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Table of Contents
CASE REPORT
Year : 2020  |  Volume : 38  |  Issue : 2  |  Page : 102-104

Nipple adenoma: A report of two cases


1 Department of Dermatology, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
2 Department of Pathology, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan
3 Department of Dermatology; Department of Dermatology, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan

Date of Submission05-May-2019
Date of Decision09-Aug-2019
Date of Acceptance19-Aug-2019
Date of Web Publication24-Apr-2020

Correspondence Address:
Dr. Cheng-Che E Lan
Department of Dermatology, Kaohsiung Medical University Hospital, Kaohsiung; No.100 , Tzyou 1st Road, Kaohsiung 807
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ds.ds_38_19

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  Abstract 


Nipple adenoma is a rare benign tumor of the nipple due to mammary proliferation of the lactiferous ducts. Common presentation of the tumor includes nipple erosion, erythema, scaling, and eczematous change of the overlying skin. Delayed diagnosis is common since its clinical presentation resembles that of other benign or malignant condition of the nipple, such as nipple eczema or Paget's disease. Timely identification with histopathological correlation is essential for appropriate management. We herein describe the clinical and histopathological characteristics of two cases of nipple adenoma.

Keywords: Lactiferous duct, myoepithelial cell, nipple adenoma, nipple erosion


How to cite this article:
Yang TT, Su YC, Lan CCE. Nipple adenoma: A report of two cases. Dermatol Sin 2020;38:102-4

How to cite this URL:
Yang TT, Su YC, Lan CCE. Nipple adenoma: A report of two cases. Dermatol Sin [serial online] 2020 [cited 2020 Jul 4];38:102-4. Available from: http://www.dermsinica.org/text.asp?2020/38/2/102/283209




  Introduction Top


Nipple adenoma is a rare benign tumor of the nipple caused by mammary proliferation of the lactiferous ducts. It often presents as an indurated nipple tumor with erythema or erosion of the overlying skin and clinically resembles Paget's disease of the nipple or nipple eczema.[1],[2],[3] In this report, we present two cases of nipple adenoma presenting as chronic nipple erosions.


  Case Report Top


Case 1

A 43-year-old woman with no significant medical history presented with an enlarging right nipple erosion for 6 years. She had visited many breast specialists for the above condition and received breast sonography and mammography, but both revealed no definite abnormalities. She was therefore referred to our dermatology department for further evaluation. On examination, superficial erosion with induration measuring up to 2 cm was noted at the inferior portion of the right nipple and surrounding areolar skin [Figure 1]. There was also blood-tinged serous discharge from the lesion. No axillary lymph nodes were palpable.
Figure 1: (a) Swelling of the right nipple with erosion. (b) Superficial ulcer with usual type ductal hyperplasia (H and E, ×20). (c) Proliferation of glands forming compact (arrowhead) and cribriform (arrow) patterns (H and E, ×40). (d) p63 highlights an intact myoepithelial layer (×40)

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A 3-mm punch biopsy was done at the junction of the erosion and adjacent areolar skin. Histopathological evaluation [Figure 1] showed a superficial ulcer with ductal hyperplasia forming compact and cribriform patterns. Cytological atypia was absent. p63 and cytokeratin (CK) 5/6 immunostaining demonstrated intact myoepithelial layer and proliferation of myoepithelial cells. The tumor cells were negative for gross cystic disease fluid protein 15 and Her-2/neu. Considering the clinical presentation and histologic features, the diagnosis favors nipple adenoma.

Since the nipple discharge significantly interfered with the patient's daily life, the patient underwent enucleation of the tumor with preservation of the rest of the nipple. Histopathological examination of the entire tumor showed nipple adenoma.

Case 2

A 51-year-old woman with no significant medical history was referred to our dermatology department with a painful chronic ulceration over the left nipple for more than 20 years. A 1.6 cm indurated ulcer causing deformity of the nipple profile was noted over the center portion of the left nipple. Some serous discharge was noted from the ulcer. Breast sonography showed no underlying tumor, and mammography showed only benign calcifications. Paget's disease was suspected.

