Effect of Topical Steroid Treatment on Idiopathic Granulomatous Mastitis: Clinical and Radiologic Evaluation

Idiopathic granulomatous mastitis (IGM) is a rare inflammatory, noncaseating, chronic granulomatous benign disease. The etiology of IGM is still unknown. It is postulated to be an autoimmune localized response. The use of a course of oral steroids provides an important regression of breast mass and skin lesions. Topical corticosteroids are used to treat many skin diseases, but no study is available on the sole use of topical steroids for treating IGM. Eleven women with IGM were treated with topical steroid and evaluated using mammography, ultrasonography, and dynamic MRI. At the end of the 12th week, patients were clinically and radiologically evaluated for the regression of breast and skin lesions. Pre‐ and post‐treatment time‐intensity curve patterns (TICs) were also compared. During the topical steroid treatment, the inflammation signs in the affected breast had markedly disappeared, the fistulas had become inactive, and the fistula orifices and/or skin erosions had closed in nine patients. The median follow‐up period was 17 months (range: 12–48 months). Recurrence was observed in two patients (2/11) at 5 and 8 months, which were treated again topically. The inflammatory findings of the breast skin completely resolved after 8 weeks of treatment, and no side effects or steroid‐related complications occurred. In the pretreatment period, TICs from enhancing areas showed a Type 1 pattern in three cases, a Type 2 pattern in five cases, and a Type 3 pattern in three cases. After topical steroid treatment, TIC was not changed only in one patient (Type 2). Type 1 patterns were determined in seven cases and Type 2 patterns in two cases. In addition, in two patients, TICs were not determined due to complete healing. Our MRI findings showed that topical steroid therapy may be useful because it affects mammary parenchyma as well as mammary skin. Further studies with a greater number of patients are needed to determine the topical steroid therapy dosing and duration, and to better understand the efficacy for treating IGM.     

Combined Long-Term Steroid and Immunosuppressive Treatment Regimen in Granulomatous Mastitis

Background

Idiopathic granulomatous mastitis (IGM) is a rare benign inflammatory disease of the breast. It is related to various etiological factors. The treatment of IGM is challenging as there is a lack of consensus in the literature and treatment options vary widely. Conservative treatment with antibiotics, glucocorticoids and immunosuppressive drugs, and surgery are used in the management of the disease. In this article we report our experience with IGM patients receiving immunosuppressive treatment.

Patients and Methods

The medical records of patients with IGM receiving systemic therapy at the Hacettepe University Hospital between October 2007 and May 2010 were reviewed. 15 cases of histopathologically proven IGM were identified. The data was examined for risk factors and success of treatment.

Results

14 patients were given prednisolone together with azathioprine, and 1 patient who was pregnant at the time of diagnosis received only prednisolone (30 mg/day). 11 (73%) patients had a complete response to systemic therapy. 2 patients had a relapse, of whom 1 required surgical drainage and 1 was treated with a higher dose of glucocorticoids.

Conclusion

Systemic therapy is a safe and effective treatment for IGM. The addition of azathioprine to glucocorticoid therapy permits quick tapering of the steroid doses and increases the treatment success.     

IgG4‐related sclerosing mastitis in a 49‐year‐old patient with multiple,tumor‐like nodules—Diagnostic accuracy of core needle biopsy

Recently, it has been reported that IgG4‐related disease may occur in the breast manifesting as nodular sclerosing interstitial mastitis. Here we report a case with multiple tumor‐like nodules in one breast. The histologic diagnosis was established on core needle biopsies, and treatment was initiated without open biopsy. Diagnosis of IgG4‐related sclerosing mastitis should be suspected in cases of tumor‐like lesions on imaging with an interstitial plasma cell‐rich sclerosing inflammation on histology.     

