Follow-up of granulomatous mastitis with monitoring versus surgery

Introduction

IGM (Idiopathic Granulomatous Mastitis) is a rare, chronic, non-malignant and non-life-threatening breast disease [1]. Clinically and radiologically, it has similar characteristics with other granulomatous diseases and breast carcinomas. It is divided two groups; idiopathic and specific. Specific granulomatous mastitis can develop after tuberculosis, sarcoidosis, fungal infections (actinomycosis, histoplasmosis, blastomycosis etc.) and parasitic infections (filariasis, schistosomiasis; etc.). The etiology of IGM is unknown, but it is generally associated with the use of oral contraceptives, lactation, hyperprolactinemia, blunt trauma, drug use (metoclopramide, ranitidine). The most common complaints of patients are unilaterally breast pain or painless masses [2,3]. There is no certain treatment application in the literature [4,5]. The options of treatment depend on the severity of the disease during the treatment [6].

Material and Methods

The patients whom diagnosed IGM according to pathology report were retrospectively evaluated between November 2014 and January 2016. The demographic properties, complaints during admission, diagnosis and treatment methods and clinical results were obtained via investigation of the patient files. The patients were called for follow-up and discussion.

All patients were informed about IGM and diagnostic methods (needle biopsy, incisional biopsy and excisional biopsy) and treatment options as non-surgery (observation, anti-biotherapy, anti-inflammatory, steroid, methotrexate, colchicine and such immunosuppression treatments) and surgery (excision, mastectomy ) were explained in detail.

Results

The patients whom diagnosed IGM were retrospectively evaluated between November 2014 and January 2016, total number of patients were 19. The average following period was 11 months (4–13 months). Fourteen patients were in the reproductive period, five patients were in the postmenopausal period. The mean age of the patients was 44 (25–77) and all female patients had a history of pregnancy and breast feeding. None of the patients were in the lactation period. Contents and results presented in Table 1.

Five of nineteen patients whom diagnosed IGM had pain complaints, three patients had erythema, five patients had a palpable mass and five patients had ulcerated wounds (Fig. 1: The patient had ulcerated wound) and suppuration complaints. The patients with a palpable mass and complaints of swelling three patients had an abscess. Sixteen patients had IGM in a single breast, only three patients had IGM history in bilateral breast, one or three years ago. These three patients had twice or triple recurrence but this is not significant statistically. The anti-biotherapy with anti-inflammatory treatments were given 13 patients who had erythema, ulcerated wounds, suppuration and abscess complaints.

Ultrasound imaging was performed all patients, mammography was performed 6 patients over 40 years of age. The patients were not imaged by magnetic resonance imaging (MRI).

The diagnostic procedures decided according to clinical findings of the patients. Core biopsy was performed on all patients but only one patient of diagnosed bilateral IGM had been drainage and incisional biopsy due to a twice developing abscess clinic and recurrence. The mean size of the masses performed excisional biopsy was 3.2 cm.

The following staining methods were performed on the tissue samples and aspiration materials obtained from the patients; Hematoxylin + eosin (H & E) staining for histopathological investigation, alkaline-acid resistance factors for tuberculosis bacteria (AARB) and Lowenstein-Jensen culture, polymerase chain reaction on tissues (PCR) Ehrlich Ziehl-Neelsen (EZN) staining and Periodic acid-Schiff (PAS) staining for the investigation of fungal infections.

One patient of diagnosed bilateral IGM had been drainage and incisional biopsy due to a twice developing abscess clinic and recurrence (Fig. 2: Recurrent abscesses in the lower inner quadrant of the left breast). As a result of rheumatological evaluations, the patient was diagnosed with autoimmune connective tissue disease. Steroid treatment recommended to patient for immunosuppression but she refused. The patient had spontaneous remission during the 12 month monitoring period.

The breast ultrasound examination and mammography performed on a 77 year old patient who complaints of breast pain. Radiologic imaging showed a fibroglandular tissue density with no distinctive border, accompanied by edema and thickening of the skin. Core biopsy performed to this patient and 5 mm diameter invasive ductal carcinoma, carcinoma in situ and granulomatous mastitis was reported. Later, modified radical mastectomy performed because there were widespread high degree carcinoma in situ foci in the non-tumor areas. Axillary dissection specimen had 18 axillary lymph nodes and ductal carcinoma metastasis was reported in 2 lymph nodes. No granulomatous structures were observed in the axillary lymph nodes. The patient is being followed in an adjuvant chemotherapy program.

