Uncommon Location of Idiopathic Granulomatous Mastitis: A Case Report: Uncommon Location of IGM

Saba Alvand; Ali  Hessami; Leila  kiani; Ahmad  Ostadali Makhmalbaf; Ahmad Elahi

doi:10.32768/abc.202293SI320-324

Saba Alvand ⁽¹⁾, Ali Hessami ⁽²⁾, Leila kiani ⁽³⁾, Ahmad Ostadali Makhmalbaf ⁽⁴⁾, Ahmad Elahi ⁽⁵⁾

(1) School of Medicine, Tehran University of Medical Sciences, Tehran, Iran, Iran, Islamic Republic of,

(2) Mostuofi pathobiology laboratory, Tehran, Iran, Iran, Islamic Republic of,

(3) Kiani Breast Imaging Center, Tehran, Iran, Iran, Islamic Republic of,

(4) Mostuofi pathobiology laboratory, Tehran, Iran, Iran, Islamic Republic of,

(5) Department of Breast Surgical Oncology, Alborz university of Medical Sciences, Karaj, Iran ; Breast Disease Research Center, Tehran University of Medical Sciences, Tehran, Iran, Iran, Islamic Republic of

https://doi.org/10.32768/abc.202293SI320-324

Issue
Vol. 9 No. 3-SI: Special issue on Granulomatosis Mastitis, July 2022

Submitted
April 19, 2022

Published
May 6, 2022

Keywords:

Accessory breast, Axilla, Granulomatous mastitis.

PDF XML

Abstract

Background Idiopathic granulomatous mastitis (IGM) is a rare benign disease involving breast parenchyma mostly in periareolar region. Women of childbearing with recent history of pregnancy and lactation are more at risk of IGM. The common locations of IGM are retro areolar and periareolar of the breast, however involvement of axillary region has never been reported elsewhere.

Case presentation A 36-year-old female with history of two times pregnancy and lactation 8 months prior to presentation, referred with pain and swelling in the right axillary area. The past medical history and habitual history were negative and she did not use oral contraceptives or other medications. Local physical examination showed normal breasts with bilateral accessory breasts. A tender mass with the size of 4x6cm was palpable in the right axillary region accompanied by erythema and a few secretory fistulas without lymphadenopathy. Routine blood test came back negative and serum prolactin was normal. Ultra-sonography (US) demonstrated a soft tissue swelling, edema, and decreased echogenicity area in the right axillary region compatible with IGM. The patient started on prednisolone 50 mg per day, and the condition has not been improved for two months. To exclude other possible etiologies due to atypical location, the patient underwent a second US and core-needle biopsy which confirmed the diagnosis of axillary IGM. Prednisolone tapered off and a non-steroidal anti-inflammatory drug (NSAID) started. All the symptoms improved in a month and fully resolved in 3 months.

Conclusion Since IGM is not fully known yet, the presentation and the location can be variable. Considering IGM as a probable diagnosis in inflammatory presentation in the axillary region in patients with accessory breasts is suggested.

Full text article

Generated from XML file

References

Haitz K, Ly A, Smith G. Idiopathic granulomatous mastitis. Cutis. 2019 Jan;103(1):38–42.

Kessler E, Wolloch Y. Granulomatous mastitis: a lesion clinically simulating carcinoma. Am J Clin Pathol. 1972 Dec;58(6):642–6. doi: 10.1093/ajcp/58.6.642.

Grover H, Grover SB, Goyal P, Hegde R, Gupta S, Malhotra S, et al. Clinical and imaging features of idiopathic granulomatous mastitis - The diagnostic challenges and a brief review. Clin Imaging. 2021 Jan;69:126–32. doi: 10.1016/j.clinimag.2020.06.022.

Chandanwale S, Naragude P, Shetty A, Sawadkar M, Raj A, Bhide A, et al. Cytomorphological Spectrum of Granulomatous Mastitis: A Study of 33 Cases. Eur J Breast Health. 2020 Apr;16(2):146–51. doi: 10.5152/ejbh.2020.5185.