Management of Idiopathic Granulomatous Mastitis Diagnosed by Core Biopsy: A Retrospective Multicenter Study

Idiopathic granulomatous mastitis (IGM) is a rare, benign, chronic inflammatory condition of the breast, which usually mimics breast carcinoma. The aim of this study was to analyze the clinical features of IGM by identifying a more reliable diagnostic protocol, and evaluating the treatment methods and patient outcomes on follow‐up. We performed a retrospective analysis of 46 patients diagnosed with IGM and managed by the same surgical team between 1999 and 2011, at three high‐volume hospitals. The median age of the patients was 33 years. The most common symptom was painful breast mass (n = 39), followed by abscess (n = 11). All patients underwent ultrasonography (USG). Mammography (MG) and magnetic resonance imaging (MRI) were also performed in 20 patients (43%) and 17 patients (37%), respectively. The mean size of the lesions was 32.8 ± 8.8 mm and ranged from 15 to 50 mm. Preoperative diagnosis of IGM was established by core needle biopsy (CNB) under USG guidance. Eighteen patients (39%) underwent complete excision of the lesion and 25 (54%) were treated with steroids. Three patients treated with steroids subsequently underwent local excision. The mean follow‐up period was 35.4 ± 30.9 months. Eight patients (17%) developed disease recurrence; three of these were successfully treated with steroids, one with surgery, and four with both steroids and surgery. CNB in conjunction with high diagnostic accuracy has a significant role in distinctive diagnosis of IGM and hence, is useful for treatment planning. Treatment can be designated according to the extent and the severity of the disease, and the patient's general health and treatment preferences. Patients with IGM must be closely followed up due to the frequency of disease recurrence.     

The effect of methotrexate monotherapy on treatment-resistant idiopathic granulomatous mastitis patients

BackgroundIdiopathic granulomatous mastitis (IGM) is a disease of unknown etiology, involving a chronic inflammatory process, characterized by noncaseating granuloma formation. IGM can mimic a tumor clinically and radiologically. Since we are a tertiary referral center, most of our patients (n = 56, 87.5%) are secondary admissions who have previously had antibiotics and steroid treatments; therefore, we accept these patients as resistant cases. Here, we aim to present our single-center series of 64 patients with resistant IGM who underwent methotrexate monotherapy. To the best of our knowledge, our study includes the highest number of patients described in the literature with IGM who have undergone this treatment.MethodsThis study included 64 patients, 56 of which were resistant cases, diagnosed with IGM between January 2013 and January 2020 at Istanbul University Cerrahpasa, Cerrahpasa Medical Faculty, General Surgery Breast Outpatient Clinic that were followed-up at least once. These patients were administered oral methotrexate monotherapy 15 mg/week for 24 weeks, and in relapsed cases, the treatment was up to 20 mg/week for 1 year. Folic acid 10 mg/week was given as a supplement to all patients.ResultsComplete recovery was observed in 52 (81.25%) of the 64 patients. Follow-up was discontinued by 4 patients. The dose was increased and the duration of treatment was extended up to 1 year when relapse was observed in 8 patients and complete response was then obtained in these cases. Only 3 patients (4.69%) experienced side effects and were switched to subcutaneous treatment due to nausea.ConclusionConsidering the high patient compliance, low recurrence, minimal side effects, and overall success of the treatment, we believe that methotrexate monotherapy may be used in treatment-resistant IGM patients and may also be the first choice for first-line treatment in the future.     

The Role of Interleukin-33 as an Inflammatory Marker in Differential Diagnosis of Idiopathic Granulomatous Mastitis and Breast Cancer

Introduction: Idiopathic granulomatous mastitis (IGM) is a rare benign inflammatory breast disease that can clinically and radiographically mimic breast carcinoma and bacterial mastitis. The aim of this study is to investigate the importance of levels of the inflammation markers, interleukin-33 (IL-33), soluble ST2 receptor of IL-33 (sST2), procalcitonin (PCT), and CRP on differential diagnosis of IGM and breast cancer (BC). Methods: 25 patients with IGM and 32 patients with primary BC who applied to General Surgery Clinic, and 30 healthy volunteer women with similar demographic condition were enrolled in the study. While the IL-33, sST2, and PCT levels were measured with ELISA method, other biochemical parameters were studied with autoanalyzer. Results: IL-33, sST2 levels were significantly higher in both BC and IGM groups than in control (p < 0.001). More importantly, the IL-33 levels of IGM group were found to be significantly higher when compared to the group with BC (p < 0.001). Accordingly the sST2 levels of the patients with IGM were significantly lower than the patients with BC (p < 0.001). There was no statistical significance between PCT levels BC and IGM groups (p > 0.05). Conclusion: The results of our study suggest that measurement of IL-33 and its receptor sST2 levels, alongside radiological studies can help distinguishing IGM from BC. Findings of our study need to be supported with additional studies.     

