Collapse
|
|
12
|
Corines MJ, Krystel-Whittemore M, Murray M, Mango V. Uncommon Tumors and Uncommon Presentations of Cancer in the Breast. CURRENT BREAST CANCER REPORTS 2021; 13:331-346. [PMID: 36589040 PMCID: PMC9798716 DOI: 10.1007/s12609-021-00435-x] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/05/2023]
Abstract
Purpose of review The purpose is to present a case series of rare diagnoses and unusual presentations of breast lesions with radiologic-pathologic correlation from a major cancer center, and to review the recent literature on each entity with a focus on radiology-pathology concordance. We present our findings and experience from cases of metastatic small cell lung carcinoma to the breast, IgG-4 related breast disease, breast implant associated anaplastic large cell lymphoma, granular cell tumor, pleomorphic sarcoma, adenomyoepithelioma, post-radiation angiosarcoma, and breast carcinoma after risk-reducing total mastectomy. Recent findings It is essential for physicians to have knowledge of rare breast diagnoses and unusual breast disease presentations to formulate a complete differential diagnosis, recognize radiological-pathological concordance of these entities and provide appropriate patient care. Summary Current literature on these rare described entities exists mainly as case reports, case series and small-scale studies. By sharing our findings, we hope to educate trainees in radiology, pathology and other fields across the continuum of care in radiologic-pathologic correlation, while also augmenting the existing literature on these rare entities.
Collapse
Affiliation(s)
- Marina J. Corines
- Memorial Sloan Kettering Cancer Center, Department of Radiology, 300 E 66th Street, New York, NY 10065,Corresponding author: Marina J. Corines, MD, 1275 York Ave, Department of Radiology, New York, New York, 10065, Telephone: (914) 319-6970,
| | | | - Melissa Murray
- Memorial Sloan Kettering Cancer Center, Department of Pathology, 1275 York Avenue, New York, NY 10065
| | - Victoria Mango
- Memorial Sloan Kettering Cancer Center, Department of Radiology, 300 E 66th Street, New York, NY 10065
| |
Collapse
|
|
13
|
Erivwo P, Turashvili G. Pathology of IgG4-related sclerosing mastitis. J Clin Pathol 2020; 74:475-482. [PMID: 33328182 DOI: 10.1136/jclinpath-2020-207029] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/18/2020] [Revised: 11/15/2020] [Accepted: 12/06/2020] [Indexed: 01/11/2023]
Abstract
Immunoglobulin G4-related sclerosing mastitis (IgG4-RM) is a recently recognised member of the IgG4-related disease (IgG4-RD) family, a multisystem fibroinflammatory condition that can affect any organ system. IgG4-RM is rare and predominantly occurs in middle-aged women. It may present with painless palpable mass and/or lymphadenopathy thereby mimicking breast cancer. Although there is an abundance of literature describing the clinicopathological characteristics of IgG4-RD in a variety of organs, data on IgG4-RM are limited due to its rarity. This review describes the manifestation of the disease in the breast based on reported cases, emphasising the clinicopathological features, pathophysiology, differential diagnosis, treatment and prognosis.
Collapse
Affiliation(s)
- Polycarp Erivwo
- Department of Pathology, Memorial University of Newfoundland, St. John's, Newfoundland, Canada
| | - Gulisa Turashvili
- Department of Pathology and Laboratory Medicine, Mount Sinai Hospital, University of Toronto, Toronto, Ontario, Canada
| |
Collapse
|
|
14
|
Erivwo P, Bharatwal V, Huang M, Godra A, Turashvili G. Classic IgG4-related sclerosing mastitis is not so classic. Breast J 2020; 26:1245-1248. [PMID: 32311182 DOI: 10.1111/tbj.13846] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/17/2020] [Accepted: 03/27/2020] [Indexed: 12/24/2022]
Abstract
IgG4-related sclerosing mastitis (IgG4-RM) is a rare, benign, mass-forming fibroinflammatory disease of the breast that can mimic a neoplastic process and pose a diagnostic challenge to clinicians and pathologists. We present a case of IgG4-RM in an 84-year-old woman and highlight the characteristic morphologic findings that should allow the pathologist to raise the possibility of this entity. We also briefly discuss pathophysiology and differential diagnosis. Awareness of clinical and morphologic features suggestive of this rare condition and its variable presentation in the breast can prevent unnecessary surgeries as well as undue patient anxiety.
Collapse
Affiliation(s)
- Polycarp Erivwo
- Department of Laboratory Medicine and Pathobiology, University of Toronto, Toronto, ON, Canada.,Memorial University of Newfoundland, St. John's, NL, Canada
| | | | | | - Anita Godra
- Department of Laboratory Medicine, Michael Garron Hospital, Toronto East Health Network, Toronto, ON, Canada
| | - Gulisa Turashvili
- Department of Laboratory Medicine and Pathobiology, University of Toronto, Toronto, ON, Canada.,Department of Pathology and Laboratory Medicine, Mount Sinai Hospital, Toronto, ON, Canada
| |
Collapse
|
|
15
|
Rosai-Dorfman Disease of the Breast With Variable IgG4+ Plasma Cells: A Diagnostic Mimicker of Other Malignant and Reactive Entities. Am J Surg Pathol 2020; 43:1653-1660. [PMID: 31436555 DOI: 10.1097/pas.0000000000001347] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
Rosai-Dorfman disease (RDD) is an uncommon disorder, characterized by an atypical expansion of histiocytes which classically shows emperipolesis and immunoreactivity with S-100 protein. RDD affects the lymph nodes as well as extranodal sites; however, RDD of the breast is exceptionally rare. Herein, we describe the histopathologic features of 22 cases of RDD occurring in the breast, with an emphasis on the differential diagnosis. All cases were notable for an exuberant lymphocytic infiltrate with and without germinal center formation, and the majority (19/22) showed numerous plasma cells: 5 to 132/high-power field (HPF). IgG and IgG4 immunohistochemical stains were available for 13 cases; in no instance were criteria for IgG4-related sclerosing disease met, though in a single case the IgG4/IgG ratio was increased to 25%. Sclerosis was present in the majority of cases (18/22), and was frequently prominent. RDD cells showing emperipolesis were present in all cases (22/22), and ranged from rare (<1/50 HPF) to numerous (>50/50 HPF). Two of the cases in our series were initially misdiagnosed as inflammatory myofibroblastic tumor and plasma cell mastitis with granulomatous inflammation. As emperipolesis can be indistinct, the presence of stromal fibrosis and a prominent lymphoplasmacytic inflammatory infiltrate should prompt a careful search for the characteristic histiocytes, which can be aided by the use of S-100 immunohistochemistry.
Collapse
|
|
16
|
Goulabchand R, Hafidi A, Van de Perre P, Millet I, Maria ATJ, Morel J, Le Quellec A, Perrochia H, Guilpain P. Mastitis in Autoimmune Diseases: Review of the Literature, Diagnostic Pathway, and Pathophysiological Key Players. J Clin Med 2020; 9:jcm9040958. [PMID: 32235676 PMCID: PMC7231219 DOI: 10.3390/jcm9040958] [Citation(s) in RCA: 50] [Impact Index Per Article: 10.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/08/2020] [Revised: 03/24/2020] [Accepted: 03/25/2020] [Indexed: 12/13/2022] Open
Abstract
Mastitis frequently affects women of childbearing age. Of all the pathological breast conditions requiring specific management, autoimmune mastitis is in the third position after infection and breast cancer. The aim of this literature review was to make a comprehensive description of autoimmune diseases targeting the mammary gland. Four main histological patterns of autoimmune mastitis are described: (i) lymphocytic infiltrates; (ii) ductal ectasia; (iii) granulomatous mastitis; and (iv) vasculitis. Our literature search found that all types of autoimmune disease may target the mammary gland: organ-specific diseases (diabetes, thyroiditis); connective tissue diseases (such as systemic erythematosus lupus or Sjögren’s syndrome); vasculitides (granulomatosis with polyangiitis, eosinophilic granulomatosis with polyangiitis, giant cell arteritis, polyarteritis nodosa, Behçet’s disease); granulomatous diseases (sarcoidosis, Crohn’s disease); and IgG4-related disease. Cases of breast-specific autoimmune diseases have also been reported, including idiopathic granulomatous mastitis. These breast-limited inflammatory diseases are sometimes the first symptom of a systemic autoimmune disease. Although autoimmune mastitis is rare, it is probably underdiagnosed or misdiagnosed. Early diagnosis may allow us to detect systemic diseases at an earlier stage, which could help to initiate a prompt, appropriate therapeutic strategy. In case of suspected autoimmune mastitis, we hereby propose a diagnostic pathway and discuss the potential pathophysiological pathways leading to autoimmune breast damage.