Yin Y, Liu X, Meng Q, Han X, Zhang H, Lv Y. Idiopathic Granulomatous Mastitis: Etiology, Clinical Manifestation, Diagnosis and Treatment. J Invest Surg. 2022 Mar;35(3):709–20. doi: 10.1080/08941939.2021.1894516.

Pandey TS, Mackinnon JC, Bressler L, Millar A, Marcus EE, Ganschow PS. Idiopathic granulomatous mastitis--a prospective study of 49 women and treatment outcomes with steroid therapy. Breast J. 2014 Jun;20(3):258–66. doi: 10.1111/tbj.12263.

Ozturk E, Akin M, Can MF, Ozerhan I, Kurt B, Yagci G, et al. Idiopathic granulomatous mastitis. Saudi Med J. 2009 Jan;30(1):45–9.

Centers for Disease Control and Prevention (CDC). Idiopathic granulomatous mastitis in Hispanic women - Indiana, 2006-2008. MMWR Morb Mortal Wkly Rep. 2009 Dec 4;58(47):1317–21.

Manogna P, Dev B, Joseph LD, Ramakrishnan R. Idiopathic granulomatous mastitis—our experience. Egyptian Journal of Radiology and Nuclear Medicine. 2020 Jan 15;51(1):15. doi: 10.1186/s43055-019-0126-4.

Benson JR, Dumitru D. Idiopathic granulomatous mastitis: presentation, investigation and management. Future Oncol. 2016 Jun;12(11):1381–94. doi: 10.2217/fon-2015-0038.

Wolfrum A, Kümmel S, Theuerkauf I, Pelz E, Reinisch M. Granulomatous Mastitis: A Therapeutic and Diagnostic Challenge. Breast Care (Basel). 2018 Dec;13(6):413–8. doi: 10.1159/000495146.

Bede K, Valente SA. Idiopathic granulomatous mastitis. 2020 [Internet]. 2020;4. Available from: https://abs.amegroups.com/article/view/6455

Yuan QQ, Xiao SX, Farouk O, Du YT, Sheybani F, Tan QT, et al. Management of granulomatous lobular mastitis: an international multidisciplinary consensus (2021 edition). Mil Med Res. 2022 Apr 26;9(1):20. doi: 10.1186/s40779-022-00380-5.

Martinez-Ramos D, Simon-Monterde L, Suelves-Piqueres C, Queralt-Martin R, Granel-Villach L, Laguna-Sastre JM, et al. Idiopathic granulomatous mastitis: A systematic review of 3060 patients. Breast J. 2019 Nov;25(6):1245–50. doi: 10.1111/tbj.13446.

Pfeifer JD, Barr RJ, Wick MR. Ectopic breast tissue and breast-like sweat gland metaplasias: an overlapping spectrum of lesions. J Cutan Pathol. 1999 Apr;26(4):190–6. doi: 10.1111/j.1600-0560.1999.tb01827.x.

Nakamura J, Tanaka F, Ohtaka K, Sato K, Iyama A. A Rare Case of Granulomatous Mastitis in the Accessory Axillary Breast of a Pregnant Woman Successfully Treated by Surgery. Eur J Breast Health. 2022 Apr;18(2):195–8. doi: 10.4274/ejbh.galenos.2021.2021-10-5.

Yılmaz MA, Kaya TI, Demirel M, Yuyucu Karabulut Y. Pregnancy-associated granulomatous mastitis of accessory breast: A novel clinical presentation. Dermatol Ther. 2021 Jan;34(1):e14729. doi: 10.1111/dth.14729.

Brennan ME, Morgan M, Heilat GB, Kanesalingam K. Granulomatous lobular mastitis: Clinical update and case study. Aust J Gen Pract. 2020 Feb;49(1–2):44–7. doi: 10.31128/AJGP-08-19-5042.

Alsaleh N. Assertive clinical practice in managing patients with idiopathic granulomatous mastitis: Review of literature. Ann Med Surg (Lond). 2021 Oct;70:102792. doi: 10.1016/j.amsu.2021.102792.

