Treatment and Management Experience of Idiopathic Granulomatous Mastitis in a Low-income Country / Experiência de tratamento e manejo da mastite granulomatosa idiopática em um país de baixa renda

Abstract Objective Reporting our experience of the management and treatment of Idiopathic granulomatous mastitis (IGM) in a low-income country by describing patients characteristics and therapy with emphasis on conservative surgical excision and postoperative care as the cornerstone of treatment. Methods A retrospective cohort of women with histopathological diagnosis of IGM from 2014 to 2018 at Instituto Nacional Materno Perinatal in Lima, Peru. Patients' characteristics, clinical presentation, treatment, management, postoperative care, and follow-up were analyzed. Results Thirty-eight patients with histopathological diagnosis of IGM were identified. Their average age was 35.9 years and 23 (60.5%) reported previous use of hormonal contraceptives. Nine (23.7%) patients had chronic mastitis with previous treatment. The time from the onset of symptoms to the first clinic consult was 5.1 months on average. Twenty-one (55.3%) patients had the lesion in the right breast, with a mean size of 6.9 cm. Conservative surgical excision was performed in all patients. Additionally, 86.8% required corticosteroids and 78.9% were treated with antibiotics. Complete remission was obtained at 141 days on average (range 44 to 292 days). Six (15.8%) women reported ipsilateral recurrence and 5 (13.2%), contralateral. The latency time was 25.5 months on average. Conclusion The conservative surgical treatment demonstrated and close follow-up made for a high cure rate, but with recurrence similar to that reported in the literature. Use of gloves is an alternative to manage post operative wounds in a low-income country. The most frequent adverse effect was breast surgical scar.

Clinicopathological features of cystic neutrophilic granulomatous mastitis / 中华普通外科杂志

Objective:To investigate the etiology, clinical manifestations, clinicopathological features of cystic neutrophil granulomatous mastitis (CNGM).Methods:From Jan 2019 to Dec 2020, 95 CNGM cases diagnosed by biopsy pathology at Chongqing Hospital of Traditional Chinese Medicine and Chongqing Liangping District Hospitol of Traditional Chinese Medicine were reviewed.Results:There were 95 female patients, aged 21 to 50 years, with a median age of 32 years. Laboratory examination showed that 56% (53/95) cases had elevated rheumatoid antibody level, 27 % (26/95) had increased level of serum thyroid antibody, 15% (14/95) had elevated antineutrophil antibody, 35% (33/95) had increased ESR, 38% (36/95) had increased C-reactive protein. The positive rate of Gram-stained bacilli was 82% (78/95). Histology: pyogenic granuloma with lobule of breast as the center, the center of granuloma was cystic vacuole.Immunohistochemistry showed that the inflammatory cells in and around granuloma were mainly CD3 + cells, and CD4 + cells were more than CD8 + cells. Conclusions:The cystic neutrophilie granulo matous mastitis is a rare type of idiopathic granulomatous mastitis. The diagnosis of CNGM is dependent on its specific pathological features.

Efficacy of local injection of triamcinolone acetonide combined with surgery in the treatment of mass-type granulomatous mastitis / 西安交通大学学报(医学版)

【Objective】 To analyze the efficacy and safety of intralesional triamcinolone acetonide in the treatment of mass granulomatous mastitis (GLM). 【Methods】 Retrospective analysis was made on 67 patients with GLM who were treated in Xijing Hospital from July 2021 to May 2022 and met the inclusion criteria. Among them, 31 patients were treated with local injection of triamcinolone acetonide, while 36 ones were treated with oral methylprednisolone. All the included patients underwent surgical treatment after their condition met the surgical criteria, and the follow-up period lasted for up to six months after surgery. The two groups were compared in improvement of symptoms and signs, treatment time, clinical effectiveness and safety, and recurrence rate. 【Results】 The effective rate of triamcinolone acetonide group and methylprednisolone group was 100%, but the clinical cure rate was significantly higher in triamcinolone acetonide group than in methylprednisolone group in stratified analysis (P<0.05). The improvement time of symptoms and signs in triamcinolone acetonide group was significantly shorter than that in methylprednisolone group (P<0.05). The incidence of side effects in triamcinolone acetonide group was also significantly reduced compared to methylprednisolone group (P<0.05). Follow-up for half a year showed no recurrence in both groups. 【Conclusion】 Compared with the traditional oral methylprednisolone group, local injection of triamcinolone acetonide in the treatment of mass granulomatous mastitis can rapidly relieve clinical symptoms and signs, shorten treatment time, and has higher efficiency and fewer side effects. Local injection of hormone combined with surgery is effective in treating mass granulomatous mastitis with low recurrence rate.