Low-Dose Methotrexate Use in Idiopathic Granulomatous Mastitis: An Alternative Treatment Method

BackgroundIdiopathic granulomatous mastitis (IGM) is a rare, recurrent and progressive breast disease with an unknown etiology. Patients with IGM will probably face stressful, time-consuming treatment procedures with side effects due to medications. There are different treatment modalities in clinical use including medical and surgical interventions.ObjectiveThe aim of this study was to present the results of using the combination therapy of low-dose methotrexate (MTX) and steroid in IGM.MethodsSeventeen patients diagnosed with IGM and treated with MTX were included into the study. Low-dose MTX at 5 mg/week and 8 mg/day prednisone were given for 2–3 months.ResultsAfter 2–3 months of treatment, 10 patients exhibited (58.5%) complete, 3 patients (17.6%) partial recovery, and no response to the treatment process was observed in 4 patients (23.5%). No side effects of MTX and recurrent events were noted in any of the patients.ConclusionLow-dose MTX and prednisone treatment for IGM patients, who did not respond to steroids alone, should be considered as an alternative treatment method instead of surgical intervention.     

浆细胞性乳腺炎外科治疗方法初步探索(附62例报告)

目的探讨不同时期浆细胞性乳腺炎的治疗方法及其疗效。方法62例浆细胞性乳腺炎患者中,根据疾病的不同阶段采取不同的处理方式:因导管扩张行溢液乳段切除术24例(38.7%),因急、慢性炎症行乳腺区段切除术11例(17.7%),因脓肿形成行B超引导下乳腺脓肿穿刺冲洗术8例(12.9%),脓肿置管引流术4例(6.5%),脓肿切开引流10例(16.1%),因瘘管或窦道形成行瘘管或窦道切除术5例(8.1%)。结果乳腺导管扩张期、急慢性炎症期行溢乳段切除、乳腺区段切除的35例均愈合,脓肿形成期的22例中,2例经3次脓肿穿刺冲洗未愈而改行切开引流术,余均痊愈,但行脓肿切开引流术的10例中复发4例(6.5%);窦道期的5例经1~10个月换药痊愈。结论对于浆细胞性乳腺炎不同的阶段应选择适当的治疗方法,避免贸然采取乳腺单纯切除术,可使患者手术创伤减小,生活质量提高。     

A Comparative Study of Conservative versus Surgical Treatment Protocols for 77 Patients with Idiopathic Granulomatous Mastitis

The purpose of this study was to analyze the clinical features and demographic data of patients with idiopathic granulomatous mastitis (IGM) and to compare the results of conservative versus surgical treatment protocols. The demographic data, clinical findings, microbiological and pathologic features, scanning and treatment methods, recurrence, and recovery rates of 77 patients were analyzed retrospectively. The patients were divided into two groups based on the type of treatment received. Core biopsies were used to diagnose 37 patients: 26 using incisional biopsies and 14 using excisional biopsies. Of the patient population with IGM, 31 were treated with surgical excision, one with a simple mastectomy, and one with a subcutaneous mastectomy combined with a breast implant, whereas 44 were treated with steroids. The recovery rates of the 44 patients who were treated conservatively were 6 (1–15) months while for the 33 patients who were treated surgically, it was 1 (1–5) month (p = 0.001). Nine patients from the conservative treatment group experienced a recurrence while there were no recurrences in the surgically treated group (p = 0.009). Among all patients, the recurrence rate was 11.7% (9/77) while the average follow‐up period was 16.57 ± 18.57 months. As a comparative study between conservative treatment protocols and surgical ones for patients with idiopathic granulomatous mastitis (IGM), this study is the largest to date. A wide surgical excision is the preferred approach for treating patients with IGM because of the low recurrence rate.     

