Introduction

Hidradenoma papilliferum (HP) is an uncommon, benign, cystic, papillary tumor that occurs primarily in the vulva of Caucasian women, thought to arise from anogenital glands, and exhibits both eccrine and apocrine differentiation, with the latter being more common.[₁] Bartholin’s abscess is also an anogenital cystic lesion caused by partial or complete ductal obstruction with abscess formation subsequently following infection.[₂] Concomitant presentation of Bartholin’s abscess and Hidradenoma papilliferum is unique. We report to the best of our knowledge the first case of Bartholin’s abscess arising in hidradenoma papilliferum.

Case Report

A 43 year-old African American woman presented with a painful cystic mass on the left labia majora. Preoperatively, the lesion was diagnosed as Bartholin’s abscess. During the excision and drainage, an additional 2.0 x 0.8 x 0.8 cm tan-brown dermal nodule was identified and submitted for histological analysis. Microscopic examination revealed multiple pieces of fibro-necrotic tissues (Fig. 1) associated with a well-circumscribed papillary neoplasm with cystic dilation (Fig. 2). The papillary projections and cystic areas were lined by basophilic cuboidal to columnar cells with outer compressed myoepithelial cells (Fig. 3). There were foci of active decapitation secretion and apocrine differentiation (Fig. 4). The diagnosis of Bartholin’s abscess arising in hidradenoma papilliferum was made. Although significant clinical correlations between these two lesions are not well understood at this point, the intimate association of hidradenoma papilliferum with Bartholin’s abscess suggests partial or complete obstruction of ductal drainage by the tumor mass.

Figure 1

Figure 1: Bartholin’s abscess wall demonstrating fibro-necrotic tissues with acute and chronic inflammation. (Hematoxylin-eosin stain; original magnification: x10)

Figure 2

Figure 2: Papillary neoplasm with cystic dilation. (Hematoxylin-eosin stain; original magnification: x10)

Figure 3

Figure 3: The papillary projections and cystic areas are lined by basophilic cuboidal to columnar cells with outer compressed myoepithelial cells. (Hematoxylin-eosin stain; original magnification: x10)

Figure 4

Figure 4: Hidradenoma papilliferum. Papillary neoplasm with foci of apocrine differentiation and cystic dilation. (Hematoxylin-eosin stain; original magnification: x10)

Discussion

Instances of pathology occurring concurrently with Bartholin's abscess or cyst do exist. Literature describes a case of cellular angiofibroma for which the preoperative diagnosis was Bartholin's glandular cyst.[₃] Another case report described a vulvar mass that was preoperatively diagnosed as a Bartholin's duct cyst but found to be leiomyosarcoma, a rare gynecologic malignancy.[₄]

Bartholin’s glands are bilaterally located at the base of the labia minora and drain through 2- to 2.5-cm-long ducts that empty into the vestibule at the 4 o’clock and 8 o’clock positions.[₅₆] Hidradenoma papilliferum usually presents as a slow-growing, small (2 mm to 3 cm) nodule which most commonly arise from the apocrine sweat glands between the labia majora and labia minora.[₇] Woodworth et al. reported 38% of hidradenoma papilliferum originate from the labia majora and 26% originate from the labia minora.[₈] Occurrence of hidradenoma papilliferum in the labia minora certainly presents the possibility of disrupting the ducts of Bartholin’s glands which are located within the vicinity.

Due to hidradenoma papilliferum’s mixed eccrine and apocrine features, cyst or abscess formation secondary to the disruption of ductal drainage should be considered.[₈] Hidradenoma papilliferum should be present in the differential in the context of recurrent cysts/abscesses in the anogenital region.[₉] The clinical differential diagnosis of a Bartholin’s abscess, which includes mucous cysts, epidermal inclusion cysts, and hidradenoma papilliferum,[₉] demonstrates the importance of maintaining an awareness of differentiation between Bartholin’s abscess and hidradenoma papilliferum.

Diagnostically, hidradenoma papilliferum tends to occur exclusively in post-pubescent white women between the ages of 30 and 70, occurring most commonly in the fourth decade.[₈] Interestingly, our diagnosis of Bartholin’s abscess arising in hidradenoma papilliferum was made in an African American woman, whereas, hidradenoma papilliferum tends to occur primarily in Caucasian women.[₉]

Though it has been suggested surgical excision of Bartholin’s abscess is unnecessary due to the low risk of Bartholin’s gland cancer,[₁₀] our finding of hidradenoma papilliferum and other mentioned neoplasms demonstrates the need for an increased level of suspicion with a preoperative diagnosis of Bartholin’s abscess. Gynecologic oncology referral is also supported in patients older than 40 years of age to rule out adenocarcinoma.[₁₁]

The clinical presentation of hidradenoma papilliferum becomes increasingly indistinct when simultaneously occurring with other cystic lesions of the vulva. A Bartholin’s abscess warrants further histological analysis when hidradenoma papilliferum and other tumors are present in the differential diagnosis.

Correspondence to

Salvatore Docimo, Jr.
Anatomy and Pathology Academic Medicine Fellow
New York College of Osteopathic Medline
Phone: 203-554-8114
Fax: 516-686-7440