Metastatic extraocular sebaceous carcinoma with an occult primary

The cytological diagnosis of sebaceous carcinoma is based on the aspiration of tumor lobules with extensive sebaceous differentiation. However, these tumors may show a spectrum of cells ranging from basaloid to well-differentiated sebaceous. Extraocular sebaceous carcinoma is an uncommon tumor. An occult primary at initial presentation has not been described previously. A 60-yr old woman was referred to the fine-needle aspiration (FNA) clinic with a submandibular mass. The cytological diagnosis was mucoepidermoid carcinoma. Histopathologically, it was a sebaceous carcinoma metastatic to the submandibular lymph node. No primary tumor was identified. Six months later, she presented with a nodule at the angle of her mouth and a recurrence of the submandibular swelling. FNA cytology (FNAC) and histopathologic examination revealed a primary cutaneous sebaceous carcinoma with submandibular lymph node metastasis. A metastatic sebaceous carcinoma in the submandibular region with limited sebaceous differentiation and occult primary is another pitfall in the cytomorphological diagnosis of mucoepidermoid carcinoma.     

Sebaceous carcinoma of the submandibular gland with high-grade malignancy: Report of a case

A case of sebaceous carcinoma arising in the left submandibular gland of a 66-year-old man is reported. The clinical and pathological examinations revealed a carcinoma, which was of salivary gland In origin, with regional lymph nodal metastases. Pathological findings showed features of highgrade sebaceous carcinoma with spindle myoepitheliomatous differentiation. Neither squamous cell nor duct epithelial-like cell differentiation was noted. lmmunohistochsmically, tumor cells were positive for cytokeratin, S-100 protein and vimentin. Lipid was demonstrated in the cytoplasm of the tumor cells. Ultrastructurally, tumor cells contained numerous intracyto-plasmic lipid droplets. Myoepitheliomatous differentiation is rare in sebaceous carcinoma of the salivary gland. Presented is the second reported case of sebaceous carcinoma arising in the submandibular gland.     

Metaplastic carcinoma of the breast with dominant squamous and sebaceous differentiation in the primary tumor and osteochondroid metaplasia in a distant metastasis: report of a case with review of sebaceous differentiation in breast tumors

Metaplastic breast carcinoma is an uncommon tumor that develops from conventional ductal mammary carcinoma, usually consisting of squamous and/or spindle cell components and/or mesenchymal elements. Although several morphological subtypes of metaplastic breast carcinoma are known, sebaceous metaplasia has not yet been described in this context. The authors report a case of an 84-year-old woman with a huge, ulcerated primary tumor in her left breast that had already been present for 10 years. Pulmonary and bone metastases and a tumor nodule in the contralateral breast were also detected at the time of admission. Left simple mastectomy was performed. Histological examination of the tumor revealed metaplastic carcinoma consisting of ductal carcinoma, which immunohistochemically exhibited a triple-negative immunoprofile, along with dominant areas of squamous and sebaceous differentiation. Adjuvant chemotherapy was subsequently given with partial regression of the systemic metastases. Seven months after surgery and diagnosis, a new, rapidly growing, large soft tissue metastatic tumor appeared in the intramuscular compartment of the patient's right thigh, which was removed and histologically examined. Morphologically this metastatic tumor showed ductal adenocarcinoma along with areas of sebaceous differentiation and, in addition, osteochondroid metaplasia. Immunohistochemically, unlike the primary, this tumor expressed HER-2. The case is presented because of its rarity, and sebaceous differentiation is also proposed as a novel type of metaplasia in this context, expanding the spectrum of the histological patterns of metaplastic breast carcinoma. The literature concerning breast sebaceous lesions is reviewed, and the hypothetical biological mechanisms responsible for the tumor pathogenesis in this case are discussed.     

