+ MODEL Asian Journal of Surgery (2015) xx,1e9

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ORIGINAL ARTICLE Bilateral idiopathic granulomatous Mehmet Velidedeoglu a, Fahrettin Kilic b, Birgul Mete c, Mucahit Yemisen c, Varol Celik a, Ertugrul Gazioglu a, Mehmet Ferahman a, Resat Ozaras c, Mehmet Halit Yilmaz b, Fatih Aydogan a,* a Department of General Surgery, Cerrahpasa Medical Faculty, Istanbul University, Istanbul, Turkey b Department of Radiology, Cerrahpasa Medical Faculty, Istanbul University, Istanbul, Turkey c Department of Infectious Diseases, Cerrahpasa Medical Faculty, Istanbul University, Istanbul, Turkey

Received 1 July 2014; received in revised form 24 February 2015; accepted 25 February 2015

KEYWORDS Summary Objectives: Idiopathic (IGM) is a benign rare inflammatory cancer; pseudotumor. Bilateral involvement of IGM has been reported in a few cases. To our knowl- cancer; edge, this study is the largest series of bilateral cases to date. The goals of this study were corticosteroids; to present clinical features of bilateral IGM and to evaluate the results of treatments. granulomatous Materials and methods: We performed a retrospective review of the idiopathic granulomatous mastitis; mastitis database from 2010 to 2013. Ten female patients who met required histologic and clin- idiopathic; ical criteria of IGM in both were included in study. Demographic data, clinical findings, tuberculosis medication history, and radiologic findings are presented. Results: The mean age at onset of the disease was 38.4 8.3 years (range: 29e52 years). Nine patients had no recurrence during a mean follow-up period of 21 months (range: 11e26 months). Additionally, the median time to second breast involvement was 15.6 months. Conclusion: Bilateral IGMs have a higher rate of more relapse and greater resistance to medical therapies than do unilateral IGMs. Surgical management should be avoided unless all medical treatment options have been exhausted. Nevertheless, expectant management seems a rational option for the treatment of bilateral IGM. Copyright ª 2015, Asian Surgical Association. Published by Elsevier Taiwan LLC. All rights reserved.

Conflicts of interest: Authors of the manuscript declare no conflict of interest. They certify that all their affiliations with or financial involvement in, within the past 5 years and foreseeable future, any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript are completely disclosed (e.g., employment, consultancies, honoraria, stock ownership or options, expert testimony, grants or patents received or pending, royalties). _ _ * Corresponding author. Istanbul U¨niversitesi Cerrahpas‚a Tıp Faku¨ltesi Yerles‚kesi, Genel Cerrahi AD 34098 Fatih/Istanbul, Turkey. E-mail address: [email protected] (F. Aydogan). http://dx.doi.org/10.1016/j.asjsur.2015.02.003 1015-9584/Copyright ª 2015, Asian Surgical Association. Published by Elsevier Taiwan LLC. All rights reserved.

Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003 + MODEL 2 M. Velidedeoglu et al.