Presentación clínica y hallazgos radiográficos de pacientes con diagnóstico histopatológico de mastitis granulomatosa idiopática / Clinical presentation and radiological findings of patients with histopathological diagnosis of idiopathic granulomatous mastitis

INTRODUCCIÓN. La mastitis granulomatosa idiopática es una patología inflamatoria benigna de mama con clínica y hallazgos imagenológicos no específicos; usualmente confundida con cáncer de mama. El síntoma más frecuente es una masa mamaria palpable. El diagnóstico es histopatológico. OBJETIVO. Describir el perfil demográfico, presentación clínica y hallazgos radiográficos de pacientes con diagnóstico histopatológico de mastitis granulomatosa idiopática. MATERIALES Y MÉTODOS. Estudio observacional, descriptivo, retrospectivo. Población de 1130 y muestra de 49 datos de historias clínicas electrónicas de pacientes con diagnóstico histológico de mastitis granulomatosa idiopática con el código CIE10 N61x Trastornos Inflamatorios de la mama, atendidas en la Unidad Técnica de Imagenología del Hospital de Especialidades Carlos Andrade Marín en la ciudad de Quito entre enero 2019 hasta diciembre 2021. El criterio de inclusión fue la confirmación histopatológica de mastitis granulomatosa idiopática. Los criterios de exclusión: antecedentes de neoplasia maligna de mama, antecedentes de HIV, patología inflamatoria sistémica como granulomatosis de Wegener, sarcoidosis, infecciones granulomatosas crónicas como tuberculosis, brucelosis, histoplasmosis, sífilis y reacciones a cuerpos extraños como material de implantes mamarios. Se analizaron datos demográficos, presentación clínica, hallazgos mamográficos, ecográficos y la categorización BIRADS. Se efectuó un análisis univarial; para las variables cualitativas se realizó frecuencias y porcentajes; para las variables cuantitativas se realizó medidas de tendencia central. La información recolectada fue analizada en el programa estadístico International Business Machines Statistical Package for the Social Sciences. RESULTADOS La mediana de la edad fue 36 años. El 94,00% de pacientes tenían por lo menos un hijo; 77,50% presentaron con una masa palpable; 55,10% se acompañaron de signos inflamatorios; 16,00% asociaron fístulas y 24,40% presentaron secreción. Solo 1 caso presentó afectación bilateral. CONCLUSIÓN En este estudio, la mastitis granulomatosa idiopática afecta a mujeres en edad reproductiva sin antecedentes de malignidad quienes presentan una masa mamaria palpable que puede estar acompañada de signos inflamatorios, colecciones y fístulas. La realización de una biopsia core eco guiada, para confirmar su diagnóstico.

INTRODUCTION. Idiopathic granulomatous mastitis is a benign inflammatory breast pathology with nonspecific clinical and imaging findings; usually mistaken for breast cancer. The most frequent symptom is a palpable breast mass. The diagnosis is histopathologic. OBJECTIVE. To describe the demographic profile, clinical presentation and radiographic findings of patients with histopathologic diagnosis of idiopathic granulomatous mastitis. MATERIALS AND METHODS. Observational, descriptive, retrospective study. Population of 1130 and sample of 49 data from electronic medical records of patients with histological diagnosis of idiopathic granulomatous mastitis with ICD10 code N61x Inflammatory disorders of the breast, attended at the Technical Imaging Unit of the Carlos Andrade Marín Specialties Hospital in the city of Quito between January 2019 and December 2021. The inclusion criterion was histopathological confirmation of idiopathic granulomatous mastitis. Exclusion criteria: history of malignant breast neoplasia, history of HIV, systemic inflammatory pathology such as Wegener's granulomatosis, sarcoidosis, chronic granulomatous infections such as tuberculosis, brucellosis, histoplasmosis, syphilis and reactions to foreign bodies such as breast implant material. Demographic data, clinical presentation, mammographic and ultrasound findings and BIRADS categorization were analyzed. Univariate analysis was performed; frequencies and percentages were used for qualitative variables; measures of central tendency were used for quantitative variables. RESULTS. The median age was 36 years. 94,00% of patients had at least one child; 77,50% presented with a palpable mass; 55,10% were accompanied by inflammatory signs; 16,00% were associated with fistulas and 24,40% presented with discharge. Only 1 case presented bilateral involvement. CONCLUSION. In this study, idiopathic granulomatous mastitis affects women of reproductive age with no history of malignancy who present with a palpable breast mass that may be accompanied by inflammatory signs, collections and fistulas. The performance of an echo-guided core biopsy to confirm the diagnosis.

Application of oncoplastic round block technique in surgical management of idiopathic granulomatous mastitis / 中华医学美学美容杂志

Objective:To explore the efficacy of the oncoplastic round block technique in surgical management of idiopathic granulomatous mastitis (IGM).Methods:From January 2014 to December 2019, a total of 18 patients (24 to 38 years old, 32.2 years in average) with IGM underwent excision of the inflammatory breast mass with oncoplastic round block technique, the postoperative clinical efficacy was summarized and analyzed.Results:All 18 patients with IGM underwent excision of the inflammatory breast mass with oncoplastic round block technique, among them 2 patients underwent round-block reduction surgery of contralateral breast at the same time. The median follow-up duration was 16.1 months (from 6 to 36 months). Incision poor healing occurred in two cases which was cured after dressing change. Recurrence occurred in one case at 6 months after operation, and then cured with conservative measures. No other severe complications occurred. All patients were satisfacted with the results.Conclusions:Application of oncoplastic round block technique in surgical management of IGM may remove more tissue in order to reduce the recurrent rate, and get a better cosmetic results.

