Evaluation of therapeutic mammoplasty techniques in the surgical management of female patients with idiopathic granulomatous mastitis with mild to moderate inflammatory symptoms in terms of recurrence and patients’ satisfaction

Abstract

BACKGROUND:

Although idiopathic granulomatous mastitis (IGM) affects young females, its surgical management usually leads to disfigurement of the breasts.

OBJECTIVES:

To assess the use of therapeutic mammoplasty techniques for management of IGM in terms of recurrence and postoperative patients’ satisfaction.

METHODS:

This prospective clinical study included thirteen patients who were diagnosed histologically as IGM. Patients with moderate to large breasts, who had a breast mass between 20–50% of the breast size with failed medical treatment or intolerability to steroids were subjected to therapeutic mammoplasty techniques. Only patients with large breasts were offered contra-lateral reduction mammoplasty to resume symmetry and achieve better aesthetic results.

RESULTS:

Early postoperative bleeding that was encountered in one patient (7.7%) was the only serious postoperative complication. Patient was re-operated and the bleeder was secured. Recurrence occurred in 2 patients (15.4%) at 16 and 24 months after the operation. Kyungpook National University Hospital (KNUH) breast reconstruction satisfaction questionnaire used to assess patients’ satisfaction 6 months after the operation and revealed that 10 patients (76.9%) were satisfied after the operation.

CONCLUSION:

Using therapeutic mammoplasty techniques in surgical management of IGM in moderate to large breasts seems justifiable with good results regarding recurrence and postoperative patients’ satisfaction.

Introduction

Idiopathic granulomatous mastitis (IGM), first described by Kessler and Wolloch [1] in 1972, is an uncommon, chronic, noninfectious inflammation of breast tissue with granuloma formation [2]. Patients suffering from this disease are relatively young women in the child-bearing age [3]. Pain, breast swelling, breast masses and inflammatory changes, resembling a breast abscess, are usually the first manifestations of this disease. A palpable breast mass, often painful, is present in virtually every patient and can vary in size from 1 to 10 cm. IGM is usually unilateral with no predilection for one side or another, and axillary lymphadenopathy is present in approximately 15% of patients [4–7]. However, presentations, imaging findings and even Fine Needle Aspiration Cytology may mimic malignancies [8].

Although there is no clear clinical consensus regarding the ideal therapeutic management of IGM, wide local excision and corticosteroids are commonly used to treat this condition [5–9]. A recent algorithm for management of IGM by Kiviani et al. [10] suggested to start initial medical treatment that is consisted of a wide spectrum antibiotic for at least 2 weeks and a nonsteroidal anti-inflammatory drug (NSAID) for at least 2 months. Systemic corticosteroids to be only prescribed after 4 weeks of intractability and for early recurrent (recurrence in the first 4 weeks) or progressive disease. Corticosteroids to be tapered after 1 week of remission and to be discontinued in 2 months. All patients should be followed up serially at least for 18 months. Further interventions, such as surgery (resection or drainage), were reserved for the persistent and completely intractable diseases (failure of medical treatment in 6 weeks and any sign of abscess formation).

Traditional type of surgical treatment of IGM aims at excision of the chronic inflammatory breast mass with enough disease-free margin and direct closure of the defect, which leads to seroma formation. Subsequent infection of this seroma may lead to disfigurement of the shape of the breast [7]. Different studies discussed plastic techniques to improve cosmetic outcome after surgical management of IGM [11–14]. However, most of these studies were case reports. They used musculocutaneous flaps only when the disease involved most of the breast tissue and mastectomy was indicated.

The aim of this study was to assess surgical treatment of IGM using therapeutic mammoplastic techniques in terms of recurrence and postoperative patients’ satisfaction.

Patients and methods

This study is a prospective study that included 13 patients with the diagnosis of IGM who were admitted to the Medical Research Institute, Alexandria University, Egypt between July 2010 and January 2013 and matched the inclusion criteria of the study.

Inclusion criteria. Patients with moderate to large breasts (brassier size B or more), who had a breast mass with or without skin involvement (mild to moderate inflammatory symptoms) between 20–50% of the breast size with failed medical treatment, (i.e., persistence of mass or skin changes in spite of corticosteroid treatment for 12 month), or occurrence of complications due to steroid therapy.

Exclusion criteria. All patients with known etiology of granulomatous mastitis (duct ectasia, sarcoidosis, Wegener’s granulomatosis, giant cell arteritis, polyarteritis nodosa, tuberculosis, syphilis, and cat-scratch disease in breast lymph node tissue) were excluded from the study. Patients with severe inflammatory symptoms who did not show any improvement after drainage and corticosteroids were excluded from the study too. Patients with small breasts or who had a breast mass less than 20% or more than 50% of the breast size were also excluded from the study and offered an alternative surgical option.