1. Introduction the shortest and longest intervals after childbirth were 1 year and 10 years, respectively. None of the patients were Idiopathic granulomatous mastitis (IGM), also known as breast-feeding. None of the patients had a history of idiopathic granulomatous lobulitis, is an uncommon, benign tuberculosis or systemic granulomatosis, but three had a inflammatory pseudotumor that was first reported by family history of tuberculosis. Kessler and Wolloch in 19721. Clinically, IGM presents as a The most common presenting symptom was a hard mass, Z palpable hard lump with erythematous skin changes or which was detected in all of the patients (n 10; 100%). lymph node involvement, which may lead to Additionally, seven presented with redness of the skin discharge or retraction, sinus formation, breast , (70%), and draining sinuses (fistulas) or discharge from the e Z and “apple jelly” scars2 8. IGM is usually unilateral and can breast skin (n 7; 70%). Nine patients had no recurrence e be seen in all quadrant regions, except for the subareolar during a mean follow-up period of 21 months (range: 11 26 area9,10. months). Only one patient had static disease, at a follow-up Few cases of bilateral involvement have been reported duration of 26 months (Fig. 1). in the literature11e13. To our knowledge, this study, which includes 10 bilateral IGM cases, is the largest series of 3.2. Radiologic findings bilateral cases to date. The goals of this study were to present the clinical features of bilateral IGM and to eval- The most useful method for evaluating the radiologic uate the results of treatment. appearance of IGM is sonography. The technique defines valuable data for infectious conditions such as effusions, 2. Materials and methods inflammation of parenchyma and fatty tissues, abscess formations, fistula tracts. These are the key findings for discriminating the suspected lesions from malignancy. In- Between 2010 and 2013, 10 female patients who met the formation of the patients’ history is essential to assess required histologic criteria of IGM in both breasts were before evaluation. Sonography examination showed a included in this retrospective study. The patients were hypoechoic collection in nine patients (90%), solid lesion in referred to our mastitis study group from the Outpatient one patient (10%), thickening of the breast skin in eight Departments of the General Surgery and Infectious Diseases patients (80%), fistula tracts in four patients (40%), and Clinics. Our study group included three infectious disease ductal fillings and postobstructive dilatation in three pa- physicians, two radiologists, two pathologists, and two tients (30%). Mammographic findings were nonspecific in breast surgeons. The diagnosis was confirmed by core bi- the three patients who underwent mammograms. Magnetic opsy of the suspicious breast lesions and biopsy specimens resonance imaging (MRI) evaluation may demonstrate the taken from the abscess wall. The pathological criteria for extension of the disease and may be used to clarify the the diagnosis of IGM were the presence of the characteristic suspected diagnosis. MRI was performed for three cases; histopathological features of a noncaseating granulomatous asymmetrical enhancement with nonspecific contrast inflammation centered on breast lobules, in the absence of enhancement kinetics and fluid collections were common. any evidence of specific underlying causes. The presence of epithelioid histiocytes, lymphocytes, plasma cells, poly- morphonuclear leukocytes, and multinucleated Langhans- 3.3. Medication history type giant cells without caseous necrosis were demon- strated in each case. Treatment outcomes were evaluated All patients (100%) had previously taken antibiotics, six by physical examination and ultrasonography (US). The patients (60%) had undergone drainage before presenting clinical data in terms of the presentation, histopathology, at our clinic and four patients (40%) (Cases 1, 7, 8, and management, recurrence, discharge from the clinic, and 10) were treated with a second course of antibiotics follow-up at monthly intervals were analyzed by a review of (Table 1). the medical records. The Ethics Committee of Istanbul We sent all the biopsy samples to the histopathology University Cerrahpasa Medical School, Istanbul, Turkey department. The characteristic caseating granulomas for approved the study. The document number is 83045809/ TB was observed only in one and this sample was sent for TB 604/02-15545/Date: June 5, 2014. culture as well. All were stained with Periodic Acid Schiff (PAS) and Ehrlich Ziehl Neelsen (EZN) to demonstrate the 3. Results microorganisms within the tissue; all remained negative. Case 1 had two recurrences and received antibiotics and meloxicam. The patient was offered steroid treat- 3.1. Demographic data and clinical findings ment but refused. Following cessation of medications, the patient had expectant conservative management followed Table 1 provides the clinical characteristics of the patients. by complete spontaneous recovery. Cases 8 and 10 showed The mean age at onset of the disease was 38.4 8.3 years complete disease resolution and no recurrence with (range: 29e52 years). The patients had no history of breast expectant management. Case 7 had a history of antitu- carcinoma and no family history of granulomatous mastitis. berculous therapy and a complete recovery of IGM in her All but two patients were of reproductive age, and all were left breast, and she was referred to us for a lump affecting parous. The parity of the patients ranged from two to five, therightbreast.Nopreviouspathologicalormicrobio- with a mean parity of three. None of the patients had a logical data regarding tuberculosis were available. Core recent history of pregnancy at the time of the presentation; needle biopsy was performed, and the specimen was

Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003 + MODEL Bilateral idiopathic granulomatous mastitis 3 confirmed to be granulomatous mastitis; bacterial culture sinuses, and scars, which are manifestations of intrinsic was negative. This patient was managed conservatively inflammatory or neoplastic breast disease1,2,8,16,21. Abscess with regular clinical and radiological surveillance. After 2 and fistula formation, ulceration, deformity of the breast, months of follow-up, the patient showed complete re- and unsightly scars are common complications of IGM. covery. One month later, she received meloxicam 15 mg Although this disease is not life-threatening such as other once per day for 1 month to treat her pain. Until 2013, malignancies, it seriously depresses the social life of the none of these four patients recurred without medication. patient3,16,17,21e24. Four (40%) patients (Cases 3, 4, 6, and 9) were treated All of our patients had hard masses as the initial with oral prednisolone for 6 weeks at an initial dose of complaint, which were painful in only two patients. Six 0.5 mg/kg/d in divided doses (4 þ 2), which were tapered (60%) patients had redness of the skin. There was a ten- slowly with clinical improvement. Of these, Case 3 dency for local recurrence and delayed wound healing. All developed recurrence when the prednisolone dose was of our patients had at least one recurrence, and most tapered. As the dose was increased, the patient seemed experienced inadequate or no response to various treat- to respond, but the medication was stopped because she ment modalities. became pregnant. She underwent expectant conservative IGM is seen mostly in females of childbearing age with a management, and experienced complete spontaneous history of pregnancy and lactation in the previous 5e6 recovery. Case 4 developed recurrence 2 months after a years9,11,25. Although the youngest and oldest females ever complete recovery following cessation of steroid therapy. reported were aged 11 years and 83 years, respectively, the Nonetheless, the recurrent disease responded well to a disease usually appears in the breast in the third or fourth second course of oral prednisolone and the dose was decade3,17,19. However, younger patients were reported in tapered slowly. However, after a short time period, she recent trials2,26e30. In our study, the mean age of patients again developed recurrence. Expectant conservative at onset of the disease was 38.4 years. management was preferred, and the patient experienced There was a notable delay between onset of symptoms complete spontaneous recovery after 5 months. Interest- and diagnosis, a median of 14 months in our study. As IGM is ingly, this patient became pregnant during this recovery rare, this disease is not routinely considered as a differ- period. Case 9 showed complete recovery after a course ential diagnosis of a simple breast abscess. Surgical of steroid therapy. Case 6 developed recurrence following drainage or US-guided aspiration is commonly preferred in cessation of steroid therapy, but the patient showed practice in place of core-needle biopsy or incisional biopsy complete recovery after a second course. Case 2 had no from the abscess wall, which may explain the prolonged clinical response to steroid treatment; however, the pa- time to diagnosis. The median time to second breast tient showed complete remission after undergoing com- involvement was 15.6 months. No response to antibiotics bined antituberculous therapy (rifampicin, ethambutol, was observed in three of the 10 unilateral mastitis cases and isoniazid) for 6 months. Case 5 had no response to before the second breast was involved. Two patients antituberculosis treatment, so she received oral prednis- showed no response to antibiotics and steroid therapy olone. She recurred twice following cessation of steroid before bilateral involvement. One patient showed a partial therapy because of intolerance, but showed a complete response to antibiotics, but bilateral involvement occurred response thereafter. After 2 years, she experienced a thereafter. Three patients showed a complete response (1 second recurrence, which is at present being treated with responded to antibiotics, 1 to antibiotics and steroids, and antibiotic therapy. There was a notable delay between 1 to antibiotics and abscess drainage), but they later onset of symptoms and diagnosis, a median of 14 months developed bilateral involvement. Only one patient showed in our study. Additionally, the median time to second complete disease resolution after antituberculosis treat- breast involvement was 15.6 months (Figs. 2e4). ment; unfortunately, the other breast showed involvement 48 months later. We concluded that bilateral IGMs were resistant to medical treatment, and tended to relapse and 4. Discussion recur. All of our patients had a history of pregnancy, were IGM is a rare, nonmalignant, chronic inflammatory breast parous, and had breast-fed. No patient experienced condition of unknown etiology that has received little symptom-onset during lactation. These results are in attention in the literature11. Almost 200 cases have been accordance with previous reports that pregnancy and reported during the past three decades, with most breastfeeding conditions are associated with an increased cases occurring in females from Mediterranean countries risk of IGM15,26,31e34. However, Azlina et al18 and Fletcher (Turkey and Jordan) and Asia (China, Arabia, and et al35 found no association with lactation. In Cases 3 and 4 Malaysia)2,3,5,11,14e18. Although no ethnic predisposition in our series, the medication was stopped because the has been documented, underdiagnosis of tuberculosis patients became pregnant. These patients had expectant mastitis might be a major reason for the increased conservative management, and experienced complete prevalence19,20. spontaneous recovery. It is unclear whether being pregnant The diagnosis and treatment of IGM remains challenging. or receiving expectant management therapy contributed to IGM can mimic breast abscess or inflammatory breast can- their recovery. Kaur et al36 reported that prolonged breast- cer in terms of physical and radiological findings. It typi- feeding might cause long-term distension of acini and cally presents as a unilateral palpable firm , ducts, causing them to rupture. They suggested that this with occasional , peau d’orange change, factor might cause a granulomatous response. Baslaim erythema, nipple inversion, breast asymmetry, draining et al2 opposed this idea; they reported that patients,

Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003 + MODEL 4 M. Velidedeoglu et al.

Table 1 Clinical characteristics of patients with bilateral IGM. Patient’s characteristics Case 1 Case 2 Case 3 Case 4 Age (y) 52 51 30 33 Parity 3 4 4 2 Past lactation Yes Yes Yes Yes Oral contraceptive use No No No No Tobacco smoking Yes No Yes Yes Trauma history to the breast No No No No Medical history DM Hashimoto’s thyroiditis No No Family history in relation No No Yes No to TB First admission to us No No No No Presentation time 8 y after 6 y after 5 y after 2.5 y after childbirth childbirth childbirth childbirth Total duration of breast- 36 17 60 7 feeding (mo) Initial complaint A hard mass A hard mass and redness A hard mass and A hard mass and of the skin redness of the skin redness of the skin Time from onset of symptoms 6241412 to diagnosis (mo) First affected breast side Right Right Left Right Duration for the second 612123 breast involvement (mo) Size and location of mass in 2 cm, Upper inner 2.5 cm, Upper inner 3 cm, lower inner 4 cm, Upper inner right breast quadrant quadrant quadrant quadrant Size and location of mass in 2.5 cm, Upper inner 2 cm, Upper outer 3 cm upper quadrant 4 cm, Upper outer left breast quadrant quadrant close to aerola quadrant Core needle biopsy Yes Yes Yes Yes Tuberculin skin test (mm) 20 Anergic 28 9 Acid fast stain Negative Negative Negative Negative Rose Bengal Negative Negative Negative Negative Radiologic tool US US US US Abscess drainage Yes Yes Yes Yes Antibiotics Amoxicillin-clavulanate Amoxicillin-clavulanate Ceftidoren pivoxil Ceftriaxon þ ornidazol Steroid therapy No Previously Yes Yes Antituberculous therapy No Yes No No Recurrence Yes Yes Yes Yes Outcome Healed Healed Healed Healed Unsightly scars Yes, bilateral Yes, bilateral Yes, bilateral Yes, bilateral DM Z diabetes mellitus; IGM Z idiopathic granulomatous mastitis; US Z ultrasonography.a The data are not available due to histopathological sampling of outer clinic. especially nursing mothers who had breast-fed from one delay recovery and extend IGM disease involvement to the breast only, developed the disease in the contralateral second breast. breast. Although unilateral IGM is typical in previous reports, The etiology of IGM is unclear; however, many factors, bilateral involvement has been included in only a maximum such as local irritants, oral contraceptives, nicotine addic- of two patients in these studies4,12,30. Bilateral involvement tion, and breast trauma, have been consid- has been reported at a lower frequency, and we found no ered2,3,9,15,16,30,37,38. No patients included in the studies by reports of a bilateral IGM series. Some authors reported Ozel et al30, Azlina et al18, or Neel et al39 used oral con- more common involvement of the right breast16,19,35, traceptive hormones. Only one of our patients used oral whereas others reported left or equal involvement30,39.In contraceptives for a short period 15 years ago. The patients our study, the left breast was the first affected in six of 10 in the study by Azlina et al18 were nonsmokers. In addition, patients. the patients had no history of local trauma to the breast in For an accurate diagnosis, IGM must be differentiated the Ozel et al30 study. In our study, six patients were from other disorders such as tuberculosis, sarcoidosis, smokers, but none reported a history of local trauma. An foreign body reaction, ductal ectasia, mycotic infection, association between periductal mastitis (but not IGM) and Wegener’s granuloma, and histoplasmosis40. Diagnostic nicotine addiction has been established, as seen in more tests for tuberculosis include the purified protein derivative than half of our patients (60%); moreover, smoking may (PPD) skin test, in vitro interferon-gamma assay and

Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003 + MODEL Bilateral idiopathic granulomatous mastitis 5

Case 5 Case 6 Case 7 Case 8 Case 9 Case 10 34 40 43 32 29 40 2452 33 Yes Yes Yes Yes Yes Yes No No For 45 d 15 y ago No No No Yes Yes No Yes No No No No No No No No No No No No No No Yes No Yes No No No

No No No No No No 5 y after childbirth 1 y after childbirth Over 2 y after 10 y after childbirth 4 y after childbirth 6 y after childbirth childbirth Over 12 mos 48 120 12 27 60

A hard mass and A painful hard A hard mass and A painful hard mass A hard mass and A hard mass and redness of the skin mass redness of the skin redness of the skin redness of the skin 15 14 30 12 4 9

Left Left Left Right Left Left 15 8 48 24 4 24

6 mm, Upper outer 1 cm, Upper outer 4 cm, Upper outer 3 cm, Upper inner 1 cm, Upper outer quadrant quadrant quadrant quadrant quadrant 3 cm, Upper inner 2 cm, Upper outer No data (left lesion 3 cm, Upper inner 3 cm, Upper outer No data(left lesion quadrant quadrant 4 y ago) quadrant quadrant 2.5 y ago) Yes Yes Yes Yes Yes Yes 17 8 20 8 9 Not performed Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative US US US US US US Yes Yes No Yes Yes Yes Amoxicillin- Amoxicillin- Fusidic acid Amoxicillin- Amoxicillin- Ciprofloxacin þ clavulanate clavulanate clavulanate clavulanate Metronidazole Yes, later Yes No No Yes No Yes, initial No Previously No No No Yes Yes Yes Yes Yes Yes Resistant Healed Healed Healed Healed Healed Yes, in left breast Yes, in left breast Yes, in right breast Yes, in right breast Yes, in left breast Yes, in left breast polymerase chain reaction. Ziehl-Neelsen (ZN) stain PPD describe the disease as inhomogeneous hypoechogenicity and routine histology studies are sometimes insufficient to with internal hypoechoic tubular lesions4,10,19,25,43e45.Itis exclude tuberculosis mastitis16,41,42. In our study, periodic possible to discriminate benign neoplastic lesions from acid-Schiff (PAS) and ZN acid-fast stains failed to identify cancer using dynamic contrast-enhanced MRI; however, this specific causative organisms in any of the 10 patients. The method has lower specificity for distinguishing granuloma- PPD skin test was not performed in one patient. Four pa- tous mastitis from . Therefore, MRI may sup- tients had positive PPD skin test results (> 10 mm in- port US and mammography findings in distinguishing benign durations). Case 2 was anergic for PPD, and she did not inflammatory lesions from malignant lesions5,45e47. MRI aids respond to steroid treatment; therefore, she commenced the surgical approach by delineating the extent of the combined antituberculous therapy (rifampicin, etham- disease, as edema of the breast skin and tissue prevent butol, and isoniazid) for 6 months and showed complete precise evaluation by US12. In our study, US revealed locu- remission. Further differential diagnoses were negative, lated fluid collections of various sizes and ill-defined including a chest X-ray, the Rose Bengal test, and the hypoechoic masses in three cases. Two patients under- antidsDNA test. went breast MRI previously in other centers. In one case, US, mammography, and MRI have limited roles in dis- ductal carcinoma in situ was suspected based on MRI; tinguishing granulomatous mastitis from malignancy. however, US-guided biopsy proved the diagnosis to be IGM. Mammographic examination may show a focal asymmetrical Consequently, there are no IGM-specific radiological find- density in dense breast parenchyma or a mass with a ings. US and mammography are used to rule out breast recognizable margin in fatty breasts. US has been used to cancer in centers that have experienced numerous cases of

Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003 + MODEL 6 M. Velidedeoglu et al.

Figure 1 The picture presents the deformation of the breast due to skin fistulas of IGM. IGM Z idiopathic granulomatous mastitis.

IGM. Diagnosis is confirmed on tissue sampling biopsy by revealing noncaseating granuloma with epithelioid histio- cytes, giant cells, plasma cells, and eosinophils confined within breast lobules48. No clear consensus exists in the literature regarding the optimal treatment approach for IGM, as none seems to be ideal. Treatment options include antibiotics, steroids, methotrexate (MTX), azathioprine, drainage of the wound, wide surgical resection, mastectomy, and expectant man- agement with close follow-up49e54. Aggressive surgery, such as wide resection or mastectomy, is an accepted treat- ment7,15,55, whereas some recommend a conservative approach17,39. In addition to being therapeutic, surgical excision provides a definitive diagnosis56. Involvement of oncoplastic surgery, such as mastectomy, followed by autologous breast reconstruction and steroids are alterna- tive treatments for severe and intractable cases33. How- ever, surgical resection can also be associated with complications, including recurrence rates as high as 50%, poor wound healing, sinus tract or abscess formation, and disfigurement9,15e17,22,33,40,57e59. None of our patients with bilateral IGM underwent surgical resection. Regarding conservative management, steroids as a pri- mary treatment have been shown to be beneficial for shrinking the lesion both pre- and postoperatively in per- sisting masses60,61. DeHertogh et al60 first reported that high-dose prednisone was more effective in recurrent or Figure 2 Common sonographic findings of IGM (AeC). (A) The refractory cases. Many clinicians recommend an initial most common finding is hypoechoic collections without well treatment of 30e60 mg prednisone orally, which is tapered defined contours; (B) skin thickening secondary to inflamma- gradually over several weeks (from 1 week to 22 tion should be compared with contralateral breast; right breast months)18,19,22,24,28,40,41,44,52,53,62. In our study, four (40%) skin was thickened compared with the left breast; and (C) cases were treated with oral prednisolone for 6 weeks at an hypoechoic fistula tracts is specific finding and should be fol- initial dose of 0.5 mg/kg/d in divided doses (4 þ 2), which lowed due to fistula formation. IGM Z idiopathic granuloma- were tapered slowly with clinical improvement. Because tous mastitis. continued high doses of steroids until complete resolution may be necessary51,60, the patients should be observed closely for side effects, such as glucose intolerance and relapsed when the steroid dose was tapered-off. Two pa- Cushing’s syndrome, as well as , avascular ne- tients developed recurrence after a complete recovery crosis, depression, and cataracts13,20,63. One should also following cessation of steroid therapy. Only one of four take into account relapses following withdrawal of the patients who was given prednisone showed complete re- therapy or tapering off the dose57,60. One of our patients covery after a course of steroid therapy, and no relapse

Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003 + MODEL Bilateral idiopathic granulomatous mastitis 7

