Case Report
 

By Dr. Saber Boutayeb , Dr. Sophia Benomar , Prof. Badreddine Hassam , Prof. Hassan Errihani
Corresponding Author Dr. Saber Boutayeb
National Institute of Oncology, - Morocco
Submitting Author Mr. Saber Boutayeb
Other Authors Dr. Sophia Benomar
Dermatology Department, - Morocco

Prof. Badreddine Hassam
Dermatology Department, - Morocco

Prof. Hassan Errihani
Medical Oncology Department, - Morocco

CANCER

Adenoma, Nipple, Breast, Male

Boutayeb S, Benomar S, Hassam B, Errihani H. Adenoma Of The Nipple In A Man: Case Report. WebmedCentral CANCER 2010;1(9):WMC00605
doi: 10.9754/journal.wmc.2010.00605
No
Submitted on: 12 Sep 2010 12:04:49 AM GMT
Published on: 12 Sep 2010 12:29:09 AM GMT

Abstract


Adenomatosis (adenoma, papillary adenoma, florid papillomatosis) of the nipple is a rare benign disorder involving the nipple witch can be mistaken clinically for Paget's disease and pathologically be misinterpreted as an adenocarcinoma. It occurs mostly in middle-aged women and is extremely rare in adolescents and children.  Adenomatosis of the nipple is a well-known in women, but extremely rare in males.
We report a 55-year-old man with an adenomatosis of the nipple evolving positively after surgery.

Introduction


Adenomatosis of the nipple (AN), also called superficial papillary adenomatosis, erosive adenomatosis or florid papillomatosis, is an uncommon benign mammary disease that occurs mostly in middle-aged women and is extremely rare in men and children. This report describes a new case of a male patient clinically and histologically diagnosed with adenoma of the nipple.
A 55-year-old man, without significant previous medical history, consulted in the department of dermatology for a mass lesion of his right nipple that appeared 6 months before and grew gradually.
Physical examination revealed an irregular nodule on the right nipple area with edema. The mass, measuring 21 mm x 13 mm, was flesh-coloured, swelling, non adherent to the right breast and with no attachment to pectoral muscle. The adjacent skin did not present eczema, inflammation or ulceration (Fig 1).
The left breast and nipple were normal (Fig.2). There were no palpable axillary nodes.  The ultrasonography showed a hypoechoic irregular nodule measuring 19.5 mm in the nipple area with no other abnormal findings on the ultrasonography of the right nipple, breast and axillary area.
A biopsy specimen revealed a proliferation of gland like structures in which the tubules were lined with a double layer of epithelial cells, an outer myoepithelial layer of cuboidal cells. These findings were consistent with the diagnosis of AN (Fig.3)
Once pre-operative blood tests were achieved, a complete resection of the nipple with reconstruction was performed. After excision of the nipple, the histological study made a definite diagnosis of AN.
The patient was pleased with the results because the architecture of the nipple was preserved (Fig. 4). Our patient has no signs of progression of the disease in 13 months of follow-up.

Case Report(s)


A 55-year-old man, without significant previous medical history, consulted in the department of dermatology for a mass lesion of his right nipple that appeared 6 months before and grew gradually.
Physical examination revealed an irregular nodule on the right nipple area with edema. The mass, measuring 21 mm x 13 mm, was flesh-coloured, swelling, non adherent to the right breast and with no attachment to pectoral muscle. The adjacent skin did not present eczema, inflammation or ulceration (Fig 1).
The left breast and nipple were normal (Fig.2). There were no palpable axillary nodes.  The ultrasonography showed a hypoechoic irregular nodule measuring 19.5 mm in the nipple area with no other abnormal findings on the ultrasonography of the right nipple, breast and axillary area.
A biopsy specimen revealed a proliferation of gland like structures in which the tubules were lined with a double layer of epithelial cells, an outer myoepithelial layer of cuboidal cells. These findings were consistent with the diagnosis of AN (Fig.3)
Once pre-operative blood tests were achieved, a complete resection of the nipple with reconstruction was performed. After excision of the nipple, the histological study made a definite diagnosis of AN.
The patient was pleased with the results because the architecture of the nipple was preserved (Fig. 4). Our patient has no signs of progression of the disease in 13 months of follow-up.

