Rare Inflammatory Disease Of Breast Research Articles

Is poor oral health a risk factor for idiopathic granulomatous mastitis?

Idiopathic granulomatous mastitis (IGM) is a rare inflammatory breast disease that can be clinically and radiologically mistaken for carcinoma. Although its etiology remains uncertain, potential associations with pregnancy, lactation, hormonal imbalances, autoimmunity, smoking, and various microorganisms have been suggested. This study aimed to evaluate the relationship between IGM and oral health. We included 42 female patients diagnosed with IGM based on histopathological evaluations conducted between September 2018 and October 2023. The reference group consisted of 47 female patients with clinically, radiologically, and laboratory-proven non-specific mastitis and 36 healthy female individuals. The oral health of all participants was evaluated by an experienced dentist using the "Decayed, Missing and Filled Teeth" (DMFT) index and the "Simplified Oral Hygiene Index" (OHI-S). The ages of IGM patients included in this study ranged from 29 to 51 years, with a mean age of 34.88 ± 4.87 years. The most common clinical findings were pain (n = 38), palpable breast mass, erythema, induration, and dermal sinus. Comparison of the OHI-S and DMFT index values among participants revealed that those diagnosed with IGM had significantly higher values than those in the reference group (P < 0.05). Our findings suggest a potential involvement of poor oral health in the etiology of IGM. Future studies should consider oral health as a factor in IGM etiology and explore the oral microbiota in samples obtained from the affected tissue.

Pathological manifestations of granulomatous lobular mastitis.

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.

İdiopatik Granülomatöz Mastitin Ameliyatsız ve Steroidsiz Tedavisi: Tek Beden Herkese Olmaz

Aim: Idiopathic granulomatous mastitis (IGM) is a rare inflammatory breast disease mimicking malignancy&#x0D; with no consensus on the optimal treatment. We aimed to present a single-center algorithm for&#x0D; IGM treatment without surgery and steroids.&#x0D; Material and Methods: This study was conducted at Bülent Ecevit University Hospital, Zonguldak,&#x0D; Turkey, between 2010 and 2020. A prospective database was reviewed to identify patients with biopsyproven&#x0D; IGM who were managed through observation and ultrasound-guided interventions over a 10-&#x0D; year period.&#x0D; Results: Seventy-eight female patients with a confirmed diagnosis of IGM via reevaluation were&#x0D; determined. Of these, 34.61% required ultrasound-guided abscess aspiration and 10.25% required&#x0D; incisional abscess drainage without surgical resection. Complete clinical resolution was achieved within&#x0D; 6 months in 58.97% cases. The overall recurrence rate was 12.82%, and surgical drainage was required&#x0D; in one patient. Univariate logistic regression analysis revealed no statistically significant association&#x0D; between recurrence and the demographic or pathologic factors evaluated. Twenty-two (28.2%) patients&#x0D; with a histopathologic diagnosis of cystic İGM who received antibiotics (doxycycline) achieved complete&#x0D; resolution within 2 weeks and experienced no recurrence.&#x0D; Conclusion: IGM is a self-limiting disease that resolves spontaneously independent of medical&#x0D; intervention and without resection in up to 18 months. After diagnosis, surgery with or without steroids as&#x0D; the first line of therapy should be replaced with close observation and ultrasound-guided interventions&#x0D; according to patients’ preferences.

Drug-free remission is an achievable target with immunosuppressive treatment in idiopathic granulomatous mastitis.

Idiopathic granulomatous mastitis (IGM) is a rare inflammatory breast disease, in which there is no clear established treatment algorithm. Several physicians keep away from using immunosuppressive(IS) treatments in routine clinical practice. This study aimed to evaluate the rates of drug-free remission of the patients with IGM in a period of 3-year follow-up. This retrospective study conducted with 55 biopsy-proven IGM patients, who were followed up between February, 2011, and November, 2021, in rheumatology outpatient clinic of Gulhane Training and Research Hospital. The demographic and clinical characteristics of the patients were obtained from patients' files. The 3-year follow-up data were assessed for long-term outcome analyses. There were 55 female patients with a mean age of 36.8 ± 6.3years. Fifty-four (98.1%) patients were in drug-free remission at the end of 3years. The median duration of drug-free remission in patients receiving methotrexate (MTX), only corticosteroid (CS), and azathioprine was 19.7, 32.9, and 14.7months, respectively. The drug-free remission duration for the patient who received cyclosporine A as IS was 28.3months. The median duration of IS treatment was 15.8months, and the median duration of treatment with CS and other IS combination was 6.7months. Recurrence was observed in 4 (80%) patients without IS therapy after surgery, of whom MTX was used in 3 (75%) patients and achieved remission. IS agents provide high rate of prolonged drug-free remission and should be considered a part of routine medical care of the patients with IGM.