The patients who had bilateral IGM diagnosis except, all patients were in remission. Bilateral IGM patients had twice or triple recurrence but this is not significant statistically. Steroid treatments was recommended for these patients but refused the treatment. One of patients bilateral IGM performed re-excision and autoimmune connective tissue disease was diagnosed following the rheumatological evaluations. The patient had spontaneous remission during the 12 month monitoring period. Two of patients bilateral IGM had no recurrence during the 8 months monitoring period. Nobody of patients hadn’t given immunosuppression treatments. Mastectomy performed only one patient.

Discussion

IGM is a non-malignant, chronic, non-life-threatening and rare breast disease. It was first defined in 1972 and its etiology could not be explained yet [1]. Many publications related to IGM have been published in many countries, it is considered that environmental factors are affecting the pathophysiology. Hormones and autoimmunity are considered to have a profound effect on pathophysiology. The majority of the patients diagnosed with IGM are breastfeeding or pregnant women in their reproductive periods [7]. It can be observed in men very rarely [8]. Fourteen patients in our patient group were in the reproductive period, five of them were in the postmenopausal period.

The common presenting symptom a mass in one breast. The other symptoms are hyperaemia, sensitivity, areolar retraction, fistula and ulceration [2,9]. The mass may mimic breast cancer by pulling the skin tissue above it, or may penetrate the pectoralis major muscle and nipple retraction and may cause lymphadenopathy. Routine radiologic evaluation, ultrasound and mammography may not discern IGM from breast cancer. Similarly, magnetic resonance imaging may not discern the inflammatory process from a tumoral process [5,9]. Irregular hypo-echoic mass, tubular hypo-echoic enlargement, parenchymal heterogeneity and multiple abscess collections are the most common ultrasonographic findings and none of them are specific findings. Since the majority of the population of patients diagnosed with granulomatous mastitis are young women, the sensitivity of mammography is low. While mammography findings are not specific, asymmetric density and skin shrinkage is observed most frequently as well as irregular bordered masses and axillar lymphadenopathy [10]. Our one patient who was diagnosed with invasive ductal carcinoma and ductal carcinoma in situ in addition to IGM, applied us with complaints of a mass at 9 o’clock of the right breast, close to the areola. She had axillary palpable 2 cm diameter lymphadenopathies. The ultrasonography was performed that fibroglandular tissue increase with no evident borders and an inflammatory image, right axillar 2  cm × 2 cm non-reactive 2 lymphadenopathies was reported (Fig. 3: Ultrasonography revealed a fibroglandular tissue increase and axillar lymphadenopathies). Mammography showed focal asymmetric opacity in the retroareolar and outer quadrant of right breast (Fig. 4, 5: Mammography showed opacity in the retroareolar and outer quadrant). Inflammatory breast carcinoma and IGM were considered during the pre-diagnosis. Core biopsy was performed. It was reported as invasive ductal carcinoma and granulomatous mastitis. Only 3 cases which IGM and breast cancer were present concurrently, have been presented in the literature [11–13]. The important aspect of this patient is 4th case whom IGM and breast cancer are present concurrently.

No common consensus exists in the treatment protocol of IGM. Alternative treatment methods vary from medical treatment, monitoring to mastectomy. The investigation of publications related to IGM show a preference for conservative methods. The first preference for patients diagnosed with IGM should be non-operative. Because repeat of surgical procedures may result with cosmetic breast problems. Steroid treatment may be initiated in patients with severe symptoms. Patients with more severe symptoms or those with frequent recurrences may receive immunosuppression or surgical treatment [4,6,14]. Three patients whom diagnosed bilateral IGM developed recurrence. Steroid treatment was recommended for these patients but our patients refused the treatment. After re-excision was performed one patient, an autoimmune connective tissue disease was diagnosed following the rheumatological evaluations. No recurrence developed during the 12 months monitoring period after the re-excision of the patient. Other two patients hadn’t developed during the 8 months monitoring period. Many recently studies have reported clinical regression of the disease just by observational monitoring after IGM diagnosis [7,15,16]. Similarly, except our three patients with recurrence development, the clinical progress of the others showed regression just by observation. The limitations regarding observation and follow-up of our study group are the low number of patients and short monitoring period of 4 months. Despite these limitations, the regression of the clinical IGM only by monitoring are good news regarding the protection of the breast against deforming procedures as well as the cosmetic preservation of the breast.

Conclusion

Patients who followed-up by observation should be explained that IGM is a chronic disease and that it may recur in certain periods. Following by observation may be supported by studies with more patients and longer follow-up duration.

Clinical, radiological and pathological investigations have to be performed together for breast cancer developing due to the chronical progresses of IGM or concurrent breast cancer.

Malignancy in IGM case can be described little more precise, as it is rare.

Conflicts of Interest

None