NOWOTWORY Journal of Oncology 2007 volume 57 Number 3 131e135e

Case report

A case of bilateral synchronous male breast cancer

Barbara Hofman1, Daniel Jajtner1, Danuta Maka2, Ewa Chudyba3, Tadeusz Leniak1
Bilateral male breast cancer is a very rare disease. The authors report a case of a 62-year-old man with bilateral breast cancer who was treated at the Beskidian Oncological Center in Bielsko-Biala. Invasive ductal carcinoma was localized in both his breasts. Before surgery the malignancy of the tumour had been confirmed only within the left breast. Lumpectomy of the right breast combined with intraoperative pathological examination revealed the presence of breast cancer. On receiving this results bilateral radical Pateys mastectomy had been performed. Surgery is the treatment of choice in male breast cancer, but it should be Key words: bilateral, male, breast cancer, invasive cancer, mastectomy, radiotherapy, chemotherapy

Introduction
Male breast cancer is a rare disease, with an incidence of 1% of all breast cancers in humans; bilateral male breast cancer is extremely rare. We will present the case of a 62-year-old man who came to the Oncological Outpatient Clinic at the Beskidian Oncological Center in Bielsko-Biaa because of retroareolar tumours in both breasts.

revealed the presence of suspected lymph nodes of about 1.5 cm in diameter. The supraclavicular lymph nodes were not examined. Additional tests In the mammography that was performed, the breast structure is predominantly fatty, the test sensitivity is high. In the left breast, retroareolarly, there was a nodule of high density and spicular shape of 15 mm in size. In the right breast, at the limit of the lateral quadrants, periareolarly, there is a visible density of about 15 mm in size, without clear signs of malignancy in the mammographic test and without the signs of tumour in the ultrasound examination. In addition, at the limit between the internal quadrants of the right breast, there was a 5 mm density of slightly uneven shape. The patient was referred for fine-needle biopsy. The results were as follows: Examination No 12258. The left breast: cellulae carcinomatosae. The image corresponds to: carcinoma mammae. The lymph node of the left axilla: in the diagnostic material, there are small lymphocytes, lymph node germinal centre cells, macrophages. No malignant cells were found. Examination No 12481. The right breast: the diagnostic material is rich in cells, the fluid is albuminous, bloody, there are many flaps of epithelial cells without signs of atypia, a few myoepithelium cells. The cytological image corresponds to a benign lesion; however, histopathological verification of the lesion is indicated. In routine ultrasound examinations of the abdominal cavity and chest X-ray, there were no lesions. In addition, ultrasound examination of the testes was performed due to the doubts in the physical examination, apart from a

Case description
A 62-year-old patient, case record No 1505/02 s, was admitted to the Department of Oncological and General Surgery at the Beskidian Oncological Center for the treatment of nodular changes in both breasts. The lesion in the right breast he noticed himself about one month before; he did not notice the lesion in the left breast. In the history he reported type 2 diabetes treated with insulin. He reported no other diseases. His family history was irrelevant. On physical examination, apart from an uneven surface of the testes, especially on the right side, no deviations from the normal condition were found. Local condition: in the subareolar area, in the left breast, there was a nodule of about 1 centimetre in diameter, below which there was a mobile infiltration of about 1.5 cm in diameter. In the right breast, there was a round, hard and compact nodule of about 1 cm in diameter. The examination of both axillary regions

1 2 3

Oddzia Chirurgii Onkologicznej i Oglnej Oddzia Onkologii Klinicznej Zakad Teleradioterapii Beskidzkie Centrum Onkologii im. Jana Pawa II w Bielsku-Biaej

132e The other parts of the breast fibrous connective tissue and fatty tissue normal structure without cancer infiltration. Lymph nodes metastases carcinomatosae 1/25. Histological type ductale , Nottingham grading G2, tumour size pT 1.7 cm, pT4 (skin infiltration), lymph node status 1/25; pN1, peripheral inflammatory infiltration (-), emboli in the blood vessels (-), radicality deep margin 0.2 cm, lateral 1.5 cm. ER (-), PR (+++), HER-2 neu (-). Clinical stage III. Examination No 30349: tumour in the right breast lesion size 1.5 cm. Post-intra diagnosis: carcinoma papillare G1 cum microinvasione. Tumour infiltration caused no inflammatory reaction in the stroma. No cancer cell emboli were found in the lumen of the blood vessels. Ca mammae. Histological type papillare cum microinvasione, tumour size pT1, lymph node status 0/14, pN0, peripheral inflammatory infiltration (-), emboli in the blood vessels (-), ER (++), PR (++), HER 2/neu (-).
Figure. 1. A 62-year-old patient. Mammography: on the left side, retroareolarly, a spicular shape of about 15 mm in size, on the right side, a lesion in the limit between the lateral quadrants, periareolarly, without clear signs of malignancy