Characteristics and Management of Granulomatous Lobular Mastitis Associated with Antipsychotics-Induced Hyperprolactinemia.

Background: Granulomatous lobular mastitis (GM) is a rare inflammatory breast disease. Reports focusing on GM caused by antipsychotic-induced hyperprolactinemia (HPRL) are very rare. Aim: To report a study of GM associated with antipsychotic-induced HPRL and discuss the mechanism and management. Materials and Methods: A retrospective review of patients with GM and psychiatric disorders were carried out. The clinical characteristics, management and outcome were collected and analyzed. The relationship between antipsychotics and GM was evaluated using the Naranjo Adverse Drug Reaction Probability Scale (Naranjo scale). Results: Nineteen female GM patients with psychiatric diseases, aged 21-39 years, who had received antipsychotics for 0.5-10.2 years were included. Most patients took multiple antipsychotics, and 10 (52.6%) took risperidone-containing regimens. Increased prolactin (PRL) was detected in all patients (range 35.15-200 ng/mL). The scores of Naranjo scale were 7-8, indicated the antipsychotics probably induced GM. All patients received systemic therapy, and were prescribed bromocriptine. Seven patients (36.8%) decreased the dose of antipsychotics, six (31.6%) switched antipsychotics, three (15.8%) continued the primary antipsychotics, and three (15.8%) discontinued antipsychotics. In addition, 14 patients (73.7%) received corticosteroid, 4 (21.1%) received antimycobacterials. PRL decreased to normal in 1 month. Seven patients (36.8%) received excisional surgery. After 12 months' follow-up (range 9-56 months), only three patients (15.8%) had a recurrence. Conclusion: Long-term use of antipsychotics may increase PRL levels, and lead to GM. It is vital to assess PRL level and reduce PRL to normal in patients with GM.

Serum C-Reactive Protein and Interleukin-6 Levels as Biomarkers for Disease Severity and Clinical Outcomes in Patients with Idiopathic Granulomatous Mastitis.

Idiopathic granulomatous mastitis (IGM) is a rare inflammatory breast disease mimicking breast cancer. Limited research has been conducted on the application of serum biomarkers. This study aims to investigate the association of serum biomarkers with disease severity in patients with IGM. From November 2011 to March 2020, medical records of patients with IGM were reviewed. Serum cytokine levels were measured in patients and healthy controls between July 2018 and March 2020. A total of 41 patients with histologically proven IGM were found. Serum interleukin (IL)-6 level was significantly higher in patients with IGM (n = 11) than healthy controls (n = 7). Serum IL-6 and C-reactive protein (CRP) levels were significantly higher in patients with severe disease than mild and moderate disease. Serum IL-6 (Spearman’s ρ = 0.855; p < 0.001) and CRP (Spearman’s ρ = 0.838; p = 0.001) levels were associated with time to resolution. A higher serum CRP level was associated with a longer time to resolution (B = 0.322; p < 0.001) in multiple linear regression analysis. Serum IL-6 and CRP levels can be used as biomarkers for the evaluation of disease severity in IGM. IL-6 may play a crucial role in the immunopathology of IGM.

Postoperative Yanghe decoction regimen improves outcomes for idiopathic granulomatous mastitis: A retrospective cohort study.

The etiology of idiopathic granulomatous mastitis (IGM), a rare inflammatory breast disease, is not understood. There is no consensus regarding the treatment of IGM. The purpose of this study was to determine the efficacy of surgery combined with traditional Chinese medicine for the treatment of IGM.We retrospectively analyzed 53 patients of IGM who were treated with surgical excision at our hospital. Group A (n = 25) included patients treated with only surgery, and Group B included patients treated with surgery combined with postoperative Yanghe decoction. The clinical data were compared between the 2 groups, including demographics, clinical characteristics, and outcomes.All patients were female with a mean age of 34.6 ± 5.9 years. There were no significant differences between the groups regarding preoperative demographics or clinical characteristics. The follow-up time was comparable between the groups (13.2 ± 10.0 vs 12.0 ± 10.2 months). Patients in Group B had shorter complete remission (CR) times than patients in Group A (76.1 ± 15.2 vs 84.0 ± 12.2 days; P < .05). The CR rate was higher in Group B than in Group A (96.4% vs 76.0%; P < .05), and the recurrence rate was lower in Group B than in Group A (0% vs 16.0%; P < .05).The postoperative Yanghe decoction regimen was associated with more rapid recovery after IGM surgery. Surgical management combined with postoperative oral Yanghe decoction treatment yielded a higher CR rate and lower recurrence rate than surgery alone. The effect of traditional Chinese medicine in IGM treatment requires further study.