Mastitis granulomatosa: una simuladora de cáncer, un gran reto diagnóstico y terapéutico / Granulomatous mastitis: a cancer simulator, a great diagnostic and therapeutic challenge

Resumen ANTECEDENTES: La mastitis granulomatosa idiopática es un padecimiento benigno de la glándula mamaria sumamente raro, simulador de cáncer de mama. Las manifestaciones clínicas más significativas son: nódulo palpable, cambios en la coloración de la piel y mastalgia. Para integrar su diagnóstico se requiere el apoyo de estudios auxiliares de imagen, sin que por ello deje de ser imprescindible la toma de biopsia para establecer el diagnóstico definitivo. No existen pautas de tratamiento debidamente establecidas, pero sí de tratamiento farmacológico y quirúrgico, ésta última se reserva para casos de recidivas, que son frecuentes. CASO CLÍNICO: Paciente de 35 años, nuligesta; acudió a consulta debido a la aparición de un tumor palpable en la glándula mamaria derecha, de siete meses de evolución, con cambios en la coloración y retracción del pezón. La mastografía y ecografía catalogaron el tumor en BI-RADS 5. La biopsia con aguja de corte y citología integraron el diagnóstico de mastitis granulomatosa idiopática. Se indicó tratamiento farmacológico y el tumor desapareció espontáneamente. CONCLUSIONES: La mastitis granulomatosa es una enfermedad rara y de diagnóstico complejo. Para cada paciente habrá un protocolo diferente para evitar el sobretratamiento. Su pronóstico es bueno, a pesar de ser un simulador de cáncer de mama. El tratamiento aún es ambiguo, pero en primera instancia se recomiendan los corticosteroides o inmunosupresores y la cirugía se reserva para casos de recidivas.

Abstract BACKGROUND: Idiopathic granulomatous mastitis is an extremely rare benign entity of the mammary gland, simulating breast cancer. The most significant clinical manifestations are: palpable nodule, skin discoloration changes and mastalgia. The diagnosis requires the support of auxiliary imaging studies, although a biopsy is essential to establish the definitive diagnosis. There are no well-established treatment guidelines, but there are guidelines for pharmacological and surgical treatment, the latter is reserved for cases of recurrence, which are frequent. CLINICAL CASE: A 35-year-old nulligesta patient came for consultation due to the appearance of a palpable tumor in the right mammary gland, of 7 months of evolution, with changes in color and nipple retraction. The mastography and ultrasound catalogued the tumor in BI-RADS 5. The biopsy with cutting needle and cytology integrated the diagnosis of idiopathic granulomatous mastitis. Pharmacological treatment was indicated, and the tumor disappeared spontaneously. CONCLUSIONS: Granulomatous mastitis is a rare disease with a complex diagnosis. For each patient there will be a different protocol to avoid overtreatment. Its prognosis is good, despite being a breast cancer simulator. Treatment is still ambiguous but, in the first instance, corticosteroids or immunosuppressants are recommended and surgical treatment is reserved for cases of recurrence.

Mastitis granulomatosa idiopática. Características clínicas, imagenológicas, patológicas y su correlación con la respuesta al tratamiento - Hospital Dr. Cosme Argerich / Idiopathic granulomatous mastitis: clinical, imagenological, pathological characteristics and their correlation with treatment response

Introducción: La mastitis granulomatosa idiopática (MGI) es una patología inflamatoria benigna que se presenta típicamente como tumoración dolorosa. Recientemente, un subconjunto con patrón histológico neutrofilico quístico (MGNQ) fue asociado al corynebacterium. Objetivo: Revisión bibliográfica y evaluación de nuestra casuística. Material y método: Se seleccionaron 24 mujeres con MGI diagnosticada entre 2000-2019. Variables analizadas: demográficas, antecedentes, clínica, imágenes, cultivos, patología, tratamientos y evolución. Resultados: Edad media: 40.7 años. 50% tuvo ≥4 gestas y 91.6% lactó. Presentación clínica: Tumor abscedado palpable 58.33%, Palpable asintomático 25%, Palpable doloroso 12.5%. Lesión no palpable 4.17%. Tamaño promedio de lesión: 3.5 cm. Imágenes BIRADS 4/5: 87.5% Cultivo de corynebacterium 25%. Patología: Polimorfonucleares intraductales (PMNID) 62.5%, Vacuolas quísticas 50% (2 casos con baci- los). Tratamiento: Antibióticoterapia 79.1%, Inmunosupresión 79.1%, Drenaje de abscesos 45.8%. Tumorectomía 41.6%. Tiempo de resolución: 5.5 meses Tiempo libre de enfermedad: 33.6 meses. Recurrencias: 31.%. Conclusiones: Debe sospecharse en toda mujer joven con tumor mamario asociado a abscesos, fistulas y/o úlceras. El diagnóstico de MGNQ y PM- NID tienen asociación estadísticamente significativa con recurrencia. Es esencial el abordaje multidisciplinario y el tratamiento multimodal