Kehribar DY, Duran TI, Polat AK, Ozgen M. Effectiveness of Methotrexate in Idiopathic Granulomatous Mastitis Treatment. Am J Med Sci. 2020 Nov;360(5):560–5. doi: 10.1016/j.amjms.2020.05.029.

Akbulut S, Yilmaz D, Bakir S. Methotrexate in the management of idiopathic granulomatous mastitis: review of 108 published cases and report of four cases. Breast J. 2011 Dec;17(6):661–8. doi: 10.1111/j.1524-4741.2011.01162.x.

Ringsted S, Friedman M. A rheumatologic approach to granulomatous mastitis: A case series and review of the literature. Int J Rheum Dis. 2021 Apr;24(4):526–32. doi: 10.1111/1756-185X.14065.

Kaviani A, Vasigh M, Omranipour R, Mahmoudzadeh H, Elahi A, Farivar L, et al. Idiopathic granulomatous mastitis: Looking for the most effective therapy with the least side effects according to the severity of the disease in 374 patients in Iran. Breast J. 2019 Jul;25(4):672–7. doi: 10.1111/tbj.13300.

Gupta N, Vats M, Garg M, Dahiya DS. Bilateral idiopathic granulomatous mastitis. BMJ Case Rep. 2020 Aug 31;13(8):e234979. doi: 10.1136/bcr-2020-234979.

Lai ECH, Chan WC, Ma TKF, Tang APY, Poon CSP, Leong HT. The role of conservative treatment in idiopathic granulomatous mastitis. Breast J. 2005 Dec;11(6):454–6. doi: 10.1111/j.1075-122X.2005.00127.x.

Zhou F, Liu L, Liu L, Yu L, Wang F, Xiang Y, et al. Comparison of Conservative versus Surgical Treatment Protocols in Treating Idiopathic Granulomatous Mastitis: A Meta-Analysis. Breast Care (Basel). 2020 Aug;15(4):415–20. doi: 10.1159/000503602.

Kaviani A, Vasigh M, Zand S. Idiopathic Granulomatosis Mastitis, Time to a Paradigm Change in Treatment. Iranian Quarterly Journal of Breast Disease. 2020 Jul 10;13(2):69–73. doi: 10.30699/ijbd.13.2.69.

Authors

Saba Alvand

School of Medicine, Tehran University of Medical Sciences, Tehran, Iran

https://orcid.org/0000-0002-8149-2056

Ali Hessami

Mostuofi pathobiology laboratory, Tehran, Iran

Leila kiani

Kiani Breast Imaging Center, Tehran, Iran

Ahmad Ostadali Makhmalbaf

Mostuofi pathobiology laboratory, Tehran, Iran

Ahmad Elahi

Department of Breast Surgical Oncology, Alborz university of Medical Sciences, Karaj, Iran ; Breast Disease Research Center, Tehran University of Medical Sciences, Tehran, Iran

https://orcid.org/0000-0003-2243-9578

elahi84@gmail.com (Primary Contact)

Alvand S, Hessami A, kiani L, Ostadali Makhmalbaf A, Elahi A. Uncommon Location of Idiopathic Granulomatous Mastitis: A Case Report: Uncommon Location of IGM. Arch Breast Cancer [Internet]. 2022 May 6 [cited 2024 Oct. 30];9(3-SI):320-4. Available from: https://archbreastcancer.com/index.php/abc/article/view/578

Download Citation

This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

Copyright©. This is an open-access article distributed under the terms of the Creative Commons Attribution-Non-Commercial 4.0 International License, which permits copy and redistribution of the material in any medium or format or adapt, remix, transform, and build upon the material for any purpose, except for commercial purposes.

