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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 50  |  Issue : 5  |  Page : 183-185

Ductal carcinoma in situ in a 35-year-old male presenting with ipslateral gynecomastia


1 Department of Surgery, Lin Shin General Hospital, Taichung City, Taiwan
2 Department of Pathology, Lin Shin General Hospital, Taichung City, Taiwan
3 Department of Surgery, Chern Ching General Hospital, Taichung, Taiwan

Date of Submission09-Jan-2017
Date of Decision15-Feb-2017
Date of Acceptance12-Mar-2017
Date of Web Publication9-Oct-2017

Correspondence Address:
Hsien-Pin Sun
Department of Surgery, Chern Ching General Hospital, Taichung City
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/fjs.fjs_1_17

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  Abstract 


Concomitant ductal carcinoma in situ (DCIS) and ipslateral gynecomastia (GM) are very rarely reported in male <40. We report an unusual case of a 35-year-old male, with no familial history of breast cancer, presented with unilateral GM, diffuse type. After subcutaneous mastectomy for his unilateral GM, a 0.7 cm × 0.3 cm DCIS lesion was incidentally found without clinical symptom. About 14%–43% of patients with DCIS have the potential of evolving into an invasive tumor in the subsequent 10–20 years. Our case emphasizes the rare incidence of DCIS in male breast and the presence of concurrent GM can frequently result in misdiagnosis. In patients with rapid growth in breast tissue should be cautious of cancer in young men with GM.

Keywords: Ductal carcinoma in situ, male breast carcinoma, ipslateral gynecomastia


How to cite this article:
Huang CJ, Chen JP, Lin HJ, Sun HP. Ductal carcinoma in situ in a 35-year-old male presenting with ipslateral gynecomastia. Formos J Surg 2017;50:183-5

How to cite this URL:
Huang CJ, Chen JP, Lin HJ, Sun HP. Ductal carcinoma in situ in a 35-year-old male presenting with ipslateral gynecomastia. Formos J Surg [serial online] 2017 [cited 2022 May 26];50:183-5. Available from: https://www.e-fjs.org/text.asp?2017/50/5/183/216231




  Introduction Top


Gynecomastia (GM), defined as a benign proliferation of male breast glandular tissue, is usually caused by increased estrogen activity, decreased testosterone activity, or the use of numerous medications. Asymptomatic GM is very common and has a trimodal age distribution, occurring in neonatal, pubertal, and elderly males. GM is usually bilateral at clinical presentation and less common in unilateral.[1] GM implies an imbalance between androgen and estrogen and may be a predisposing factor of male breast carcinoma.[2] Although most male breast carcinomas are clinically apparent, distinguishing early breast cancer from GM is considered a difficult task. Ductal carcinoma in situ (DCIS) in male patients is found in 5% of all male breast malignancy and associated with GM in 2%–11%.[3] Synchronous DCIS and GM in the young adult population are very rare. To the best of our knowledge, only 14 cases, < than 40 years, with DCIS and concurrent GM were described in literature.[2],[4],[5] Here, we present a 35-year-old male patient with the coexistence of ipslateral GM and DCIS with no familial history of breast malignancy.


  Case Report Top


A 35-year-old male had a 2-year history of a painful lump in the periareolar region on the right breast with no history of hormonal medications. He was otherwise fit and healthy with negative familial history for breast cancer. Physical examination noted that the right breast was larger than the left one and no axillary lymphadenopathy. Breast ultrasound reported a diffuse type GM pattern on the right side [Figure 1]a and normal pattern on the left side [Figure 1]b. A right subcutaneous mastectomy was planned and performed. A 7.7 cm × 7 cm × 2.4 cm, 68 g, breast tissue was excised. Histological examination revealed epithelial intraductal hyperplasia with the proliferation of periductal stromal fibrosis, consistency with GM. In addition, a 0.7 cm × 0.3 cm lesion with Grade 2/3 DCIS, papillary, and cribriform pattern [Figure 2]a,[Figure 2]b,[Figure 2]c was found in the resected breast tissue incidentally. No invasive cancer was seen and the resection margin was <1 cm. The myoepithelial stain (p63) revealed nearly total loss in the proliferation area and the outer rim of myoepithelial cells was preserved [Figure 2]d. Estrogen receptor stain revealed 95% staining [Figure 2]e.
Figure 1: (a) 1.09 cm of anterior-posterior depth with mixed echogenic breast tissue consistent with diffuse type gynecomastia on the right breast. (b) Ultrasound study showed no gynecomastia on the left breast

