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Apocrine cystadenoma of the vulva
498
Journal of the American Academy of Dermatology September 1994
Brief communications
Apocrine cystadenoma of the vulva Earl J. Glusac, MD,a, b Michael S. Hendrickson, Stanford, California
MD,a
and Bruce R. Smoller,
Apocrine cystadenoma CAe) is an uncommon tumor considered by some to be a proliferative variant of apocrine hidrocystoma.' The tumor is almost always solitary and occurs with greatest frequency on the face and other areas of the head and neck.? U nusuallocations include the thorax- 4 and anus.v 6 Rare cases on the penis have been reported?' 8; however, some have argued that these represent median raphe cysts." Surprisingly, no cases on the female genitalia have been reported to date. 10
MDa, b
'.
CASE REPORT A 34-year-old woman had a freely mobile, 3 em, fluctuant, dermal nodule on the left anterior labium major. The lesion was non tender and had been present for several years without enlargement. Microscopic examination of the excised specimen revealed a well-circumscribed, unilocular, cystic tumor located 3 mm beneath the vulvar epidermis. Surrounding the tumor was a layer of loosely woven fibrous tissue oriented parallel to the cyst wall. The cystic lining was plump columnar to focally cuboidal, well-differentiated apocrine epithelium (Fig. 1). The epithelial nuclei were uniform, round, basally situated, and contained prominent central nucleoli. The cytoplasm was abundant, eosinophilic, and exhibited prominent decapitation secretion. Focally, the epithelium formed small tufts and larger rounded papillary proliferations with delicate fibrovascular cores (Fig. 2). Beneath the epithelial layer was a focally discontinuous layer of elongated to cuboidal myoepithelial cells. Diastase-treated sections stained with periodic acid-Schiff reagent revealed red-staining granules within the apices of the apocrine epi thelial cells. This stain also highlighted the presence of a well-formed basement membrane zone. Prussian blue staining revealed blue granules within the apical portion of the epithelial cells. Immunoperoxidase labeling of paraffin-embedded sections was performed, employing a standard avidin-biotin-peroxidase technique.'! Antibodies to the breast/apocrine marker From the Departments of Pathology" and Dermatology," Stanford University Medical Center. Reprint requests: Bruce R. Smaller, MD, Department of Pathology, Stanford University Medical Center, 300 Pasteur Dr., Stanford, CA 94305. J AM ACAD DERMATOL 1994;31 :498-9. Copyright ® 1994 by the American Academy of Dermatology, Inc. 0190-9622(94 $3.00 + 0 16(54(54919
Fig. 1. Apocrine cystadenoma lined by columnar to cuboidal cells with decapitation secretion. A loose but organized fibrous stroma surrounds cyst wall. (Hematoxylin-eosin stain; X 100.)
Fig. 2. Delicate papillary proliferation within apocrine cystadenoma. Fine fibrovascular cores are lined by plump apocrine epithelial cells. (Hematoxylin-eosinstain; X250.)
GCDFP-15 (gross cystic fluid disease protein) labeled the apocrine epithelial cells. These cells did not label with antibodies to carcinoembryonic antigen or factor XlIIa, which was used as a control. DISCUSSION
Our case shows features consistent with previously reported cases of AC. Microscopically, the lesion is a cyst lined by columnar to cuboidal eosino-
Journal of the American Academy of Dermatology Volume 31, Number 3, Part I
philic cells with prominent decapitation secretion. 1 Focal papillary proliferations are typically present as well as a discontinuous myoepithelial layer as in this case. Labeling with GCDFP·15 supports the lesion's apocrine origin and is consistent with previously reported findings. 12 To our knowledge, this is the first case of AC reported to occur on the vulva. Ahmed and Jones 7 have speculated that AC has not been reported to arise on the female genitalia because neoplasia of the apocrine glands of the vulva takes the form of hidradenoma papilliferum (HP).7 However, this lesion differs from HP in several ways. HP is more densely proliferative and is composed of prominent curved and linear interlacing papillae lined by closely approximated tall columnar epithelial cells.P: 14 The stroma within the papillae of HP is more densely collagenous. The overall appearance of HP has been likened to a mammary intraductal papilloma. 10 By contrast, AC is more cystic and less proliferative. The papillary projections in our case and other reported cases of AC are more delicate and flowerlike in arrangement. The appearance is reminiscent of an ovarian cystadenoma. The papillary stroma in AC is also more delicate and poorly collagenized, and the epithelial cells are more plump with an abundance of granular cytoplasm. AC has been reported to arise in other areas rich in apocrine glands such as the face, chest, and anus. Why these lesions have not been reported to have arisen on the vulva, which is also rich in apocrine glands, is unclear. Controversy has arisen regarding the terms AC and apocrine hidrocystoma. Some authors have used the terms interchangeably and considered these lesions to be retention cysts. In contrast, Mehregan I has argued that A C is a distinct organoid growth rather than a simple retention cyst. His arguments are (I) the tumor is composed of high columnar
Brief communications
499
apocrine cells without flattening, (2) the tumor exhibits true papillomatous hyperplasia with central fibrovascular cores, and (3) the lesion has a circumferential, organized fibrous stroma characteristic of all cutaneous adnexal tumors. These features are present in our case, and tend to support Mehregan's supposition.
