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LETTER TO THE EDITOR
Year : 2018  |  Volume : 9  |  Issue : 5  |  Page : 346-348  

Tubular apocrine adenoma on vulva: An unusual location


Department of Dermatology, Seth GS Medical College and KEM Hospital, Mumbai, Maharashtra, India

Date of Web Publication4-Sep-2018

Correspondence Address:
Atul M Dongre
Department of Dermatology, Seth GS Medical College and KEM Hospital, Mumbai - 400 012, Maharashtr
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/idoj.IDOJ_343_17

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How to cite this article:
Jain S, Valiulla K, Dongre AM, Malkar BV. Tubular apocrine adenoma on vulva: An unusual location. Indian Dermatol Online J 2018;9:346-8

How to cite this URL:
Jain S, Valiulla K, Dongre AM, Malkar BV. Tubular apocrine adenoma on vulva: An unusual location. Indian Dermatol Online J [serial online] 2018 [cited 2021 Apr 18];9:346-8. Available from: https://www.idoj.in/text.asp?2018/9/5/346/240532



Sir,

A 36-year-old female presented with an asymptomatic lesion over the vulval area for the last 2 years. Cutaneous examination revealed a single, well-circumscribed smooth surfaced, skin colored, firm, nontender, 0.5 cm × 0.5 cm × 0.5 cm nodule located at the right interlabial sulcus [Figure 1]. Inguinal lymph nodes were not enlarged on either side. Our differential diagnoses were epidermoid cyst and hidradenoma papilliferum. Histopathological examination showed a well-demarcated, unencapsulated neoplasm in the dermis and subcutis, extending up to the base of biopsy specimen [Figure 2]a. It was composed of multiple neoplastic cell nests that contained oval and irregular tubules [Figure 2]b. These tubules were lined by an epithelium, which was mainly two-layered, but also multilayered at some places. The inner layer was formed by cuboidal cells that showed decapitation secretion, and the outer layer was composed of two types of cells: flattened and cuboidal [Figure 2]c. The intervening stroma was minimal and fibrous in nature, with virtually no inflammatory infiltrate. On clinicopathological correlation, the diagnosis of tubular apocrine adenoma was made and complete surgical excision was advised.
Figure 1: A smooth surfaced, skin-colored, rounded nodule located at the right interlabial sulcus

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Figure 2: (a) Well-demarcated, unencapsulated neoplasm in the dermis and subcutis, composed of scattered neoplastic cell nests (H and E, ×25). (b) Neoplastic cell nests contained oval and irregular tubules, with minimal intervening fibrous stroma with no inflammatory infiltrate (H and E, ×100). (c) Tubules lined by two-layered epithelium, inner cuboidal (showing decapitation secretion), and outer flattened cells (H and E, ×200)

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Tubular apocrine adenomais a rare benign adnexal neoplasm that exhibits apocrine differentiation. It was first described by Landry and Winkelmann in 1972.[1] It usually presents as a slow growing well-circumscribed nodule. The most common site of involvement is the scalp. Although it has also been reported to occur at other sites like face, neck, axilla, trunk, upper and lower extremities, but female genitalia is a rarely documented site.[2]

Recently, anogenital mammary-like glands have been identified as a type of cutaneous adnexal gland, localized to the anogenital area of both sexes, with characteristics of modified eccrine and apocrine glands. It has been suggested that the lesions arising in anogenital area like apocrine adenoma, fibroadenoma, hidrocystoma, and hidradenoma papilliferum may originate from these glands. Hence, tubular apocine adenoma is likely a type of adenoma of anogenital mammary-like glands.[3]

Histopathologically, it is a well-demarcated, unencapsulated neoplasm located in the dermis, sometimes extending in the subcutis. It is composed of scattered neoplastic cell nests that contain round, oval, or irregular tubules, lined by two-layer or multilayered epithelium. The inner layer is formed by cuboidal or columnar epithelial cells that often show decapitation secretion and the outer layer is composed of basal cells or myoepithelial cells. The stroma is minimal and fibrous in nature, with paucity of inflammatory cell infiltrate.[4]

Tubular apocrine adenoma, syringocystadenoma papilliferum, and apocrine cystadenoma (apocrine hidrocystoma) constitute neoplasms with apocrine differentiation. As per recent study by Ansai et al., all these three neoplasms have similar components, but they show different architecture. The authors also described composite entities that showed features of two or three of these neoplasms, which they labeled as tubulopapillary cystic adenoma with apocrine differentiation. They concluded that these three neoplasms represent parts of the same spectrum.[4]

Syringocystadenoma papilliferum is an endophytic neoplasm. It shows cystic invaginations from the surface epidermis, which contain papillary projections that are lined by two-layer epithelium, inner columnar, and outer cuboidal or flattened cells. Inner columnar cells show decapitation secretion. The stroma usually contains numerous plasma cells. Apocrine cystadenoma is characterized by unilocular or multilocular cysts, lined by two-layer epithelium, inner cuboidal or columnar with decapitation secretion, and outer cuboidal or elongated myoepithelial cells.

