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Microcystic Adnexal Carcinoma Arising Within a Nevus Sebaceus

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Microcystic Adnexal Carcinoma Arising Within a Nevus Sebaceus Microcystic Adnexal Carcinoma Arising Within a Nevus Sebaceus Nektarios Lountzis, MD; Jacqueline Junkins-Hopkins, MD; Marie Uberti-Benz, MD; Rosalie Elenitsas, MD Nevus sebaceus (NS) is a congenital skin lesion aris- verrucous hyperplasia. The final stage is marked by ing on the face and scalp that has been linked to the a propensity for both benign and malignant tumor development of various carcinomas. We describe growth.2-5 Benign neoplasms, such as syringocystad- a case of microcystic adnexal carcinoma (MAC) enoma papilliferum and trichoblastoma, are more arising in an NS on the scalp of a 62-year-old man. likely to occur, whereas malignant neoplasms gener- Excisional skin biopsy and hematoxylin and eosin ally are rare.3 With this in mind, we report a case stains were performed to examine the specimen. of a microcystic adnexal carcinoma (MAC) arising Serial sections revealed papillomatosis typical of within an NS. NS, with focal changes consistent with syringocys- tadenoma papilliferum. Adjacent to the syringocys- Case Report tadenoma papilliferum was an area containing small A 62-year-old white man presented to his dermatolo- epithelial islands that extended focally into the sub- gist for reevaluation of a scalp growth. The patient ini- cutaneous layer. The cystic islands were embedded tially was evaluated in 1998 for the same lesion, which in a desmoplastic stroma with poor circumscription, had been present since birth. A biopsy was performed consistent with MAC. This case presents a rare find- and the results were interpreted elsewhere as mul- ing of MAC within an NS. tiple follicular infundibular cysts. The lesion gradually Cutis. 2007;80:352-356. recurred, but a complete excision was declined by the patient. In 2002, the growth was treated locally with carbonic acid by another dermatologist. The lesion adassohn1 first described nevus sebaceus (NS) in continued to grow, and the patient presented to us for 1895 as a hamartomatous locus of embryologically a reassessment. His past medical history was notable for J defective pilosebaceous units. Because this lesion myelodysplasia and splenectomy. There was a family involves more than just a sebaceous component, history of colon cancer. the more encompassing term organoid nevus also Physical examination revealed a yellow-beige has been coined. Clinically, the lesion presents in verrucous plaque on the patient’s left temple. An 3 stages. Initially, it is a well-circumscribed, flat, round excisional biopsy was performed. Hematoxylin and to oval, hairless patch that progresses into a raised, eosin–stained sections revealed a large lesion with papillated, yellow plaque under the hormonal influ- epidermal acanthosis and papillomatosis (Figure 1). ences of puberty. The lesion arises on the face and Arising in the dermis were endophytic epithelial scalp of children, rarely on the trunk and extremi- islands that formed glandular spaces, with decapitation ties, and usually is present at birth. During puberty, secretion. Together these features were believed to be hormones influence the apocrine glands to mature, most consistent with syringocystadenoma papilliferum sebaceous glands to enlarge, and epidermis to undergo arising in an NS. Adjacent to the syringocystadenoma papilliferum was a separate dermal adnexal neoplasm that was composed of large and small cystic structures Accepted for publication December 6, 2006. Dr. Lountzis is from Geisinger Medical Center, Danville, filled with an amorphous material (Figure 2). This Pennsylvania. Drs. Junkins-Hopkins, Uberti-Benz, and proliferation extended deeply into the reticular dermis Elenitsas are from the Hospital of the University of and focally into the subcutaneous layer (Figures 3 Pennsylvania, Philadelphia. and 4). The small islands were embedded in a desmo- The authors report no conflict of interest. plastic stroma with poor circumscription that extended Reprints: Rosalie Elenitsas, MD, University of Pennsylvania, Department of Dermatology, 3600 Spruce St, 2 Maloney Bldg, beyond the lateral margins of the specimen. There was Philadelphia, PA 19104-6142 focal neurotropism (Figure 5). The luminal surface (e-mail: [email protected]). of the cystic areas stained positively with epithelial 352 CUTIS® Microcystic Adnexal Carcinoma Figure 1. A large lesion with irregular verrucous epidermal hyperplasia with variably sized cystic and glandular struc- tures in the dermis (H&E, original magnification 312.5). membrane and carcinoembryonic antigens. This atypi- cal sclerosing lesion was most consistent with features of MAC. A reexcision subsequently was performed, with pathology that revealed a residual MAC with clear margins. Comment With