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 Table of Contents  
ORIGINAL ARTICLE
Year : 2016  |  Volume : 6  |  Issue : 2  |  Page : 100-104

Chronic granulomatous mastitis: A therapeutic dilemma revisited


Department of General Surgery, DY Patil Medical College, Pune, Maharashtra, India

Date of Web Publication30-Nov-2016

Correspondence Address:
Vinayak V Kshirsagar
17/1, Durga Colony No. 3, Ganesh Nagar, Thergaon, Pune - 411033, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2278-9596.194983

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  Abstract 

Background: Idiopathic granulomatous mastitis (IGM) is a rare chronic inflammatory disease. Because of obscure etiology and diagnosis, its management remains a challenge. Rareness and varied presentation of IGM makes it difficult to standardize treatment protocol. The aim of this study was to analyze the clinical presentations, imaging findings, clinical course, and management of patients who were treated for IGM.
Patients and Methods: In this study, a retrospective review of 15 patients with IGM between January 2012 and December 2014 was performed based on clinical, radiological, and pathological findings; follow-up information and recurrence were obtained from records.
Results: The median age of presentation was 30 years (age range: 24–42). All patients were married and parous with a history of breast feeding. Breast mass with or without skin changes was a symptom in all patients. While parenchymal heterogeneity, abscess, and mass were the findings of ultrasound, increased asymmetric density was the main finding of mammography. Wide local excision was performed in 7 (46.7%) patients and incisional biopsy with abscess drainage in 4 (26.7%) patients. Medical treatment was given to 4 (26.7%) patients. Median follow-up was 24 (range: 10–34) months. Eleven patients (73%) were recurrence free.
Conclusions: Granulomatous mastitis often presents as a breast lump with or without skin changes. The diagnosis is done with pathological examination of the surgical specimen or biopsy. The main treatment should be guided according to the clinical presentation either surgical or medical or both.

Keywords: Diagnostic dilemma, granulomatous mastitis, steroids


How to cite this article:
Kshirsagar VV, Male PV, Bendre M, Chavan S, Rathod S. Chronic granulomatous mastitis: A therapeutic dilemma revisited. Arch Int Surg 2016;6:100-4

How to cite this URL:
Kshirsagar VV, Male PV, Bendre M, Chavan S, Rathod S. Chronic granulomatous mastitis: A therapeutic dilemma revisited. Arch Int Surg [serial online] 2016 [cited 2019 Dec 12];6:100-4. Available from: http://www.archintsurg.org/text.asp?2016/6/2/100/194983


  Introduction Top


Granulomatous mastitis is a benign inflammatory disease of the breast which was first described in 1972.[1] It presents with varied local presentations.[2] Although easily confused with tuberculosis or malignancy, diagnosis is confirmed only on histopathological examination.[3] The etiology of idiopathic granulomatous mastitis (IGM) is unknown, however, some authors have speculated that the cause may be an autoimmune process, undetected microorganisms, use of oral contraceptives, or a reaction to childbirth.[4] IGM is characterized pathologically by the presence of chronic granulomatous lobulitis in the absence of an obvious etiology.[1],[5]

An optimal treatment is not established. Medical therapy, wide local excision, and abscess drainage are currently the favored treatment options for IGM.[6] Incidence of IGM is increasing; surgeons and pathologists are commonly not aware of IGM.[7],[8] It is often misdiagnosed by the primary care physician. It leads to diagnostic confusion and invariably morbidity to the patient. Surgical excision, antibiotics, incision drainage, and use of steroids are the only treatment options available with no specific guidelines.[1],[9],[10]

Wide local excision with or without steroid therapy is the most commonly recommended treatment.[1],[10],[11]

The aim of this study was to analyze the clinical presentations, imaging findings, clinical course, and management of the patients who were treated for IGM.


  Patients and Methods Top


In this study, a retrospective review of 15 patients with IGM between January 2012 and December 2014 was performed based on clinical, radiological, and pathological findings; follow-up information and recurrence were obtained from records. The ethical committee clearance was obtained.