Collapse
Affiliation(s)
- Radjiv Goulabchand
- St Eloi Hospital, Department of Internal Medicine and Multi-Organic Diseases, Local Referral Center for Systemic and Autoimmune Diseases, 80 Avenue Augustin Fliche, F-34295 Montpellier, France; (R.G.); (A.T.J.M.); (A.L.Q.)
- Internal Medicine Department, Caremeau University Hospital, 30029 Nimes, France
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
- Inserm U1183, Institute for Regenerative Medicine and Biotherapy, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295 Montpellier, France
| | - Assia Hafidi
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
- Gui de Chauliac Hospital, Pathology Department, 80 Avenue Augustin Fliche, 34295 Montpellier, France
| | - Philippe Van de Perre
- Pathogenesis and Control of Chronic Infections, Univ Montpellier, INSERM, EFS, Montpellier University Hospital, 34394 Montpellier, France;
| | - Ingrid Millet
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
- Lapeyronie Hospital, Montpellier University, Medical Imaging Department, 371 Avenue du Doyen Gaston Giraud, 34295 Montpellier, France
| | - Alexandre Thibault Jacques Maria
- St Eloi Hospital, Department of Internal Medicine and Multi-Organic Diseases, Local Referral Center for Systemic and Autoimmune Diseases, 80 Avenue Augustin Fliche, F-34295 Montpellier, France; (R.G.); (A.T.J.M.); (A.L.Q.)
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
- Inserm U1183, Institute for Regenerative Medicine and Biotherapy, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295 Montpellier, France
| | - Jacques Morel
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
- Department of Rheumatology, CHU and University of Montpellier, 34295 Montpellier, France
| | - Alain Le Quellec
- St Eloi Hospital, Department of Internal Medicine and Multi-Organic Diseases, Local Referral Center for Systemic and Autoimmune Diseases, 80 Avenue Augustin Fliche, F-34295 Montpellier, France; (R.G.); (A.T.J.M.); (A.L.Q.)
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
| | - Hélène Perrochia
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
- Gui de Chauliac Hospital, Pathology Department, 80 Avenue Augustin Fliche, 34295 Montpellier, France
| | - Philippe Guilpain
- St Eloi Hospital, Department of Internal Medicine and Multi-Organic Diseases, Local Referral Center for Systemic and Autoimmune Diseases, 80 Avenue Augustin Fliche, F-34295 Montpellier, France; (R.G.); (A.T.J.M.); (A.L.Q.)
- Montpellier School of Medicine, University of Montpellier, 34967 Montpellier, France (I.M.); (J.M.); (H.P.)
- Inserm U1183, Institute for Regenerative Medicine and Biotherapy, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295 Montpellier, France
- Correspondence: ; Tel.: +33-467-337332
| |
Collapse
|
|
17
|
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.
Collapse
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
| |
Collapse
|
|
18
|
Immunoglobulin G4 related mastitis: A case report. MARMARA MEDICAL JOURNAL 2020. [DOI: 10.5472/marumj.681980] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/18/2022]
|
|
19
|
Matsubayashi H, Ishiwatari H, Imai K, Kishida Y, Ito S, Hotta K, Yabuuchi Y, Yoshida M, Kakushima N, Takizawa K, Kawata N, Ono H. Steroid Therapy and Steroid Response in Autoimmune Pancreatitis. Int J Mol Sci 2019; 21:E257. [PMID: 31905944 PMCID: PMC6981453 DOI: 10.3390/ijms21010257] [Citation(s) in RCA: 35] [Impact Index Per Article: 5.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/25/2019] [Revised: 12/18/2019] [Accepted: 12/25/2019] [Indexed: 12/11/2022] Open
Abstract
Autoimmune pancreatitis (AIP), a unique subtype of pancreatitis, is often accompanied by systemic inflammatory disorders. AIP is classified into two distinct subtypes on the basis of the histological subtype: immunoglobulin G4 (IgG4)-related lymphoplasmacytic sclerosing pancreatitis (type 1) and idiopathic duct-centric pancreatitis (type 2). Type 1 AIP is often accompanied by systemic lesions, biliary strictures, hepatic inflammatory pseudotumors, interstitial pneumonia and nephritis, dacryoadenitis, and sialadenitis. Type 2 AIP is associated with inflammatory bowel diseases in approximately 30% of cases. Standard therapy for AIP is oral corticosteroid administration. Steroid treatment is generally indicated for symptomatic cases and is exceptionally applied for cases with diagnostic difficulty (diagnostic steroid trial) after a negative workup for malignancy. More than 90% of patients respond to steroid treatment within 1 month, and most within 2 weeks. The steroid response can be confirmed on clinical images (computed tomography, ultrasonography, endoscopic ultrasonography, magnetic resonance imaging, and 18F-fluorodeoxyglucose-positron emission tomography). Hence, the steroid response is included as an optional diagnostic item of AIP. Steroid treatment results in normalization of serological markers, including IgG4. Short- and long-term corticosteroid treatment may induce adverse events, including chronic glycometabolism, obesity, an immunocompromised status against infection, cataracts, glaucoma, osteoporosis, and myopathy. AIP is common in old age and is often associated with diabetes mellitus (33-78%). Thus, there is an argument for corticosteroid therapy in diabetes patients with no symptoms. With low-dose steroid treatment or treatment withdrawal, there is a high incidence of AIP recurrence (24-52%). Therefore, there is a need for long-term steroid maintenance therapy and/or steroid-sparing agents (immunomodulators and rituximab). Corticosteroids play a critical role in the diagnosis and treatment of AIP.
Collapse
Affiliation(s)
- Hiroyuki Matsubayashi
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
- Genetic Medicine Promotion, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan
| | - Hirotoshi Ishiwatari
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Kenichiro Imai
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Yoshihiro Kishida
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Sayo Ito
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Kinichi Hotta
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Yohei Yabuuchi
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Masao Yoshida
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Naomi Kakushima
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Kohei Takizawa
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Noboru Kawata
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| | - Hiroyuki Ono
- Division of Endoscopy, Shizuoka Cancer Center 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka 411-8777, Japan; (H.I.); (K.I.); (Y.K.); (S.I.); (K.H.); (Y.Y.); (M.Y.); (N.K.); (K.T.); (N.K.); (H.O.)
| |
Collapse
|
|
20
|
Bajad S, Tanna D, Ekbote G, Bindroo M, Kaur K, Gupta R. Immunoglobulin G4-Related Mastitis: An Unusual Case of Recurrent Breast Abscess. Arch Rheumatol 2019; 35:623-626. [PMID: 33758821 PMCID: PMC7945706 DOI: 10.46497/archrheumatol.2020.7567] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/24/2019] [Accepted: 08/17/2019] [Indexed: 01/13/2023] Open
Abstract
Immunoglobulin G4-related mastitis (IgG4-RM) is an uncommon entity in clinical practice which has an evolving spectrum of manifestations and presently is of high clinical interest among rheumatologists. Since its closest differential remains breast carcinoma, the importance of describing this entity is in the fact that early diagnosis, awareness and timely management can save the patient from unnecessary surgical intervention and its complications. Tumefactive lesions are considered hallmark of this disease, but rarely abscess may also be the presenting feature. In this article, we describe a 24-year-old female patient of a very unusual case of IgG4-RM presenting as recurrent breast abscess, its successful management and discussion about novel treatment strategies.
