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Ectopic hidradenoma papilliferum* * Work conducted at Dermatology Service, Hospital Universitário Regional do Norte do Paraná; Universidade Estadual de Londrina, Londrina (PR), Brazil.

Dear Editor,

We report a case of a 36-year-old male patient with a two-month history of an asymptomatic, erythematous, 1 x 1cm papule of fibroelastic consistency on the left areola (Figure 1). The left axillary lymph nodes were not enlarged.

Figure 1
A - An erythematous papule on left areola . B - Detail of the lesion

We performed an excisional biopsy of the papule for diagnosis. Histopathology revealed a dermal tumor with no connection to the overlying epidermis. The lesion contained cells with papillary folds, tubules, cystically dilated spaces, and lumen lined by columnar cells with decapitation secretion, compatible with hidradenoma papilliferum (HP) (Figures 2 and 3).

Figure 2
A - Microscopic examination: dermal tumor with papillary folds and no connection to the overlying epidermis (Hematoxylin & eosin, X100). B - Lumen lined by columnar cells with decapitation secretion. (Hematoxylin & eosin, X400)
Figure 3
Histopathological: transverse section (Hematoxylin & eosin, X400)

HP is a rare, benign, adnexal tumor that occurs almost exclusively in the anogenital region of female patients.11 Tanaka M, Shimizu S. Hidradenoma papilliferum occurring on the chest of a man. J Am Acad Dermatol. 2003;48:S20-1.

2 Virgili A, Marzola A, Corazza M. Vulvar Hidradenoma Papilliferum. A review of 10.5-year's experience. J Reprod Med. 2000 ;45:616-8.

3 Handa Y, Yamanaka N, Inagaki H, Tomita Y.. Large Ulcerated Perianal Hidradenoma Papilliferum in a Young Female. Dermatol Surg. 2003;29:790-2.
-44 Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5. HP appears as solitary, asymptomatic, well-circumscribed, skin-colored or reddish-brown nodules measuring from 0.5 to 1cm. HP lesions that are not located in the anogenital area are referred to as ectopic HP.11 Tanaka M, Shimizu S. Hidradenoma papilliferum occurring on the chest of a man. J Am Acad Dermatol. 2003;48:S20-1.,44 Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5.,55 Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9.

Many cases of HP have been reported since it was first described by Werth in 1878.55 Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9. However, very few cases of ectopic HP have been reported in male patients.44 Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5. Ectopic HP can occur on the eyelids, orbit, nose, breast, chest, abdomen, and scalp. 44 Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5.,55 Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9.

The histogenesis for both anogenital and ectopic HP remains unclear. Although the presence of human papillomavirus (HPV) DNA has been identified in a few cases, it does not seem to play a role in the pathogenesis of HP. Histopathological findings show dermal tumors with no connection to the overlying epidermis, with papillary and tubular formations, cystically dilated spaces, and lumen lined by columnar cells with decapitation secretion. Such histopathology findings are similar to those observed in intraductal papilloma of the breast and some authors believe that accessory mammary-like glands may have a role in the histogenesis of the tumor. 55 Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9.

The actual incidence and prevalence of ectopic HP is unknown due to the low number of cases reported. Kim et al. compiled the clinical data available from all 19 patients previously reported. Despite the small sample, some aspects are wotth mentioning. The mean age of patients with ectopic HP ranged from 40 to 59 years (10 to 20 years more than the mean age for patients with anogenital HP). In contrast to anogenital HP, nearly one half of the patients with ectopic HP are men and lesions are located mainly on the head and neck. 44 Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5.

Histological examination is required for the accurate diagnosis of HP.55 Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9. The differential diagnosis for ectopic HP includes benign adnexal tumors, such as apocrine hidrocystoma and syringocystadenoma papilliferum.55 Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9. In our case, we included clinical differential diagnosis of basal cell carcinoma, epidermal cyst, and pyogenic granuloma.

Malignant transformation is very rare. Five cases of ductal carcinoma in situ arising within a pre-existing hidradenoma papilliferum have been documented, and two cases have reported invasive carcinomas arising from HP (malignant perianal papillary hidradenoma and vulvar adenosquamous carcinoma).55 Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9.

The treatment of choice for ectopic HP is complete excision of the tumor,44 Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5. as in the present case, and recurrence is typically attributed to incomplete excision. 11 Tanaka M, Shimizu S. Hidradenoma papilliferum occurring on the chest of a man. J Am Acad Dermatol. 2003;48:S20-1.,44 Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5.

  • Financial support: None.
  • *
    Work conducted at Dermatology Service, Hospital Universitário Regional do Norte do Paraná; Universidade Estadual de Londrina, Londrina (PR), Brazil.

REFERENCES

  • 1
    Tanaka M, Shimizu S. Hidradenoma papilliferum occurring on the chest of a man. J Am Acad Dermatol. 2003;48:S20-1.
  • 2
    Virgili A, Marzola A, Corazza M. Vulvar Hidradenoma Papilliferum. A review of 10.5-year's experience. J Reprod Med. 2000 ;45:616-8.
  • 3
    Handa Y, Yamanaka N, Inagaki H, Tomita Y.. Large Ulcerated Perianal Hidradenoma Papilliferum in a Young Female. Dermatol Surg. 2003;29:790-2.
  • 4
    Kim YJ, Lee JW, Choi SJ, Kim SJ, Kim YJ, Jeon YS, ET AL. Ectopic Hidradenoma Papilliferum of the Breast: Ultrasound Finding. J Breast Cancer. 2011;14:153-5.
  • 5
    Theodosiou G, Zafeiriadou V, Papageorgiou M, Mandekou-Lefaki I. An unusual lesion in the right place. Dermatol Pract Concept. 2016;6:7-9.

Publication Dates

  • Publication in this collection
    May-Jun 2018

History

  • Received
    22 Aug 2017
  • Accepted
    26 Nov 2017
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