乳腺皮脂腺样癌临床病理诊断

目的探讨乳腺皮脂腺样癌的临床病理学特征及其诊断和鉴别诊断要点。方法对1例乳腺皮脂腺样癌进行光镜观察和免疫组化标记。结果癌组织由两型细胞构成：一型为未分化细胞,较基底细胞胞质丰富;另一型为较分化细胞,胞界清楚,胞体宽大,胞质丰富,有小空泡,两型细胞均可见1～3个小红核仁。癌细胞排列成不规则小叶,各小叶中两型细胞的数目和分布不一,部分似皮脂腺小叶结构。可见癌组织与残存小叶内导管上皮移行结构,部分区域伴鳞化。结论皮脂腺样癌为罕见的特殊类型乳腺癌,可能源自小叶内导管上皮皮脂腺化生。确诊须符合下列标准：①肿物位于乳腺实质内;②具备皮脂腺分化特点及恶性特征;③可见癌组织与乳腺导管上皮移行结构。     

Update on sebaceous neoplasia: the morphologic spectrum and molecular genetic drivers of carcinoma

Tumors with sebaceous differentiation represent a challenge to diagnose, classify and occasionally to treat. The histopathologic spectrum of sebaceous neoplasia includes sebaceous adenoma, sebaceoma, and sebaceous carcinoma, while sebaceous hyperplasia represents hyperplasia of benign sebaceous glands surrounding a hair follicle. While often recognizable on morphologic grounds alone, sebaceous lineage also be informed by the application of immunohistochemical studies, including antibodies for adipophilin and Factor XIIIa (AC-1A1 clone). Sebaceous tumors are important to recognize given the relationship to the Muir-Tore autosomal dominant cancer predisposition syndrome. However, although it is well accepted that a sebaceous tumor might exhibit defects in mismatch repair, whether such a defect is related to germline defects in mismatch repair genes (or are due to somatically acquired alterations limited to the tumor cells) remains a critical clinical question with significant implications for the patient. Finally, recently published molecular studies have elaborated a molecular-genetic framework by which to understand the drivers of sebaceous carcinomas arising in different anatomic locations.     

眼睑皮脂腺癌的穿刺细胞学诊断及鉴别诊断

目的 探讨眼睑皮脂腺癌的穿刺方法、细胞学特点和鉴别诊断。方法 4例眼睑肿物行细针穿刺细胞学(FNAC)检查,穿刺涂片行常规HE染色以及脂肪染色(苏丹Ⅲ染色),光镜观察其细胞学特点;与活检结果对照,并与霰粒肿、眼睑毛母质瘤和基底细胞癌的细胞学特点进行比较。结果 4例肿物穿刺结果考虑为皮脂腺癌。涂片内细胞丰富,特征明显,可见2种肿瘤细胞,一种为胞质丰富呈泡沫样的皮脂腺分化细胞,一种为核大深染、异型明显的低分化癌细胞,脂肪染色显示肿瘤细胞胞质内及背景中可见橘红色脂滴。穿刺诊断与活检结果基本一致。而霰粒肿细胞学表现为肉芽肿性炎症,毛母质瘤内见无异型的基底样细胞、嗜碱性细胞、影细胞及钙化,基底细胞癌表现为核致密深染、排列紧密的细胞团,与皮脂腺癌易于区别。结论 眼睑肿物穿刺细胞学检查是一种安全有效的检查方法,有助于眼睑皮脂腺癌的早期诊断及答别诊断。     

Sebaceous carcinoma of the parotid gland

Summary Sebaceous carcinoma of salivary gland origin is extremely rare and, because of its rarity, the clinicopathological characteristics and the histogenesis are not fully understood. We present a case of sebaceous carcinoma of the parotid gland which brings the total number of reported cases to 22.The tumor showed epithelial cell nests which were mainly composed of sebaceous cells with marked cellular atypia. In most of the nests, glandular spaces lined by ductal epithelium were present. Scattered mucous cells and flattened eosinophilic cells at the periphery of the nests were also seen. Ultrastructural and immunohistochemical observations of the tumour revealed coexistence of sebaceous and glandular differentiations in some tumour cells. Tumour cells with lipid granules often participated in the formation of glandular structures or exhibited intracytoplasmic lumina, and immunohistochemical localization of lactoferrin and secretory component, the functional markers of ductal epithelium of salivary gland, was demonstrated not only in duct-forming tumour cells but also in many sebaceous tumour cells.It seems likely that sebaceous carcinoma originates from pluripotential duct cells which can differentiate into sebaceous, ductal and mucous cells.     

Sebaceous gland and sweat gland carcinomas of the skin. Clinicopathological study and significance of c-erbB-2 oncoprotein expression.