Collapse
Affiliation(s)
- Shruti Bajad
- Medanta-The Medicity, Rheumatology and Clinical Immunology, Gurugram, India
| | - Dhaval Tanna
- Medanta-The Medicity, Rheumatology and Clinical Immunology, Gurugram, India
| | - Gayatri Ekbote
- Medanta-The Medicity, Rheumatology and Clinical Immunology, Gurugram, India
| | - Muzaffar Bindroo
- Medanta-The Medicity, Rheumatology and Clinical Immunology, Gurugram, India
| | - Kanchan Kaur
- Medanta-The Medicity, Rheumatology and Clinical Immunology, Gurugram, India
| | - Rajiva Gupta
- Medanta-The Medicity, Rheumatology and Clinical Immunology, Gurugram, India
| |
Collapse
|
|
21
|
Tsuda B, Kumaki N, Ishida R, Mizuno M, Yokoyama K, Oshitanai R, Terao M, Morioka T, Okamura T, Saito Y, Suzuki Y, Niikura N. Distinction of IgG4-related mastitis from breast cancer: a case report. Surg Case Rep 2019; 5:123. [PMID: 31367855 PMCID: PMC6669223 DOI: 10.1186/s40792-019-0681-y] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/04/2018] [Accepted: 07/22/2019] [Indexed: 12/24/2022] Open
Abstract
BACKGROUND Immunoglobulin (Ig) G4-related sclerosing disease is a pathological concept proposed in Japan during the early 2000s. This lesion-forming disease may exhibit characteristics of a systemic disease but often affects a single organ. To date, IgG4-related sclerosing disease in the mammary gland, or IgG4-related mastitis (IgG4-RM), has rarely been reported. CASE PRESENTATION Here, we describe the case of a female patient who was admitted to our hospital with the main complaints of left breast and axillary lymphadenopathy. A careful diagnostic imaging examination led to an initial suspicion of breast cancer. However, a needle biopsy led to a diagnosis of IgG4-RM. Subsequently, the patient was successfully treated with predonin. CONCLUSIONS The treatment requirements for breast cancer and IgG4-RM differ considerably. This is a good example of a case wherein unnecessary surgical treatment, which is indicated for breast cancer, was avoided by needle biopsy. Accordingly, the patient was appropriately treated with steroids following a correct diagnosis.
Collapse
Affiliation(s)
- Banri Tsuda
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan.
| | - Nobue Kumaki
- Department of Pathology, Tokai University School of Medicine, Kanagawa, Japan
| | - Rie Ishida
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan
| | - Mari Mizuno
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan
| | - Kozue Yokoyama
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan
| | - Risa Oshitanai
- Department of Breast and Endocrine Surgery, Tokai University Hachioji Hospital, Hachioji, Japan
| | - Mayako Terao
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan
| | - Toru Morioka
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan
| | - Takuho Okamura
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan
| | - Yuki Saito
- Department of Breast and Endocrine Surgery, Tokai University Hachioji Hospital, Hachioji, Japan
| | - Yasuhiro Suzuki
- Department of Breast and Endocrine Surgery, Tokai University Hachioji Hospital, Hachioji, Japan
| | - Naoki Niikura
- Department of Breast and Endocrine Surgery, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa, 259-1193, Japan
| |
Collapse
|
|
22
|
Kriegsmann M, Gomez C, Heil J, Schäfgen B, Gutjahr E, Kommoss FKF, Kriegsmann K, Flechtenmacher C, Goeppert B, Sinn H. IgG4‐related sclerosing mastitis in a 49‐year‐old patient with multiple, tumor‐like nodules—Diagnostic accuracy of core needle biopsy. Breast J 2019; 25:1251-1253. [DOI: 10.1111/tbj.13436] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/03/2019] [Accepted: 04/03/2019] [Indexed: 12/24/2022]
Affiliation(s)
- Mark Kriegsmann
- Department of Pathology University Hospital Heidelberg Germany
| | - Christina Gomez
- Department of Obstetrics and Gynaecology University Hospital Heidelberg Germany
| | - Jörg Heil
- Department of Obstetrics and Gynaecology University Hospital Heidelberg Germany
| | - Benedikt Schäfgen
- Department of Obstetrics and Gynaecology University Hospital Heidelberg Germany
| | | | | | - Katharina Kriegsmann
- Department of Haematology, Oncology and Rheumatology University Hospital Heidelberg Germany
| | | | | | - Hans‐Peter Sinn
- Department of Pathology University Hospital Heidelberg Germany
| |
Collapse
|
|
23
|
Saied GM, Moustafa KG. Satisfactorily responds to antituberculous treatment: Surgery has no role in the management of tuberculous mastitis in Egyptian women population. A sample study. Breast J 2019; 25:328-330. [PMID: 30738000 DOI: 10.1111/tbj.13220] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/13/2017] [Revised: 11/03/2017] [Accepted: 11/07/2017] [Indexed: 12/22/2022]
Affiliation(s)
- Gamal M Saied
- Department of General Surgery, Faculty of Medicine, Cairo University, Cairo, Egypt
| | - Karim G Moustafa
- Department of Reproductive Health, National Research Centre, Giza, Egypt
| |
Collapse
|
|
24
|
Yokoe T, Hayashida T, Kikuchi M, Watanuki R, Nakashoji A, Maeda H, Toyota T, Seki T, Takahashi M, Iwasaki E, Mikami S, Kameyama K, Kitagawa Y. IgG4-related mastopathy: A case report and literature review. Clin Case Rep 2018; 6:1549-1553. [PMID: 30147902 PMCID: PMC6099038 DOI: 10.1002/ccr3.1657] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/20/2018] [Revised: 05/21/2018] [Accepted: 05/30/2018] [Indexed: 12/24/2022] Open
Abstract
IgG4-related sclerosing disease (IgG4-RD) occasionally involves breast entity, which is often difficult to distinguish from malignant tumor, as both radiologically resembles. We report a case of a breast mass diagnosed as IgG4-related mastopathy (IgG4-RM) through needle biopsy, which responded well to glucocorticoid therapy. Unnecessary excision should be avoided.
