IJD
Indian Journal of Dermatology
  Publication of IADVL, WB
  Official organ of AADV
Indexed with Science Citation Index (E) , Web of Science and PubMed
 
Users online: 1388  
Home About  Editorial Board  Current Issue Archives Online Early Coming Soon Guidelines Subscriptions  e-Alerts    Login  
    Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
Table of Contents 
CORRESPONDENCE
Year : 2020  |  Volume : 65  |  Issue : 2  |  Page : 167-169
Diagnostic clues for pagetoid Bowen's disease


Department of Dermatology, Jichi Medical University, Shimotsuke, Tochigi 329-0498, Japan

Date of Web Publication25-Feb-2020

Correspondence Address:
Binluen Chiang
Department of Dermatology, Jichi Medical University, Shimotsuke, Tochigi 329-0498
Japan
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_362_18

Rights and Permissions



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 2020 Nov 28];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

Click here to view
Figure 2: Dermoscopy showing irregular white reticular structure with various blood vessels

Click here to view
Figure 3: Histopathology showing pagetoid cells in all layers of the epidermis (H and E, ×200)

Click here to view
Figure 4: By immunohistochemistry, pagetoid cells were positive for CK5/6 (Left), (×400) and p63 (Right), (×400)

Click here to view
Figure 5: Multinucleated giant cells were also observed (H and E, ×400)

Click here to view


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 Top

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.  Back to cited text no. 1
    
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.  Back to cited text no. 2
    
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.  Back to cited text no. 3
    
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.  Back to cited text no. 4
    
5.
Karakas C. Paget's disease of the breast. J Carcinog 2011;10:31.  Back to cited text no. 5
[PUBMED]  [Full text]  


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
    Article in PDF (1,929 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    References
    Article Figures

 Article Access Statistics
    Viewed524    
    Printed3    
    Emailed0    
    PDF Downloaded18    
    Comments [Add]    

Recommend this journal