© 2003 Arányi Lajos Foundation PATHOLOGY ONCOLOGY RESEARCH Vol 9, No 3, 2003 Article is available online at http://www.webio.hu/por/2003/9/3/0196 CASE REPORT Vulvar Syringoma Aggravated by Pregnancy Nebil BAL, 1 Erdogan ASLAN, 2 Fazilet KAYASELÇUK, 1 Ebru TARIM, 2 Ilhan TUNCER 1 Departments of 1 Pathology and 2 Gynecology and Obstetric, Baskent University Faculty of Medicine, Adana Teaching and Medical Research Center, Adana, Turkey Introduction Syringomas are benign tumors of eccrine sweat gland derivation that occur more frequently in women. These neoplasms usually develop at puberty. Clinically, they appear as multiple, tiny, firm, skin-colored papules. The sites of predilection are the eyelids, malar regions, neck, and chest. 1-8 Syringoma limited to the vulva is rare. Most syringomas on the face are symptomatic and permanent, whereas most vulvar syringomas are asymptomatic and therefore overlooked. Uncommonly, vulvar syringomas may itch. Vulvar syringoma should be included in the dif- ferential diagnosis for itchy and papular lesions of vulvae. 1-3,6 We present the case of a 24-year-old woman with vulvar syringoma that was exacerbated during pregnancy. Case Report The patient was a 24-year-old gravida 1, para 1 female who presented with the complaint of genital itching. She had experienced intermittent vulvar pruritus for 12 years, and had noted no change in her symptoms during menstruation. She had no history of eczema or contact hypersensitivity. Physical examination revealed multiple, soft, yellowish- to-skin-colored, 1-2 mm-diameter papules on the vulva. Received: May 9, 2003; accepted: June 12, 2003 Correspondence: Nebil BAL, MD, Baskent Üniversitesi, Patoloji A.D., Adana Hastanesi, Yüredir 01250, Adana Turkey Tel:+90 322 327 2727 Fax: +90 322 3271276 E-mail: nebilbal@yahoo.com or nbal@baskent-adn.edu.tr Syringoma is a benign tumors of eccrine sweat gland. They appear as multiple, tiny, firm, skin-col- ored papules. Vulvar involvement of syringoma is rare. Only 24 cases with vulvar syringoma have been previously reported in the literature. The majority of patients with vulvar syringomas are asymptomatic. A case of syringoma of the vulva exacerbated during pregnancy is presented. The case appears remarkable for the experienced aggra- vated pruritic symptoms of the patient during her pregnancy. (Pathology Oncology Research Vol 9, No 3, 196–197, 2003) Keywords: pregnancy, syringoma, vulva No similar lesions were detected elsewhere on the body. The patient reported that the papular lesions intensified during her pregnancy, and that they were reduced in size and number after that pregnancy was terminated. The results of laboratory tests were normal. A biopsy of the lesion was performed after pregnancy was terminated and microscopic examination revealed the typical features of syringoma. There were numerous tubu- lar structures embedded in fibrous stroma in the papillary and reticular dermis (Figure 1). Each of these ductal struc- tures had a central lumen that was lined by two rows of epithelial cells. The ductal lumina contained amorphous debris (Figure 2). Based on these findings, the patient was diagnosed with vulvar syringoma. Immunohistochemical studies revealed no estrogen and progesterone receptors in the tumor cells. Discussion Syringomas are fairly common tumors of eccrine sweat ducts, but genital syringoma is rare. The first genital case of syringoma to be published was a penile tumor. Corneino et al. were the first to report a case of vulvar syringoma. 1-3 Most of these neoplasms are asymptomatic, and are detect- ed on routine gynecologic examination. 1 Clinically, the lesions appear as multiple, tiny, firm, skin-colored papules, each approximately 1-3 mm in diameter. Some are yellow- ish in color. In typical vulvar syringomas, the papules are bilateral and are symmetrically distributed. 1-5 Vulvar syringoma is an unusual clinical variant of syringoma. It is unusual for the vulvar form to present as pruritus vulvae; ¸ ¸