Discussion


Adenomatosis of the nipple (AN) is a complex benign mammary proliferation, first described in 1955 as "florid papillomatosis of the nipple duct" by Jones and al [1].  
AN occurs mostly in middle-aged women and is extremely rare in men and children. Most patients with AN  are in their fourth or fifth decade of life.
In their review of literature, Montemarano and al reported only five cases in men [2, 3].
The lesion may be asymptomatic or characterized by a serous or serosanguinous discharge, tenderness, crusting, pruritus, erythema, swelling or induration. Nipple adenoma is often clinically misdiagnosed as Paget’s disease of the breast [3, 4].
In the World Health Organization classification of breast tumour, established in 2003, AN is defined as “a compact proliferation of small tubules lined by epithelial and myoepithelial cells, with or without proliferation of the epithelial component, around the collecting ducts of the nipple.” [5].
Histologically, it is sometimes difficult to distinguish nipple adenoma from carcinoma arising in the nipple. The presence of a myoepithelial cell layer in neoplastic ducts is thought to be the most important histological finding for distinguishing adenoma from carcinoma [6].
Dermatoses of the nipple are rare, and because gross appearances of these lesions are very similar, differential diagnosis is of great clinical importance. Early lesions are scaly and erythematous, and they can be misdiagnosed as eczema or inflammatory skin disorders of the nipple and treated with topical medication.
Benign tumours may include mammary duct ectasia, nipple calcifications, abscess of the Montgomery gland, and rarely nipple adenoma. Malignant abnormalities may include Paget disease and primary lymphoma as well as carcinoma of the breast.
For most authors, the treatment of choice for AN is a limited local excision. Complete or partial resection of the nipple depending on the size and extent of the tumour. Only patients with large lesions need a complete resection of the nipple. In some cases, nipple reconstruction should be done [7].
Handley and al advocated the total excision of the nipple and the areola with an underlying wedge of breast. However, such procedures seem to be overly aggressive for a benign disease. [8]. Kuflik and al described successful treatment with cryosurgery [9].
The prognosis of AN is excellent. A complete adequate excision of the lesion is curative without any risk of recurrence or development of malignancy. At the opposite, recurrence can occur if initial excision is incomplete [7, 9].
The majority of nipple adenomas are entirely benign, although rare examples have shown coincidental presentation with breast carcinoma [10].
Our patient is one of the rare men specimens. Curiously, the adjacent skin of the nipple was no ulcerated despite the tumour size. Concerning the treatment, our surgical team chooses the option of complete resection of the nipple, conserving the areola, with an immediate reconstruction. The histological examination of the excised nipple showed a complete histological resection.  Thirteen months later, the follow-up didn’t show any recurrence
Although AN is a rare disease, this diagnosis should be evocated in case of mass’s nipple in man.

References


1. Jones DB. Florid papillomatosis of the nipple ducts. Cancer 1955; 8: 315–19
2. Montemarano AD, Sau P, James WD. Superficial papillary adenomatosis of the nipple: a case report and review of the literature. J Am Acad Dermatol 1995; 33:871-5.
3. Ishii N,  Kusuhara M, Yasumoto S,  Hashimoto T. Adenoma of the nipple in a Japanese man. Clin Exp Dermatol 2007 ;32:448-9.
4. 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
5. Moulin G, Darbon P, Balme B, Frappart L. Erosive adenomatosis of the nipple: report of 10 cases with immunohistochemistry. Ann Dermatol Venereol 1990; 117:537-45.
6. Miller L, Tyler W, Maroon M. Erosive adenomatosis of the nipple: a benign imitator of malignant breast disease. Cutis 1997 Feb; 59:91-2.
7.  Hye JL, Kee YC. Erosive adenomatosis of the nipple: conservation of nipple by Mohs micrographic surgery. J Am Acad Dermatol  2002; 47: 578–80.
8.  Handley RS, Thackray AC. Adenoma of nipple. Br J Cancer 16: 187-194, 1962.
9. Kuflik EG. Erosive adenomatosis of the nipple treated with cryosurgery. J Am Acad Dermatol 1998; 38: 270–1.  
10. Kono S, Kurosumi M, Simooka H, Kawanowa K, Takei H, Suemasu K. Nipple Adenoma Found in a Mastectomy Specimen: Report of a Case with Special Regard to the Proliferation Pattern.  Breast Cancer. 2007; 14 :234-8.

Source(s) of Funding


none

Competing Interests


The authors have indicated no significant interest with commercial supporters and no conflict of interest.

Disclaimer


This article has been downloaded from WebmedCentral. With our unique author driven post publication peer review, contents posted on this web portal do not undergo any prepublication peer or editorial review. It is completely the responsibility of the authors to ensure not only scientific and ethical standards of the manuscript but also its grammatical accuracy. Authors must ensure that they obtain all the necessary permissions before submitting any information that requires obtaining a consent or approval from a third party. Authors should also ensure not to submit any information which they do not have the copyright of or of which they have transferred the copyrights to a third party.
Contents on WebmedCentral are purely for biomedical researchers and scientists. They are not meant to cater to the needs of an individual patient. The web portal or any content(s) therein is neither designed to support, nor replace, the relationship that exists between a patient/site visitor and his/her physician. Your use of the WebmedCentral site and its contents is entirely at your own risk. We do not take any responsibility for any harm that you may suffer or inflict on a third person by following the contents of this website.

Reviews
1 review posted so far

Adenoma of the nipple in a man: case report
Posted by Prof. Meral Guzey on 20 Jul 2012 12:17:29 PM GMT

Comments
0 comments posted so far

Please use this functionality to flag objectionable, inappropriate, inaccurate, and offensive content to WebmedCentral Team and the authors.

 

Author Comments
0 comments posted so far

 

What is article Popularity?

Article popularity is calculated by considering the scores: age of the article
Popularity = (P - 1) / (T + 2)^1.5
Where
P : points is the sum of individual scores, which includes article Views, Downloads, Reviews, Comments and their weightage

Scores   Weightage
Views Points X 1
Download Points X 2
Comment Points X 5
Review Points X 10
Points= sum(Views Points + Download Points + Comment Points + Review Points)
T : time since submission in hours.
P is subtracted by 1 to negate submitter's vote.
Age factor is (time since submission in hours plus two) to the power of 1.5.factor.

How Article Quality Works?

For each article Authors/Readers, Reviewers and WMC Editors can review/rate the articles. These ratings are used to determine Feedback Scores.

In most cases, article receive ratings in the range of 0 to 10. We calculate average of all the ratings and consider it as article quality.

Quality=Average(Authors/Readers Ratings + Reviewers Ratings + WMC Editor Ratings)