Most read articles by the same author(s)

Ramesh Omranipour, Ahmad Elahi, Implication of Intramammary Lymph Node Metastasis in the Breast: A Review of the Literature , Archives of Breast Cancer: Vol 4, No 1: February 2017
A. Ali Assarian, Ahmad Elahi, Contralateral Axillary Lymph Node Metastasis in Breast Cancer, an Unusual Clinical Scenario: A Case Report and Review of the Literature , Archives of Breast Cancer: Vol 3, No 3: August 2016
Farhad Shahi, Mehrzad Mirzania, Mahdi Aghili, Mohammadreza Dabiri, Sharareh Seifi, Alireza Bary, Nafiseh Ansarinejad, Alireza Rezvani, Soroush Rad, Amirali Shahi, Ahmad Elahi, Ahmad Kaviani, Modifications in Breast Cancer Guidelines in COVID-19 Pandemic; An Iranian Consensus , Archives of Breast Cancer: Vol 7, No 1: February 2020
Amin Safavi, Ahmad Kaviani, Narjes Mohammadzadeh, Sanaz Zand, Ahmad Elahi, David Krag, Breast Cancer Prognostication by Pathologic Node Staging (pN-staging) System Versus Lymph Node Ratio (LNR): A Critical Review of Conflicts With Number of Nodes, Z-0011 Trial , Staging Cut-points, Neo-adjuvant Therapy, and Survival Estimation , Archives of Breast Cancer: Vol 4, No 4: November 2017
Ramesh Omranipour, Bita Eslami, Masoume Najafi, Habibollah Mahmoudzadeh , Mahtab Vasigh, Ahmad Elahi, Management of Carcinoma of Unknown Primary Involving Axillary Lymph Node (CUPAx) , Archives of Breast Cancer: Vol 6, No 3: August 2019
Ahmad Elahi, Gholamreza Toogeh, Habibollah Mahmoodzadeh, Behnaz Jahanbin, Farhad Shahi, Bita Kalaghchi, Nahid Sedighi, Ductal Carcinoma In Situ Close to the Inked Margin: A Case Presented in Multidisciplinary Session With Clinical Discussion and Decision Making , Archives of Breast Cancer: Vol 5, No 2: May 2018
Ahmad Kaviani, Amir Ashraf-Ganjouei, Sanaz Zand, Ahmad Elahi, Mahtab Vasigh, Hamid Ahmadi, Marzieh Kord Zanganeh, Samira Rahmani, Remy Salmon, Cross Oncoplastic Breast Surgery Technique; A Good Choice for Tumors Located Far From Nipple Areola Complex , Archives of Breast Cancer: Vol 6, No 3: August 2019
Marzieh Mohammadi Zavieh, Farid Azmude Ardalan, Farnaz Karimi, Nahid Sedighi, Ramesh Omranipour, Ahmad Elahi, Papillary Lesion of the Breast in a Young Girl Suspicious to Juvenile Papillomatosis, Clinical Decision Making in a Multi-disciplinary Team and Review of the Literature , Archives of Breast Cancer: Vol 7, No 3: August 2020
Forough Yazdanian, Mahtab Vasigh, Sanaz Zand, Farid Azmoudeh Ardalan, Maryam Rahmani, Negar Mashoori, Soheil Saadat, Ahmad Elahi, Idiopathic Granulomatous Mastitis; Radio-pathologic findings and Grading based on Clinical Presentation and treatment outcome in 224 patients , Archives of Breast Cancer: Vol. 9 No. 3-SI: Special issue on Granulomatosis Mastitis, July 2022
Mahsa Tavakol, Saba Alvand, Farid Azmoudeh Ardalan, Abdolali Assarian, Idiopathic Granulomatous Mastitis with Incidental Lobular Carcinoma In Situ: A Case Report , Archives of Breast Cancer: Vol. 9 No. 3-SI: Special issue on Granulomatosis Mastitis, July 2022