Methotrexate in the management of idiopathic granulomatous mastitis: review of 108 published cases and report of four cases

This study aimed to discuss the role of agents, such as steroids and methotrexate (MTX), in the treatment of patients with idiopathic granulomatous mastitis (IGM). Using Pubmed and Google Scholar data bases, a retrospective study was carried out on IGM cases treated with steroids and/or MTX between 1972 and 2010. Four IGM cases treated with MTX at our clinic were also summarized in this study. A total of 541 IGM cases since 1972, including ours, were retrospectively analyzed. Steroid treatment 5-85 mg was administered over 5 days-22 months to 112 patients aged 21-48 years. Recurrence occurred in 22 patients, steroid-induced diabetes mellitus in 5 patients, no response to treatment was observed in 4 patients, in 2 patients, the mass decreased in size, and static disease was observed in one. The steroid treatment was changed to MTX treatment in 4 patients who had recurrence, 5 with steroid-induced DM and in 4 who were nonrespondents. Three patients were started on steroid together with MTX as a primary treatment. Of the patients treated with MTX, a satisfactory result was obtained in 14 cases and in 2, mastectomy was performed because of recurrence despite the treatment with MTX. IGM is a troublesome condition that presents management problems due to the side effects of steroids. Our study demonstrates that the use of MTX in IGM cases has been effective in preventing complications, in resolving the inflammatory process, and in limiting side effects of corticosteroids.     

Mastitis,a Radiographic,Clinical, and Histopathologic Review

Mastitis is a benign inflammatory process of the breast with heterogeneous histopathological findings, which clinically and radiographically may mimic a mammary carcinoma. We undertook a retrospective study on 37 cases of mastitis in our institution to correlate the radiographic imaging features and the clinical presentation with the histopathological findings. Histologically, there were 21 granulomatous, 7 fibrous, 3 plasma cell, 3 lupus, 2 lymphocytic, and 1 case of acute mastitis. Radiographically, 16/25 (64%) patients with ultrasound studies showed irregular hypoechoic masses suspicious for malignancy. Clinically, 38% of patients had an associated systemic disease.     

Idiopathic granulomatous mastitis with erythema nodosum: Is it a variant of clinical presentation indicating treatment resistance? A retrospective cohort study

Idiopathic granulomatous mastitis (IGM) rarely occurs with erythema nodosum (EN) as a systemic finding. However, the impact of their coexistence on disease severity and response to steroids has not been investigated yet. Patients diagnosed with IGM between September 2014 and October 2018 were divided into two groups according to the presence or absence of EN during the first admission retrospectively. The IGM was more severe in patients with EN as it was presented more often as bilateral and diffuse involvement of the breast. Findings of mastitis did not resolve with steroids in 50% of this group. Repetitive excisions and mastectomy with reconstructions were required to control the disease. Coexistence of EN and IGM was found to be related to bilateral and aggressive involvement, which could be associated with insufficient response to steroids. Associated patients should be informed in terms of the aggressive course, and surgery can be highlighted as a first‐line treatment.     

A Novel First-Line Treatment Alternative for Noncomplicated Idiopathic Granulomatous Mastitis: Combined İntralesional Steroid İnjection with Topical Steroid Administration

