Correspondence

Ectopic sebaceous glands: a rare cause of vaginal swelling

Editor, A 46-year-old woman was referred with vulval pruritus. She was noted to have hyperpigmented areas on the labia minora, the histology of which confirmed benign melanosis, with no evidence of inflammatory change. Incidentally, vulval examination also revealed a firm, indurated, asymptomatic swelling 1 cm in diameter, which was visible at the lower end of the vagina, near the introitus. Histological examination showed keratinizing squamous epithelium with clusters of sebaceous glands associated with vestigial follicular structures within the superficial dermis (Fig. 1). Sebaceous glands are commonly found in association with hair follicles as a pilosebaceous unit. They can occur independently in non-hair bearing areas, such as on the buccal and labial mucosae as Fordyce spots, the nipple as Montgomery’s tubercles, and the penis as Tyson’s glands. The presence of sebaceous glands in the cervix and vagina, however, is rare given the difference in embryological origin. Sebaceous glands develop with hair follicles from the germinal layer of the epidermis and are ectodermal in origin. The upper third of the vagina, uterus, and cervical canal are derived from mesoderm. The remainder of the vaginal canal develops from the sinu–vaginal plate at the back of the urogenital sinus, which is of endodermal origin.

Two theories to explain this rare finding have been suggested.1 According to the first, this represents a metaplastic change of vaginal epithelial cells following prolonged irritation and/or inflammation. The second theory proposes that there is a developmental anomaly and aberrant differentiation occurs in utero. Evidence to support either hypothesis is sparse but favors the former given the wellknown ability of the cervix and vagina to undergo change following local trauma. To our knowledge, the presence of sebaceous glands in the vagina has been reported only twice before and not in the presence of ectopic skin. In the first case, sebaceous glands were found in the upper vagina on histological examination in a patient who had undergone surgery for cervical cancer.2 An ill-defined white area was noted on the upper vagina on internal vaginal examination in the second case.3 Both patients presented to gynecology, and in both cases, findings pertained to the upper vagina and thus would not have been obvious to a dermatologist on external vulval examination. The present case appears to be unique because sebaceous glands were found in the lower vagina and presented as an asymptomatic swelling on vulval examination. Ectopic sebaceous glands in the vagina are highly unusual and may present to both gynecologists and dermatologists. Although rare, the presence of such glands is worth considering as a cause of an asymptomatic vaginal swelling.

Shaheen S. Haque Hussain, MBBS, BSc, MRCP Pamela M. Todd, MBBS, BSc, FRCP, MEd Department of Dermatology Cambridge University Hospitals National Health Service (NHS) Foundation Trust Addenbrookes Hospital Cambridge UK E-mail: [email protected]

Figure 1 Histopathology of biopsy material from a 46-yearold woman with an asymptomatic swelling in the lower vagina shows keratinizing squamous epithelium with numerous sebaceous glands. (Hematoxylin and eosin stain; original magnification 94)

Ed Rytina, MBBS, PhD, FRCPath Department of Pathology Cambridge University Hospitals National Health Service (NHS) Foundation Trust Addenbrookes Hospital Cambridge UK

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ª 2014 The International Society of Dermatology

Correspondence

References 1 Ionescu DN, Mohan D, Carter G, et al. Epidermoid metaplasia of the cervix. Arch Pathol Lab Med 2004; 128: 1052–1053. 2 Belousova IE, Kazakov DV, Michal M. Ectopic sebaceous glands in the vagina. Int J Gynecol Pathol 2005; 24: 193– 195. 3 Rose M, Moore G. Epidermalization of cervix and vagina: an unsolved dilemma. J Low Genit Tract Dis 2008; 12: 217–219.

Hereditary benign telangiectasia in monozygotic twins with no family history

We present the case of 3-year-old twin girls (monochorionic diamniotic) with no past family or personal history of interest who presented asymptomatic skin lesions that appeared almost simultaneously a year before the consultation. The number and size of the lesions had increased since initial onset. The parents reported no previous trauma and no family history of similar lesions. Neither of the twins had a history of mucosal bleeding (oral or gastrointestinal bleeding or epistaxis). Physical examination showed erythematous pink macules with superficial telangiectasias on the neck, trunk, and upper extremities, the borders were burred, and the distribution was random, bilateral, and asymmetric (Fig. 1). Some lesions were surrounded by anemic halos (Fig. 1c). The lesions did not disappear when pressure was applied. No mucosal lesions were detected. Histological examination showed a normal epidermis and thinwalled dilated capillaries and postcapillary venules in the superficial dermis (Fig. 2). Eighteen months later, the lesions were stable and remained asymptomatic. Hereditary benign telangiectasia (HBT), a rare autosomal dominant genetic disorder first described by Wells and Dowling1 in 1971, is classified as a primary telangiectasia and considered a benign form of hereditary hemorrhagic telangiectasia.2 The lesions are acquired in childhood by patients with no history of hemorrhage or

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systemic disease. The cutaneous presentation can be very polymorphic (ranging from erythematous maculae to spider or punctiform telangiectasias), and the mucosas are not affected. The main histologic feature is dilatation of the vessels in the subpapillary venous plexus. The lesions are invariably asymptomatic but may constitute a cosmetic problem. Brancati et al.3 identified a link between HBT and the locus for capillary malformation (CM; named CMC1) on chromosome 5q14. The differential diagnosis should primarily include CMs. There is some debate as to whether CMs and HBT constitute two separate disorders or represent two forms of a single entity: an overlap between these two phenotypes has been reported in many families, a link with the same locus (CMC1) on chromosome 5 has been identified in both diseases, and the histologic features are the same in both conditions. These findings have led some authors to posit that CMs and HBT are in fact different presentations of a single disorder.3 Acquired port wine stain lesions that are not present at birth are relatively rare and have generally been associated with previous trauma. Furthermore, such lesions usually have a unilateral distribution and appear on the face or neck. Families in which

Figure 2 Dilated capillaries in the upper dermis (Hematoxylin-eosin, 9200)

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Figure 1 (a,b) Erythematous macules with telangiectasias on the chin and arm of the first twin. (c) Telangiectatic lesion with anemic halo on the back of the second twin. The arrow shows the site of skin biopsy ª 2014 The International Society of Dermatology

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Ectopic sebaceous glands: a rare cause of vaginal swelling.

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