Introduction: Idiopathic granulomatous mastitis (IGM) is a benign inflammatory disease that typically presents as a painful mass. Recently, a subset with cystic neutrophilic histological pattern (CNGM) was related to corynebacterium. Objetive: Bibliographic review and evaluation of our casuistry. Material and method: During 2000-2019, 24 women diagnosed with IGM were selected. Variables analyzed: Demographic, background, clinical, images, cultures, pathology, treatment and evolution. Results: Average age: 40.7 years. 50% had ≥ 4 gestations and 91.6% lactated. Clinical presentation: Palpable abscessed tumor 58.33%, Palpable asymptomatic 25%, Palpable painful 12.5%. Non palpable lesion 4.17%. Average lesion size: 3.5 cm. BIRADS images 4/5: 87.5% Corynebacterium culture 25%. Findings: Intraductal polymorphonuclear cells (IDPMNC) (62.5% ), Cystic vacuoles (50%) (With bacilli in 2 cases). Treatment: Antibiotic therapy 79.1%, Immunosuppression 79.1%, Drainage of abscesses 45.8%. Lumpectomy 41.6%. Resolution time: 5.5 months Diseasefree time: 33.6 months. Recurrences: 31.8%. Conclusions: It should be suspected in any young woman with a breast tumor associated with abscesses, fistulas and/or ulcers. The diagnosis of CNGM and IDPMNC has statistically significant association with recurrence. A multidisciplinary approach and multimodal treatment is essential

Mastitis granulomatosa idiopática: Caso clínico y revisión literaria / Idiopathic granulomatose mastitis: Case report and literary review

Se describe el caso clínico de una paciente con diagnóstico histológico de mastitis granulomatosa idiopática, enfermedad poco frecuente en la que se utilizan inmunosupresores como tratamiento descripto y puede confundirse con otras entidades atendidas en la especialidad. Se describe el caso y se realiza una revisión de la literatura.

The clinical case of a patient with a histological diagnosis of idiopathic granulomatous mastitis is described, a rare disease in which immunosuppressants are used as the described treatment and can be confused with other entities treated in the specialty. The case is described and a literature review is carried out.

Corynebacterium kroppenstedtii y patología mamaria / Corynebacterium kroppenstedtii and breast pathology

Resumen Corynebacterium kroppenstedtii es un bacilo gram positivo corineforme lipofílico, poco frecuente en la clínica humana. Forma parte de la microbiota cutánea de los seres humanos y, por esta razón, su interpretación clínica es compleja. La mastitis granulomatosa es una enfermedad inflamatoria de origen incierto con baja incidencia. Se presentan dos casos clínicos en los que se describe la asociación de C. kroppenstedtii con mastitis granulomatosa. El tejido mamario es rico en lípidos. El carácter lipofílico de este microorganismo podría explicar su presencia en dicho tejido.

Abstract Corynebacterium kroppenstedtii is a rare lipophilic coryneform gram-positive bacillus. It is part of the human skin microbiota and, for this reason, its clinical interpretation is complex. Granulomatous mastitis is an inflammatory disease of uncertain origin with a low incidence. The association of C. kroppenstedtii with granulomatous mastitis was described in two clinical case reports. The lipophilic characteristics of this microorganism explains why it can be found in lipid-rich breast tissue.

Resumo Corynebacterium kroppenstedtii é um bacilo gram-positivo corineforme lipofílico poco frecuente. Faz parte da microbiota do seres humanos, por isso sua interpretação clínica é complexa. A mastite granulomatosa é uma doença inflamatória de origem incerta com baixa incidência. Foram apresentados dois casos clínicos nos quais é descrita a associação de C. kroppenstedtii com mastite granulomatosa. O tecido mamário é rico em lipídios. O caráter lipofílico desse microrganismo pode explicar sua presença em tal tecido.

Mastitis granulomatosa: caso clínico y revisión de la literatura / Granulomatous mastitis: case report and literature review

OBJETIVO: La mastitis granulomatosa es una patología mamaria benigna y crónica de baja incidencia, cuyo diagnóstico es complejo y su tratamiento no está del todo establecido. Se presenta un caso clínico con el objetivo de hacer una revisión de la bibliografía sobre esta patología, su diagnóstico y tratamiento. MATERIAL Y MÉTODOS: Se presenta el caso de una paciente de 30 años con una mastitis refractaria a tratamiento antibiótico a la que finalmente se diagnosticó una mastitis granulomatosa idiopática gracias a la biopsia excisional. El tratamiento con corticoides a dosis altas tuvo buenos resultados en este caso. Se realizó una búsqueda bibliográfica en las bases de datos Medline vía PubMed, EMBASE y SciELO y también en libros de texto en papel. RESULTADOS: Se hallaron 598 referencias, la mayoría de ellas revisiones sistemáticas y casos clínicos. CONCLUSIONES: La mastitis granulomatosa es una patología poco frecuente cuyo diagnóstico se logra mediante estudio histológico de la misma. No hay consenso sobre cuál es el tratamiento óptimo, pero la tendencia actual es el tratamiento conservador con corticoides a altas dosis. Cada vez es más frecuente el uso de metotrexato cuando no hay buena respuesta con el tratamiento con corticoides.