Collapse
Affiliation(s)
- Takamichi Yokoe
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Tetsu Hayashida
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Masayuki Kikuchi
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Rurina Watanuki
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Ayako Nakashoji
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Hinako Maeda
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Tomoka Toyota
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Tomoko Seki
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Maiko Takahashi
- Department of SurgeryKeio University School of MedicineTokyoJapan
| | - Eisuke Iwasaki
- Division of Gastroenterology and HepatologyDepartment of Internal MedicineKeio University School of MedicineTokyoJapan
| | - Shuji Mikami
- Diagnostic PathologyKeio University School of MedicineTokyoJapan
| | - Kaori Kameyama
- Diagnostic PathologyKeio University School of MedicineTokyoJapan
| | - Yuko Kitagawa
- Department of SurgeryKeio University School of MedicineTokyoJapan
| |
Collapse
|
|
25
|
Bledsoe JR, Della-Torre E, Rovati L, Deshpande V. IgG4-related disease: review of the histopathologic features, differential diagnosis, and therapeutic approach. APMIS 2018; 126:459-476. [PMID: 29924455 DOI: 10.1111/apm.12845] [Citation(s) in RCA: 81] [Impact Index Per Article: 11.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/31/2017] [Accepted: 04/20/2018] [Indexed: 02/06/2023]
Abstract
Immunoglobulin G4-related disease (IgG4-RD) is an uncommon disorder that demonstrates characteristic clinicopathologic features including sclerosing lesions with storiform fibrosis, increased IgG4+ plasma cells with an increased IgG4+/IgG+ plasma cell ratio, obliterative phlebitis, and often an increased serum IgG4 level. This review summarizes the characteristic histopathologic and clinical features of IgG4-RD with detailed discussion of the histopathologic characteristics of the most commonly involved anatomic sites. We also present recent advances in our understanding of the pathophysiologic mechanisms of IgG4-RD and discuss updates on the treatment, prognosis, and outcomes of this rare disease, including discussion of the possible association between IgG4-RD and malignancy.
Collapse
Affiliation(s)
- Jacob R Bledsoe
- Department of Pathology, UMass Memorial Medical Center, University of Massachusetts, Worcester, MA, USA
| | - Emanuel Della-Torre
- Unit of Immunology, Rheumatology, Allergy, and Rare Diseases (UnIRAR), Università Vita-Salute San Raffaele - San Raffaele Scientific Institute, Milan, Italy.,Ragon Institute of MGH, MIT, and Harvard University, Cambridge, MA, USA
| | - Lucrezia Rovati
- Unit of Immunology, Rheumatology, Allergy, and Rare Diseases (UnIRAR), Università Vita-Salute San Raffaele - San Raffaele Scientific Institute, Milan, Italy.,Ragon Institute of MGH, MIT, and Harvard University, Cambridge, MA, USA
| | - Vikram Deshpande
- The James Homer Wright Pathology Laboratories of the Massachusetts General Hospital, Boston, MA, USA
| |
Collapse
|
|
26
|
Abstract
Immunoglobulin G4-related disease is a fibroinflammatory systemic disease that is characterized by focal or diffuse organ infiltration by immunoglobulin G4-bearing plasma cells. Immunoglobulin G4-related disease may affect any organ, and a high index of suspicion is necessary for early detection to avoid irreversible fibrosis, organ dysfunction, and death. Tumor-forming lesions are common radiological features of immunoglobulin G4-related disease that need to be differentiated from malignancies. The diagnostic approach requires the integration of clinical, biochemical, and radiographic manifestations with classic histopathologic features, which remain crucial to diagnosis. The histology of immunoglobulin G4-related disease is determined by a dense lymphoplasmacytic infiltrate, storiform fibrosis, and obliterative phlebitis in the presence of increased immunoglobulin G4-positve plasma cells. Although immunoglobulin G4-related disease forms a distinct, clinically independent disease category, many questions and problems remain unanswered, especially on its pathogenesis and the role of immunoglobulin G4. Advances in the understanding of immunoglobulin G4-related disease are likely to change the diagnostic approach in the future and create potential targets for therapeutic purposes. Here we describe the concept of immunoglobulin G4-related disease and the most recent knowledge in the clinico-pathological characteristics on this emerging disease. This study can guide clinicians in early diagnosis and prevent unnecessary surgical resections.
Collapse
Affiliation(s)
| | | | - Metin Özdemirli
- Department of Pathology, Medstar Georgetown University Hospital, Washington, USA
| |
Collapse
|
|
27
|
Al-Mujaini A, Al-Khabori M, Shenoy K, Wali U. Immunoglobulin G4-Related Disease: An Update. Oman Med J 2018; 33:97-103. [PMID: 29657677 DOI: 10.5001/omj.2018.20] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022] Open
Abstract
Immunoglobulin G4-related disease (IgG4-RD) is an increasingly recognized immune-mediated condition comprised of a collection of disorders that share specific pathological, serological, and clinical features. IgG4-RD is a fibroinflammatory condition with a tendency to form tumors with inflammatory infiltrate with IgG4 rich plasma cells and elevation of serum IgG4, which may affect virtually every organ and tissue. IgG4-related ophthalmic disease may present as dacryoadenitis, myositis, or involvement of other orbital tissue. Hypophysitis or pachymeningitis may manifest as cranial neuropathies. The diagnosis of IgG4-RD is based on a typical clinical scenario, supportive laboratory test, expected radiological characteristics, and distinct histopathological and immunohistochemical features. Corticosteroids and immunosuppressives form the mainline treatment.
Collapse
Affiliation(s)
- Abdullah Al-Mujaini
- Department of Ophthalmology, College of Medicine and Health Sciences, Sultan Qaboos University Hospital, Muscat, Oman
| | - Murtadha Al-Khabori
- Department of Hematology, College of Medicine and Health Sciences, Sultan Qaboos University Hospital, Muscat, Oman
| | - Kashinatha Shenoy
- Department of Ophthalmology, College of Medicine and Health Sciences, Sultan Qaboos University Hospital, Muscat, Oman
| | - Upender Wali
- Department of Ophthalmology, College of Medicine and Health Sciences, Sultan Qaboos University Hospital, Muscat, Oman
| |
Collapse
|
|
28
|
Goulabchand R, Hafidi A, Millet I, Morel J, Lukas C, Humbert S, Rivière S, Gény C, Jorgensen C, Le Quellec A, Perrochia H, Guilpain P. Mastitis associated with Sjögren's syndrome: a series of nine cases. Immunol Res 2018; 65:218-229. [PMID: 27561784 DOI: 10.1007/s12026-016-8830-x] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/29/2023]
Abstract
Sjögren's syndrome is well known to target exocrine glands, especially lacrimal and salivary glands, which share with mammary glands anatomical, histological, and immunological features. Herein, we investigated the mammary involvement in patients with Sjögren's syndrome and compared the histological findings with minor salivary gland involvement. We reviewed the charts of patients with Sjögren's syndrome (followed in Montpellier University Hospital, between January 2000 and January 2015), in whom minor salivary gland and mammary tissues were available. Two expert pathologists analysed retrospectively these tissues in order to identify inflammatory patterns. Immunohistochemical stainings were performed to precise leucocyte distribution. Sixteen Sjögren's syndrome patients with available salivary and breast tissue samples were included. All were women, with a median age of 60.1 ± 11.3 years at Sjögren's syndrome diagnosis. Mammary biopsy was conducted because of breast symptoms in 6 patients and following imaging screening strategies for breast cancer in 10 patients. Nine patients exhibited an inflammatory breast pattern (lymphocytic infiltrates or duct ectasia), close to minor salivary gland histological findings. Immunohistochemical stainings (n = 5) revealed B and T cell infiltrates within breast tissue, with a higher proportion of T CD4+ cells, but no IgG4-secreting plasma cells were found. This is the first series to describe breast inflammatory patterns in Sjögren's syndrome. Mastitis is in line with the classical involvement of exocrine glands in this disease. These findings are consistent with the literature data considering Sjögren's syndrome as an "autoimmune epithelitis".
