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Idiopathic Granulomatous Mastitis

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Idiopathic Granulomatous Mastitis CASE REPORT Idiopathic Granulomatous Mastitis Karyn Haitz, MD; Amy Ly, MD; Gideon Smith, MD, PhD that hyperprolactinemia2 or an immune response to local PRACTICE POINTS lobular secretions might play a role in pathogenesis. Early • Idiopathic granulomatous mastitis (IGM) is a painful misdiagnosis as bacterial mastitis is common, prompting and scarring rare granulomatous breast disorder that multiple antibiotic regimens. When antibiotics fail, patients can have a prolonged time to diagnosis that delays are worked up for inflammatory breast cancer, given the proper treatment. nonhealing breast nodules. Mammography, ultrasonog- • The pathogenesis of IGM remains poorly understood. raphy, and fine-needle aspiration often are unable to rule The temporal association of the disorder with breast- out carcinoma, warranting excisional biopsies of nodules. feeding suggests that hyperprolactinemia or an immune The patient is then referred to rheumatology for potential response to local lobular secretions might play a role. sarcoidosis or to dermatology for IGM. In either case, the • Although many cases of IGM resolve without treat- workup should be similar, but additional history focused ment, more severe cases can persist for a long on behavior and medications is essential in suspected IGM, period before adequate symptomatic treatment is copy given the association with hyperprolactinemia. provided with methotrexate, corticosteroids, or surgi- Because IGM is rare, there are no randomized, cal excision. placebo-controlled trials of treatment efficacy. In many • Before any of these therapies are applied, however, contributing factors, such as long-term breastfeeding cases,not patients undergo complete mastectomy, which and drugs that induce hyperprolactinemia, should be is curative but may be psychologically and physically identified and withdrawn. impactful in young women. In some cases, high-dose cor- ticosteroids have been successful; however, because the IGM process can last longer than 2 years, patients treated Doin this manner are exposed to steroid morbidities.1 Idiopathic granulomatous mastitis (IGM) is a rare, poorly understood We report 3 cases of IGM that add to the literature condition that presents as inflammatory nodules of the breast. It is on possible contributing factors, clinical presentations, often initially misdiagnosed as furunculosis or cellulitis. Despite the and treatments for this disease. We also demonstrate painful, scarring, and debilitating nature of the disease, patients often have a delay in accurate diagnosis and treatment. Even when IGM is that appropriate trigger identification and steroid-sparing considered as a diagnosis, it is one of exclusion, with the differential agents, specifically methotrexate, can be breast-saving as diagnosis including serious conditions such as breast cancer, sar- they can alleviate this debilitating condition, obviating the coidosis, and cutaneous tuberculosis. Therefore, appropriate workup need for radical surgical intervention. is important. Given that IGM is a diseaseCUTIS of the skin, it is important for dermatologists to be familiar with its presentation. We describe CASE REPORTS 3 cases of young women with this condition and demonstrate that identification of triggers or methotrexate treatment is highly success- Patient 1 ful in sparing patients the drastic surgical alternative of mastectomy. A 40-year-old woman with a 4-year history of breastfeed- Cutis. 2019;103:38-42. ing noted a grape-sized nodule on the left breast that grew to the size of a grapefruit after 2 weeks. Ulceration and drainage periodically occurred, forming pink plaques diopathic granulomatous mastitis (IGM) is rare during along the lateral aspects of the breast after healing. Her pregnancy; it typically is seen in women of childbearing primary care provider suspected infectious mastitis; she Ipotential from 6 months to 6 years postpartum.1 Because was given an oral antibiotic (cephalexin) and intravenous of a temporal association with breastfeeding, it is believed antibiotics without improvement. Dr. Haitz is from the Department of Dermatology and Cutaneous Surgery, University of Miami, Florida. Drs. Ly and Smith are from Massachusetts General Hospital, Boston. Dr. Ly is from the Department of Pathology, and Dr. Smith is from the Department of Dermatology. The authors report no conflict of interest. The eTable is available in the Appendix online at www.mdedge.com/cutis. Correspondence: Karyn Haitz, MD, University of Miami, Department of Dermatology and Cutaneous Surgery, 1600 NW 10th Ave, RMSB 2023A, Miami, FL 33136 ([email protected]). 