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Figure 2: (a) Section show breast tissue with ductal expansion with monotonous ductal epithelial cells proliferation, involving at least three ducts, and gynecomastia with epithelial intraductal hyperplasia and proliferation of periductal collangenous connective tissue (×40). (b) Monotonous cells proliferation form cribriform and papillary structures in which moderate pleomorphism and mild cellular atypia are noted (×100). (c) Close up shows moderate pleomorphism and mild cellular atypia (×400). (d) The myoepithelial stain (p63) reveals nearly total loss in the proliferation. However, the outer rim of myoepithelial cells is preserved. A diagnosis of ductal carcinoma in situ is confirmed (×100). (e) ER stain reveals 95% staining (×100)

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The plan of treatment for this patient was discussed in our breast multidisciplinary team meeting, and the need for removing the nipple areolar complex, local radiotherapy on areolar region, or adjuvant hormonal therapy was suggested to reduce the risk of local tumor recurrence.


  Discussion Top


GM is enlarged male breast resulting from the proliferation of glandular component of the breast, triggered by conditions that block the effects of testosterone or increase the effects of estrogen. Physiological GM occurs in neonates, at puberty, and with obesity and aging. Several other things can also upset the hormone balance, such as antianxiety medications, alcohol, and certain health conditions, including liver and kidney disease, hormonally active tumors, and Klinefelter's syndrome. Three patterns of GM have been described: nodular type (early and reversible phase, characterized by increase number of ducts and epithelial proliferation with loose cellular stroma and edematous changes), dendritic type (fibrotic phase, characterized by dilated duct, moderate epithelial proliferation, and fibrosis), and diffuse type (proliferative changes with lobule formation).[6] In the diffuse type, ultrasound shows an anterior-posterior breast tissue depth in the subareolar region more than 1 cm as our presented case.

Men with GM may suffer from absolute or relative estrogen excess and their risk of different malignancies increase.[7] However, a study by Lapid et al. analyzed the total of 5113 histopathology results of surgically excised breast specimens with the diagnosis of GM showed an overall prevalence of invasive carcinomas as only 0.11% and of in situ carcinomas, 0.18%. The prevalence of malignancies in GM resection specimens was very low.[8] Another study by Senger et al. also revealed no significant pathological findings in GM resection specimens by a cohort study of 452 cases with 2178 slides. The authors suggested that the routine pathological evaluation of tissue from GM might not be necessary for not increasing further on the current health-care budget.[9]

Generally speaking, the prevalence of malignancies in GM resection specimens is low and whether GM associates a significant risk factor for breast malignancy or not is still controversial.[2],[10] The differential diagnosis between the GM and breast cancer in men or the exclusion of concurrent cancer lesion with GM represents the primary aim for the clinician in daily practice.

Male breast cancer is estimated as 1% of all breast carcinomas and 0.17% of all cancer cases in men. The rare DCIS of male breast represents approximately only 5% (reported range, 0.5%–17%) of all male breast carcinomas.[3] Due to the male breast characteristically lacks lobules (terminal duct lobular units) in which a majority of DCIS cases originate in the female breast, DCIS in the male breast displays significantly different distribution of morphologic subtypes compared with its female counterpart; most common in intraductal papillary type, 46%; less common in papillary with a cribriform pattern, 27%. The majority of pure DCIS of men, 57%, are found to be low grade (Grade 1, no cellular atypia, no necrosis), 43% DCIS of men are intermediate grade (Grade 2, mild to moderate cellular atypia with or without necrosis). High-grade DCIS (Grade 3, severe cellular atypia with or without necrosis) appears to be a very rare lesion in the pure DCIS of male breast except in patients with invasive ductal cancer.[3]

The tumor grades were low or intermediate in the all these 14 cases with GM and concomitant DCIS, <40-year-old, collected from the literatures and our presented patient. It was interesting to find in these 14 cases that at least three cases had bilateral GM and synchronous bilateral DCIS.[2],[4],[5] These 14 cases including, our presented patient, all had no clinical symptom related to DCIS. Most of these 14 cases with DCIS were bilateral GM, and unilateral GM was seen only in one case that had a strong familial history of breast malignant disease. It was different from our patient with ipslateral GM and no familial history of breast malignant disease.