Po s t - o p e r a t i v e c o u r s e Post-operative wound healing was normal. Combined bilateral drainage did not exceed 500 ml in the first day, and 300 ml in subsequent days. In the first day post surgery, follow-up blood cell counts showed anaemia (erythrocytes RBC, 2.71 x106/mm3; haemoglobin Hb, 10.0 g/dl; haematocrit Ht, 29.4%; platelets PLT, 121,000 mm3). The patient was transfused with two units of red blood cell concentrate without any complications. Two days later, the catheter was removed from the right ulnar region because of inflammation developing in this area. On the same day, in the evening, the patient had fever up to 39.2. Infection at the venous access site was diagnosed. The end of the catheter was sent for bacteriological test. A broad-spectrum antibiotic was administered orally, an incision was made and the subcutaneous tissue abscess was drained in the area of previous venous access; the symptoms gradually resolved. Punctures of the operated areas were performed daily there was moderate lymph retention no signs of infection in the post-operative wounds. The patient was discharged from the hospital in good general condition with normally healing wounds.

slight amount of fluid in the capsules of both testes, the examination was normal. Laboratory tests. Complete blood cell counts on admission: slight anaemia (RBC 3.31 x106/mm3; haemoglobin Hb, 12.8 g/dl; haematocrit Ht, 36.2%; platelets PLT, 110,000/mm3). In the biochemical tests: low total serum protein, 5.84 g/dl; no other abnormalities. The patient was consulted anaesthesiologically twice for blood pressure fluctuations. He was referred for surgery, operated in perispinal anaesthesia, then, after intraoperative examination of the nodule in the right breast, in general anaesthesia. Surgery The tumour in the right breast was removed and an intraoperative histopathological examination was performed. Result of examination No 30349: 1. smear cellulae carcinomatosae, 2. intra carcinoma invasivum. Following the intraoperative examination, bilateral Pateys mastectomy was performed. Re s u l t o f t h e h i s t o p a t h o l o g i c a l t e s t Examination No 30374: the left breast with tumour of 1.7 cm in diameter, infiltrating the nipple carcinoma invasivum G2. The cancer invades the skin. In the vessels, there are the emboli of the tumour cells, there is no reactive inflammatory reaction in the stroma. Deep margin 0.2 cm no infiltration. Lateral margin 1.5 cm no infiltration.

Adjuvant treatment
In adjuvant treatment, according to a decision of the expert team, the patient was referred for CMF chemotherapy (cyclophosphamide, methotrexate, 5-fluorouracil), then radiotherapy and hormonal therapy. The date of the first treatment cycle was scheduled about four weeks after surgery. However, the patient received no chemotherapy on that day due thrombocytopaenia WHO grade 2. After a week, with normal