REFERENCES 1. Mehregan AH. Apocrine cystadenoma: a clinicopathologic study with special reference to the pigmented variety. Arch Dermatol 1964;90:274-9. 2. Smith JD, Chernosky M E. Apocrine hidrocystoma (cystadenoma). Arch Dermatol 1974;109:700-2. 3. Benich B,Peison B.Apocrine hidrocystoma of'theshoulder. Arch Dermatol 1977;113:71-2. 4. Ter Poorten HJ. Apocrine hidrocystoma of the right scapula [Letter]. Arch DermatoI1977;13:1730. 5. Von Seebach HV, Stumm D. Misch P, et al, Hidrocystoma and adenoma of apocrine anal glands. Virchows Arch [A] Pathol Anat Histopathol 1980;386:231-7. 6. Weigand DV. Burgdorf WHC. Perianal apocrine gland adenoma. Arch Dermatol 1980;116:1051-3. 7. Ahmed A, Jones AW. Apocrine cystadenoma. Br.T Dermatol 1969;81:899-901. 8. Powell RF, Palmer CH, Smith EB. Apocrine cystadenoma of the penile shaft. Arch Dermatol 1977; 113:1250-1. 9. Hashimoto K, Mehregan AH. Kumakiri M. Tumors of skin appendages. Boston: Butterworths, 1987:146. 10. Santa Cruz DJ. Tumors of sweat gland differentiation. In: Farmer ER, Hood AF, eds. Pathology of the skin. Norwalk: Appleton & Lange, 1990:641. 11. Bindl JM, Warnke RA. Advantages of detecting monoclonal antibody binding to tissue sections with biotin and avidin reagents in Coplin jars. Am J Clin Pathol 1986; 85:490-3. 12. Tsubura A, Senzaki H, Sasaki M, et al. Immunohistochemical demonstration of breast-derived and/or carcinoma-associated glycoproteins in normal skin appendages and their tumors. J Cutan Pathol 1992; 19:73-8. 13. Woodworth H. Dockerty MD, Wilson RB, et al. Papillary hidradenoma of the vulva: a clinicopathologic study of 69 cases. Am J Obstet Gynecol 1971;110:501-8. 14. Meeker JH, Neubecker RD, Helwig EB. Hidradenoma papilliferum. Am J Clin Pathol 1962;37:182-95.
Journal of the American Academy of Dermatology September 1994
Brief communications
Apocrine cystadenoma of the vulva Earl J. Glusac, MD,a, b Michael S. Hendrickson, Stanford, California
MD,a
and Bruce R. Smoller,
Apocrine cystadenoma CAe) is an uncommon tumor considered by some to be a proliferative variant of apocrine hidrocystoma.' The tumor is almost always solitary and occurs with greatest frequency on the face and other areas of the head and neck.? U nusuallocations include the thorax- 4 and anus.v 6 Rare cases on the penis have been reported?' 8; however, some have argued that these represent median raphe cysts." Surprisingly, no cases on the female genitalia have been reported to date. 10
MDa, b
'.
CASE REPORT A 34-year-old woman had a freely mobile, 3 em, fluctuant, dermal nodule on the left anterior labium major. The lesion was non tender and had been present for several years without enlargement. Microscopic examination of the excised specimen revealed a well-circumscribed, unilocular, cystic tumor located 3 mm beneath the vulvar epidermis. Surrounding the tumor was a layer of loosely woven fibrous tissue oriented parallel to the cyst wall. The cystic lining was plump columnar to focally cuboidal, well-differentiated apocrine epithelium (Fig. 1). The epithelial nuclei were uniform, round, basally situated, and contained prominent central nucleoli. The cytoplasm was abundant, eosinophilic, and exhibited prominent decapitation secretion. Focally, the epithelium formed small tufts and larger rounded papillary proliferations with delicate fibrovascular cores (Fig. 2). Beneath the epithelial layer was a focally discontinuous layer of elongated to cuboidal myoepithelial cells. Diastase-treated sections stained with periodic acid-Schiff reagent revealed red-staining granules within the apices of the apocrine epi thelial cells. This stain also highlighted the presence of a well-formed basement membrane zone. Prussian blue staining revealed blue granules within the apical portion of the epithelial cells. Immunoperoxidase labeling of paraffin-embedded sections was performed, employing a standard avidin-biotin-peroxidase technique.'! Antibodies to the breast/apocrine marker From the Departments of Pathology" and Dermatology," Stanford University Medical Center. Reprint requests: Bruce R. Smaller, MD, Department of Pathology, Stanford University Medical Center, 300 Pasteur Dr., Stanford, CA 94305. J AM ACAD DERMATOL 1994;31 :498-9. Copyright ® 1994 by the American Academy of Dermatology, Inc. 0190-9622(94 $3.00 + 0 16(54(54919
Fig. 1. Apocrine cystadenoma lined by columnar to cuboidal cells with decapitation secretion. A loose but organized fibrous stroma surrounds cyst wall. (Hematoxylin-eosin stain; X 100.)