Detailed history regarding presenting symptoms, history of lactation, use of contraceptives, follow-up information, and recurrence were recorded from clinical records. All patients underwent clinical breast examination. Ultrasonography of breast was performed in all patients. Mammography was performed in 3 patients (age 35, 38, and 42, respectively) and magnetic resonance imaging (MRI) of the breast in 1 patient. Histopathological diagnosis was obtained from incisional or wide local excisional biopsies. Wide local excision was performed. After drainage of abscess, incisional biopsy was taken from the abscess cavity. Inflammatory reaction with granulomas which were composed of epitheloid histiocytes, Langhans giant cells accompanied by lymphocytes, plasma cells, and occasional eosinophils centered on lobules was seen on histopathologic examination.

In case of abscess, Ziehl–Neelsen staining was done for tuberculosis. Cultures for aerobic and anaerobic bacteria were also obtained. Ultrasonography and physical examination were performed every month until resolution of lesions was confirmed.


  Results Top


Fifteen patients who were diagnosed with IGM histopathologically and had complete follow-up data were evaluated and included in this study.

The mean age was 30 years, ranging from 24 to 42 years. 93.33% of patients (14 out of 15) were in their reproductive ages. All patients had children and had a history of breast feeding. The left breast was affected in 8 (53.3%) patients, right breast in 6 (40%) patients, and bilateral in 1 (6.3%) patient [Table 1]. The lesions were located in the upper external quadrant in 2 (13%) patients, in upper internal quadrant in 3 patients (20%), in lower internal quadrant in 1 patient (6.6%), in lower external quadrant in 1 (6.6%) patients, in periareolar region in 5 patients (33.33%), and diffuse involvement was seen in 3 patients (20%) [Table 2].
Table 1: Site of lesion in the breast

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Table 2: Location of lesions in the breast

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Painful, firm, and ill-defined mass were the symptoms in all the patients [Figure 1]. Palpable mass lesions were observed in 6 (40%) patients [Table 3].
Figure 1: Painful swelling in breast

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Table 3: Patients' symptoms

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Parenchymal heterogeneity with no discrete mass was established in 6 patients (40%), irregular mass in 4 patients (26.66%), abscess formation in 2 patients (13.33%), irregular mass with heterogeneity in 1 patient (6.66%), heterogeneity with abscess formation in 1 patient (6.6%), and heterogeneity, abscess formation, and mass in 1 patient (6.6%) on ultrasonography [Table 4].
Table4: US findings of patients with idiopathic granulomatous mastitis ultrasound findings

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Associated enlarged axillary lymph nodes were present in 4 (26.7%) patients. Mammogram was obtained from 3 (20%) patients. Mammography detected increased asymmetric density in 1 (6.66%) patient and asymmetric density with skin thickening in 2 patients. MRI was performed in 1 patient where involvement was diffuse and bilateral. Wide local excision was performed in 7 (46.6%) patients, incisional biopsy with abscess drainage in 4 (26.66%), and medical treatment in 4 patients. Each case demonstrated the presence of epithelioid histiocytes, lymphocytes, plasma cells, polymorphonuclear leukocytes, and multinucleated Langhans-type giant cells without caseous necrosis [Figure 2]. Serologic and bacterial tests were negative in all patients. Patients with abscess formation received empirical treatment. Patients' follow-up was done monthly till recovery.
Figure 2: The inflammatory response in granulomatous mastitis is cell-mediated and consists of epithelioid histiocytes, Langhans giant cells, lymphocytes, plasma cells, and a few eosinophils. Areas of fat necrosis and even microabscesses may be seen

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Treatment with wide local excision was successfully performed in 7 patients. There was recurrence after 4 months in one patient in the same quadrant of the breast which was treated with reexcision. In 4 patients, we started medical treatment after trucut biopsy report. Tablet prednisolone (0.5–1 mg/kg) was given for 2 weeks with a gradual tapering dose. Only 1 patient had recurrence after medical treatment due noncompliance of the patient. The average time for resolution of symptom after incision and drainage was 5 months. Out of 4 patients treated with incision and drainage, 2 had recurrence that was treated medically [Table 5] and [Table 6].
Table 5: Treatment received