INTRODUCTION

Granulomatous mastitis (GM) is a rare benign nonnecrotizing granulomatous condition that involves breast parenchyma, which is grouped into two main categories of primary or idiopathic (IGM) and secondary. Secondary GM can be due to infections like histoplasmosis, actinomycosis, and autoimmune diseases such as sarcoidosis, granulomatosis with polyangiitis, IgG4-RD mastitis, foreign body reaction, and fat necrosis. 1 IGM was firstly introduced by Kessler and Wolloch in 1972. 2,3 Only 0.44-1.6% of the breast biopsies are diagnosed with IGM. 2 It mostly involves women of childbearing age and occurs a few years after pregnancy and lactation. 3 Although the exact pathology is not well-known, there are observations suggesting that Hispanic origins and people from middle-east are predisposed to developing IGM. 4 5 6 The pathogenesis may be due to autoimmunity, infection, hormonal imbalance, smoking, antitrypsin deficiency, and oral contraceptives; thus, the proper exclusion of possible etiologies should be taken into account. 4 The incidence of IGM has been reported to be 0.37% in the United States, but unfortunately there are no clear reports about the incidence in the Middle east. 7 IGM mostly presents as a tender unilateral mass in the retro areolar of the breast; bilateral masses are rare. Erythema, skin changes, nipple contraction, abscess, fistula, and lymphadenopathy may also be present. 8 9 10 11 Early diagnosis of IGM is important because of its high similarity with breast cancer. 10 Because of similar findings of breast cancer and tuberculosis on presentation and radiological assessments, histological investigation is required. Besides, the secondary GM should be excluded via history and paraclinical assessment, making the final diagnosis faster. 1 An international multidisciplinary consensus in 2021 agreed on comprehensive workup by clinicians, radiologists and pathologists and the use of history taking, physical examination, imaging and laboratory modalities to finalize the diagnosis. 12 The delay in diagnosis on average of 4-5 months is common and reported in a systematic review on 3060 cases. 13 In this case report, we present an unusual location of IGM in the accessory breast in the axilla region, which delayed the process of diagnosis.

CASE PRESENTATION

A 36-year-old female presented with pain and swelling in the right axillary area for 3 months. She had two children and breastfed both of them for two years for 8 months prior the presentation. She denied any other specific disease in the past medical history and did not have a family history of breast cancer. She had never used alcohol and cigarettes and did not use oral contraceptives and other medications.

The systemic examination was unremarkable. On local physical examination, the left breast was intact and bilateral accessory breasts were prominent. A tender mass with ill-defined borders of an approximate of 4x6cm was palpable in the right axillary region accompanied by erythema and a few secretory fistulas. Lymph nodes were not palpable. Figure 1 demonstrates the lesion.

In the blood test, the cell blood count, fasting blood glucose, HbA1C, and serum prolactin were normal. In bilateral full digital mammography, focal asymmetry was seen at the right axillary region which required ultra-sonography (US) for better assessment. The US at the time of presentation found soft tissue swelling, edema, and a decreased echogenicity area in the right axillary region without detectable fluid collection and abscess, suggesting subacute cellulitis. The core needle biopsy was performed showing a breast tissue in the axilla with an inflammatory process containing mixed infiltrates of inflammatory cells as well as granuloma formation which was suggestive of granulomatous mastitis. The specimens were then sent for antibiogram and culture in growth media and PCR for TB and fungus in another test tube. The growth media did not detect bacterial colonization, and PCR for TB and fungus were also negative. With an impression of IGM, prednisolone 50mg per day was prescribed for the patient. In the 2-month follow-up, the condition did not improved. Since the location of the lesion was atypical for IGM and the patient did not respond to prednisolone, another US and a core-needle biopsy were performed to exclude other possible etiologies. The second US reported a 42x15mm hetero echoic structure thick collection in the right axillary region associated with skin thickening which was consistent with focal granulomatous mastitis with BIRAD III (Figure 2). A core-needle biopsy reported similar findings to the previous biopsy which was suggestive of lobar (idiopathic) granulomatous mastitis ( Figures 3&4).

With the diagnosis of corticosteroid resistant IGM, the prednisolone tapered off and naproxen 500mg was prescribed twice per day. After a month, the erythema and secretion decreased and the mass became softer than the first presentation. In the 3-month follow-up, the erythema and mass completely resolved, and the scars of previous fistulas appeared; thus, naproxen was discontinued. Nine months after the full remission, the patient was followed for the possible relapse. The breast was intact with normal shoulder movement and scars of resolved fistulas.