Collapse
Affiliation(s)
- Radjiv Goulabchand
- Department of Internal Medicine, Maladies Multi-Organiques, Centre de compétence "maladies systémiques et auto-immunes rares", St Eloi Hospital, CHRU de Montpellier, 80 Avenue Augustin Fliche, 34295, Montpellier Cedex 5, France
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
| | - Assia Hafidi
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
- Pathology Department, Gui de Chauliac Hospital, Montpellier University, 80 Avenue Augustin Fliche, 34295, Montpellier, France
| | - Ingrid Millet
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
- Medical Imaging Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France
| | - Jacques Morel
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
- Rheumatology Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France
| | - Cédric Lukas
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
- Rheumatology Department, Lapeyronie Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France
| | - Sébastien Humbert
- Department of Internal Medicine, Maladies Multi-Organiques, Centre de compétence "maladies systémiques et auto-immunes rares", St Eloi Hospital, CHRU de Montpellier, 80 Avenue Augustin Fliche, 34295, Montpellier Cedex 5, France
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
- Internal Medicine Department, Jean Minjoz Hospital, Besançon University, 3 Boulevard Fleming, 25030, Besançon, France
| | - Sophie Rivière
- Department of Internal Medicine, Maladies Multi-Organiques, Centre de compétence "maladies systémiques et auto-immunes rares", St Eloi Hospital, CHRU de Montpellier, 80 Avenue Augustin Fliche, 34295, Montpellier Cedex 5, France
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
| | - Christian Gény
- Neurology Department, Gui de Chauliac Hospital, Montpellier University, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France
| | - Christian Jorgensen
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
- Clinical Immunology and Osteoarticular Diseases Therapeutic Unit, Lapeyronie Hospital, 371 Avenue du Doyen Gaston Giraud, 34295, Montpellier, France
- Inserm U1183, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295, Montpellier, France
| | - Alain Le Quellec
- Department of Internal Medicine, Maladies Multi-Organiques, Centre de compétence "maladies systémiques et auto-immunes rares", St Eloi Hospital, CHRU de Montpellier, 80 Avenue Augustin Fliche, 34295, Montpellier Cedex 5, France
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
| | - Hélène Perrochia
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France
- Pathology Department, Gui de Chauliac Hospital, Montpellier University, 80 Avenue Augustin Fliche, 34295, Montpellier, France
| | - Philippe Guilpain
- Department of Internal Medicine, Maladies Multi-Organiques, Centre de compétence "maladies systémiques et auto-immunes rares", St Eloi Hospital, CHRU de Montpellier, 80 Avenue Augustin Fliche, 34295, Montpellier Cedex 5, France.
- UFR Médecine (Medical School), Montpellier University, 34295, Montpellier, France.
- Inserm U1183, St Eloi Hospital, 80 Avenue Augustin Fliche, 34295, Montpellier, France.
| |
Collapse
|
|
29
|
Tan ES, Friesen B, Loh SF, Fox J. Immunoglobulin-G4 related mastitis: A case report. Int J Surg Case Rep 2017; 37:169-172. [PMID: 28735232 PMCID: PMC5522915 DOI: 10.1016/j.ijscr.2017.06.012] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/08/2017] [Revised: 06/11/2017] [Accepted: 06/11/2017] [Indexed: 12/11/2022] Open
Abstract
IgG4-RM is exceedingly rare with only ten reported cases in the literatreu. Diagnosis of IgG4-RM is based exclusively on histological analysis. It is a benign chronic inflammatory process that can be treated sufficiently with excision or steroid. Extensive whole body imaging is generally not recommended unless the patient is symptomatic. Introduction IgG4-related mastitis (IgG4-RM) is exceedingly rare with only ten cases reported in the literature to date. Organs that are affected with IgG4-related disease (IgG4-RD) all share the same histopathological hallmarks consisting of dense lymphocytic infiltration, storiform fibrosis and obliterative phlebitis. Presentation of case This case report highlights a case of IgG4-RM found incidentally in a 52-year-old woman during a routine breast screen and it explores the current literature about IgG4-RM and IgG4-RD. Discussion IgG4-RM and IgG4-RD, in general, is a new entity in the field of medicine and its aetiology is not well understood. In the literature, IgG4-RM often presents as a painless palpable breast lump in isolation or with other systemic manifestations. IgG4-RM is considered benign and has excellent prognosis post-conservative treatment with steroid or surgical excision. Conclusion IgG4-RM is diagnosed exclusively on histological analysis. It is hard to distinguish IgG4-RD from malignant breast lesions purely on clinical examination and imaging studies. Increasing awareness of this condition among clinicians will assist them in managing patients better. Extensive whole body imaging is not recommended unless symptomatic.
Collapse
Affiliation(s)
- Ee Syn Tan
- Department of General Surgery (Monash Health), 135-145 David Street, Dandenong, VIC 3175, Australia.
| | - Brendon Friesen
- Lake Imaging, St John of God Hospital, Geelong, VIC, Australia
| | - Seow Foong Loh
- Department of Breast Surgery (Monash Health), Moorabbin Hospital, 823-865 Centre Rd, Bentleigh East, VIC 3165, Australia
| | - Jane Fox
- Department of Breast Surgery (Monash Health), Moorabbin Hospital, 823-865 Centre Rd, Bentleigh East, VIC 3165, Australia
| |
Collapse
|
|
30
|
Vitkovski T, Marder GS, Filardi DA, Gupta E, Breuer F. IgG4-Related Sclerosing Disease of the Breast in a Male Patient. Int J Surg Pathol 2017; 25:711-715. [PMID: 28612666 DOI: 10.1177/1066896917714324] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/11/2023]
Abstract
IgG4-related sclerosing disease of the breast is a rare entity with 10 reports in the literature. We report the first case in a male patient. A 48-year-old male presented with 4-week history of palpable right upper outer quadrant breast mass associated with skin puckering. He reported a family history of breast cancer in his aunt. Ultrasound and mammography showed a spiculated 2.5-cm mass associated with skin retraction with extension to the pectoralis muscle. Ultrasound-guided core biopsy was performed. The findings were interpreted as acute and chronic inflammatory process. The patient subsequently denied improvement of the mass, and an excisional biopsy was performed. Histologic examination showed fibrosis and dense lymphoplasmacytic inflammation in the breast mass and muscle biopsy. Focal obliterative phlebitis was noted. IgG4-positive plasma cells were increased with counts of over 50 per high-power field. The diagnosis was confirmed as sclerosing IgG4-related disease of breast.
Collapse
Affiliation(s)
- Taisia Vitkovski
- 1 Department of Pathology and Laboratory Medicine, Hofstra Northwell Health, New Hyde Park, NY, USA
| | - Galina S Marder
- 2 Department of Internal Medicine Division of Rheumatology, Hofstra Northwell Health, New Hyde Park, NY, USA
| | - Dominic A Filardi
- 3 Department of Surgery, Hofstra Northwell Health, New Hyde Park, NY, USA
| | - Ekta Gupta
- 4 Department of Radiology, Hofstra Northwell Health, New Hyde Park, NY, USA
| | - Frank Breuer
- 1 Department of Pathology and Laboratory Medicine, Hofstra Northwell Health, New Hyde Park, NY, USA
| |
Collapse
|
|
31
|
Erdheim-Chester Disease of the Breast Without Systemic Involvement. J Clin Rheumatol 2017; 23:228-230. [PMID: 28538279 DOI: 10.1097/rhu.0000000000000464] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
|
|
32
|
Zalaquett E, Razmilic D, Oddo D. Immunoglobulin G4 –related Sclerosing Mastitis:AIRP Best Cases in Radiologic-Pathologic Correlation. Radiographics 2016; 36:959-62. [DOI: 10.1148/rg.2016150235] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022]
|
|
33
|
Allen SG, Soliman AS, Toy K, Omar OS, Youssef T, Karkouri M, Ayad E, Abdel-Aziz A, Hablas A, Tahri A, Oltean HN, Kleer CG, Merajver SD. Chronic Mastitis in Egypt and Morocco: Differentiating between Idiopathic Granulomatous Mastitis and IgG4-Related Disease. Breast J 2016; 22:501-9. [PMID: 27279578 PMCID: PMC5007188 DOI: 10.1111/tbj.12628] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
Idiopathic granulomatous mastitis (IGM) is a benign, frequently severe chronic inflammatory lesion of the breast. Its etiology remains unknown and reported cases vary in their presentation and histologic findings with an optimal treatment algorithm yet to be described owing mainly to the disease's heterogeneity. IgG4‐related disease (IgG4‐RD) is a newly recognized systemic fibroinflammatory condition characterized by a dense lymphoplasmacytic infiltrate with many IgG4‐positive plasma cells, storiform fibrosis, and obliterative phlebitis. Immunosuppressive therapy is considered to be an effective first‐line therapy for IgG4‐RD. We sought to clarify and classify chronic mastitis according to the histologic findings of IgG4‐RD mastitis with respect to IGM and to develop a robust diagnostic framework to help select patients for optimal treatment strategies. Using the largest collection to date (43 cases from Egypt and Morocco), we show that despite sharing many features, IGM and IgG4‐RD mastitis are separate diseases. To diagnostically separate the diseases, we created a classification schema—termed the Michigan Classification—based upon our large series of cases, the consensus statement on IgG4‐RD, and the histologic description of IGM in the literature. Using our classification, we discerned 17 cases of IgG4‐RD and 8 cases of IGM among the 43 chronic mastitis cases, with 18 indeterminate cases. Thus, our Michigan Classification can form the basis of rational stratification of chronic mastitis patients between these two clinically and histopathologically heterogeneous diseases.