Fig. 2. Delicate papillary proliferation within apocrine cystadenoma. Fine fibrovascular cores are lined by plump apocrine epithelial cells. (Hematoxylin-eosinstain; X250.)
GCDFP-15 (gross cystic fluid disease protein) labeled the apocrine epithelial cells. These cells did not label with antibodies to carcinoembryonic antigen or factor XlIIa, which was used as a control. DISCUSSION
Our case shows features consistent with previously reported cases of AC. Microscopically, the lesion is a cyst lined by columnar to cuboidal eosino-
Journal of the American Academy of Dermatology Volume 31, Number 3, Part I
philic cells with prominent decapitation secretion. 1 Focal papillary proliferations are typically present as well as a discontinuous myoepithelial layer as in this case. Labeling with GCDFP·15 supports the lesion's apocrine origin and is consistent with previously reported findings. 12 To our knowledge, this is the first case of AC reported to occur on the vulva. Ahmed and Jones 7 have speculated that AC has not been reported to arise on the female genitalia because neoplasia of the apocrine glands of the vulva takes the form of hidradenoma papilliferum (HP).7 However, this lesion differs from HP in several ways. HP is more densely proliferative and is composed of prominent curved and linear interlacing papillae lined by closely approximated tall columnar epithelial cells.P: 14 The stroma within the papillae of HP is more densely collagenous. The overall appearance of HP has been likened to a mammary intraductal papilloma. 10 By contrast, AC is more cystic and less proliferative. The papillary projections in our case and other reported cases of AC are more delicate and flowerlike in arrangement. The appearance is reminiscent of an ovarian cystadenoma. The papillary stroma in AC is also more delicate and poorly collagenized, and the epithelial cells are more plump with an abundance of granular cytoplasm. AC has been reported to arise in other areas rich in apocrine glands such as the face, chest, and anus. Why these lesions have not been reported to have arisen on the vulva, which is also rich in apocrine glands, is unclear. Controversy has arisen regarding the terms AC and apocrine hidrocystoma. Some authors have used the terms interchangeably and considered these lesions to be retention cysts. In contrast, Mehregan I has argued that A C is a distinct organoid growth rather than a simple retention cyst. His arguments are (I) the tumor is composed of high columnar
Brief communications
499
apocrine cells without flattening, (2) the tumor exhibits true papillomatous hyperplasia with central fibrovascular cores, and (3) the lesion has a circumferential, organized fibrous stroma characteristic of all cutaneous adnexal tumors. These features are present in our case, and tend to support Mehregan's supposition.
REFERENCES 1. Mehregan AH. Apocrine cystadenoma: a clinicopathologic study with special reference to the pigmented variety. Arch Dermatol 1964;90:274-9. 2. Smith JD, Chernosky M E. Apocrine hidrocystoma (cystadenoma). Arch Dermatol 1974;109:700-2. 3. Benich B,Peison B.Apocrine hidrocystoma of'theshoulder. Arch Dermatol 1977;113:71-2. 4. Ter Poorten HJ. Apocrine hidrocystoma of the right scapula [Letter]. Arch DermatoI1977;13:1730. 5. Von Seebach HV, Stumm D. Misch P, et al, Hidrocystoma and adenoma of apocrine anal glands. Virchows Arch [A] Pathol Anat Histopathol 1980;386:231-7. 6. Weigand DV. Burgdorf WHC. Perianal apocrine gland adenoma. Arch Dermatol 1980;116:1051-3. 7. Ahmed A, Jones AW. Apocrine cystadenoma. Br.T Dermatol 1969;81:899-901. 8. Powell RF, Palmer CH, Smith EB. Apocrine cystadenoma of the penile shaft. Arch Dermatol 1977; 113:1250-1. 9. Hashimoto K, Mehregan AH. Kumakiri M. Tumors of skin appendages. Boston: Butterworths, 1987:146. 10. Santa Cruz DJ. Tumors of sweat gland differentiation. In: Farmer ER, Hood AF, eds. Pathology of the skin. Norwalk: Appleton & Lange, 1990:641. 11. Bindl JM, Warnke RA. Advantages of detecting monoclonal antibody binding to tissue sections with biotin and avidin reagents in Coplin jars. Am J Clin Pathol 1986; 85:490-3. 12. Tsubura A, Senzaki H, Sasaki M, et al. Immunohistochemical demonstration of breast-derived and/or carcinoma-associated glycoproteins in normal skin appendages and their tumors. J Cutan Pathol 1992; 19:73-8. 13. Woodworth H. Dockerty MD, Wilson RB, et al. Papillary hidradenoma of the vulva: a clinicopathologic study of 69 cases. Am J Obstet Gynecol 1971;110:501-8. 14. Meeker JH, Neubecker RD, Helwig EB. Hidradenoma papilliferum. Am J Clin Pathol 1962;37:182-95.