OBJECTIVE: Granulomatous mastitis is a benign and chronic breast pathology with a low incidence, whose diagnosis is complex and its treatment is not fully established. A case report is presented with the aim of reviewing the literature on this pathology, its diagnosis and its treatment. MATERIAL AND METHODS: The patient is a 30-year-old woman with a mastitis refractory to antibiotic treatment. Finally, idiopathic granulomatous mastitis was diagnosed through excisional biopsy. Treatment with high-dose corticosteroids had good results in this patient. A literature search was performed in the Medline databases via PubMed, EMBASE and SciELO and also in paper textbooks. RESULTS: 598 references were found, most of them systematic reviews and case reports. CONCLUSIONS: Granulomatous mastitis is an uncommon pathology whose definitive diagnosis is achieved by histological study. Treatment is not fully established, but conservative treatment with high-dose corticosteroids is the current trend. The use of methotrexate has risen when there is no good evolution with corticosteroid treatment.

Mastitis quística granulomatosa neutrofílica asociada a corinebacterias: Reporte de caso

La mastitis granulomatosa neutrofílica quística (MGNQ) es una entidad recientemente caracterizada, con detalles histopatológicos específicos que la diferencian de otros tipos de mastitis crónicas idiopáticas. La presencia de bacilos grampositivos dentro de espacios de apariencia quística rodeados de neutrófilos, en un contexto de inflamación granulomatosa supurativa, definen esta entidad. La importancia de su reconocimiento en el reporte diagnóstico recae en su asociación a la infección por especies de corinebacterias, de esa manera se puede direccionar el tratamiento con antibióticos, más allá del tratamiento antiinflamatorio que suele administrarse en mastitis idiopáticas. Se describe el caso de una paciente de sexo femenino, de 35 años de edad, sin antecedentes relacionados, con dolor mamario intermitente, asociado a la presencia de múltiples nodulaciones palpables, duras, en ambas mamas, con resultados de múltiples biopsias particulares previas que describieron mastitis crónica granulomatosa con reacción de tipo tuberculoso, por lo cual recibió tratamiento antituberculoso. Pese a ello, las tumoraciones cedieron sólo parcialmente. Se revisó las láminas histológicas en la institución y se estableció el diagnóstico de MGNQ. Se consideró importante la presentación de este caso debido a su escaso reconocimiento entre patólogos, pese a presentar características ya definidas en estudios previos.

Cystic neutrophilic granulomatous mastitis (CNGM) is a recently characterized entity, with specific histopathological details that differentiate it from other types of chronic idiopathic mastitis. The presence of gram-positive bacilli within cystic-like spaces surrounded by neutrophils, in a context of suppurative granulomatous inflammation, define this entity. The importance of its recognition in the diagnostic report lies in its association with infection by corynebacterial species, so that treatment with antibiotics can be targeted, beyond the anti-inflammatory treatment that is usually administered to idiopathic mastitis. We describe the case of a 35-year-old female patient with no related history, with intermittent breast pain, associated with the presence of multiple hard, palpable nodules in both breasts, with results from multiple previous private biopsies that described chronic granulomatous mastitis with a tubercular reaction, for which she received treatment for tuberculosis. Despite this, the clinical response was partial. Histological slides were reviewed at the institution and the diagnosis of CNGM was then established. The presentation of this case was considered important due to its low recognition among pathologists, despite presenting characteristics already defined by previous studies.

Mastitis Quística Granulomatosa Neutrofílica, Reporte De Caso / Granulomatous Neutrophilic Cystic Mastitis, Case Report

La mastitis granulomatosa neutrofílica quística (MGNQ) es una entidad recientemente caracterizada, con detalles histopatológicos específicos que la diferencian de otros tipos de mastitis crónicas idiopáticas. La presencia de bacilos grampositivos dentro de espacios de apariencia quística rodeados de neutrófilos, en un contexto de inflamación granulomatosa supurativa, definen esta entidad. La importancia de su reconocimiento en el reporte diagnóstico recae en su asociación a la infección por especies de corinebacterias, de esa manera se puede direccionar el tratamiento con antibióticos, más allá del tratamiento antiinflamatorio que suele administrarse en mastitis idiopáticas. Se describe el caso de una paciente de sexo femenino, de 35 años de edad, sin antecedentes relacionados, con dolor mamario intermitente, asociado a la presencia de múltiples nodulaciones palpables, duras, en ambas mamas, con resultados de múltiples biopsias particulares previas que describieron mastitis crónica granulomatosa con reacción de tipo tuberculoso, por lo cual recibió tratamiento antituberculoso. Pese a ello, las tumoraciones cedieron sólo parcialmente. Se revisó las láminas histológicas en la institución y se estableció el diagnóstico de MGNQ. Se consideró importante la presentación de este caso debido a su escaso reconocimiento entre patólogos, pese a presentar características ya definidas en estudios previos.

