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in vivo 30 : 727-732 (2016) doi:10.21873/invivo.10987 Review Plasma Cell Mastitis in Men: A Single-center Experience and Review of the Literature ANDREA PALMIERI 1, VALERIO D’ORAZI 2, GIOVANNI MARTINO 1, FEDERICO FRUSONE 1, DANIELE CROCETTI 3, MARIA IDA AMABILE 1 and MARCO MONTI 4 1Department of Surgical Sciences, 3Pietro Valdoni Department of Surgery, and 4Department of Gynecological, Obstetrics and Urologic Sciences, Sapienza University of Rome, Rome, Italy; 2Department of General Microsurgery and Hand Surgery, Fabia Mater Hospital, Rome, Italy Abstract. Plasma cell mastitis is an inflammatory disease of in men was documented in 1974 by Tedeschi and Mc Carthy the breast parenchyma, rare in males. In the last 40 years, few (9). Our recent observation and treatment of male patients cases have been described in literature. Our recent treatment affected by plasma cell mastitis raised a series of diagnostic, of male patients affected by plasma cell mastitis raised a series etiological and therapeutic issues persuading us to carry out of issues which led us to carry out a critical review of the a critical review of the literature. literature. Plasma cell mastitis is often not well defined and is difficult to assess by clinical examination and radiological Our Single-center Experience investigation alone. An understanding of the pathogenesis and the mechanisms behind plasma cell mastitis may help improve Out of a total of 411 breast surgeries performed between the diagnostic and therapeutic course of the disease, leading 2011 and 2015 at the Department of Surgical Sciences, to a more targeted and less invasive treatment. Sapienza University of Rome, Italy, we recorded six cases of breast surgeries in men (1.46% of the total). In three cases Plasma cell mastitis is an inflammatory disease of the breast out of these six (0.73% of the total), the final histological parenchyma, frequent in females, characterized by a diagnosis was gynecomastia, in one case it was ductal periductal phlogistic reaction, accompanied by ductal ectasia. carcinoma, and in two cases (0.48% of the total) the final It was described for the first time by Burkitt in 1850 (1). In histological diagnosis was plasma cell mastitis. the literature, this disease is also known as granulomatous The first case of plasma cell mastitis concerned a 56-year- mastitis, obstructing mastitis, comedo mastitis (2), periductal old man who complained of the appearance of a hard mastitis (3, 4), and mammary duct ectasia (5-7). Plasma cell retroareolar lump, with smooth edges, which was quite mastitis is more frequent in young women and in women painful, and without nipple secretions. A bilateral breast who have not had pregnancy nor breastfed (8). It is ultrasound examination was performed which showed a solid, characterized by occasional mastodynia with ductal ectasia non-homogeneous lesion, with irregular edges and diameter and usually presents as a retroareolar lump with inverted of 17 mm (Figure 1). An ultrasound-assisted core biopsy was nipple and secretions. The most common complication is the performed due to suspicion of a neoplastic lesion, with the onset of a fistula (7). The first case of plasma cell mastitis aim of characterizing the lesion. The histological examination revealed intense chronic inflammation, mostly plasmacellular, and focal steatonecrosis, without neoplastic cells. Therefore, the patient did not undergo surgery. An appropriate anti- Τhis article is freely accessible online. inflammatory therapy was performed and the patient healed in about 3 weeks. At further follow-up evaluation, no signs Correspondence to: Maria Ida Amabile, MD, Ph.D., Department of of breast disease were observed, as previously described (10). Surgical Sciences Sapienza University of Rome, Viale Regina Elena The second case concerned an otherwise healthy 76-year- 324, 00161 Rome, Italy. Tel/Fax: +39 0649972449, e-mail: [email protected] old man who reported the presence of a hard retroareolar lump, which was quite painful, without nipple secretions. The Key Words: Plasma cell mastitis, male breast, periductal phlogistic breast ultrasound examination showed a hypoechoic lesion reaction, review. with poorly defined edges and perilesional/ intralesional 0258-851X/2016 $2.00+.40 727 in vivo 30 : 727-732 (2016) Figure 1. Ultrasound examination of the left breast revealing the presence of a retroareolar solid lump with a diameter of approximately 17 mm. vascularization. Ultrasound-assisted fine-needle aspiration of the terminal ducts (12). In 1984, Chan and Lau described cytology obtained hematic material which was not useful in the case of a patient who had a breast lump treated with diagnosis. The patient refused any other invasive excision of