Collapse
Affiliation(s)
- Steven G Allen
- Internal Medicine, University of Michigan, Ann Arbor, Michigan
| | - Amr S Soliman
- Epidemiology, University of Nebraska, Omaha, Nebraska
| | - Kathleen Toy
- Pathology, University of Michigan, Ann Arbor, Michigan
| | | | - Tamer Youssef
- Surgical Oncology, Mansoura University, Mansoura, Egypt
| | - Mehdi Karkouri
- Pathology, Hassan the Second University, Casablanca, Morocco
| | - Essam Ayad
- Pathology, Cairo University, Cairo, Egypt
| | | | | | - Ali Tahri
- Centre Hospitalier Universitaire-Mohamed VI, Marrakesh, Morocco
| | - Hanna N Oltean
- Epidemiology, University of Michigan, Ann Arbor, Michigan
| | | | | |
Collapse
|
|
34
|
Troxell ML, Gordon NT, Doggett JS, Ballard M, Vetto JT, Pommier RF, Naik AM. Cystic Neutrophilic Granulomatous Mastitis: Association With Gram-Positive Bacilli and Corynebacterium. Am J Clin Pathol 2016; 145:635-45. [PMID: 27247368 DOI: 10.1093/ajcp/aqw046] [Citation(s) in RCA: 33] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/11/2022] Open
Abstract
OBJECTIVES To determine whether cystic neutrophilic granulomatous mastitis (CNGM) can be associated with Gram-positive bacilli and Corynebacterium METHODS We reviewed our experience with 35 granulomatous mastitis patients over a 10-year period, including histologic pattern, Gram stain and other microbiologic data, clinical presentation, treatment and outcome. RESULTS Biopsies from 19 patients demonstrated CNGM, while 16 patients had other patterns of granulomatous mastitis. Gram-positive organisms were seen within microcystic spaces in 16/19 CNGM, but 0/16 non-CNGM patients (P = .000). Culture or molecular studies demonstrated Corynebacterium species in three, all CNGM. Patients with CNGM were more likely to be younger, of Hispanic ethnicity, and born outside of the United States. Granulomatous mastitis resolved after a protracted course with widely variable treatment (antibiotics, surgery, steroids). CONCLUSIONS Our data further support CNGM as an infectious disease; further study of Corynebacterium-directed therapy in CNGM is needed.
Collapse
Affiliation(s)
- Megan L Troxell
- From the Department of Pathology Department of Pathology, Stanford University, Stanford, CA.
| | | | - J Stone Doggett
- Division of Infectious Diseases, Portland Veterans Affairs Medical Center, Portland, OR
| | - Morgan Ballard
- Department of Pathology, Stanford University, Stanford, CA
| | - John T Vetto
- Department of Surgery, Division of Surgical Oncology, Oregon Health & Science University, Portland
| | - Rodney F Pommier
- Department of Surgery, Division of Surgical Oncology, Oregon Health & Science University, Portland
| | - Arpana M Naik
- Department of Surgery, Division of Surgical Oncology, Oregon Health & Science University, Portland
| |
Collapse
|
|
35
|
Yamada R, Horiguchi SI, Yamashita T, Kamisawa T. IgG4-related mastitis, a rare disease, can radiologically and histologically mimic malignancy. BMJ Case Rep 2016; 2016:bcr-2016-214870. [PMID: 27009197 DOI: 10.1136/bcr-2016-214870] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/16/2022] Open
Abstract
IgG4-related disease (IgG4-RD) is characterised by high serum concentrations of IgG4, dense lymphoplasmacytic infiltrates, storiform fibrosis and increased IgG4-positive plasma cells in tissues. This systemic disease occurs in various organs metachronously, but IgG4-related mastitis appears extremely rare. We report a case of IgG4-related mastitis, radiologically considered to represent breast cancer mainly composed of intraductal component and requiring histological differentiation from mucosa-associated lymphoid tissue (MALT) lymphoma. The breast mass disappeared with steroid therapy. When patients have a breast mass, regardless of the presence or absence of IgG4-RD, IgG4-related mastitis should be considered in addition to breast cancer. If histological findings show dense lymphoplasmacytic infiltrates, IgG4-related mastitis should be suspected in addition to malignant lymphoma, and lack of monoclonality should be confirmed. To avoid unnecessary surgery or chemotherapy, knowledge and accurate diagnosis of the entity of IgG4-related mastitis is necessary.
Collapse
Affiliation(s)
- Rin Yamada
- Department of Pathology, Tokyo Metropolitan Komagome Hospital, Bunkyo-ku, Tokyo, Japan
| | - Shin-ichiro Horiguchi
- Department of Pathology, Tokyo Metropolitan Komagome Hospital, Bunkyo-ku, Tokyo, Japan
| | - Toshinari Yamashita
- Department of Breast Surgery, Tokyo Metropolitan Komagome Hospital, Bunkyo-ku, Tokyo, Japan
| | - Terumi Kamisawa
- Department of Internal Medicine, Tokyo Metropolitan Komagome Hospital, Bunkyo-ku, Tokyo, Japan
| |
Collapse
|
|
36
|
Abstract
IgG4-related disease (IgG4-RD) is a fibroinflammatory disease of unknown etiology, which is characterized by a tendency to form tumefactive lesions, increased serum levels of IgG4, and massive infiltration of IgG4-positive plasma cells with storiform fibrosis and/or obliterative phlebitis. Patients with IgG4-RD have frequently multiorgan involvements such as the pancreas, biliary tree, salivary glands, periorbital tissues, kidneys, lungs, lymph nodes, and retroperitoneum. IgG4-RD mainly affects middle-aged to elderly men except for involvement in lachrymal and salivary glands, so-called Mikulicz's disease. The clinical manifestations of IgG4-RD depend on individually involved organs and respond well to steroid, but the prognosis still remains unclear. Some patients develop serious complications such as obstructive jaundice due to hepatic, gallbladder, or pancreatic lesions; hydronephrosis due to retroperitoneal fibrosis; or respiratory symptoms due to pulmonary lesions. Nomenclatures of individual organ manifestation of IgG4-RD have been internationally consented.