133e blood cell counts, chemotherapy was started according to the previous regimen with oral cyclophosphamide. The second dose of the 1st cycle of chemotherapy was administered in reduced dose because the patient experienced anaemia and thrombocytopaenia (WHO grade 2). At the time of the 2nd course, the number of leucocytes in one mm3 of the patients blood was 2.2 x 103. The chemotherapy was rescheduled once more. Despite the introduction of medication, the blood cell counts only allowed the treatment to be introduced after 6 weeks. It was decided again to change chemotherapy to AC (doxorubicin, cyclophosphamide) as the regimen associated with lower toxicity, including myelotoxicity. The patient received the drugs in the appropriate doses. At the time of the 2nd cycle of AC, the patient returned with symptoms of weakness and dyspnoea. The RBC count was 1.6 x106/mm3; haemoglobin concentration 6.1 g/dl, and WBC count was 1.9 x103/mm3. The patient required transfusion of 4 units of RBC concentrate, and antibiotics administration. Bone marrow trepanobiopsy was also performed twice in the iliac ala for histological evaluation of bone marrow. Examinations No 32735 and 41056 did not show any cancer infiltration in the bone marrow in the course of breast cancer. Due to haematological complications and the impossibility to maintain the chemotherapy rhythm, it was discontinued. The patient started hormonal therapy with tamoxifen 20 mg/day. Then the patient was transferred to the Department of Teleradiotherapy for irradiation of the wall of the chest with the scar as well as axillary and supraclavicular lymph nodes on the left side, according to the previously accepted treatment regimen. The supraclavicular-axillary lymph system was irradiated with gamma Co60 photons from typical fields, and the chest wall with the scar postmastectomy was irradiated with gamma Co60 photons from tangential fields df 2.25 Gy in 20 fractions once daily up to Dc 45 Gy. During irradiation, leucopenia WHO grade 1, thrombocytopaenia and anaemia were observed; they did not require radiation discontinuation. Early skin reaction was evaluated as WHO grade 1. When the irradiation was complete, haematological disorders exacerbated in the form of pancytopaenia: Hb 7.8 g/dl, WBC 1,900/mm3, PLT 67,000/mm3. The patient received 2 units of RBC concentrate with shortterm improvement. The patient was referred to the Haematology Outpatient Clinic where he was diagnosed. However, the diagnostics were not completed. The patient died suddenly at home. An autopsy has not been performed. cancer occurs in men is believed to be 60.6 years [3, 5-8]. The risk factors include: testicular diseases, benign lesions in the breasts (including gynaecomastia), genetic disorders with gynaecomastia (Klinefelter syndrome), metabolic diseases, oestrogen inactivation disorders in men caused by liver diseases (alcohol cirrhosis, viral hepatitis) particularly important in the Polish population, genetic mutations (BRCA2 gene carriers) [1, 9-17] and other, often controversial risk factors such as: exposure to ionising radiation, geographical and ethnic factors, using certain drugs spironolactone [9], and: overweight, diabetes, hypertension, hyperlipidaemia [17]. Clinically, male breast cancer usually occurs (98%) in the form of retroareolar, painless tumour [3, 5, 6, 8, 17] of about 1 cm in diameter. Despite this small size, skin can be retracted, suggesting early infiltration [8]. The clinical course can differ, from slow disease progression to a disease with early metastases [2]. Mammographic and/or ultrasound examinations usually differentiate between cancer and gynaecomastia [8]. In histopathological examination, male breast cancer is similar to female tumours, although male tumours are more often estrogen-positive. Histologically, in the male population, invasive ductal carcinoma is prevalent [13], and lobular carcinoma is rare [8]. The treatment of choice remains surgical treatment in the form of modified radical Pateys mastectomy. In the past, the Halstedts procedure was preferred, consisting in the removal of the greater pectoral muscle in a block with the breast. This was related to a higher stage of tumours detected in men and smaller size of the breast, and earlier infiltration of the greater pectoral muscle with the tumour mass. The procedure caused a significant limitation in the movements of the shoulder girdle and asymmetry of the wall of the chest; therefore, with the advance in irradiation techniques, this method has been discontinued. The surgical treatment of the primary lesion should be associated with adjuvant treatment: radiation therapy, chemotherapy or hormonal therapy [13, 18]. The following factors have a significant effect on the type of adjuvant treatment: lymph node status, tumour size, the presence of hormonal receptors on the surface of the tumour cells and the patients general condition [18]. However, the most important prognostic factor remains the lymph node status and the disease stage [5]. In systemic chemotherapy, the most commonly used schedule is CMF (cyclophosphamide, methotrexate, 5-fluorouracile) or regimens with anthracyclines [8, 13]. Chemotherapy is recommended for men with affected lymph nodes, primary tumour larger than 1 cm in diameter, hormonally-negative metastases, as well as hormone-refractory tumours. Due to common presence of hormonal receptors on the cell surface, hormonal therapy is used [17]. The first-line drug is tamoxifen.

Conclusion
Male breast cancer is a rare disease [1, 2]. The incidence is 0.2 to 1.5% of malignant cases in men and 15.7% of all breast tumours [3, 4]. The mean age at which breast

134e

Unilateral breast mass

Complete history and physical examination

Bilateral

Unilateral

Yes

Does the patent take any medication?

The following are associated with gynecomastia cimetidine thiazide diuretics omeprazol tricyclic antidepressants spironolactone diazepam anabolic steroids estrogen marijuana

No

Consider the patent's age

Age <25 yr

Age >50 yr

Stigmata of systemic illness

Pathologic hormone imbalances noted on personal and family history and examination

Yes

No

Yes

No

Consider cirrhosis chronic renal insufciency or other conditions

Positive family history or excess exposure to estrogen

Consider: Klinefelters syndrome undescended testes orchitis other causes of hipogonadism

Yes

No

Consider physiological changes associated with puberty

Consider work up if no improvement in 6 months

Consider work-up (bilateral cancers reported in 5% of causes)

Stop the suspected medication and repeat examination in 4-6 months

Yearly follow-up because of greater risk of breast cancer

Mammography

Breast mass in men (in 60% men >70 yr)

Figure 2. Suggested diagnostic procedure in the case of bilateral lesions in the male breasts

The second-line treatment remains orchidectomy or the use of gonadoliberin analogues with or without anti-androgens [18]. Hormonal therapy should be administered for five years. In the case of patients with histologically confirmed metastases and positive hormonal receptors, neoadjuvant hormonal treatment and systemic chemotherapy should be introduced both in the cases of receptor-negative patients and in the cases of hormone-refractory tumours [19]. Although rare, male breast cancer is a surreptitious disease. Men are rarely aware that it can affect them. No screening has been carried out for early detection; on the other hand, as the disease can be easily detected on palpation, screening does not seem necessary. However, the most important conclusion is the need of palpation examination of the male breasts during prophylactic tests. The patients themselves should also be made aware of the most subtle symptoms from the breast, and seek specialist help should they occur [6].

Figure 2 presents the suggested diagnostic procedure in the case of bilateral lesions in the male breasts [20].
Dr med. Barbara Hofman ul. Traugutta 24/36 43-502 Czechowice-Dziedzice

References
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Paper received: 27 July 2006 Accepted: 11 December 2006