the nodule (13), and in 1985 Ashworth et al. investigations and an excisional open biopsy was performed presented a similar case, but treated with excision of the major by periareolar access, without the need to position a metallic ducts (14). In 1996, Downs and Fisher described two cases of guide wire, as it is necessary instead in cases of female breast breast lumps in human immunodeficiency virus-positive lesions with similar dimensions and characteristics (11). Entire patients, the first treated with excisional biopsy, while the excision of the lump was carried out. The postoperative second with antibiotic therapy (15). In 2001, Al-Masad definitive histological examination documented a diagnosis of documented two cases treated with excisional biopsy, one of plasma cell mastitis. No further therapies were necessary. which was concomitant with Behçet's disease, suggesting that In both cases, further investigations were accomplished in some cases of plasma cell mastitis are a sign of an underlying order to exclude plasma cell mastitis as being an epiphenomenon autoimmune disease (16). In 2005, Reddy et al. described the of an unknown autoimmune disease. The immunological profile case of a man with a retroareolar breast lump who underwent was within the normal range in both cases. a fine-needle aspiration and, subsequently, an excisional biopsy (17). In 2011, Gupta et al. treated a man with a breast Review of the Literature lump, which was initially thought to be a lobular carcinoma, after cytological examination by means of fine-needle Plasma cell mastitis is a disease relatively common in aspiration, and therefore treated with a simple mastectomy women, but quite rare in men. This is probably why between (18). The definitive histological examination, however, 1974 and 2016, only 14 cases have been described in the showed the presence of plasma cell mastitis, as in all of the literature, as can be seen in Table I. previous cases. In the case dating back to 2012, described by The first case of plasma cell mastitis described in a man Kim et al. , a documented retroareolar breast lump in a 37- dates back to 1974, and referred to a 57-year-old man with a year-old man was hypothesized to be breast carcinoma (19). lump of the left breast, who underwent a simple mastectomy The patient underwent mammography, breast ultrasound, and (9). In 1979, Mansel and Morgan documented three cases of subsequently an ultrasound-guided biopsy, the histological plasma cell mastitis (12). In the first, a breast lump with examination of which gave a diagnosis of plasma cell mastitis. inverted nipple and secretion was treated with an attempt at drainage and, subsequently, with a total ductectomy. In the Discussion second case, the patient had recurrent abscesses of the right breast that were treated initially with a ductectomy, and then It is difficult to differentiate between plasma cell mastitis, with a simple mastectomy (12). In the third case, a breast described for the first time during the last century (1) and lump was treated with subcutaneous mastectomy and excision later better defined by Bloodgood in 1921 (20) and by 728 Palmieri et al : Plasma Cell Mastitis in Men (Review) Table I. Literature review and patient characteristics. Author (Ref) Cases Patient Diagnostic Histology Therapy procedure Tedeschi and Mc Case 1 57-Year-old Excisional Chronic inflammatory infiltrate; Simple mastectomy Carthy (1 case) (9) man with left biopsy lymphocytes and plasma cells with breast lump desquamation of the duct epithelium Mansel and Morgan Case 2 62-Year-old man Mammography, Bilateral chronic inflammatory Bilateral drainage of (3 cases) (12) with left breast biopsy, infiltrate with abscess evolution the breast abscess with nodule, inverted microbial total ductectomy nipples and cultural discontinuous examination of secretion the secretion Case 3 47-Year-old man Cytological Chronic inflammatory infiltrate with Drainage of the abscess, with recurrent examination of marked focal granulomatous reaction total duct excision, right breast nipple secretion, repeated incisions and abscesses biopsy drainage, simple mastectomy Case 4 52-Year-old man Cytological Chronic inflammatory infiltrate Subcutaneous mastectomy with left examination of nipple with terminal duct excision breast lump secretion, biopsy Chan and Lau Case 5 34-Year-old man Biopsy Inflammatory infiltrate with Excision of the nodule (1 case) (13) with right lymphocytes and plasma cells breast lump Ashworth et al. Case 6 50-Year-old man Mammography, Inflammatory infiltrate with Excision of the major ducts (1 case) (14) with right biopsy lymphocytes and plasma cells breast lump Downs et al. Case 7 30-Year-old Fine-needle Mostly plasmacellular chronic Excisional biopsy (2 cases) (15) HIV-positive man aspiration