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CORRESPONDENCE |
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| Year : 2020 | Volume
: 65
| Issue : 2 | Page : 167-169 |
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| Diagnostic clues for pagetoid Bowen's disease |
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Binluen Chiang, Koji Kamiya, Takeo Maekawa, Mayumi Komine, Satoru Murata, Mamitaro Ohtsuki
Department of Dermatology, Jichi Medical University, Shimotsuke, Tochigi 329-0498, Japan
| Date of Web Publication | 25-Feb-2020 |
Correspondence Address:
Binluen Chiang
Department of Dermatology, Jichi Medical University, Shimotsuke, Tochigi 329-0498
Japan
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/ijd.IJD_362_18
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How to cite this article:
Chiang B, Kamiya K, Maekawa T, Komine M, Murata S, Ohtsuki M. Diagnostic clues for pagetoid Bowen's disease. Indian J Dermatol 2020;65:167-9 |
How to cite this URL:
Chiang B, Kamiya K, Maekawa T, Komine M, Murata S, Ohtsuki M. Diagnostic clues for pagetoid Bowen's disease. Indian J Dermatol [serial online] 2020 [cited 2022 Jun 26];65:167-9. Available from: https://www.e-ijd.org/text.asp?2020/65/2/167/279193 |
Sir,
An 82-year-old Japanese woman presented with a 2-year history of a nonhealing ulcer on the left breast without pain or pruritus. There is no history of arsenic exposure. Physical examination revealed a well-demarcated erythematous plaque with erosions on the areola [Figure 1]. Dermoscopic findings showed irregular white reticular structure with various blood vessels [Figure 2]. The skin on the nipple was intact. No palpable lymph nodes (axillary and others) were noted. Histopathological findings revealed pagetoid cells in all layers of the epidermis [Figure 3]. On immunohistochemical staining, the pagetoid cells were positive for CK5/6 and p63 [Figure 4] but negative for CK7, CK20, GCDFP15, PAS, S-100 protein, Melan A, and HMB-45. Therefore, pagetoid Bowen's disease (PBD) was suspected. After the complete excision, the specimen showed more typical areas of Bowen's disease that displayed dyskeratotic cells and multinucleated giant cells in the epidermis [Figure 5]. Immunohistochemistry showed the same findings as the biopsy one. Consequently, the diagnosis of PBD was confirmed. | Figure 1: A well-demarcated erythematous plaque with erosions on the areola with intact skin of the nipple
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 | Figure 2: Dermoscopy showing irregular white reticular structure with various blood vessels
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 | Figure 3: Histopathology showing pagetoid cells in all layers of the epidermis (H and E, ×200)
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 | Figure 4: By immunohistochemistry, pagetoid cells were positive for CK5/6 (Left), (×400) and p63 (Right), (×400)
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Bowen's disease is a form of cutaneous squamous cell carcinoma in situ more commonly developing on su n-exposed sites. Bowen's disease of the breast is extremely rare. With only 9 cases reported in the English literature so far, and none of them is PBD on nipple-areola complex.[1] PBD, in which pagetoid cells are observed histologically, accounts for 5% of Bowen's disease.[2] The pagetoid growth pattern mimics mammary Paget's disease and superficial spreading melanoma.[3] The treatment, surgical margin of excision, and the follow-up period are different, so it is crucial to make the exact diagnosis. Histopathological features for PBD include the existence of dyskeratotic cells, multinucleated giant cells, desmosomes among pagetoid cells, and pagetoid cells existing in all layers of the epidermis. In contrast, pagetoid cells tend to exist only in the lower layer of epidermis in mammary Paget's disease and malignant melanoma. Nevertheless, it is sometimes difficult to differentiate from one another without the help of immunohistochemistry.[4] In PBD, pagetoid cells typically stain for CK 5/6 and p63. GCDFP15 and PAS expression are observed in mammary Paget's disease. S-100 and HMB-45 are useful markers for malignant melanoma. Notably, CK7 expression is almost always observed in mammary Paget's disease. However, cases of PBD with positive CK7 expression have been reported. Although our case shows negative expression for CK7, CK7 expression is not always a helpful marker to differentiate PBD from mammary Paget's disease.[3] Mammary Paget's disease is frequently associated with underlying breast carcinoma, which often originates from lactiferous ducts.[5] Therefore, the clinical lesion usually starts from the nipple, then it extends to the areola and surrounding skin. In contrast, in our case of PBD, the lesion only involved the areola and left the skin of the nipple uninvolved. The clinical appearance without nipple involvement actually gave us a hint that this disease might not arise from the nipple or the underlying lactiferous ducts. Consequently, the existence of clinical nipple involvement can also be a clue to tell the difference between PBD and mammary Paget's disease.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
 References | |  |
| 1. | Zhao Z, Tay TK, Agrawal R, Tan VK, Tan YY, Tan PH. Intraepidermal malignancy in breast skin: A tale of two tumours. Hum Pathol Case Rep 2018;14:33-7.  |
| 2. | Raju RR, Goldblum JR, Hart WR. Pagetoid squamous cell carcinoma in situ (pagetoid Bowen's disease) of the external genitalia. Int J Gynecol Pathol 2003;22:127-35. |
| 3. | Williamson JD, Colome MI, Sahin A, Ayala AG, Medeiros LJ. Pagetoid bowen disease: A report of 2 cases that express cytokeratin 7. Arch Pathol Lab Med 2000;124:427-30. |
| 4. | Elbendary A, Xue R, Valdebran M, Torres KM, Parikh K, Elattar I, et al. Diagnostic criteria in intraepithelial pagetoid neoplasms: A histopathologic study and evaluation of select features in Paget disease, Bowen disease, and melanoma in situ. Am J Dermatopathol 2017;39:419-27. |
| 5. | Karakas C. Paget's disease of the breast. J Carcinog 2011;10:31. [ PUBMED] [Full text] |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5] |
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Extramammary Paget’s Disease: Diagnosis, Pathogenesis, and Treatment with Focus on Recent Developments |
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| Shoichiro Ishizuki, Yoshiyuki Nakamura | | Current Oncology. 2021; 28(4): 2969 | | [Pubmed] | [DOI] | | | 3 |
Differentiating pagetoid Bowen disease from Paget disease on the nipple-areola complex: Two unique, challenging cases |
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| Leire Barrutia, Gerardo Martínez-García, Alba Santamarina-Albertos, Esther Leticia Garabito Solovera, Víctor Volo, Daniel Ruíz-Sánchez, Pilar Manchado López | | Journal of Cutaneous Pathology. 2021; 48(11): 1416 | | [Pubmed] | [DOI] | |
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