Thirteen sebaceous gland carcinomas and 10 sweat gland carcinomas were examined to elucidate any important histological parameters influencing their prognosis, and the relationship between immunohistochemical expressions of c-erbB-2 oncoprotein and survival of the patients was analyzed. Sebaceous gland carcinomas with vacuolated cytoplasm in more than 50% of whole tumor area, with necrosis, and without lymphoid cell infiltration in tumor nests and stroma had a higher incidence of tumor recurrence and tumor-related death than tumors with vacuolated cytoplasm in 50% or less of whole tumor area (p < 0.01), without necrosis, and with lymphoid cell infiltration in tumor nest and stroma (p < 0.05). Sweat gland carcinomas of all cases with fatal outcomes demonstrated tubular differentiation in 20% or less of whole tumor area, lymphatic permeation and desmoplastic reaction. Three sebaceous gland carcinomas and three sweat gland carcinomas were positive for c-erbB-2 oncoprotein. Two of three sebaceous gland carcinomas, and all three sweat gland carcinomas developed tumor recurrence and ended in tumor-related deaths. Sweat gland carcinomas with c-erbB-2 expression had significantly shorter survival than those with negative immunostain (p < 0.01). Cytoplasmic appearance, tumor necrosis, and lymphoid cell infiltration in tumor nests and stroma of sebaceous gland carcinoma, and tubular differentiation, lymphatic permeation, and growth patterns of sweat gland carcinoma are considered to closely correlate to the prognosis. Immunohistochemically detected c-erbB-2 oncoprotein may be an indicator of bad prognosis.     

Sebaceous carcinoma of the breast

We report on a rare distinctive variant of infiltrating ductal carcinoma characterized by sebaceous differentiation of tumor cells. The neoplasm was identified in a lumpectomy specimen from a 45-year-old woman with extensive metastatic disease. In addition to conventional in situ and invasive ductal components, approximately half of the tumor cells exhibited a phenotype resembling tumors of the sebaceous skin appendage with coarsely vacuolated cytoplasm and peripherally displaced nuclei. The sebaceous moiety was also present in the distant metastatic deposits. There was no evidence of mucin production by tumor cells. Ultrastructurally, empty-appearing non-membrane bound vacuoles attested to the sebaceous cells' lipid content. The immunoprofile of the lesion included positivity for cytokeratin and epithelial membrane antigen. Vimentin, S100 protein and carcinoembryonic antigen were not expressed. Most tumor cell nuclei reacted with antibodies to oestrogen and progesterone receptors but failed to show overexpression of the HER2/neu protein. The MIB-1 labeling index averaged 16%. At variance with sebaceous breast carcinomas on record, the present case is notable for its prolonged clinical course.     

Sebaceous Gland and Sweat Gland Carcinomas of the Skin Clinicopathological Study and Significance of c-erbB-2 Oncoprotein Expression

Thirteen sebaceous gland carcinomas and 10 sweat gland carcinomas were examined to elucidate any important histological parameters influencing their prognosis, and the relationship between immunohistochemical expressions of c-erbB-2 oncoprotein and survival of the patients was analyzed. Sebaceous gland carcinomas with vacuolated cytoplasm in more than 50% of whole tumor area, with necrosis, and without lymphoid cell infiltration in tumor nests and stroma had a higher incidence of tumor recurrence and tumor-related death than tumors with vacuolated cytoplasm in 50% or less of whole tumor area (p < 0.01), without necrosis, and with lymphoid cell infiltration in tumor nest and stroma (p < 0.05). Sweat gland carcinomas of all cases with fatal outcomes demonstrated tubular differentiation in 20% or less of whole tumor area, lymphatic permeation and desmoplastic reaction. Three sebaceous gland carcinomas and three sweat gland carcinomas were positive for c-erbB-2 oncoprotein. Two of three sebaceous gland carcinomas, and all three sweat gland carcinomas developed tumor recurrence and ended in tumor-related deaths. Sweat gland carcinomas with c erbB 2 expression had significantly shorter survival than those with negative immunostain (p < 0.01). Cytoplasmic appearance, tumor necrosis, and lymphoid cell infiltration in tumor nests and stroma of sebaceous gland carcinoma, and tubular differentiation, lymphatic permeation, and growth patterns of sweat gland carcinoma are considered to closely correlate to the prognosis. Immunohisto-chemically detected c erbB 2 oncoprotein may be an indicator of bad prognosis. Acta Pathol Jpn 42: 585–594, 1992.     