A punch biopsy was obtained from the center of the erosion. Histopathological examination [Figure 2] showed proliferating glands within a sclerosing background with some glands extending to the epidermis causing erosion. The myoepithelium was highlighted by both CK 5/6 and p63. The histologic findings indicated nipple adenoma. Complete excision was suggested, but the patient refused any further treatment. No significant change of the lesion was reported by the patient at 2-year follow-up.
Figure 2: (a) Glandular proliferation causing superficial ulcer (H and E, ×100). (b) p63 highlights intact myoepithelium (×100). (c) Glandular epithelium and myoepithelial cells positive for cytokeratin 5/6 (×100)

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  Discussion Top


Nipple adenoma is a rare benign tumor of the nipple due to proliferation of the lactiferous ducts. It is also known by different names such as florid papillomatosis, erosive adenomatosis, and superficial papillary adenomatosis of the nipple.[4] It usually occurs in middle-aged women but also rarely occurs in men and children.[5],[6],[7] Nipple adenoma may present as an indurated tumor with or without erosion.[1],[2],[4],[8] Erythema, scaling, and eczematous change of the overlying skin are also commonly present.[1],[2],[4],[8] Same as both of our patients, 37% of patients report having nipple discharge.[8] Clinical symptoms include irritation, pruritus, or pain.[2],[4],[8] Consequently, it may be confused with nipple eczema, Paget's disease of the nipple, or other breast malignancies clinically. A skin biopsy is mandatory to make a correct diagnosis.

Since the tissue density of nipple adenoma is similar to that of normal breast tissue, it is rarely detected on conventional imaging modalities of the breast.[4],[8] Both of our cases showed no visible tumor on either breast sonography or mammography. More advanced imaging modalities, such as breast magnetic resonance imaging (MRI), may be used to assess the extent of involvement of the tumor.[9],[10] However, the results of contrast-enhanced MRI should be interpret with caution since the dynamic enhancement pattern of nipple adenoma may be confused with breast malignancy.[9]

The definite diagnosis of nipple adenoma relies on histopathological examination.[4] Histologically, adenoma of the nipple is characterized by a well-circumscribed tumor with proliferation of glandular and tubular structures lined with an inner layer of epithelial cells and an outer layer of myoepithelial cells.[2],[4],[8] Mitoses are sometimes seen, but cytological atypia is absent.[2],[4],[8] As observed in the two cases presented in this report, the glands may extend to the overlying epidermis and cause erosion or even ulceration. The presence of a myoepithelial cell layer is the key to differentiate nipple adenoma from invasive carcinoma of the breast.[2],[11] Immunohistostaining such as p63, caldesmon 1, calponin 1, smooth muscle actin, CK 5/6, and CK 8/18 can be used to highlight the myoepithelial cells and aid in the differential diagnosis.[2],[4],[8] For both of our patients, intact myoepithelial cell layers are demonstrated by positive p63 and CK 5/6 immunohistostaining. According to the WHO Classification of Tumours of the Breast, there exists four histologic subtypes: sclerosing papillomatosis, papillomatosis, adenosis, and mixed type.[11],[12] The papillomatosis subtype and mixed type are most commonly confused with Paget's disease or carcinoma clinically since surface ulceration and inflammation are commonly present.[4],[13] Both of our patients belong to the papillomatosis subtype and have ulceration over the nipple on presentation. However, overlapping of the subtypes is common and the histologic classification does not provide any prognostic significance.[12]

Complete surgical resection under local anesthesia is considered curative for nipple adenoma, but local recurrence may occur.[4],[8],[14] Successful management with minimally invasive procedures, including Mohs micrographic surgery or cryosurgery, has also been report but are restricted to early detected cases.[15],[16],[17],[18],[19] Without surgical excision, nipple adenoma will continue to enlarge and cause local destruction of the nipple architecture. Symptoms such as discharge and irritation of the nipple are also disturbing and severely affect the quality of life of patients. However, delayed diagnosis is common. The duration from lesion detection to diagnosis has been reported to range from 2 months to 10 years.[3] Destruction of the nipple architecture and requirement of extensive excision will result from late diagnosis.

Co-existence of nipple adenoma and breast cancer is well documented in the literature.[11],[12],[20],[21] Although breast cancer appears to be an incidental finding in most cases, carcinoma arising from the same location has been reported.[11],[12],[20],[21] The association between nipple adenoma and breast cancer still remains unclear. Regular follow-up of patients with nipple adenoma to exclude breast cancer is recommended.

In summary, nipple adenoma is a rare benign tumor which clinically mimics nipple eczema or malignant conditions of the breast. Both of the cases in this report were initially diagnosed as Paget's disease. Timely diagnosis with histopathological correlation is important since it allows for minimally invasive surgical methods, such as Mohs micrographic surgery, which provides adequate treatment efficacy and satisfactory cosmetic results.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patients have given their consent for 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.
Healy CE, Dijkstra B, Walsh M, Hill AD, Murphy J. Nipple adenoma: A differential diagnosis for Paget's disease. Breast J 2003;9:325-6.  Back to cited text no. 1
    
2.
Di Bonito M, Cantile M, Collina F, D'Aiuto M, Liguori G, De Cecio R, et al. Adenoma of the nipple: A clinicopathological report of 13 cases. Oncol Lett 2014;7:1839-42.  Back to cited text no. 2
    