38 I CUTIS® WWW.MDEDGE.COM/CUTIS Copyright Cutis 2019. No part of this publication may be reproduced, stored, or transmitted without the prior written permission of the Publisher. IDIOPATHIC GRANULOMATOUS MASTITIS Imaging—Subsequent magnetic resonance imaging peau d’orange change to the epidermis (Figure 2A). Based revealed a large, irregular, enhancing mass within the on pathologic analysis, she was worked up for a possible outer left breast (6.5 cm at greatest dimension) with granulomatous etiology. Negative purified protein deriva- additional surrounding amorphous enhancement highly tive (tuberculin)(PPD) and a normal chest radiograph suspicious for malignancy. There also were multiple ruled out tuberculosis. Normal chest radiography, serum prominent left axillary lymph nodes, with the largest Ca2+ and angiotensin-converting enzyme (ACE) levels, demonstrating a cortical thickness of 8 mm. and ophthalmology examination ruled out sarcoidosis. Biopsy—Core breast biopsy showed benign tissue The patient reported she continued breastfeeding her with fat necrosis. Fine-needle aspiration revealed few 4-year-old son. Additionally, she had been started on benign ductal cells and rare histiocytes; because these trazadone and buspirone for alcohol abuse recovery, then findings were nondiagnostic and cancer was still a con- switched to and maintained on fluoxetine 1 year before sideration, the patient underwent excisional biopsy. developing these symptoms. Histologic sections of breast tissue showed exten- Buspirone, fluoxetine, and prolonged breastfeeding sive lobulocentric inflammation comprising histiocytes all contribute to hyperprolactinemia, a possible trigger of and lymphocytes, with neutrophils admixed and forming IGM. The patient was therefore advised to stop breast- microabscesses (Figure 1A). Multinucleated giant cells and feeding and to be switched from fluoxetine to a medica- single-cell necrosis were seen, but true caseous necro- tion that would not increase the prolactin level. She did sis was absent (Figure 1B). Duct spaces often contained not require methotrexate treatment because her condi- inflammatory cells or secretions. Special stains for fungal tion resolved rapidly after breastfeeding and fluoxetine and acid-fast bacterial microorganisms were negative. were discontinued (Figure 2B). Referral to Dermatology—Granulomatous lobular mas- titis was diagnosed, and the patient was referred to der- Patient 2 matology. On presentation to dermatology, the left breast A 40-year-old womancopy with no history of breastfeeding showed a 6-cm area of firm induration and overlying who gave birth 4.5 years prior presented to her primary care provider with a painful breast lump and rash on the right breast of 2 months’ duration. Infectious mastitis was not Do A CUTIS A B B FIGURE 1. A, Histopathology showed breast tissue with inflamma- tion in a lobulocentric distribution (H&E, original magnification ×40). FIGURE 2. A, Firm induration and overlying peau d’orange change B, Neutrophilic microabscess (left side) and multinucleated cells also to the epidermis at presentation to the dermatology department. were seen (H&E, original magnification ×400). B, Resolution after discontinuation of breastfeeding and fluoxetine. WWW.MDEDGE.COM/CUTIS VOL. 103 NO. 1 I JANUARY 2019 39 Copyright Cutis 2019. No part of this publication may be reproduced, stored, or transmitted without the prior written permission of the Publisher. IDIOPATHIC GRANULOMATOUS MASTITIS suspected; she was given cephalexin and clindamycin 8 months compared to prior unremitting pain and drain- without improvement of symptoms. age. She was then tapered from methotrexate over Imaging—Mammography and ultrasonography 6 weeks without additional flares. were nondiagnostic. Biopsy—Breast biopsy showed tissue with large Patient 3 expanses of histiocytes, neutrophils, lymphocytes, plasma A 27-year-old woman who gave birth 2 years prior and cells, and multinucleated giant cells (Figure 3A). Many discontinued breastfeeding 6 weeks after delivery noted discrete granulomas were seen against this mixed inflam- bilateral breast rashes for several months. The lesions matory background, associated with focal fat necrosis were growing in size, tender, and draining. Her primary (Figure 3B). Special stains were negative for microorgan- care provider suspected infectious mastitis and prescribed isms. Histologic findings were consistent with granulo- antibiotics, which were ineffective. matous mastitis. Biopsy—Breast core biopsy showed histologic findings Referral to Dermatology—On presentation to dermatol- similar to patients 1 and 2, including lobulocentric mixed ogy, the patient was worked up for a possible granuloma- inflammation, neutrophilic microabscesses, and scattered tous etiology, which included a negative PPD, as well as a discrete granulomas. Microorganisms were not found normal chest radiograph, serum Ca2+ and
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