Clinicopathological and immunohistochemical features of a sebaceous carcinoma arising within a benign dermoid cyst of the ovary

Pure sebaceous neoplasms arising in dermoid cysts of the ovary are exceedingly rare. A 63-year-old female with abdominal swelling and pain underwent a right salpingo-oophorectomy that showed a unilocular cyst weighing 830 g and measuring 15×12×10 cm, filled with sebaceous material containing a few hair shafts. The cyst wall exhibited plaques protruding into the cavity of the cyst. Microscopy revealed a dermoid cyst with nests and lobules of atypical and infiltrating sebaceous cells surrounded by basaloid cells. The tumor cells stained diffusely for high-molecular-weight cytokeratins and focally for cytokeratin 7, cytokeratin 19, epithelial membrane antigen and carcinoembryonic antigen in the immunohistochemistry study. Low-molecular-weight cytokeratins, cytokeratin 20, vimentin, S100, p63, estrogen receptor, progesterone receptor, p53 and c-erbB-2 were negative in tumoral cells. The proliferative labeling index (Ki67 and proliferating cell nuclear antigen) was low. Basal cell carcinoma with sebaceous differentiation and sebaceoma must be considered in the differential diagnosis. However, the presence of obvious malignant sebaceous differentiation in nearly every tumor nest and lack of peripheral palisading and peri-tumoral myxoid stroma excluded these diagnoses. Some histogenetic concepts relevant to this case are discussed along with a brief review of this neoplasm. To our knowledge, this is the sixth case report of a sebaceous carcinoma arising in a mature cystic teratoma of the ovary.     

Cytologic features of metastatic sebaceous carcinoma: report of two cases with comparison to three cases of basal cell carcinoma.

The cytologic findings of two cases of metastatic sebaceous carcinoma are described and compared to three cases of locally recurrent basal cell carcinoma. Morphological findings for sebaceous carcinoma in fine-needle aspiration biopsy (FNAB) smears included cellular, loosely cohesive cell clusters with central necrosis, squamous pearl formation, and adjacent keratin debris. The tumor cells had moderate amounts of vacuolated cytoplasm, round to oval vesicular nuclei with clumped chromatin, nucleoli, some nuclear overlap, and numerous mitotic figures. An interesting finding was the presence of numerous multinucleated giant cells, probably responding to extravasated lipid or keratin material. In contrast, the FNAB smears of basal cell carcinoma typically were less cellular, with more tightly cohesive and smaller clusters of uniform hyperchromatic basaloid cells with high nuclear to cytoplasmic ratios, and a narrow rim of cytoplasm without vacuolization. The morphologic features of sebaceous carcinoma in FNAB smears appear to be distinct from those of basal cell carcinoma. FNAB can be a useful preoperative diagnostic technique to distinguish these two cutaneous malignancies.     

Sebaceous carcinoma of the eyelids: thirty cases from Japan

Sebaceous carcinoma of the eyelids is rare in Western countries but not uncommon in Asian countries. Diagnosis is difficult both clinically and histologically. Thirty cases of sebaceous carcinoma of the eyelids treated at Tokyo Medical University Hospital were reviewed to elicit characteristic features of pathological findings. The tumor cells were infiltrating in a lobular pattern that consisted mainly of large atypical germinative cells. Sebocytes seen in the lobules had conspicuous nucleolus associated with perinucleolar halo. In 17 cases (57%) there was foamy histiocyte infiltration in and around the tumor nests. Sebaceous duct differentiation, namely holocrine secretion indicating a specific type of coagulation necrosis maintaining a cellular framework or maintaining a bubbly cytoplasm associated with nuclear debris in the periphery, was seen in 24 cases (80%). Although unequivocal squamous differentiation was limited to only 11 carcinomas, scattered individual necrosis with nuclear debris in the background of germinative cells appeared in 29 cases (96.7%). Expression of epithelial membrane antigen, perilipin and adipophilin were detected in all cases. In conclusion, to detect sebaceous differentiation in sebaceous carcinoma, it would be helpful to focus on holocrine secretion, squamous differentiation and foamy macrophage infiltration, and to utilize immunohistochemistry involving anti-perilipin and anti-adipophilin stain.     