3.
Brownstein MH, Phelps RG, Magnin PH. Papillary adenoma of the nipple: Analysis of fifteen new cases. J Am Acad Dermatol 1985;12:707-15.  Back to cited text no. 3
    
4.
Spohn GP, Trotter SC, Tozbikian G, Povoski SP. Nipple adenoma in a female patient presenting with persistent erythema of the right nipple skin: Case report, review of the literature, clinical implications, and relevancy to health care providers who evaluate and treat patients with dermatologic conditions of the breast skin. BMC Dermatol 2016;16:4.  Back to cited text no. 4
    
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Burdick C, Rinehart RM, Matsumoto T, O'Connell TJ, Heisterkamp CW. Nipple adenoma and Paget's disease in a man. Arch Surg 1965;91:835-9.  Back to cited text no. 5
    
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Fujisawa K, Kato M, Kono T, Utsunomiya H, Watanabe A, Watanabe S. Nipple adenoma in a 2-year-old boy. Pediatr Dermatol 2018;35:e184-5.  Back to cited text no. 6
    
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Albers SE, Barnard M, Thorner P, Krafchik BR. Erosive adenomatosis of the nipple in an eight-year-old girl. J Am Acad Dermatol 1999;40:834-7.  Back to cited text no. 7
    
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Wang C, Wang X, Ma R. Diagnosis and surgical treatment of nipple adenoma. ANZ J Surg 2015;85:444-7.  Back to cited text no. 8
    
9.
Adusumilli S, Siegelman ES, Schnall MD. MR findings of nipple adenoma. AJR Am J Roentgenol 2002;179:803-4.  Back to cited text no. 9
    
10.
Tsushimi T, Enoki T, Takemoto Y, Harada E, Hayashi M, Furuya T, et al. Adenoma of the nipple, focusing on the contrast-enhanced magnetic resonance imaging findings: Report of a case. Surg Today 2011;41:1138-41.  Back to cited text no. 10
    
11.
Eusebi V, Lester S. Tumours of the nipple. In: Lakhani SR, Ellis IO, Schnitt SJ, Tan PH, van de Vijver MJ, editors. WHO Classification of Tumours of the Breast. Vol. 4. Ch. 12. Lyon, France: IARC; 2012.  Back to cited text no. 11
    
12.
Rosen PP, Caicco JA. Florid papillomatosis of the nipple. A study of 51 patients, including nine with mammary carcinoma. Am J Surg Pathol 1986;10:87-101.  Back to cited text no. 12
    
13.
Rosen PP. Rosen's Breast Pathology. 3rd ed. Philadelphia: Lippincott Williams and Wilkins; 2009.  Back to cited text no. 13
    
14.
Fujii T, Yajima R, Morita H, Yamaguchi S, Tsutsumi S, Asao T, et al. Adenoma of the nipple projecting out of the nipple: Curative resection without excision of the nipple. World J Surg Oncol 2014;12:91.  Back to cited text no. 14
    
15.
Van Mierlo PL, Geelen GM, Neumann HA. Mohs micrographic surgery for an erosive adenomatosis of the nipple. Dermatol Surg 1998;24:681-3.  Back to cited text no. 15
    
16.
Barco I, Vidal M, Fraile M, Masferrer E, Barco L, Barco D, et al. MOHS micrographic surgery for treating erosive adenoma of the nipple: A case report and review of the literature. Int J Dermatol 2017;56:1451-4.  Back to cited text no. 16
    
17.
Owen JL, Krunic AL. Successful treatment of nipple adenoma using Mohs micrographic surgery to preserve the nipple-areolar complex Dermatol Surg doi: 10.1097/DSS.0000000000001839. [published online ahead of print February 14, 2019].  Back to cited text no. 17
    
18.
Kuflik EG. Erosive adenomatosis of the nipple treated with cryosurgery. J Am Acad Dermatol 1998;38:270-1.  Back to cited text no. 18
    
19.
Pasquali P, Freites-Martinez A, Fortuño A. Nipple adenoma: New images and cryosurgery treatment. Breast J 2016;22:584-5.  Back to cited text no. 19
    
20.
Jones MW, Tavassoli FA. Coexistence of nipple duct adenoma and breast carcinoma: A clinicopathologic study of five cases and review of the literature. Mod Pathol 1995;8:633-6.  Back to cited text no. 20
    
21.
Sasi W, Banerjee D, Mokbel K, Sharma AK. Bilateral florid papillomatosis of the nipple: An unusual indicator for metachronous breast cancer development-a case report. Case Rep Oncol Med 2014;2014:432609.  Back to cited text no. 21
    


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