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Pleomorphic lobular carcinoma of the male breast with axillary lymph node involvement: a case report and review of literature
© Zahir et al.; licensee BioMed Central Ltd. 2014
Received: 7 November 2013
Accepted: 17 April 2014
Published: 27 April 2014
Carcinoma of the male breast is responsible for less than 1% of all malignancies in men but the incidence is rising. Invasive ductal carcinoma is the most common histological subtype while invasive lobular carcinoma is responsible for only 1.5% of the total cases of which pleomorpic lobular carcinoma is an extremely rare variant. We report the case of a gentleman with node positive, pleomorphic lobular carcinoma of the breast.
An elderly gentleman with a past history of type 2 diabetes and long term ethanol use presented to us with a self-discovered palpable lump in the left breast. Physical examination revealed bilateral gynaecomastia along with a well circumscribed subareolar mass and palpable lymphadenopathy in the ipsilateral axilla. The breast nodule revealed atypical cells on fine needle aspiration biopsy and the patient underwent a modified radical mastectomy after systemic surveillance was negative for metastatic disease. The lesion was reported as grade III pleomorphic lobular carcinoma with a lack of E-cadherin expression on immunohistochemistry and the neoplastic cells exhibited strong positivity for estrogen receptor in the absence of Her2 gene amplification. Six out of the eleven dissected regional lymph nodes showed evidence of disease. The patient completed 4 cycles of adjuvant chemotherapy without evidence of recurrent disease and was subsequently lost to follow up.
Although invasive lobular carcinomas comprise 12% of all female breast cancers, they are very rare in males due to lack of acini and lobules in the normal male breast. Pleomorphic lobular carcinoma, an aggressive variant of ILC is even rarer in males.
Chronic consumption of ethanol by our patient may have resulted in some degree of hepatic impairment with resultant hyperestrogenism. This in theory may have been the cause of his gynaecomastia, resultant breast cancer and is a plausible explanation for development of the invasive lobular subtype in a male. The prognosis and clinicopatholocial features of pleomorphic lobular carcinoma in men are less clearly defined due to its rarity. Additional studies are hence necessary to improve our understanding of this disease in males.
Carcinoma of the male breast is a rare entity accounting for 0.7% of all breast cancers . Although the disease is also responsible for less than 1% of all malignancies in men, the incidence of male breast cancer has seen a rise of about 26% over the past quarter of a century [1, 2]. Risk factors similar to those observed for female breast cancers are responsible for pathogenesis. All histopathological variants seen in female breast cancer have been observed [3, 4]. Invasive ductal carcinoma is the most common histological subtype accounting for approximately 85% of all cases. Male breast cancers are significantly more likely to exhibit hormone receptor expression in comparison to female breast cancers [1, 2, 5]. Data from the Surveillance, Epidemiology, and End Results (SEER) database shows that only 1.5% of male breast cancers are of the invasive lobular subtype while the same histological variant is responsible for approximately 12% of carcinomas of the female breast .
Herein we report the case of an elderly gentleman diagnosed with the pleomorphic variant of invasive lobular carcinoma along with involvement of the ipsilateral axillary lymph nodes. Only three cases of this histopathological subtype have ever been reported in the male breast and none thus far had shown metastasis to the regional draining lymph nodes.
A 68 year old Pakistani male was referred to the outpatient oncology clinic at our center with a three month history of a self-discovered, progressively increasing palpable lump in the left breast. He had sought medical attention for increasing size of the lump with recent development of pain. His past history was significant for type 2 diabetes and long term ethanol use. There was no family history of breast disease.
Physical examination revealed bilateral gynaecomastia along with a 3 × 3 cm sized, relatively well circumscribed subareolar mass which was firm and mildly tender. The overlying skin appeared normal. There was evidence of palpable lymphadenopathy in the ipsilateral axilla.
The patient was recommended adjuvant chemotherapy to be followed by chest wall and axillary irradiation and hormonal therapy. He completed 4 cycles of adjuvant chemotherapy without any evidence of recurrence and was subsequently lost to followup.
Although the majority of men with breast cancer have no identifiable risk factors, some risk factors that are modestly unique to men include never being married, gynecomastia, Klinefelter’s syndrome and a history of testicular or liver pathology .
Breast cancers in men typically presents as a painless, firm mass that is usually subareolar. The left breast is involved slightly more often than the right, and less than one percent of cases are bilateral [7, 8]. Male breast cancers tend to present sooner because of scarcity of breast tissue and hence most subjects have early stage disease at presentation.
Invasive lobular carcinoma (ILC) of the breast is a histopathologically distinct entity from the much more common invasive ductal carcinoma (IDC). Although invasive lobular carcinomas comprise 12% of all female breast cancers, they are very rare in the male breast . The rarity of a lobular histologic subtype of breast cancer is due to lack of acini and lobules in the normal male breast . Endogenous or exogenous estrogenic stimulation may induce the development of acini and lobules in the male breast and hence subsequently increases the theoretical risk of development of invasive lobular carcinoma (ILC). ILC typically lacks E-cadherin expression on immunohistochemical (IHC) staining and negative expression is now used as an aid in diagnosis .
Clinical and histopathological features of pleomorphic lobular carcinoma of the male breast
Maly et al.
Rohini et al.
Ishida et al.
Year of report
2.5 × 2.0
3.0 × 2.5
3.0 × 2.5
2.8 × 2.5
Estrogen receptor expression
Progesterone receptor expression
Her2 gene amplification
Ki-67 proliferative index (%)
Lymph node involvement
Disease free at 2 years
Disease free at 1 year
Disease free at 2 months
Disease free at 3.5 months
The present case merits discussion on several key points. Ethanol use by this gentleman may have resulted in some degree of hepatic impairment. It is an established fact that liver dysfunction results in hyperestrogenism which may have been the cause of his gynaecomastia and resultant breast cancer . The hyperestrogenism theory also explains the development of the invasive lobular subtype in this particular gentleman. The typical morphologic features of pleomorphic lobular carcinoma and lack of E-cadherin expression aided in this rare histopathological diagnosis. The clinical presentation, hormone receptor expression and the absence of Her2 gene amplification was typical of what is seen in male breast cancer. To the best of our knowledge, this is the first reported case of pleomorpic lobular carcinoma in the male breast with regional lymph node involvement. This may be attributed to a delay on the part of the patient to seek appropriate medical care in a resource poor country like Pakistan where all medical expenses are incurred by patients themselves.
ILC and particularly pleomorphic lobular carcinoma in the male breast is a rare occurrence and reminds us to tailor management according to subtype as this variant is known to be clinically aggressive in females. The prognosis and clinicopatholocial features of this variant in men are less clearly defined due to its rarity. Additional studies are hence necessary to improve our understanding of this disease in males.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
We would like to thank Dr. Tayyaba Z. Ansari who provided important intellectual input during the drafting phase of the manuscript. We would also like to thank Dr. Sidra Arshad who helped procure the histopathological images.
The authors did not receive any research support for this manuscript and do not have any financial disclosures to make.
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