Bowen's Disease of the Nipple and Areola in an Old Man.

Sir,

Bowen's disease (squamous cell carcinoma in situ) usually develops on sun-exposed areas and its occurrence on the breast is rare. We herein describe a case of Bowen's disease of the nipple and areola in an old man.

A 76-year-old man visited our department, complaining of a plaque on his right breast that had been first noted some years previously, and had enlarged slowly. He had no significant medical history. He denied previous histories of treatment with arsenic containing drugs. Also, he had neither occupational exposure suggestive of arsenic toxicity nor history of chronic arsenicism in his neighborhood or family via ground water poisoning. The lesion was a slightly keratotic, irregularly elevated, well-circumscribed, and sized 26 × 19 mm brownish plaque [Figure 1]. The base of the firm tumor was not adhesive to the underling tissue. The right axillary lymph nodes were not palpable. A biopsy specimen showed irregular epidermal hyperplasia and diffuse interstitial infiltration at the papillary dermis [Figure 2]. Most of the inflammatory cells were lymphocytes. In the epidermis, there were many anaplastic keratinocytes with nuclei of irregular size and form. We also found clumping cells and dyskeratotic cells, and many atypical mitotic figures. The basement membrane was not invaded. Results of immunohistochemistry showed strong expression of 34βE-12, and moderate expression of AE1/AE3 and p63 [Figure 3]. Other markers, such as CK7, CK20, CEA, GCDFP-15, and CAM5.2, were all negative. The residual tumor was totally removed with a 5-mm margin under local anesthesia.

Figure 1

Nipple lesion of the right breast

Figure 2

Histological features show irregular hyperplasia of the epidermis, and many of anaplastic keratinocytes, clumping cells, dyskeratotic cells, and atypical mitotic figures (hematoxylin and eosin stain). The basement membrane was not invaded

Figure 3

Immunohistochemistry showing positive findings of p63

Kossard and Rosen reported that the most common site of Bowen's disease was head and neck (440 among 1,001 cases of Bowen's disease), and that the least common site was the torso (65/1001).[1] It is especially rare for Bowen's disease to develop on the nipple. To date, only six cases of Bowen's disease involving the nipple have been reported including our case.[23456] The age of patients ranged from 41 to 84-years-old, and male patient was observed in only three cases including our case. Our case was unique in that Bowen's disease occurred involving the nipple and areola in an elderly male patient.

Histological features of Bowen's disease of the nipple and mammary Paget's disease are sometimes similar, but treatments of them are decisively different. It is therefore very important to differentiate those diseases. Immunohistochemical findings are useful for differentiation. Irregular epidermal hyperplasia is a histological feature of Bowen's disease, and that of mammary Paget's disease is clear cells (Paget cell) that sometimes conglomerates in the epidermis. Immunohistochemically, neoplastic cells are positive for p63 and 34βE-12 in Bowen's disease, while Paget cells are positive for CEA, CAM5.2, and GCDFP-15. Thus, in many cases, it is not so difficult to differentiate these two diseases. However, the same is not true for Pagetoid Bowen's disease, a subtype of Bowen's disease. Pagetoid Bowen's disease shows clear cells in epidermis which increase and form conglomerates, but clear cells are not always seen in Pagetoid Bowen's disease, unlike Paget's disease. As the immunohistochemical findings of Pagetoid Bowen's disease are the same as those of Bowen's disease, examination by using antibodies; in particular against p63, GCDFP-15, and 34βE-12; is useful and important for differentiation of those diseases. In conclusion, we presented with a case of Bowen's disease arising on a rare site, and successfully treated by surgical operation.