Due to the very small number of cases of men with DCIS and a limited number of literature with the management of male breast cancer, the management of DCIS in male patients is based on the evidence derived from the data from female patients. Therefore, the DCIS in male is expected to have a good prognosis with simple mastectomy only and no axillary sentinel node biopsy or chemotherapy needed as in the female patient. Radiotherapy may be recommended for a male patient with DCIS treated by lumpectomy or patients with involved resected margin for reducing the local recurrence. Preservation of nipple areolar complex by subcutaneous mastectomy or nipple sparing mastectomy may show a long-term low local recurrence with a clear retroareolar resection margin in male patients with DCIS.

We described a very rare case of concurrent ipslateral GM and incidental DCIS lesion of a young adult male with no familial history of breast cancer. Eventually, the male and female patients with DCIS, 14%–43%, may develop invasive cancer in the subsequent 10–20 years [11] and the DCIS incidence in the general population may be higher than predicted. Our case emphasizes the rare incidence of DCIS in male breast and the presence of concurrent GM can frequently result in misdiagnosis. In patients with rapid growth in breast tissue necessitate consideration of diagnoses beyond GM and should further investigate completely in patients with a familial history of breast malignancy.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Bembo SA, Carlson HE. Gynecomastia: Its features, and when and how to treat it. Cleve Clin J Med 2004;71:511-7.  Back to cited text no. 1
    
2.
Shirah BH, Shirah HA. Incidental unilateral and bilateral ductal carcinoma in situ encountered in the surgical management of young male gynecomastia. Breast Dis 2016;36:103-10.  Back to cited text no. 2
    
3.
Hittmair AP, Lininger RA, Tavassoli FA. Ductal carcinoma in situ (DCIS) in the male breast: A morphologic study of 84 cases of pure DCIS and 30 cases of DCIS associated with invasive carcinoma – a preliminary report. Cancer 1998;83:2139-49.  Back to cited text no. 3
    
4.
Cutuli B, Dilhuydy JM, De Lafontan B, Berlie J, Lacroze M, Lesaunier F, et al. Ductal carcinoma in situ of the male breast. Analysis of 31 cases. Eur J Cancer 1997;33:35-8.  Back to cited text no. 4
    
5.
Coroneos CJ, Hamm C. Ductal carcinoma in situ in a 25-year-old man presenting with apparent unilateral gynecomastia. Curr Oncol 2010;17:133-7.  Back to cited text no. 5
    
6.
Michels LG, Gold RH, Arndt RD. Radiography of gynecomastia and other disorders of the male breast. Radiology 1977;122:117-22.  Back to cited text no. 6
    
7.
Volpe CM, Raffetto JD, Collure DW, Hoover EL, Doerr RJ. Unilateral male breast masses: Cancer risk and their evaluation and management. Am Surg 1999;65:250-3.  Back to cited text no. 7
    
8.
Lapid O, Jolink F, Meijer SL. Pathological findings in gynecomastia: Analysis of 5113 breasts. Ann Plast Surg 2015;74:163-6.  Back to cited text no. 8
    
9.
Senger JL, Chandran G, Kanthan R. Is routine pathological evaluation of tissue from gynecomastia necessary? A 15-year retrospective pathological and literature review. Ann Plast Surg 2014;22:112-6.  Back to cited text no. 9
    
10.
Thomas DB, Jimenez LM, McTiernan A, Rosenblatt K, Stalsberg H, Stemhagen A, et al. Breast cancer in men: Risk factors with hormonal implications. Am J Epidemiol 1992;135:734-48.  Back to cited text no. 10
    
11.
Collins LC, Tamimi RM, Baer HJ, Connolly JL, Colditz GA, Schnitt SJ. Outcome of patients with ductal carcinoma in situ untreated after diagnostic biopsy: Results from the Nurses' Health Study. Cancer 2005;103:1778-84.  Back to cited text no. 11
    


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