Die Bedeutung von Talgzellen und Talgdrüsen für Parotisgeschwülste

Zusammenfassung Es wird über drei Parotisgeschwülste bei einer 45jährigen Frau, 41jährigen Frau und einem 28jährigen Mann berichtet, deren umgebendes tumorfreies Parotisparenchym heterotope freie Talgdrüsen in wechselnder Menge enthält. Ihr vermutlicher Entwicklungsgang: Einzeltalgzelle — Talgdrüsenknospe — reife Talgdrüse wird aufgezeigt.Im 1. Fall liegt ein pleomorphes Adenom (sog. Mischgeschwulst) vor, am Rande mit (tumorinduzierter?) Hypertrophie von Talgdrüsen ähnlich den senilen Talgdrüsennaevi der Haut, zentral mit ausgedehntem Übergang in Talgdrüsencarcinom, streckenweise auch in parakeratorisch verhornendes Plattenepithelcarcinom.Beim 2. Fall liegt ein kleines, teils solides, teils kleindrüsiges Adenom vor, das gelegentlich talgzellenähnliche Einzelzellen enthält.Der 3. Fall wird als reines Talgdrüsencarcinom der Parotis aufgefaßt.Die Bedeutung der holokrinen Elemente für das Tumorgeschehen, insbesonders hinsichtlich der Erweiterung des morphologischen Spektrums der Parotisadenome durch talgzellenartige Einzelzellen, talgdrüsenartige Strukturen und Carcinome mit talgdrüsenartigem Bau wird unterstrichen.Reine Talgdrüsencarcinome lassen sich gleichfalls zwanglos von den Talgelementen der Parotis ableiten.

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The significance of sebaceous cells and sebaceous glands in parotid tumors

Summary Three parotid tumors, in a 45-year-old woman, a 41-year-old woman and a 28-year-old man, are described. The surrounding tumor-free parotid parenchyma contained heterotopic free sebaceous glands in varying quantities. Their probable course of development: individual sebaceous cell—sebaceous-gland bud—mature sebaceous gland, is presented. The first case involved a pleomorphic adenoma (so-called mixed tumor) with (tumor induced?) hypertrophy of the sebaceous glands on the outside resembling the senile sebaceous-gland nevi of the skin, and centrally extensive transition to sebaceous gland carcinoma and in some places, to parakeratotic squamous cell carcinoma. The second case involved a small, partly solid and partly small-glandular adenoma containing occasional individual cells resembling sebaceous cells. The third case was classified as a pure sebaceous-gland carcinoma of the parotid. The significance of the holocrine elements for the tumor event is emphasized, particularly with regard to the widening of the morphological spectrum of the parotid adenoma through individual sebaceous-like cells, structures resembling sebaceous glands and carcinomas with structures similar to sebaceous glands. Pure sebaceous-gland carcinoma can also readily be derived from parotid sebaceous elements.

     

Hypopharyngeal sebaceous carcinoma: a case report

A hypopharyngeal squamous-cell carcinoma with sebaceous differentiation is reported. In the primary as well as the metastatic lymph nodes, the tumor showed basaloid, squamous, and sebaceous cells. In addition, immunostaining for S-100 protein and vimentin manifested scattered cells showing cytoplasmic processes suggesting myoepithelial cells. An exhaustive review of the literature revealed only one similar case previously reported. The probable origin from the minor salivary glands is discussed.     

Hautadnex- vs. Speicheldrüsentumoren

Despite major discrepancies in basic microscopic anatomy, remarkable similarities are manifest within the wide spectrum of cutaneous adnexal and salivary gland tumors. In this study salivary gland and adnexal tumors were identified and investigated with respect to similarities in histology, terminology and pathogenesis. Histological similarities of certain types of salivary gland tumors relate to eccrine, apocrine and rarely sebaceous (but not trichofollicular) types of adnexal tumors. The most striking similarity was found with salivary gland pleomorphic adenoma and cutaneous mixed tumor. Multistep carcinogenesis starting with intraductal carcinoma, identified in carcinoma ex pleomorphic adenoma is identical to that found in cutaneous carcinoma ex spiradenoma. Further histological and terminological similarities are shown for mucinous and mucoepidermoid carcinoma, for lymphadenoma and lymphoepithelial carcinoma, for sebaceous adenoma and carcinoma, for adenoid-cystic carcinoma, as well as for salivary gland basal cell adenoma versus cutaneous spiradenoma. Manifest diagnostic problems related to histologically similar salivary gland and adnexal tumors are rare and are topographically limited to the parotid and oral areas.     