Figure 4 The picture presents the bilateral synchronous involvement in a 34-year-old woman.

has been considered an alternative option in cases of recurrence, resistance, and side effects of predni- sone26,53,57,65. We did not prefer this medication for our patients. Complicated and resistant cases might benefit from steroids after excision19. Gurleyik et al58 recommended consecutive surgical excision of the remaining tissue after steroids and claimed that this treatment would provide better cosmesis and lower recurrence. The surgeon as well as the patient should have patience during the course of the disease because it is characterized by slow resolution and tends to gradually resolve spontaneously2. Observation without any therapy, known as expectant conservative management, is an effective treatment approach63; how- ever, close follow-up is essential. If untreated, spontaneous regression in up to half of cases has been reported8. Thus Lai et al3 reported complete disease resolution and no recurrence in 50% of their patients with expectant man- Figure 3 (A,B) MRI images of bilateral IGM. (A) Contrast nd agement. Correlatively, six (60%) of our patients underwent enhanced T1 weighted reformat substraction (2 minute) expectant conservative management and showed sponta- image shows wide asymmetrical nonmass enhancement in right neous complete recovery, as mentioned above. In the breast and retroareolar disease in left. (B) Corresponding color absence of inflammatory signs in mild disease, expectant overlay T1 weighted axial scan provided by the computer-aided management is preferred. If the condition worsens clini- diagnosis (CAD) system represents nonspecific contrast cally and radiologically, steroids are administered for 6e8 Z enhancement kinetics in both breasts. IGM idiopathic weeks at an initial dose of 0.5 mg/kg/d in divided doses Z granulomatous mastitis; MRI magnetic resonance imaging. (4 þ 2), which are tapered slowly with clinical improve- ment. As there is a strong tendency for persistence or occurred. However, no relationship was detected between recurrence, surgery should be considered in untreatable the second breast involvement and the withdrawal of the cases. steroid therapy or tapering off the dose in our patients. For follow-up, all of the patients were invited to our As steroids exacerbate infectious disease, all suspicious mastitis study group for examination once or twice per infectious etiology should be excluded before commencing week until the symptoms resolved. The disease course was steroids33,64. If there is a likelihood of a superinfection followed by routine US at 1- and 3-month intervals. After because of the presence of inflammatory signs, antibiotics significant wound healing, 6-month follow-up US is as an initial treatment may be offered until a definitive preferred. Finally, when the disease had resolved diagnosis is reached, otherwise antibiotics have no thera- completely, annual examination and screening were peutic value for true cases of IGM62. Our policy is to advised. perform surgical drainage and offer antibiotics prior to Although the diagnosis and management of IGM remain starting systemic corticosteroids if the disease is compli- challenging, interdisciplinary approaches to this disorder cated by abscess. Even though it was rarely investigated, are essential. Unnecessary intervention and delays in the immunosuppressive therapy including MTX and azathioprine diagnosis and treatment can be avoided by precise

Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003 + MODEL 8 M. Velidedeoglu et al. radiological and pathological evaluation. In conclusion, 13. Mohammed S, Statz A, Lacross JS, et al. Granulomatous bilateral IGMs have a higher rate of more relapse and mastitis: a 10 year experience from a large inner city county greater resistance to medical therapies than do unilateral hospital. J Surg Res. 2013;184:299e303. IGMs. Surgical management should be avoided unless all 14. Lin CH, Hsu CW, Tsao TY, Chou J. Idiopathic granulomatous medical treatment options have been exhausted. Never- mastitis associated with risperidone-induced hyper- prolactinemia. Diagn Pathol. 2012;7:1e6. theless, expectant management seems a rational option for 15. Asoglu O, Ozmen V, Karanlik H, et al. Feasibility of surgical the treatment of bilateral IGM. 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Please cite this article in press as: Velidedeoglu M, et al., Bilateral idiopathic granulomatous mastitis, Asian Journal of Surgery (2015), http://dx.doi.org/10.1016/j.asjsur.2015.02.003