Collapse
|
|
37
|
Farooq TA, Mudhar H, Sandramouli S. A case of non-lacrimal immunoglobulin G4 (IgG4)-related orbital disease with mastitis. Orbit 2015; 35:16-9. [PMID: 26700191 DOI: 10.3109/01676830.2015.1099690] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Abstract
IgG4-related orbital disease is a recognised cause for orbital inflammation. As its awareness increases and diagnostic accuracy improves there will be an increased number of cases being identified. This unique case demonstrates for the first time, with histological evidence, a case of a non-lacrimal IgG4-related orbital disease with concurrent IgG4-related mastitis. We describe a 47 year old who presented with a supraorbital swelling and mass. This was initially successfully treated with oral steroids and was later excised on recurrence. Immunohistochemical and blood serum analysis confirmed IgG4-related orbital disease. On systemic enquiry she was found to have a mass of the breast, which was shown to be IgG4-related mastitis. She is currently asymptomatic with no sign of recurrence and is under long-term surveillance. This case highlights the importance of systemic work up in patients presenting with orbital foci of IgG4 disease.
Collapse
Affiliation(s)
- Tahir Ali Farooq
- a Wolverhampton Eye Infirmary, Ophthalmology , Wolverhampton , United Kingdom.,b BMEC, Ophthalmology, City Hospital , Birmingham , United Kingdom
| | - Hardeep Mudhar
- c Royal Hallamshire Hospital, Sheffield Teaching Hospitals , Sheffield , United Kingdom
| | - S Sandramouli
- d Wolverhampton Eye Infirmary, Ophthalmology, New Cross Hospital , Wolverhampton , United Kingdom
| |
Collapse
|
|
38
|
González-Moreno J, Losada López I, Ortego Centeno N. [IgG4-related disease]. Med Clin (Barc) 2015; 145:539-44. [PMID: 25726306 DOI: 10.1016/j.medcli.2014.12.017] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/28/2014] [Revised: 12/17/2014] [Accepted: 12/19/2014] [Indexed: 02/08/2023]
Abstract
IgG4-related disease is a recently described clinicopathological entity showing a wide spectrum of clinical manifestations that share a common pathology. Its most characteristic feature is the formation of inflammatory tumors in different organs, which makes differentiation mainly with neoplastic diseases fundamental. The inflammatory process is typically comprised of IgG4 lymphoplasmacytic cells. The pathophysiological role of the immunoglobulin is not clear. The treatment of choice is corticosteroids. This article aims to summarize the main features of the disease.
Collapse
Affiliation(s)
| | - Inés Losada López
- Servicio de Medicina Interna, Hospital Son Llàtzer, Palma de Mallorca, España
| | - Norberto Ortego Centeno
- Unidad de Enfermedades Autoinmunes Sistémicas, Hospital Clínico San Cecilio, Granada, España
| |
Collapse
|
|
39
|
IgG4 related sclerosing mastitis: expanding the morphological spectrum of IgG4 related diseases. Pathology 2015; 47:27-33. [PMID: 25474510 DOI: 10.1097/pat.0000000000000187] [Citation(s) in RCA: 29] [Impact Index Per Article: 2.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022]
Abstract
IgG4 related disease (IgG4RD) is a recently recognised condition characterised by mass forming lesions associated with storiform fibrosis, obliterative phlebitis, lymphoplasmacytic infiltrate rich in IgG4 positive plasma cells and elevated serum IgG4 levels. Although rare, mammary involvement has been reported as IgG4 related sclerosing mastitis, the morphological counterpart of a growing family of IgG4 related diseases. A total of 17 cases belonging to mass forming benign inflammatory breast lesions such as plasma cell mastitis, granulomatous lobular mastitis, non-specific mastitis and inflammatory pseudotumour were investigated as a possible member of IgG4 related sclerosing mastitis. Clinical, radiological, histopathological and immunohistochemistry findings were noted in all cases. Cases diagnosed as inflammatory pseudotumour showed all the histopathological features of IgG4RD along with increased number of IgG4 positive plasma cells and IgG4/IgG ratio >40%. However, only a few IgG4 positive cells were seen in plasma cell mastitis, granulomatous lobular mastitis and non-specific mastitis cases. These cases also did not fulfill the morphological criteria for the diagnosis of IgG4 related diseases. IgG4RD should be excluded in plasma cell rich lesions diagnosed on core biopsies by IgG4 immunostaining. This can avoid unnecessary surgery as IgG4 related diseases respond to simple and effective steroid treatment.
Collapse
|
|
40
|
Affiliation(s)
- Kanae Kubo
- Department of Allergy and Rheumatology; The University of Tokyo Hospital; Tokyo Japan
| | - Kazuhiko Yamamoto
- Department of Allergy and Rheumatology; The University of Tokyo Hospital; Tokyo Japan
| |
Collapse
|
|
41
|
Brierley D, Kohlhardt SR, Fernando M. IgG4 related disease presenting in breast tissue. ACTA ACUST UNITED AC 2015. [DOI: 10.1016/j.mpdhp.2015.06.019] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022]
|
|
42
|
Abstract
IgG4-related disease (IgG4-RD) is relatively a new growing entity of immune-mediated origin, characterized by a mass-forming lesion, the infiltration of IgG4-positive plasma cells and occasionally elevated serum IgG4. It is considered to be both a systemic inflammation and sclerosing disease. The most common manifestations are parotid and lacrimal swelling, lymphadenopathy and autoimmune pancreatitis. Sclerosing cholangitis and retroperitoneal fibrosis are among the other mentioned frequent manifestations. The diagnosis should be approved histo-pathologically but other conditions such as lymphoma should be carefully excluded. Patients with IgG4-RD respond beneficially to glucocorticoid therapy especially when given at early onset stages. In some cases, the combination of immunosuppressive agents is required.
Collapse
|
|
43
|
Berg AN, Soma L, Clark BZ, Swerdlow SH, Roth CG. Evaluating breast lymphoplasmacytic infiltrates: a multiparameter immunohistochemical study, including assessment of IgG4. Hum Pathol 2015; 46:1162-70. [PMID: 26026200 DOI: 10.1016/j.humpath.2015.04.006] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/20/2015] [Revised: 04/16/2015] [Accepted: 04/17/2015] [Indexed: 12/24/2022]
Abstract
Lymphoplasmacytic infiltrates in the breast, a modified skin appendage, include lymphocytic lobulitis, other nonspecific benign proliferations, and mucosa-associated lymphoid tissue (MALT)-type lymphoma. Distinguishing these entities, all of which may be B-cell rich and may have associated sclerosis, can be difficult. In addition, the proportion that represents IgG4-related disease is unknown, and the similarity of MALT lymphomas to primary cutaneous marginal zone lymphoma is uncertain. To address these questions, the clinical, histologic, and immunohistochemical features of 50 benign and malignant breast lymphoplasmacytic infiltrates (10 lymphocytic lobulitis, 1 granulomatous, 19 not otherwise specified, 20 MALT lymphomas) were evaluated. Compared with the MALT lymphomas, benign cases had a less dense infiltrate (P < .001), fewer but more histologically apparent germinal centers (P < .001), and more marked fibrosis (P < .0001). Greater than 60% B cells were present in 23% (7/30) benign cases versus 75% (15/20) MALT lymphomas (P = .0003). Plasma cells were predominantly IgG+ in 83% (24/29) benign cases and predominantly IgM+ in 73% (14/19) MALT lymphomas (P < .0001). None of the benign cases had greater than 50 IgG4+ plasma cells/high-power field, and only 1 lymphocytic lobulitis case had an IgG4/IgG ratio exceeding 40% and no clinical evidence for extramammary IgG4-related disease. Although there may be some overlapping features, routine histopathology together with limited immunohistochemical stains can distinguish benign from neoplastic lymphoplasmacytic infiltrates in the breast. Despite frequent sclerosis, the breast is not a common site of unrecognized IgG4-related sclerosing disease. Although there are similarities, breast MALT lymphomas can be separated from cutaneous marginal zone lymphoma.