BackgroundIdiopathic granulomatous mastitis (IGM) is a rare form of nonlactational mastitis. Due to the small number of case series and consequently inadequate prospective studies, there is still no consensus on the optimal treatment of IGM. In this study, we aimed to compare the efficacy of intralesional steroid injection with concomitant topical steroids to systemic steroid therapy only in the treatment of noncomplicated IGM.MethodsBetween June 2015 and April 2018, the patients'' data was prospectively collected and analyzed retrospectively. The study included a total of 78 female patients diagnosed with IGM. Patients were divided into 2 groups: the local steroid treatment group (intralesional steroid injection with topical steroid administration; group 1, n = 46) and the peroral systemic steroid treatment group (group 2, n = 32). Response to the therapy, side effects, recurrence, the need for surgical treatment, and complication rates were compared.ResultsForty-three patients (93.5%) in group 1 achieved a partial or complete response compared to 23 patients (71.9%) in group 2 after 3 months; this difference was significant (p = 0.012). The recurrence rates were significantly lower in group 1 (8.7%) compared to group 2 (46.9%; p = 0.001), and the need for surgical treatment was significantly less in group 1 (2.2%) than in group 2 (9.4%; p = 0.001). While the complication rates were similar between groups, a higher rate of systemic side effects was observed in group 2.ConclusionBased on the results of our study, combined steroid injection and topical steroid treatment in IGM is as effective as systemic steroid treatment. We suggest that this combination therapy of topical steroids and local steroid injection should be used as first-line therapy in patients with noncomplicated IGM.     

Granulomatous Lobular Mastitis Following Drug-Induced Galactorrhea and Blunt Trauma

Abstract: We report a single case of chronic granulomatous lobular mastitis following metoclopramide-related galactorrhea and a blunt trauma in a young parous woman who underwent two conservative operations before becoming symptom-free. We have found only two other literature cases associated with hyperprolactinemia, and our case could be another of this etiologic group. The absence of well-formed granulomas in the first histology specimen in the present case was misleading; it was reinterpreted as granulomatous mastitis only after the second specimen was examined. Reinterpretation was based on the lobular distribution of a lymphoplasmocytic infiltrate (nonspecific chronic lobulitis) and the presence of epithelioid cell sheets and neutrophils in the absence of well-formed granulomas. The case lends further support to the theory of a local immune response initiated by the secreted material or by one of its components in the formation of granulomas. However, contributory factors such as the trauma in this case (a blow from a shovel handle) or systemic disease in others may play a role in the development of the disease, which in some instances may represent a pattern of tissue reactions to different noxious agents.     

Association between Hyperprolactinemia and Granulomatous Mastitis

Granulomatous mastitis (GM) is a relatively uncommon inflammatory breast lesion with multiple suggested etiologies. Although most GM cases show association with lactation and pregnancy, a minority of cases have been linked to hyperprolactinemia caused by either dopamine antagonist medications or with intracranial lesions, such as pituitary adenoma. The goal of this study is to review the GM cases reported in the literature with a specific emphasis on those cases associated with hyperprolactinemia and prolactinomas and to identify cases of GM seen at the Cleveland Clinic Florida which demonstrate co‐occurrences of GM and intracranial lesions. CoPath and Epic data bases at Cleveland Clinic Florida were searched for cases describing inflammatory breast lesions in patients with pituitary pathology. Chart reviews were conducted and pertinent medical history was extracted for case reports. H&E‐stained paraffin‐embedded sections retrieved from Cleveland Clinic Florida pathology storage were evaluated by light microscopy. Four cases showing a co‐occurrence of GM and hyperprolactinemia were consequently identified. A prolactin‐secreting pituitary adenoma was present in two of the three GM cases. The third case demonstrated a concomitant craniopharyngioma, which was also associated with a rise in serum prolactin. This phenomenon was presumably attributable to compression, resulting in compromised transport of dopamine to the adenohypophysis and subsequent disinhibition of prolactin secretion by lactotrophs. The fourth patient with GM had a similar history of elevated prolactin. Classical histopathological features of GM were found in all four cases, including noncaseating granulomas, multinucleated giant cells, epithelioid histiocytes, and chronic inflammation. Intriguingly, complete resolution of inflammatory breast lesions along with normalization of prolactin levels occurred following the surgical excision of the craniopharyngioma, suggesting that intracranial lesion–induced hyperprolactinemia might be directly causal in GM. Therefore, the authors would suggest screening for pituitary tumors and evaluate prolactin levels in the workup of GM patients without a recent history of lactation and pregnancy and no other identified etiology.     