Idiopathic granulomatous mastitis in a pregnant woman

Introduction: Idiopathic granulomatous mastitis (IGM) is a rare inflammatory breast disease of unknown etiology. It was described in 1972 by Kessler and Wolloch, who reported cases of granulomatous lobulitis with no established relationship with trauma, foreign body, or specific infection. This study aimed at reporting a case of IGM in a pregnant patient. Clinical case: A 36-year-old pregnant woman, with a gestational age of 27 weeks, was admitted with a complaint of right breast pain for 7 days. She denied comorbidities. Clinical examination showed a dense area in the junction of the right inner quadrants, associated with mild hyperemia without signs of fluctuation. Lymphadenopathy was not identified. Ultrasound revealed a non-homogeneous fluid of 4.9x1.7x5.2 cm in the right upper inner quadrant, with a consequent surgical approach and collection of fragments for analysis. Cultures with antibiogram and for fungi and the venereal disease research laboratory (VDRL) tested negative. Histopathological examination showed a chronic inflammatory process with abundant granulation tissue and isolated giant cells, suggesting granulomatous mastitis. Corticosteroid therapy was indicated after the birth, with prednisone 40 mg/day for 3 months, with complete resolution of the symptoms. Discussion: Usually, IGM affects women aged 17 to 42 years. Recent history of pregnancy is common, with identified cases ranging from 2 months to 15 years after the last delivery, rarely occurring during gestation. It manifests as a breast mass of 0.5 to 9 cm in diameter, often unilateral and on the left side. Signs of inflammation and local lymphadenopathy can also be identified. Mammography is not ideal for detecting IGM, with findings of low specificity, and false negatives are frequent in dense breasts. The most frequent ultrasound findings are irregular hypoechoic mass and tubular hypoechoic lesion. IGM can be confirmed by detecting a histological pattern of chronic granulomatous lobulitis, non-caseating granuloma with infiltration of histiocytes, few polymorphonuclear cells, and multinucleated giant cells. Ruling out syphilis, fungi, and breast tuberculosis is important. The recommended treatment is excisional biopsy, preferably a wide excision. However, surgical management has a negative cosmetic impact and is associated with up to 50% of cases of persistence and recurrence of the disease, in addition to complications such as abscesses, fistulas, and ulceration. Corticosteroids are an effective option for controlling the disease, but active surveillance can also be chosen. Considering the above, we underline the importance of early diagnosis with appropriate scientific and technical foundations to provide a better prognosis for patients.

Idiopathic Granulomatous Mastitis - a new approach in diagnostics and treatment.

Idiopathic granulomatous mastitis (IGM) is a rare inflammatory breast disease mimicking carcinoma and puerperal or non-puerperal mastitis. The primary purpose of this prospectively performed case control study was to compare clinical and imaging signs of IGM with the reference group of nonspecific, non- puerperal mastitis (NM) to identify the most typical clinical and imaging signs essential for a correct differential diagnosis. The secondary purpose was to present a new approach to non-invasive treatment. Thirty-nine women with histologically proven IGM and twenty-six patients with nonspecific mastitis underwent clinical examination, breast ultrasound (US), mammography (MG) and MRI examination. The most typical signs were selected for each group, and method and were statistically evaluated. The effectivity of colchicine, vitamin E and ribwort plantain tincture in treatment was assessed by clinical examination and imaging. Typical clinical signs of IGM included unilateral acute onset of breast edema, redness, palpable masses, missing fever, lymphadenopathy, no response to antibiotics or surgical interventions. Ultrasound revealed: "finger-like" structures (100%), ductectasias (76.9%), abscesses (76.9%), and lymphadenopathy (15.4%), while in MRI skin and tissue edema (100%), multicentric lesions (100%), abscesses (76.9%), ring enhancement (84.6%), lymphadenopathy (15.4%) and small enhancing lymph nodes (38.5%) were observed. Among the clinical signs, fistulas, hypoechoic mass, ductectasias and diffusion weighted images (DWI) restriction were significantly more frequent in patients with IGM than in those with NM. Treatment effectivity yielded 100% with a complete response between 6-19 months, depending on the disease extent. Targeted questions together with imaging can speed up selection for proper treatment with colchicine, vitamin E and local treatment. Long lasting use of antibiotics and repeated surgical interventions should be avoided.