Preoperative work up

All patients with a provisional diagnosis of IGM were subjected to thorough history taking and clinical examination. Radiological investigations in the form of mammography (Fig. 1) and ultrasonography. All patients were subjected to true-cut biopsy and histopathological examination. The final diagnosis of IGM was made only when the characteristic histopathologic features of a noncaseating granulomatous inflammation, centered on breast lobules, were demonstrated in each case, in the absence of any evidence of specific underlying causes (receiving antidepressant therapy [15], TB, Fungi and Corynebacteria).

All patients were initially treated by prednisolone [16]. The initial dose was determined by the physician. The median initial dose was 0.5 mg/kg/day and then the dose was tapered by 0.1 mg/kg/day, weekly or biweekly according to the clinical and radiological improvement.

An informed consent was obtained from all participant patients regarding the procedure as well as the research.

Operative work up

All patients were operated by the same team of surgeons who are experts in the field of breast surgery and various plastic techniques. The operation started by excision of the inflammatory mass with all infected tissue (Figs 2 and 3). The technique of closure of the defect was tailored according to the location of the inflammatory mass as follows [11,17]:

Inflammatory masses at the upper half of the breast were subjected to inferior pedicle therapeutic mammoplasty technique (Fig. 4) or round block.

Inflammatory masses at the lower half of the breast were subjected to superior pedicle therapeutic mammoplasty techniques and round block.

Inflammatory masses at the peri-areolar region were subjected to round block.

Regarding the contralateral breast, only patients with brassiere sizes C or D (10 patients) were offered contra-lateral reduction mammoplasty to resume symmetry and achieve better esthetic results. Seven patients accepted the procedure, while 3 patients refused.

Post operative work up

Patients were kept hospitalized for 48 hours after surgery to exclude the possibility of postoperative bleeding and for control of pain. They received medication in the form of antibiotics (third generation cephalosporin) and non steroidal anti inflammatory drugs (NSAIDs).

Outcomes

Primary endpoints

Recurrence was assessed clinically by the surgeons during the follow up visits at outpatient clinics at 3, 6, 12, 18 and 24 months. Recurrence was considered only after true-cut biopsy and histopathological confirmation.

Patients’ satisfaction was assessed by asking the patients to fill the Kyungpook National University Hospital (KNUH) breast reconstruction satisfaction questionnaire [18] during the follow up visit at 6 months after surgery. Each question was graded on a 5-point Likert scale ranging from “very satisfied (5)” to “very dissatisfied (1)” (Table 1). Then, patients were divided into two groups, with a rating of “satisfied” for mean of total scores ≥4 and “dissatisfied” for all others.

Secondary end points

Operative time measured (in minutes) by the operative nurse during the operation.

Early postoperative complications (bleeding, early wound infection, seroma or diastasis of surgical wound) detected by the surgeons at the outpatient clinic during the early postoperative period (at 14 days and one month after surgery).

Statistical analysis for patients’ data was done using the Statistical Package for Social Sciences (SPSS version 22) for data entry and analysis. To test significance of differences, independent sample t test was applied for quantitative data while the Chi-Square test was applied for qualitative data (Fisher exact test was used when appropriate). A statistically significant p -value was considered at p < 0.05.

Number of trial registry ACTRN12615000440527

http://www.ANZCTR.org.au/ACTRN12615000440527.aspx

Results

The study included 13 female patients with the histopathological diagnosis of IGM and with a mass size between 20% and 50% of the breast size. Twelve patients (92.3%) were in their reproductive age period, while one patient (7.7%) was post-menopausal. All patients had a history of pregnancy and breastfeeding for a period ranging from 12 to 21 months. Three patients (23.1%) reported using contraceptive pills, 2 patients were smokers (15.4%) and 2 patients were diabetic (15.4%). Patients showed a wide spectrum of manifestations resembling cancer, nonspecific or specific inflammation. Preoperative clinical data of the patients are shown in (Table 2).

All patients were subjected to surgical excision of the mass and therapeutic reduction mammoplastic techniques according to the location of the excised mass within the breast. Recurrence occurred in 2 patients (15.4%) 16 and 24 months after the operation. In their visit 6 months after the operation, most of the patients (76.9%) were satisfied after the operation. Operative and postoperative data are shown in (Table 3).

Univariate analysis revealed that there were no factors affecting recurrence. Multivariate analysis could not be performed due to low sample size. Recurrence was found to be a significant factor affecting patient satisfaction (Table 4).

Discussion

Idiopathic Granulomatous mastitis is an uncommon benign inflammatory disease of the breast. It is characterized by the presence of chronic granulomatous lobulitis in the absence of an obvious etiology [19]. Many studies described the association of some risk factors for IGM, e.g., young age, history of pregnancy, long breast feeding periods, receiving oral contraceptive pills and smoking [20–24]. Some of these risk factors were present among our patients, e.g., young age (mean age 35.5 years) and history of pregnancy and lactation. On the other hand, the relations of IGM to use of oral contraceptive pills and smoking were not strongly supported in our study.