Chondroblastic osteosarcoma of the temporal region: A diagnostic dilemma

Chondroblastic osteosarcoma (OS) accounts for about 25% of all cases of OS. It can pose diagnostic difficulty on cytology, as the tumor cells may resemble the carcinomatous cells. We present a case of a 35‐year‐old female patient who presented with a firm swelling in the left temporal region. On fine‐needle aspiration, the smears revealed abundant cellularity with tumor cells showing significant nuclear pleomorphism and multinucleation. The tumor cells had abundant foamy to vacuolated cytoplasm, thereby indicating sebaceous differentiation and it was cytologically interpreted as sebaceous carcinoma. However, on subsequent histopathology, similar tumor cells were seen lying down abundant amount of osteoid material along with foci of chondroid differentiation and was diagnosed as chondroblastic OS. Pitfalls in the cytodiagnosis of this case along with differential diagnosis on cytology are discussed. Diagn. Cytopathol. 2011. © 2010 Wiley‐Liss, Inc.     

Meibomian gland carcinoma seeding intracranial soft tissues

Meibomian gland carcinoma is a rare sebaceous gland malignant tumor of the eyelid. Local recurrence and regional lymph node metastases are common among this group of eyelid neoplasms. Distant liver metastases have been mentioned in one case report, but this represents an unusual finding. In addition to local recurrence and regional lymph node metastases, this autopsy case report demonstrates meibomian gland carcinoma with extension to the trigeminal ganglia and metastases to the cerebellum.     

Sebaceous carcinoma of the breast: case report and review of the literature

Sebaceous differentiation has been described in only limited examples of benign and malignant epithelial lesions of the breast. We report a rare case of mammary sebaceous carcinoma to further delineate its morphologic features. Microscopically, the tumor, arising in the right mammary gland of a 63-year-old woman, was composed of well-defined solid sheets or lobules of atypical epithelial cells including many large pale or clear cells with often scalloped nuclei and coarsely vacuolated cytoplasm, in which abundant lipid droplets were identified with oil-red-O staining. Immunohistochemical expressions of cytokeratin, epithelial membrane antigen, and receptors of estrogen and progesterone were detected, whereas GCDFP-15, S-100 protein, vimentin, α-smooth muscle actin, p63, androgen receptor, and the HER2/neu protein were not expressed. Besides, a subset of the tumor cells co-expressed synaptophysin, neurofilament, and PGP9.5, suggesting neuroendocrine differentiation that is a hitherto undescribed phenomenon in the mammary tumors with sebaceous features. This case would expand the morphologic diversity of carcinoma of the breast.     

A histochemical and immunohistochemical study of extra-ocular sebaceous carcinoma

A histochemical and immunohistochemical study of five cases of extra-ocular sebaceous carcinoma was performed using formalin-fixed and paraffin-embedded tissue specimens. Histochemically, the clear cells of sebaceous carcinomas were negative with periodic acid-Schiff and alcian blue staining. Immunohistochemically, the tumour cells of sebaceous carcinomas showed positive reactions for epithelial membrane antigen, human milk fat globules subclass 1, human milk fat globules subclass 2 and Leu M1, but did not express carcinoembryonic antigen, breast carcinoma associated antigen, S-100 protein, gross cystic disease fluid protein-15 or Dako M1. These histochemical and immunohistochemical findings were compared with those of other skin cancers which must be distinguished histopathologically from sebaceous carcinoma. We conclude that sebaceous carcinoma can be distinguished from eccrine porocarcinoma, malignant clear cell hidradenoma, extramammary Paget's disease, malignant trichilemmoma, squamous cell carcinoma and basal cell carcinoma by histochemical and immunohistochemical techniques using formalin-fixed and paraffin-embedded tissue specimens.