Cystic neutrophilic granulomatous mastitis associated with Corynebacterium including Corynebacterium kroppenstedtii

Granulomatous (lobular) mastitis is a rare inflammatory breast disease affecting parous reproductive-aged women. Once considered idiopathic, there is growing evidence of an association with corynebacteria infection, especially in the setting of a distinct histological pattern termed cystic neutrophilic granulomatous mastitis (CNGM). We describe 15 cases with histological features either confirming (n=12) or suggesting (n=3) CNGM, and concurrent microbiological evidence of Corynebacterium species. The organism was detected by culture or 16S rRNA gene sequencing of specimens obtained at surgery or fine needle aspiration. In seven cases, Gram-positive organisms were seen within vacuolated spaces. Speciation was performed in nine cases, with Corynebacterium kroppenstedtii subsequently identified. These cases provide further evidence in support of this association and in doing sohighlight the importance of recognising these histological clues as well as the limitations of Gram stain and microbiological culture in detecting this previously under-recognised disease process.

Imaging Findings in Patients with Granulomatous Mastitis.

BackgroundGranulomatous mastitis (GM) is a rare inflammatory breast disease that may mimic the clinical characteristics and radiologic imaging findings of breast carcinoma. Considering the importance of making a correct diagnosis, careful radiologic evaluations and recognition of imaging features are necessary.ObjectivesThe aim of this study was to review the radiological findings and diagnostic value of the imaging in GM.Patients and MethodsThis retrospective study involved a total of 29 patients who were diagnosed with GM between 2009 and 2013 and who underwent mammography (MG) and/or ultrasound (US) examination in addition to magnetic resonance imaging (MRI) before diagnosis.ResultsAmong 14 patients over 35 years of age who underwent MG imaging, focal asymmetric, ill-defined nodular, or diffusely increased densities were detected in nine (64.3%), two (14.3%), and one (7.1%) subjects, respectively, while there were no pathological findings in two (14.3%) patients. In the overall group of 29 patients, US showed heterogeneous hypoechoic lesions with tubular extensions in 16 (55.2%), well-demarcated heterogeneous hypoechoic lesions in eight (27.6%), parenchymal heterogeneous appearance in three (10.3%), and a heterogeneous hypoechoic lesion with irregular margins in one (3.4%), with another (3.4%) patient having normal US findings. MRI findings included lesions consistent with solitary or multiple separate or confluent abscesses with marked peripheral ring enhancement in 25 (86.2%) patients, accompanied by intensity changes suggesting edematous inflammation in the peripheral parenchyma, as well as non-mass-like heterogeneous segmental and regional contrast enhancement. Four (13.8%) patients had non-mass-like segmental and regional contrast enhancement only. A histopathological diagnosis of GM was established in all patients with biopsy.ConclusionGM presents with a wide range of conventional radiological findings, hampering the diagnosis. In patients with inconclusive conventional findings, MRI may assist in the differential diagnosis and assessment of the extent of disease. However, a definitive diagnosis and relevant treatment require histopathological confirmation.

Idiopathic granulomatous mastitis associated with risperidone-induced hyperprolactinemia

Idiopathic granulomatous mastitis (IGM) is a rare inflammatory breast disease. The etiology and treatment options of IGM remain controversial. Previous case reports have suggested that hyperprolactinemia may be associated with IGM. In the present report, we describe the first case of IGM associated with risperidone-induced hyperprolactinemia.Virtual slidesThe virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/8120093785928228