Though tubular apocrine adenoma is a benign neoplasm; however it may be locally aggressive sometimes, as evidenced by reports of perineural invasion.[5] A case of invasive apocrine adenocarcinoma developing in a benign apocrine adenoma located at perianal region has been reported in a 45-year-old woman.[6] The differentiation of tubular apocrine adenoma from apocrine adenocarcinoma is based on the lack of infiltration of surrounding tissues, the absence of cellular atypia and the presence of myoepithelial cells in the former.

Treatment of this neoplasm is complete surgical excision. Successful management of this neoplasm by Mohs micrographic surgery has also been reported.[7] The latter treatment has been justified in cases where the neoplasm has ill-defined margins. In our case, the neoplasm showed well-defined margins and hence we advised complete surgical excision in our patient.

In 2008, El Demellaway et al. reported a case of tubular apocrine adenoma in a 58-year-old female, who presented with a well-circumscribed nodule on left labia.[8] Subsequently, Ross et al. in 2010, documented an 8-year-old girl with tubular apocrine adenoma, who had a single, dark red-brown papule on posterior vaginal wall.[9] Recently, Ansai et al. reported seven cases of tubular apocrine adenoma on genitalia (including both male and female) in a large series, of which four cases were pure tubular apocrine adenoma and three were composite lesions (one showing features of both tubular apocrine adenoma and syringocystadenoma papilliferum, and two with features of both tubular apocrine adenoma and apocrine cystadenoma).[4]

Tubular apocrine adenoma is rarely reported on female genitalia. There are about 11 reports of this neoplasm occurring on genitalia, including both male and female, in the literature till date. We report this case due to unfamiliarity of this rare neoplasm and its development on a less commonly documented site.

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.
Landry M, Winkelmann RK. An unusual tubular apocrine adenoma. Arch Dermatol 1972;105:869-79.  Back to cited text no. 1
    
2.
El Demellawy D, Daya D, Alowami S. Vulvar apocrine tubular adenoma: An unusual location. Int J Gynecol Pathol 2008;27:301-3.  Back to cited text no. 2
    
3.
Ahmed S, Campbell RM, Li JH, Wang LJ, Robinson-Bostom L. Adenoma of anogenital mammary-like glands. J Am Acad Dermatol 2007;57:896-8.  Back to cited text no. 3
    
4.
Ansai SI, Anan T, Fukumoto T, Saeki H. Tubulopapillary cystic adenoma with apocrine differentiation: A unifying concept for syringocystadenoma papilliferum, apocrine gland cyst, and tubular papillary adenoma. Am J Dermatopathol 2017;39:129-37.  Back to cited text no. 4
    
5.
Burket JM, Zelickson AS. Tubular apocrine adenoma with perineural invasion. J Am Acad Dermatol 1984;11:639-42.  Back to cited text no. 5
    
6.
MacNeill KN, Riddell RH, Ghazarian D. Perianal apocrine adenocarcinoma arising in a benign apocrine adenoma; first case report and review of the literature. J Clin Pathol 2005;58:217-9.  Back to cited text no. 6
    
7.
Martinelli PT, Cohen PR, Schulze KE, Tschen JA, Nelson BR. Mohs micrographic surgery for tubular apocrine adenoma. Int J Dermatol 2006;45:1377-8.  Back to cited text no. 7
    
8.
El Demellawy D, Daya D, Alowami S. Vulvar apocrine tubular adenoma: An unusual location. Int J Gynecol Pathol 2008;27:301-3.  Back to cited text no. 8
    
9.
Ross I, Shwayder T. Tubular apocrine adenoma: Presentation in the vaginal introitus of an eight-year-old. Pediatr Dermatol 2010;27:200-1.  Back to cited text no. 9
    


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  [Figure 1], [Figure 2]



 

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