Breast cancer is a relatively rare entity in males accounting for less than 1% of all cancer in men and 0.5–0.7% of all breast cancers. [1] Bilateral synchronous breast cancers are extremely uncommon in men. Hence they have not been studied deeply from investigation and management point of view. In this report we describe a rare case of synchronous bilateral breast cancer in an elderly male without any significant risk factors.

A 75-year-old male was presented to our center with complaint of mass in the right breast. On acquisition of detailed medical history, there was no history of hormonal therapy, testicular disease and occupational exposure to radiation in his lifetime. He was nonsmoker and nonalcoholic. There was no personal and family history of breast cancer. On physical examination, there was a mass in upper outer quadrant of size 4x3 cm under nipple with evidence of bloody nipple discharge. External genitalia examination was normal. Mammography revealed a 3-cm radiopacity with regular margins in retroareolar region. Fine-needle aspiration cytology showed presence of malignant cells. Contra lateral breast was normal on physical examination. The presence of distant metastasis was excluded by computed tomography scan and abdominal ultrasound scan. Laboratory work-up including hematology, liver, kidney and thyroid function tests was within normal limits. Patient underwent modified radical mastectomy with axillary lymph node resection. On gross examination, tumor was of size 4x3x3 cm. Histopathological examination revealed features of invasive duct carcinoma, grade II. Nipple and cut margins of specimen were free of tumor. Also there was no evidence of vascular and perineural invasion. Three of seven axillary lymph nodes were positive for the presence of malignant cells. As per tumor node metastasis (TNM) classification tumor was of stage II. On immunohistochemistry analysis, tumor cells were positive for estrogen receptor (60%) and progesterone receptor (50%).

Patient received six cycles of adjuvant chemotherapy with cyclophosphamide, 5-fluorouracil, epirubicin. After surgical management external radiotherapy was administered to chest wall and axilla with doses of 50 Gy (2 Gy daily with five weekly fractions) for a period of five weeks. Also patient was advised for daily oral tamoxifen 20 mg for 5 years. But after three months during routine follow-up clinician felt a new lump in left breast which was of size 2x2 cm and on fine-needle aspiration cytology showed presence of malignant cells. Patient was managed with modified radical mastectomy with axillary lymph node resection. Histopathologically, tumor was infiltrating duct carcinoma, grade II and 2 of 6 lymph nodes showed presence of malignant cells. Hence, it was stage II carcinoma of breast. Then patient underwent orchiectomy. Again patient received adjuvant chemotherapy and radiotherapy for five weeks and continued with oral tamoxifen 20 mg daily. Now since 18 months patient is disease free.

Bilateral breast cancer in male is an infrequent condition representing 0.5–0.6% of all breast cancers. Recent studies have shown an increased incidence of male breast cancer from 0.86 to 1.08 per 10,000 men. [2] In India Mitra et al. reported incidence of male breast cancer in a tertiary care hospital 0.6% of all cancers in men and 2.5% of all cases of breast cancer. [3] Risk factors for male breast cancer include family history of cancer of breast, obesity, gynecomastia, exposure to radiation, testicular diseases like undescended testis, orchitis, orchiectomy, Klinefelter syndrome (47,XXY) and hormonal therapy. [1] Also hyperestrogenic states due to liver diseases and hypoandrogenic conditions like infertility contribute to breast cancer in men. Gene mutations of BRCA2, BRCA1 and androgen receptor gene predispose to such condition. [4] [5] Our patient was not willing for such genetic studies. No significant risk factors were identified in the present case. Hence this unusual synchronous bilateral breast cancer presentation may be a sporadic event.

Invasive duct carcinoma is the most common type of malignancy of breast observed in male with strong positivity for estrogen and progesterone receptors in 63–90% of cases. [6] In our case also patient had this common type of breast cancer, but within a period of three months he developed same type of tumor on contra lateral side. We managed the case with bilateral modified radical mastectomy, radiotherapy, adjuvant chemotherapy and hormonal therapy with tamoxifen. For hormone receptor positive breast tumor tamoxifen is the first choice of therapy that can lead to improved overall survival and outcome of the patient. Management in male and female breast cancer to some extent is similar.

Because of rarity of bilateral breast cancer in male there is dearth of such reports in literature. Giordano et al. reported only two cases of bilateral breast cancer among 2537 men with breast cancer. [4] As in males no screening strategies are designed usually they are presented in late stage. Kahla et al. reported a case of hormone receptor positive bilateral synchronous breast cancer in a 71-year-old male with 17 months disease free survival managed with bilateral modified radical mastectomy (MRM) and adjuvant chemo-endocrine treatment. [7] Lambley et al. described a 84-year-old male with bilateral infiltrating duct carcinoma managed by bilateral MRM and tamoxifen but not radiotherapy. [8] Small sample of the cases are available for study because of rarity. Shah et al. reported only 32 cases of male breast cancer over 24 years in their retrospective study. [9] Rai et al. retrospectively analyzed 30 cases of male breast cancer treated at a single center in India over 24 years. [10] They observed occurrence of loco regional recurrence in three cases and distant metastasis in nine patients. But there was no evidence of bilateral breast cancer in a single case. Some studies have shown a worse outcome for male breast cancer compared to female breast cancer, but findings are inconsistent. In our patient, there was a latent period of four months between two incidences of breast cancer.

Woo-Young Sun et al. reported a case of a 54-year-old male with simultaneous bilateral breast cancer in Korea with invasive duct carcinoma in right breast and ductal carcinoma in situ in left breast. Out of 24 cases of synchronous bilateral male breast cancer reported in literature hormonal receptor status (ER or PR) was positive in 87.5% cases. [11]

Proper clinical evaluation, mammography and fine-needle aspiration cytology play important role in the diagnosis of male breast cancer. Presence of lump, nipple retraction and discharge, ulceration and palpable axillary or supraclavicular lymph nodes should lead to a suspicion of malignancy of breast in male. [12] Agrawal et al. emphasized the important psychological aspect of male having ‘cancer of female’ which should be managed by clinician and counselor. [13] In male risk of developing second breast cancer after first breast cancer is much high compared to women. [14] Men with breast cancer have 93-fold greater risk of developing contra lateral breast cancer. [1] If investigated in depth and treated, early patients will definitely benefit with optimum outcome.

Purpose of reporting this case from India was to study and compare clinicopathological profile and management of male breast cancer cases reported in literature. This case report also adds to collection of resources for study of male breast cancer because of synchronous presentation of bilateral breast cancer with latent period of four months. Also this will increase awareness of patients and clinicians about this rare condition. Recently, Jonathan White et al. reported rising number of male breast cancer patients from all countries. But there is limited epidemiological and clinical data related to this disease. Most of the information used in understanding and managing male breast cancer comes from data of female breast cancer which may not always provide an accurate picture. [15]

Although rare, incidence of male breast cancer is increasing and survivors are at risk of development of subsequent primary cancers. Despite absence of major risk factor our patient developed synchronous bilateral breast cancer, but was managed successfully with bilateral modified radical mastectomy, adjuvant chemotherapy, hormonal therapy and radiotherapy. Future in-depth studies focusing on etiopathology of the disease will help to optimize the management of male breast cancer patients.

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