Idiopathic Granulomatous Mastitis: Surgery, Treatment, and Reconstruction

Idiopathic granulomatous mastitis is a rare inflammatory breast disease that mimics breast diseases such as inflammatory breast carcinoma, infective mastitis, and inflammatory breast disease of known cause. It is a diagnosis made only after excluding other causes, and although the disease is nonmalignant it may be both locally aggressive and recurrent. Definitive treatment may require radical excision and adjunctive treatment with immunosuppressants. Reconstruction following excision of disease has not been previously described. In those patients who undergo reconstruction, both the surgeon and the patient must be aware of complications associated with residual disease, and the potential involvement of donor sites. Treatment should be undertaken as part of a multidisciplinary team including surgeons and physicians with an interest in inflammatory breast disease. We present two patients diagnosed with idiopathic granulomatous mastitis who were referred to our unit for consideration of reconstruction. One patient underwent autologous breast reconstruction and the other contra lateral surgery to achieve symmetry.

Diagnostic Dilemma Of A Rapidly Progressive Ulcer Of The Breast: A Case Report

Idiopathic granulomatous mastitis is a rare inflammatory breast disease of unknown etiology. It tends to occur in young females usually with a history of recent childbirth or oral contraceptive usage. The clinical presentation is varied and may mimic malignancy. It may be identified by presence of epithelioid histiocytes, lymphocytes, plasma cells, neutrophils and multinucleated Langhans-type giant cells usually without any caseation or necrosis.

Feasibility of Surgical Management in Patients with Granulomatous Mastitis

Granulomatous mastitis (GM) is a rare inflammatory breast disease of unknown etiology. Although it usually presents with sinus formation and abscesses, it may mimic the clinical characteristics of breast cancer. The aim of this study was to identify the clinical, radiologic, and pathologic characteristics of patients with GM and to show the results of surgical treatment in these patients. A chart review was performed for patients that were treated with a diagnosis of GM at the Breast Unit, Department of Surgery, Istanbul Medical Faculty, University of Istanbul, between September 1998 and January 2003. Eighteen patients were eligible for this study. The median age was 41.5 years (range 16-80 years). Seventeen patients were evaluated by both ultrasonography and mammography; whereas one young patient only had ultrasonography. Three patients were further examined with color Doppler ultrasonography and magnetic resonance imaging (MRI). Fourteen patients (78%) presented with a mass as the chief symptom, with a median size of 3.9 cm (range 1-8 cm), whereas four patients presented with fistula in their breasts. None of the radiologic techniques distinguished benign disease from cancer in any of the 14 patients that presented with a mass except one patient with normal mammography findings. Ultrasonography was only helpful to localize the abscess associated with a fistula tract in one patient. Therefore fine-needle aspiration biopsy (FNAB) was performed in six patients, followed by surgical excisional biopsy. The remaining eight patients with a clinical suspicion of malignancy underwent wide surgical excision with frozen section analysis under general anesthesia. All of the FNAB and frozen section evaluations revealed benign findings. All of the 18 patients underwent a wide excisional biopsy and had a definitive histopathologic diagnosis of GM. The median follow-up was 36 months (range 6-60 months). Only one patient had a recurrent disease, which was diagnosed at 12 months. GM is a rare breast disease that mimics cancer in terms of clinical findings. Preoperative radiologic diagnosis might be difficult. Complete surgical excision is the treatment of choice.

Characteristic mammographic and sonographic findings in the diagnosis of granulomatous mastitis

Objective: Granulomatous mastitis (GM) is a rare inflammatory breast disease of unknown etiology that can clinically mimic infectious mastitis or carcinoma. We report the mammographic (MM) and sonographic (US) findings of GM that can aid in the differential diagnosis.

Idiopathic Granulomatous Mastitis: Case Report and Review of the Literature

We report a case of idiopathic granulomatous mastitis in a 35-year-old Japanese woman, who came to our hospital complaining of a tender mass in her right breast. Because the results of initial aspiration cytology were considered highly suspicious for carcinoma, modified radical mastectomy was performed. However, the final histological diagnosis was granulomatous lobular mastitis with no evidence of malignancy. Idiopathic granulomatous mastitis is a rare inflammatory breast disease of unknown etiology. Since the clinical manifestations are similar to those of mammary carcinoma, this condition has been misdiagnosed as carcinoma and treated as such. A review of the literature revealed that idiopathic granulomatous mastitis has tended to occur in young patients with a history of childbirth or oral contraceptive usage. Clinical or imaging diagnosis has often been difficult. Complete resection or corticosteroid therapy can be recommended as the optimal treatment. Since 38% of patients experience recurrence, long-term follow-up is indicated.