Cystic neutrophilic granulomatous mastitis (CNGM) is a recently characterized entity, with specific histopathological details that differentiate it from other types of chronic idiopathic mastitis. The presence of gram-positive bacilli within cystic-like spaces surrounded by neutrophils, in a context of suppurative granulomatous inflammation, define this entity. The importance of its recognition in the diagnostic report lies in its association with infection by corynebacterial species, so that treatment with antibiotics can be targeted, beyond the anti-inflammatory treatment that is usually administered to idiopathic mastitis. We describe the case of a 35-year-old female patient with no related history, with intermittent breast pain, associated with the presence of multiple hard, palpable nodules in both breasts, with results from multiple previous private biopsies that described chronic granulomatous mastitis with a tubercular reaction, for which she received treatment for tuberculosis. Despite this, the clinical response was partial. Histological slides were reviewed at the institution and the diagnosis of CNGM was then established. The presentation of this case was considered important due to its low recognition among pathologists, despite presenting characteristics already defined by previous studies.

Mastitis granulomatosa / Granulomatous mastitis / Mastite granulomatosa

La mastitis granulomatosa es una enfermedad rara, de etiología desconocida y de diagnóstico de exclusión luego de descartar otras causas de mastitis, como las asociadas a infecciones bacterianas y a gérmenes atípicos como hongos y tuberculosis. La incidencia ha sido difícil de determinar, pero varia del 0.3% al 1.8%. Más frecuente en mujeres en edad reproductiva, la presentación clínica más común de este tipo de mastitis es una masa mamaria de consistencia dura, unilateral, asociada a dolor, eritema, calor e hinchazón, sin fiebre. Tiende a formar abscesos únicos o múltiples con fistulización, lo que hace que se confunda con carcinoma inflamatorio o localmente avanzado. Además, tiende a ser recurrente. El diagnóstico solo puede confirmarse por histopatología, en la que se evidencian granulomas no caseificantes concentrados en lobulillos mamarios, con infiltrado linfocitario crónico, necrosis y, con frecuencia, ectasia ductal. El tratamiento es controvertido, con opciones que van desde la observación, antibióticoterapia, terapia con corticosteroides, medicamentos inmunosupresores como el metotrexato, a la cirugía con escisión local amplia.

Granulomatous mastitis is a rare disease of unknown etiology, and the diagnosis is of exclusion after ruling out other causes of mastitis such as those associated with bacterial infectious diseases and by atypical germs such as fungi and tuberculosis. the incidence has been difficult to determine but varies from 0.3 to 1.8%. More common in women of reproductive age, the most common clinical presentation is a breast mass of hard, unilateral consistency, associated with pain, erythema, heat, swelling, and without fever. It tends to form single or multiple abscesses with fistulization, which makes it possible to be confused with an inflammatory or locally advanced carcinoma. Also with tendency to recurrence. The diagnosis can only be confirmed by histopathology, it is characterized by non-caseifying granulomas concentrated in breast lobules with chronic lymphocyte infiltrate, necrosis and often ductal ectasia. The treatment is controversial, with options ranging from observation, antibiotic therapy, corticosteroid therapy, immunosuppressive medications such as methotrexate, to surgery such as wide local excision.

A mastite granulomatosa é uma doença rara, de etiologia desconhecida e de diagnóstico de exclusão logo de descartar outras causas de mastite, como as associadas a infecções bacterianas e a germens atípicos como fungos e tuberculose. A incidência há sido difícil de determinar, mas varia de 0.3% a 1.8%. Mais frequente em mulheres em idade reprodutiva, a apresentação clínica mais comum deste tipo de mastite é uma massa mamária de consistência dura, unilateral, associada a dor, eritema, calor e inchaço, sem febre. Tende a formar abscessos únicos ou múltiplos com fistulização, o que faz confundir com carcinoma inflamatório ou localmente avançado. Além disso, tende a ser recorrente. O diagnóstico só se pode confirmar por histopatologia, na qual se evidenciam granulomas não caseificantes concentrados em lóbulos mamários, com infiltrado linfocitário crônico, necrose e, com frequência, extasia ductal. O tratamento é controvertido, com opções que vão desde a observação, antibióticoterapia, terapia com corticosteroides, medicamentos imunossupressores como o metotrexato, à cirurgia com incisão local ampla.

Tuberculous mastitis: a rare case report

Tuberculous mastitis is a granulomatous mastitis due to infection by mycobacterium tuberculosis. It’s a rare entity witch is often confused with breast carcinoma or pyogenic breast abscess because of its clinical and radiological aspect. The diagnosis is histological with identification of an epithelioid cell granulomas and necrosis. The treatment is based on anti-tuberculous therapy with a good clinical issue. Authors report a case of tubercular mastitis in a post-menopausal female diagnosed on adenectomy.