Collapse
Affiliation(s)
- Aaron N Berg
- Department of Pathology, Division of Hematopathology, University of Pittsburgh School of Medicine, Pittsburgh, PA 15213
| | - Lorinda Soma
- Department of Laboratory Medicine, University of Washington, Seattle, WA 98195
| | - Beth Z Clark
- Department of Pathology, Division of Gynecologic and Breast Cancer Pathology, University of Pittsburgh School of Medicine, Pittsburgh, PA 15213
| | - Steven H Swerdlow
- Department of Pathology, Division of Hematopathology, University of Pittsburgh School of Medicine, Pittsburgh, PA 15213
| | - Christine G Roth
- Department of Pathology, Division of Hematopathology, University of Pittsburgh School of Medicine, Pittsburgh, PA 15213.
| |
Collapse
|
|
44
|
Cheng L, Reddy V, Solmos G, Watkins L, Cimbaluk D, Bitterman P, Ghai R, Gattuso P. Mastitis, a Radiographic, Clinical, and Histopathologic Review. Breast J 2015; 21:403-9. [PMID: 25940456 DOI: 10.1111/tbj.12430] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/17/2023]
Affiliation(s)
- Lin Cheng
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| | - Vijaya Reddy
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| | - Gene Solmos
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| | - Latanja Watkins
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| | - David Cimbaluk
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| | - Pincas Bitterman
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| | - Ritu Ghai
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| | - Paolo Gattuso
- Department of Pathology; Rush University Medical Center; Chicago Illinois
| |
Collapse
|
|
45
|
Herrera van Oostdam DA, Jaimes Piñón T, Martínez-Martínez MU, Oros-Ovalle C, Aléman-Sánchez N, Abud-Mendoza C. IgG4-related disease, retrospective histopathological diagnosis. Prevalence in a University Hospital. ACTA ACUST UNITED AC 2015; 11:335-9. [PMID: 25797707 DOI: 10.1016/j.reuma.2014.12.007] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/26/2014] [Revised: 11/24/2014] [Accepted: 12/12/2014] [Indexed: 02/08/2023]
Abstract
INTRODUCTION IgG4 related diseases (IgG4-RD) are characterized mainly by organic dysfunction and inflammation with lymphoplasmacytic cells infiltration. METHODS We conducted a retrospective study. We analyzed patients with a diagnosis of IgG4-RD through histopathologic registries. We divided the study into three phases: (i)extraction of data from the registries of the Pathology Department, including specimens reported with: non-specific inflammation with plasmatic cell infiltration, inflammatory pseudo-tumors and storiform fibrosis, and excluding any report of cancer or infection; (ii)from the selected specimens, three pathologists microscopically re-analyzed these biopsies and included only those who had at least two of the inclusion criteria cited above; (iii)finally, immunostaining was performed in the specimens selected in the second phase. The selected biopsies were catalogued as compatible for IgG4-RD if they had at least 3 inclusion criteria and as probable if they had 2 inclusion criteria. RESULTS On the first phase of the study we analyzed 23,720 biopsies, from which we included 71 and excluded 29 specimens; the rest of the specimens (n=41) underwent immunostaining. From the biopsies included, 41.4% (n=17/71) were positive to IgG4, with the most common histological diagnosis for the positive specimens being granulomatous mastitis, which represented 12.1% of the specimens catalogued initially as probable. The rest of the positive biopsies were from aortitis, dacrioadenitis and/or sialoadenitis, lung pseudo-inflammatory tumor, pericarditis and chronic pancreatitis. CONCLUSIONS The suspicion of IgG4 related disease should not be based solely on clinical manifestations or serology. In the present study we confirm the characteristic changes of IgG4-RD in patients without initial clinical suspicion.
Collapse
Affiliation(s)
| | - Tonatiu Jaimes Piñón
- Unidad Regional de Reumatología y Osteoporosis, Hospital Central, San Luis Potosí, México
| | | | - Cuahutémoc Oros-Ovalle
- Unidad Regional de Reumatología y Osteoporosis, Hospital Central, San Luis Potosí, México
| | - Natalia Aléman-Sánchez
- Unidad Regional de Reumatología y Osteoporosis, Hospital Central, San Luis Potosí, México
| | - Carlos Abud-Mendoza
- Unidad Regional de Reumatología y Osteoporosis, Hospital Central, San Luis Potosí, México.
| |
Collapse
|
|
46
|
IgG4-related disease of the breast: a systemic disease whose mammary manifestations mimic breast cancer. Int Cancer Conf J 2015. [DOI: 10.1007/s13691-015-0209-7] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022] Open
|
|
47
|
|
|
48
|
Abstract
IgG4-related disease (IgG4-RD) is an emerging immune-mediated disease with the capability of involving essentially any organ. The epidemiology of this disease has not been explored in detail. A majority of patients reported in the literature to date are from Japan, but the condition has been described all across the world and there is no strong evidence to suggest a predilection for Asian populations. The mean age at diagnosis is approximately 60 years and there is a decided male predominance for many clinical features, with an overall male:female ratio of 8:3. A cardinal feature of IgG4-RD is single or multiple organ swelling that often raises concern for malignancy. IgG4-RD should be suspected in patients presenting with unexplained enlargement or swelling of one or more organs. Presenting features vary substantially according to the specialty to which patients present first; in addition, the disease can be diagnosed unexpectedly in pathological specimens or identified incidentally on radiology studies. Involvement of major organs is common and IgG4-RD may lead to organ failure, particularly in the pancreas, liver and biliary tree, kidneys, thyroid gland, lungs, and aorta. The diagnosis of IgG4-RD relies on the coexistence of various clinical, laboratory and histopathological findings, although none is pathognomonic by itself.
Collapse
|
|
49
|
Pieringer H, Parzer I, Wöhrer A, Reis P, Oppl B, Zwerina J. IgG4- related disease: an orphan disease with many faces. Orphanet J Rare Dis 2014; 9:110. [PMID: 25026959 PMCID: PMC4223520 DOI: 10.1186/s13023-014-0110-z] [Citation(s) in RCA: 69] [Impact Index Per Article: 6.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/08/2014] [Accepted: 07/03/2014] [Indexed: 02/07/2023] Open
Abstract
Immunoglobulin G4- related disease (IgG4-RD) is a rare systemic fibro-inflammatory disorder (ORPHA284264). Although patients have been described more than 100 years ago, the systemic nature of this disease has been recognized in the 21st century only. Type 1 autoimmune pancreatitis is the most frequent manifestation of IgG4-RD. However, IgG4-RD can affect any organ such as salivary glands, orbits, retroperitoneum and many others. Recent research enabled a clear clinical and histopathological description of IgG4-RD. Typically, lymphoplasmacellular inflammation, storiform fibrosis and obliterative phlebitis are found in IgG4-RD biopsies and the tissue invading plasma cells largely produce IgG4. Elevated serum IgG4 levels are found in many but not all patients. Consequently, diagnostic criteria for IgG4-RD have been proposed recently. Treatment is largely based on clinical experience and retrospective case series. Glucocorticoids are the mainstay of therapy, although adjunctive immunosuppressive agents are used in relapsing patients. This review summarizes current knowledge on clinical manifestations, pathophysiology and treatment of IgG4-RD.
Collapse
|
|
50
|
Extrapancreatic findings of IgG4-related disease. Clin Radiol 2014; 69:209-18. [DOI: 10.1016/j.crad.2013.09.021] [Citation(s) in RCA: 29] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/01/2013] [Revised: 09/18/2013] [Accepted: 09/23/2013] [Indexed: 01/06/2023]
|