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Table 6: Recurrence

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  Discussion Top


IGM is an exceedingly rare disease with nonspecific clinical findings, mostly seen in females in their reproductive age.[6] In our study, all patients were in their reproductive age and all patients were parous. Many agents, such as local irritants, oral contraceptive pills, viruses, parasitic infections, hyperprolactinemia, diabetes mellitus, smoking, alpha 1 antitrypsin deficiency, and autoimmunity have been proposed to explain the etiology of IGM, however, these have never been proven,[6],[12],[13],[14],[15],[16] It is a diagnosis of exclusion made after malignancy and other known granulomatous diseases such as mycobacterial infections and sarcoidosis have been ruled out.[17]

Studies have shown that IGM is associated with a history of childbirth and breastfeeding within the previous 5 years,[16],[18] which is consistent with our patients' history. IGM presents most commonly with a painful, firm, tender, ill-defined mass in the breast and unilateral.[1],[16]

The lesions may be located in any quadrant of the breast.[16] In consequence of granulomatous inflammation, IGM can cause skin thickness, sinus and abscess formation, axillary lymphadenopathy, and nipple retraction, which may be clinically mistaken for breast carcinoma.[1],[6],[16] All patients were admitted with mass or accompanied skin changes. The lesions were located in any location, however, there were the tendency of periaerolar and diffuse involvement in 53.3%. Bilateral involvement is reported very rarely.[11],[14],[16] There was only 1 patient in our series with bilateral and diffuse involvement. All masses were firm and ill-defined with or without axillary lymphadenoopathy. However, all of these enlarged nodes were established to be reactive and without any suspicion for malignancy on ultrasound. The information obtained from ultrasound and mammography is nonspecific, and hence the lack of specificity to diagnose IGM or to exclude breast carcinoma. The most common mammographic appearance of the lesion is an asymmetrically increased density.

Ultrasound findings include a mass-like appearance, tubular/nodular hypoechoic structures, and focal decreased parenchymal echogenicity with acoustic shadowing.[19]

In other studies, parenchymal heterogeneity and areas of mixed echo pattern have been reported.[15] In our study, the most common ultrasound findings were parenchymal heterogeneity, irregular hypoechoic mass, and abscess formation. In our opinion, while physical examination indicates a locally advanced breast carcinoma, the appearance of parenchymal heterogeneity and abscess formation on ultrasound, especially with enlarged reactive axillary lymph nodes, suggest the presence of an inflammatory granulomatous process. However, these findings are nonspecific and do not exclude carcinoma. Therefore, histological confirmation is mandatory to rule out malignancy.

An ill-defined mass, asymmetrically increased density without parenchymal distortion, or microcalcification are the most common findings on mammography.[15],[20] Similar findings of mammography were also noted in our study.

Dursun et al. reported that round, smooth-contoured mass-like lesion with rim enhancement, or segmental nonmass-like lesion on MRI is the most common features of the disease.[20] However, MRI does not play a role in the differential diagnosis between other inflammatory and granulomatous diseases and IGM.[1],[16],[20]

There is still no accepted management strategy for IGM. Oral corticosteroids and surgery have both been used as treatment options. Limited excision alone has little benefit because there is a strong tendency of recurrence. Although initial excision of breast tissue ensures negative margins for inflammatory granulamatous tissue, it may have unfavorable cosmetic results. Oral corticosteroids have most often been used in recurrent cases in previous studies.[13],[14],[21] Initial medical treatment was started with oral steroid in 4 patients. Proper treatment is evident by the regression of skin lesions and parenchymal induration in nearly all patients after 2 weeks [Figure 3]. The residual lesion resolved over a period of 6–11 months. The recurrence rate has been reported to be 16–50% in the literature.[21],[22] In our follow-up period, IGM is seen to recur in 4 patients (26.7%). One was treated with wide local excision and the rest by medical treatment.
Figure 3: Follow-up case of abscess drainage in idiopathic granulomatous mastitis patient

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  Conclusion Top


IGM is an inflammatory breast disease found in young women of reproductive age group. It presents with a large painful irregular mass, which mimics carcinoma, as a physical change. Breast imaging modalities are not helpful in differentiating IGM from invasive cancer. The correct diagnosis is established by cytological or histological examination. Wide local excision is good when an isolated mass is present. Medical treatment with corticosteroids is preferred when irregular, diffuse mass with skin changes or with abscess formation is observed. High index of suspicion with a multidisciplinary approach by the surgeon, radiologist, and pathologist is required to reduce social and psychological morbidity of the patient.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

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    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4], [Table 5], [Table 6]


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