Granulomatous Lobular Mastitis: a Rare Chronic Inflammatory Disease of the Breast which Can Mimic Breast Carcinoma

Granulomatous lobular mastitis is a rare chronic inflammatory disease of the breast. The differential diagnosis with malign breast disease is often not easy. In most cases a surgical biopsy is needed for correct diagnosis. Idiopathic granulomatous mastitis is an exclusion diagnosis, based on the demonstration of a characteristic histological pattern, combined with the exclusion of other possible causes of granulomatous breast lesions. There is still no generally accepted optimal treatment. If surgery forms part of the treatment, a conservative approach seems to be adequate in most cases. Another option is a long-term steroid treatment. It is mandatory to exclude infectious causes of granulomatous mastitis before corticoid therapy is started.     

真空辅助微创活检治疗慢性乳腺炎的临床研究

目的探讨真空辅助微创活检加置管冲洗引流治疗慢性乳腺炎的操作技巧和临床优势。方法2010年6月～2012年5月,超声引导下应用真空辅助微创活检系统加旋切刀对肿块型与脓肿型慢性乳腺炎局部病灶进行切除,残腔冲洗及置管引流,观察其治愈率、治愈时间和复发率。结果32例一期治愈率96．9％（31／32）,仅I例在手术后5天换药仍可见脓液溢出,为术后引流不畅,扩大引流口后22天痊愈。随访时间2—24个月,中位数10个月,局部复发率3．1％（1／32）。结论应用真空辅助微创活检系统可在超声引导下对慢性乳腺炎局部病灶进行大范围甚至完全切除,辅以置管冲洗引流,可很快获得良好的治疗效果。     

Idiopathic Granulomatous Mastitis—A Prospective Study of 49 Women and Treatment Outcomes with Steroid Therapy

Idiopathic granulomatous mastitis (IGM) is an infrequently reported benign breast disease of unknown etiology. Surgical treatment has been widely advocated but can be disfiguring. We describe demographic and clinico‐pathologic features of women with IGM in a safety net hospital, and evaluate steroid therapy as a breast‐conserving modality of treatment. We also examine a possible ethnic predominance in Hispanic women. We conducted an observational prospective cohort study of all women with biopsy‐proven granulomatous mastitis in the breast clinics of an urban safety net public hospital from 2006 to 2010. Demographic, ethnic and clinical data, treatment history, and response to treatment were collected. Patients were followed up prospectively to determine the type of treatment prescribed, complete resolution of disease, and median time to resolution. A nested case–control study was conducted to examine Hispanic prevalence using chi‐square statistic. The mean age was 35 years. 80% were Hispanic. 80% presented with a painful breast mass. 59% initially received antibiotics with incomplete resolution. 90% women were prescribed oral steroids, 3% underwent surgical treatment, and 6% remained under observation with spontaneous resolution. Of those who received steroid, 80% had complete resolution of disease with a median time to complete resolution of 159 days (IQR 120–241 days). Ethnicity data in a nested case–control study revealed that women in the IGM group were more likely to be Hispanic than in the control group with an odds ratio of 3 (95% CI 1.42–6.24, p‐value 0.0032). IGM is a benign but locally aggressive breast disease. Treatment with steroids is an effective breast‐conserving option. Predominance in Hispanic women of childbearing age suggests a common genetic, environmental, immunologic, or infectious etiology and warrants further study with a multi‐disciplinary approach.     

Granulomatous Mastitis,Erythema Nodosum,and Oligoarthritis in a Pregnant Woman

Abstract: Granulomatous mastitis (GM) is an uncommon chronic inflammation of the breast and erythema nodosum (EN), is a rare extramammary manifestation of this entity. We describe a case of GM with EN and arthritis in a young pregnant woman. Review of literature showed rare similar cases.     

Idiopathic granulomatous mastitis associated with hyperprolactinemia: A nonoperative approach

There is increasing evidence associating idiopathic granulomatous mastitis (IGM) with hyperprolactinemia. All documented cases have involved the patient having at least one operative procedure before the association has been made. We present a 55 year old female with IGM associated with risperidone induced hyperprolactinemia. She was successfully treated with a dopamine agonist, bromocriptine. We demonstrated that complete resolution can be achieved without surgical intervention, by targeting serum prolactin levels. We hope this will increase awareness of this rare clinically entity and avoid potentially unnecessary surgery.