Mastitis granulomatosa idiopática con respuesta al tratamiento inmunosupresor / Idiopathic Granulomatous Mastitis with Responses to Immunosuppressive Treatment: Two Case Report

La mastitis granulomatosa idiopática es una enfermedad inflamatoria crónica de causa desconocida y presentación infrecuente, que afecta a mujeres en edad reproductiva. Su presentación clínica similar al carcinoma de mama, sumada al desconocimiento de esta entidad, genera confusión, errores y retrasos en el diagnóstico en gran proporción de los casos, por lo que la histopatología es un requisito fundamental para el diagnóstico correcto de esta enfermedad. Presentamos dos casos de pacientes femeninas con cuadro clínico de mastitis crónica en quienes, tras descartar causas infecciosas y neoplásicas, se estableció el diagnóstico de mastitis granulomatosa idiopática. Se administró terapia inmunosupresora con prednisolona, metotrexate y ácido fólico. Las pacientes tuvieron una adecuada respuesta y se evidenció la resolución del cuadro clínico a las 3 semanas de tratamiento(AU)

Idiopathic granulomatous mastitis is a chronic inflammatory disease of unknown etiology and infrequent presentation. Confusion, delayed and mistaken in diagnosis has been atribuided to the similar clinical manifestation with breast carcinoma, added to the ignorance of this entity, that's why histopathology is a fundamental requirement for the correct diagnosis of this disease. We present two cases of female patients with clinical symptoms of chronic mastitis, in whom a diagnosis of idiopathic granulomatous mastitis was made after ruling out infectious and neoplastic causes(AU)

Management of idiopathic granulomatous mastitis: a surgical experience of ten cases

Granulomatous mastitis is a chronic inflammatory condition of the breast that is rarely described in literature. It mimics breast abscess on clinical presentation giving dilemma to the surgeon. The etiology can be infectious, autoimmune or idiopathic. The various modalities of treatment for idiopathic granulomatous mastitis includes drainage of abscess, antibiotics and corticosteroid therapy. This is a case series of ten cases of diagnosed with granulomatous mastitis treated in the period of 2 years from 2018 and 2019 in Department of General Surgery, MGM Hospital Navi Mumbai. The data of all the patients were collected from records. They were following at regular intervals in the outpatient department to assess recurrence. The mean age of the patients was 41 years (range 26–47 years). Six patients gave history of breast-feeding in the last 5 years, one was lactating at the time of presentation. One of the patients was treated empirically for Tuberculous mastitis for the opposite breast. Others had no history suggestive of tuberculosis or other connective tissue diseases, such as rheumatoid arthritis or sarcoidosis USG breast was performed in all cases. Hypoechoic abscess was seen in 5 cases. The time taken for complete resolution of the disease was ranging from 2 weeks to 2 months. All patients had an uneventful recovery and no recurrences reported till now. GM is a rare benign breast disease that is difficult to distinguish from other inflammatory breast diseases or cancer. The diagnosis of GM must be based on a multidisciplinary approach. Surgical management followed by antibiotics was found to be sufficient to treat the condition in our patients. Corticosteroids need not be administered in all cases of this benign breast disease.

Bilateral idiopathic granulomatous mastitis a rare benign inflammatory disease of breast: case report

Idiopathic Granulomatous Mastitis (IGM) is an uncommon benign inflammatory disorder of breast. Peri-ductal mastitis is thought to be the initial inflicting factor for its development. IGM usually presents as painful swelling, abscess or sinus/fistula in breast. There are no specific findings on ultrasound or mammographic imaging, and moreover it looks BIRADS-IV on mammography. Histopathology provides the final diagnosis. Here authors report a case of bilateral IGM which was successfully treated with surgery.

Mastitis granulomatosa idiopática: características demográficas, clínicas e imagenológicas en una serie de casos / Idiopathic granulomatous mastitis: demographic, clinical and imaging characteristics of a series of cases

Resumen: OBJETIVO: Realizar una caracterización demográfica, clínica e imagenológica de una serie de casos de mastitis granulomatosa idiopática. MATERIALES Y MÉTODOS: Estudio observacional, retrospectivo y transversal de una serie de casos de pacientes con diagnóstico de mastitis granulomatosa idiopática atendidas en el Hospital Universitario Dr. José Eleuterio González, Monterrey, NL, entre enero 2015 y diciembre 2019. Criterio de inclusión: pacientes mayores de 18 años con diagnóstico histológico de mastitis granulomatosa idiopática en ausencia de un agente etiológico específico. Criterios de exclusión: pacientes con cuadro de mastitis relacionado con una patología específica o con algún antecedente de traumatismo-laceración reciente en la mama. Variables de estudio: información demográfica (edad, escolaridad, estado civil, etc.), proceso-método diagnóstico, hallazgos por imagen y tratamientos indicados. RESULTADOS: Se incluyeron 20 pacientes con edad promedio de 32 años, 17 en edad fértil, y el resto en la posmenopausia. Las 20 pacientes tuvieron, al menos, 1 embarazo de término sumado a un periodo variable de lactancia. 19 tenían enfermedad unilateral, la mayoría en la mama izquierda (60%). El hallazgo principal al establecer el diagnóstico: en 20 una tumoración, 15 de 20 eritema y 11 mastalgia. El reporte radiológico mediante BIRADS fue de IVA en 12, IVB en 6 y 1 tanto para II y III. En todas las pacientes se comprobó el diagnóstico con estudio histopatológico. Doce recibieron tratamiento inicial con prednisona, 7 con antibióticos y AINES y un caso con escisión quirúrgica. CONCLUSIONES: La mastitis granulomatosa idiopática es una enfermedad rara de prevalencia aún desconocida, con un comportamiento benigno de tipo inflamatorio crónico, cuya patogenia permanece aún desconocida. Al ser una enfermedad con un diagnóstico por exclusión, obliga al estudio histopatológico para establecer el diagnóstico definitivo, idealmente mediante la toma de una biopsia con Tru-cut. Aunque se han planteado varias alternativas terapéuticas, en la actualidad no existe un protocolo universal que justifique un tratamiento en forma generalizada. La resección de la lesión con márgenes amplios asociada o no con corticoides orales, sigue siendo el tratamiento más indicado.