DISCUSSION

In this case report, we presented an unusual location of IGM, which was not responsive to a twomonth consumption of steroids. Although IGM is mostly present in the periareolar of the breast, the involvement of other locations especially in presence of accessory breasts is not unexpected. Cancer, mastitis, fibroadenoma, phyllodes tumor, and fibrocystic change are the most common diseases that can involve accessory breasts, but IGM should be kept in mind as a possible disease as well. 14 Two case reports have presented IGM in accessory breast tissue in axilla recently, but both cases were in pregnant women. 15 16 Physicians should also consider that suppurative hidradenitis can mimic the same presentation with IGM in the axillary region. Moreover, our patient has a normal prolactin level, which suggests the upregulation of prolactin receptors or their hypersensitivity to prolactin in this case. 12 IGM is a self-limited disease that resolves within 2 years, but the long period of convalescence is disturbing to many patients. 10 11 17 To date, treatment options have been developed and improved in recent years. Observation is the first approach to treatment in IGM patients. While many clinicians prefer medications for severe and prolonged cases in the first place and start the treatment with oral steroids. The presence of fistula in our patient suggested an advanced disease which required treatment and thus started on corticosteroids. 12 Although steroids can be effective in nearly 80% of the cases, other patients may require alternative treatments. Immunosuppressive agents such as Methotrexate and Azathioprine are the two most common medications in unresponsive cases or patients who have developed complications due to steroids. 18 A study found a combination therapy of methotrexate and steroids with better response in IGM patients than steroids alone. 19 Also, Methotrexate had a high effectiveness methotrexate in patients resistant to corticosteroids. 20 The rate of remission with methotrexate is estimated from 58% to 100% in cases with other autoimmune presentations plus IGM. 21 The response to steroids and immunosuppressor agents is another indication that IGM is an autoimmune disease; however, the potential adverse effects and the need for close monitoring in treatment with immunosuppressive agents discourage some physicians from using them. 3 To date, non-steroidal anti-inflammatory drugs (NSAIDs) have been prescribed for IGM in recent years and a good response has been observed in a study on 374 patients in Iran. 22 In the presented patient, the undesired complications of immunosuppressors make her refuse to take immunosuppressors. Due to the success of NSAID in the mentioned study on Iranians, we chose to put the patients on NSAID. We found the same result in the mentioned case. In a meta-analysis, surgery as a less favorable treatment option in IGM was not found to be different in relapse rate from nonsurgical treatment. Fistula formation, late wound healing, and disfigurement are also some other possible complications. 23 24 25 Patients with IGM can experience several relapses for a long time; thus, using medications like NSAIDs with fewer complications than surgery and other non-surgical options should be taken into account, making it a priority especially in patients with limited access to health facilities to monitor the possible complications of medications, and also reluctant to take immunosuppressors. 26

CONCLUSION

In conclusion, IGM is a rare inflammatory disease. IGM should be considered as a probable diagnosis in inflammatory presentations in uncommon regions like milk line due to the existence of accessory breasts and can facilitate diagnosis in unusual presentations.

ETHICAL CONSIDERATIONS

The patient has provided written informed consent to the publication of this case (including the publication of images).

Right axillary region with secretory fistulas

US of the right axillary region

A granulomatous reaction in lobular granulomatous mastitis x400 Hematoxylin Eosin stain

Lobulocenteric pattern of inflammation in lobular granulomatous mastitis x200 Hematoxylin Eosin stain

Read Counter : 416 Download : 17

Share Now

Uncommon Location of Idiopathic Granulomatous Mastitis: A Case Report Uncommon Location of IGM

Abstract

Full text article

References

Authors

Most read articles by the same author(s)

Important Links

Contact Info

Article Sidebar

Abstract

Full text article

References

Authors

Article Details

Most read articles by the same author(s)

Important Links

Contact Info