a penchant for developing both benign and malignant neoplasms as well as an association with Figure 2. Microcystic adnexal carcinoma. An infiltra- NS syndrome, NS has held a traditionally small tive dermal adnexal neoplasm, which is adjacent to the threshold for prophylactic excision.6,7 Neoplasms syringocystadenoma papilliferum (H&E, original occur mostly in the fourth decade of life in approxi- magnification 35). mately 10% to 30% of lesions, with trichoblas- toma and syringocystadenoma papilliferum being the most common tumors.4,6,8 Although rare, NS carcinomas arising in NS that could have features also may experience multiple growths.9,10 The prema- that overlap with our case.16,17 lignant nature of NS was first described in 1962 by MAC is an infiltrative adnexal neoplasm occur- Michalowski,11 and basal cell carcinoma (BCC) was ring on the centrofacial region. The median age of believed to be the most common tumor. However, onset is 56 years, with an incidence equal in men and some researchers theorized that the most BCCs found women.18 Predominance is seen in the white popula- within NS were in fact benign trichoblastomas,12 and tion, but a few cases in the black population have been a study by Cribier et al3 confirmed this finding using documented.19-22 Sun exposure, genetics, immunosup- silhouette analysis and stromal examination. Cur- pression, and radiation have been implicated as risks rently, trichoblastomas are believed to be the most for MAC.18,19,23-25 Numerous cases of MAC (8%–12%) common follicular tumors associated with NS.3,8 involve patients with previous radiation exposure Nevertheless, malignant tumors do occur in NS, for treatment of acne, tinea capitis, or thyroid car- including squamous cell carcinoma (SCC), malig- cinoma several years prior to presentation.18,26-28 nant melanoma, apocrine carcinoma, and sebaceous Only one death has been attributed to MAC.29 carcinoma.4,13 Underlying malignancy within NS is Metastases also are uncharacteristic, but cases have suggested by the acute appearance of large, discrete, been reported.24,29,30 Clinically, the lesion presents as ulcerating nodules within the lesion.4,6,14 Primary a flesh-colored to yellow nodule, cyst, or plaque, with epithelial germ cell defects may explain the abnor- a strong predilection for the lips (52%) and nasal malities found in follicular, apocrine, and sebaceous area (14%).18 Lesions of the axilla and trunk rarely structures, and the tendency for neoplastic growth. have been reported.19,31,32 Delay of several years from Moreover, few cases of MAC have been reported onset to diagnosis is often observed because of the within an NS; this is the second documented case lesion’s slow growth and bland clinical appearance to date.15 However, there are reports of adnexal of MAC.18,30,33 VOLUME 80, OCTOBER 2007 353 Microcystic Adnexal Carcinoma Figure 3. Microcystic adnexal carcinoma. Small infiltrative epithelial islands and ducts extending into the subcutaneous tissue (H&E, original magnification 3200). Microscopically, MAC has a deceivingly banal resemble sweat glands, with both components appearance without cytologic atypia, mitoses, or present in variable proportions.32,36 Some authors necrosis. However, it is a locally destructive neoplasm also contend that an apocrine or sebaceous dif- with a high recurrence rate that is secondary to hid- ferentiation is plausible.15,18,37 However, immu- den invasion of deeper perineural, perichondrial, nohistochemistry has been used to identify the perimuscular, and perivascular structures.18,27,31,34 origin of MAC, which has shown a strong pen- Moreover, microscopic borders can extend to up to chant for carcinoembryonic antigen, epithelial twice the size of clinically visible margins, which membrane antigen, and cytokeratin, which suggest makes simple local excision difficult.23 Biopsy results an eccrine and follicular origin, as speculated by generally reveal poorly demarcated epithelial nests, Goldstein et al.36 MAC also shows immunoreac- strands, and cords of tumor cells that infiltrate deep tivity with cystic fibrosis-1, a monoclonal anti- into the dermis and subcutaneous layer, with areas of body specific for eccrine ducts, and acrosyringium.32 keratinized cysts and well-differentiated ductules. No Electron microscopy has further supported the variation in histology is seen between irradiated and notion of follicular and eccrine differentiation.18 nonirradiated skin.35 Light microscopy is the gold standard for diag- Differentiation of MAC is controversial; nosis, with a differential that includes trichoad- Goldstein et al36 originally hypothesized both a enoma; morpheaform BCC; syringoma; eccrine follicular and eccrine origin. Squamoid aggrega- carcinoma; desmoplastic SCC; metastatic breast tions with keratin cysts resemble hair matrix dif- cancer; and desmoplastic
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