Abstract: OBJECTIVE: To perform a demographic, clinical and imaging characterization of a series of cases of idiopathic granulomatous mastitis. MATERIALS AND METHODS: Observational, retrospective and cross-sectional study of a series of cases of patients diagnosed with idiopathic granulomatous mastitis, at Hospital Universitario "Dr. José Eleuterio González in Monterrey, Mexico between January 2015 and December 2019. The study comprised all patients older than 18 years old who had histological diagnosis of idiopathic granulomatous mastitis in the absence of a specific agent. Patients with mastitis related to a specific pathology or with a history of recent breast trauma/laceration were excluded. Variables to determine are demographic information (age, education, marital status, etc.), diagnostic process/method, imaging findings and treatment. RESULTS: 20 patients with an average age of 32 years were included, 17 of childbearing age, and the rest at menopause. All patients had at least one full-term pregnancy and a variable lactation period. 19 presented the disease unilaterally, 60% in the left breast. Clinical presentation: 20 presented tumor, 15 erythema and 11 mastalgia. Imaging: BIRADS IVA in 12, IVB in 6 , III in 1 and II in 1. Diagnostic confirmation was performed in all patients by histopathological study. 12 received initial treatment with prednisone, 7 with antibiotics and NSAIDs, and one case with surgical excision. CONCLUSIONS: Idiopathic granulomatous mastitis is a rare benign and chronic inflammatory disease of unknown prevalence and pathogenesis. It mainly affects women of reproductive age. Imaging studies usually report nonspecific findings. It requires histopathological study to establish a definitive diagnosis, as it is a disease with diagnosis by exclusion, ideally by taking a Tru-cut biopsy. Although several therapeutic alternatives have been proposed, currently there is no universal protocol for treatment, however, resection of the lesion with wide margins associated or not with oral corticosteroids prevails as the most widely used treatment nowadays.

Idiopathic granulomatous mastitis: case series / Mastite granulomatosa idiopática: série de casos

Idiopathic granulomatous mastitis is a rare disease that mimics other pathological conditions, including breast adenocarcinoma, breast tuberculosis, and abscess. Three patients diagnosed with idiopathic granulomatous mastitis were analyzed, receiving corticosteroid treatment, antibiotic therapy in cases of abscesses, and, in one case, resection of the affected breast segment. All patients exhibited regression of symptoms.

A mastite granulomatosa idiopática é uma doença rara que mimetiza outras condições patológicas, incluindo adenocarcinoma de mama, tuberculose de mama e abscesso. Foram analisados três pacientes com diagnóstico de mastite granulomatosa idiopática, que receberam como tratamento corticosteroide, antibioticoterapia em casos de abscessos e, em um caso, ressecção do segmento mamário afetado. Todas as pacientes evoluíram com regressão dos sintomas.

Granulomatous lobular mastitis: a clinicopathological analysis of 300 cases / 中华病理学杂志

Objective@#To investigate the clinicopathologic features and possible causes of granulomatous lobular mastitis(GLM).@*Methods@#Three hundred cases of GLM were collected from surgical specimens diagnosed at Longhua Hospital, Shanghai University of Traditional Chinese Medicine from January 2015 to November 2017. Morphologic features were reviewed using HE staining. A total of 116 cases were investigated by Gram staining. The expression of CD3, CD20, CD68, IgG, IgG4, CD38 and CD138 was detected by immunohistochemical staining.@*Results@#The age of the patients was 23 to 47 years and the median age was 32 years. All patients were female, 96.7% (290/300) had a history of lactation.There were 143 cases of left breasts, 138 cases of right breast and 19 cases of bilateral breasts. Serum prolactin increased in 39.7%(119/300) patients. Within 15.7%(47/300) of patients were associated with nodular erythema or joint swelling and pain of the lower extremities. Pathological observation showed that lobular-centric suppurative granulomatous inflammation, accompanied by dilatation of intralobular and interlobular ducts. There were 16 cases accompanied with duct ectasia. Immunohistochemistry showed CD3-positive lymphocytes were more than CD20-positive lymphocytes in the peripheral aggregation zone of neutrophils within granulomatous lesions. Gram positive bacteria were found in the lipid vacuoles of the 51.7%(60/116) patients.@*Conclusions@#GLM has distinctive histologic features. It may be related to corynebacterium infection, or accompanied by the increase of serum prolactin and erythrocyte sedimentation rate. The age, location and history of the disease are importance in the diagnosis and differential diagnosis.