Diagnosis of IGM is a real challenge that is usually done by exclusion. All causes of acute and chronic inflammation and infective lesions of the breast should be excluded first. This may include a long list of diseases [7,8,25,26]. Only 3 patients in our study (23.1%) presented with a picture of specific inflammation. That is why, in our study, diagnosis of IGM was confirmed preoperatively by histopathological examination of core needle biopsy to all patients.

A very important cause for underestimation of the magnitude of the problem and even misdiagnosis is the lack of awareness of IGM among physicians and pathologists. Variable clinical picture and radiological findings of IGM, which sometimes mimic inflammation or malignancy, represent another challenge for the correct diagnosis [7,8].

Management of IGM remains controversial [20,22,24,27]. Steroid is a treatment of choice as a conservative management or as a concomitant therapy with surgical excision [22,24], as it decreases lesion dimension and augments complete healing after excision [7]. However, timing and dosage of steroid therapy are still controversial. Some reports suggest it may be necessary to continue high doses of corticosteroids until complete resolution following a diagnosis of IGM [28]. Others suggest that steroids should be used either after excision or before surgery. For complicated and resistant cases, steroids should be administered after excision [7,9,18,20,24,29].

Regarding surgical options, wide excision of the mass was performed traditionally, but the recurrence rate in surgical treatment without steroids was reported to be higher than steroid treatment in some studies [30,31].

According to the policy of our institute, steroids were used preoperatively in an initial dose that was determined by the physician, which was tapered afterwards according to the clinical and radiological improvement. Only patients in whom corticosteroids failed to control their disease were subjected to surgical excision. Efforts were done to exclude any infectious conditions in the breast or somewhere else in the body as these conditions may be exacerbated by the use of steroid therapy.

Aesthetic surgery of healthy breasts has become a major industry over the past half century. Some of the indications have medical bases, but much of the demand has arisen from more complex psychosocial, economic, and reproductive pressure of human male to human female [32,33]. An important goal of breast reconstruction is to restore the breast as normally and as attractively as possible while minimizing visible scars [34]. However, on reviewing the literature, no reports were found regarding results of therapeutic reduction mammoplasty techniques for management of IGM. This may be due to paucity of both patients and studies concerning with reconstruction after excision of IGM. We believe that patients with IGM are good candidates for plastic surgical techniques. Most of these patients are in their young with active reproductive life and are much concerned with cosmetic results after surgical excision, especially that the condition they suffer is generally a benign condition.

Moreover, the nature of the disease may include collections and sometimes sinus formation with consequent scars. All of which may be avoided by prompt excision. Also, the principle for success of surgery is generous excision of the inflammatory mass to avoid recurrence of the disease. Nevertheless, this may leave a wide defect that, if not properly managed with plastic techniques, may collect seroma and lead to disfigurement of the breast.

In our patients, early postoperative complications were mostly minor. Seroma was encountered in 2 patients (15.4%) and were treated with repeated aspiration under complete aseptic techniques. Superficial wound infection occurred in 3 patients (23.1%) in the form of erythema within 1 cm from the wound and were treated successfully with conservative measures. One patient (7.7%) had diastases of the wound and was re-sutured under local anesthesia. The only serious early postoperative complication was early postoperative bleeding that was encountered in one patient. She was re-operated and the bleeder was secured.

In this study, recurrence occurred in 2 patients (15.4%). Recurrence was considered as recurrence of inflammatory mass clinically radiologically and was confirmed by tru-cut biopsy. One after 16 months while the other after 24 months after the operation. In literature, different recurrence rates were reported after excision of IGM (range, 16–50%) [20,24,35]. We believe that combining preoperative steroids with wide surgical excision may be the cause of the low recurrence rate compared to the literature. Postoperative cosmetic appearance may create a hidden pressure on the surgeon that would prevent him from performing the proper wide excision for his patients. Applying plastic techniques allows the surgeon to excise the mass with good margin without affecting his concerns about the postoperative cosmetic results. However, having a recurrence at 24 months after surgery may necessitate prolonging the required period of follow up after surgical management of IGM in future studies. No risk factors could be detected statistically to affect recurrence in this study.

Our patients’ satisfaction was assessed 6 months after surgical management. Ten patients (76.9%) were satisfied, while 3 patients (23.1%) were not satisfied. Two of the three patients who were not satisfied had recurrence. It is noted that the questionnaire was filled by the patients 6 months after surgery, i.e., before the recurrence which occurred 16 and 24 months for these patients. So, we cannot allege that the recurrence influenced patients’ satisfaction. There may be a subclinical activity of the disease that influenced the patients’ satisfaction without any evident clinical signs. Further studies may be necessary to clarify this finding.

Small number of patients, lack of control for the effect of confounding variables, limited long-term follow-up and being non comparative are considered the main limitations of this study.

Conclusions

Using therapeutic mammoplasty techniques in surgical management of IGM in moderate to large breasts seems justifiable with good results regarding recurrence and postoperative patients’ satisfaction. However, multicenter studies with larger number of patients and a longer follow up period may be necessary to support our results.