Journal of Pathology and Translational Medicine

Case Study

Development of Six Tumors in a Sebaceus Nevus of Jadassohn: Report of a Case: Serap Gozel, Melahat Donmez, Noyan Can Akdur, Hulya Yikilkan; Korean J Pathol. 2013;47(6):569-574. Published online December 24, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.6.569

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Nevus sebaceus of Jadassohn is a congenital cutaneous hamartoma comprised of multiple skin structures. It has the potential to develop into variety of neoplasms of various epidermal adnexal origins. While multiple tumors may occasionally arise, it is unusual for more than four tumors to arise simultaneously within a single sebaceus nevus. Here in, we report a case of a 70-year-old woman with six neoplastic proliferations including a syringocystadenoma papilliferum, pigmented trichoblastoma, tubular apocrine adenoma, sebaceoma, tumors of follicular infundibulum and superficial epithelioma with sebaceus differentiation arising in a long standing nevus sebaceus on the scalp. Our case is extraordinary because a single nevus sebaceus contained six neoplastic proliferations with differentiation toward the folliculosebaceous-apocrine unit.

Citations

Citations to this article as recorded by

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Original Article

Expression of bcl-2 and c-myc Proteins in Epidermal and Melanocytic Tumors.: Young Ha Oh, Chan Kum Park, Jung Dal Lee; Korean J Pathol. 1996;30(9):810-818.

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Abstract PDF: bcl-2 and c-myc protein expression were studied in 44 epidermal (8 seborrheic keratoses, 21 squamous cell carcinomas, and 15 basal cell carcinomas), and 26 melanocytic tumors(8 nevi, and malignant melanomas) by immunohistochemistry using the specific anti-bcl-2 and anti-c-myc monoclonal antibodies. 14 out of 15 basal cell carcinomas(BCC) (93.3%) showed expression of bcl-2 protein, 12 of which (85.7%) showed coexpression of c-myc protein. In the melanocytic tumors, 7 out of 8 nevi showed bcl-2 expression (87.5%). Five of these 7 cases (62.5%) also showed c-myc protein expression. Eight of 18 malignant melanomas(MM) (44.4%) showed expression of bcl-2 protein and 7 of these 8 cases (38.9%) also showed c-myc protein expression. All seborrheic keratoses and squamous cell carcinoma(SCC) were negative for bcl-2 proteins. 12 of 15 SCCs(80%) were positive for c-myc protein. In conclusion, bcl-2 and c-myc proteins were coexpressed in BCCs, nevi, and MMs. Coexpression of bcl-2 and c-myc proteins in these tumors was statistically significant(p<0.01), while no considerable differences of bcl-2 and c-myc expression were found between nevi and MMs. These results suggests that bcl-2 may cooperate with c-myc to promote tumorigenesis of BCCs, nevi, and MMs(p<0.01).

Case Reports

Complex Conjunctival Choristoma in Linear Nevus Sebaceous Syndrome: A case report.: Jinyoung Yoo, Jae Kyung Koh, Seok Jin Kang, Byung Kee Kim; Korean J Pathol. 2000;34(9):680-684.

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Abstract PDF: Linear nevus sebaceous syndrome is an uncommon neurocutaneous disorder characterized by nevus sebaceus, seizures, and mental retardation, and is often accompanied by ocular anormalies. We recently experienced a case of conjunctival choristoma associated with linear nevus sebaceous syndrome in a 17-month-old boy. The child was the product of a full-term uncomplicated pregnancy and normal delivery with normal karyotype of 46, XY. He was noted at birth to have a conjunctival mass on the right eye and linear nevi over the midline facial region and right temporoparietal scalp. Ophthalmologic examination revealed optic atrophy, peripapillary staphyloma, macular hypogenesis, and osseous choristoma of the choroid. A computed tomography scan demonstrated widening of the subarachnoid space and moderate atrophy of the cerebral and cerebellar hemispheres. He has been followed by ophthalmology and completely free of seizures since birth. The patient underwent excision of the conjunctival mass and biopsies of the skin lesion at 17 months of age. Histologic examination of the skin biopsies was characteristic for the linear sebaceous nevus of Jadassohn. Histologic findings of the conjunctival lesion showed a complex choristoma that contained lacrimal gland tissue and cartilage.

Nevus Cell Inclusions in the Lymph Node: A Report of Two Cases.: Ji Sun Song, Tae Woong Noh, Yoon Mee Kim, Sang Ho Cho, Kwang Gil Lee; Korean J Pathol. 2001;35(3):245-247.

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Abstract PDF: Nevus cell inclusion in the lymph node is an uncommon histologic finding and usually is an incidental finding in the capsule, fibrous trabeculae, perinodal adipose tissue, and parenchyma of the axillary, inguinal, or cervical lymph nodes which are removed as part of cancer diagnostic or therapeutic procedures. The aggregated pigmented nevus cells in the lymph node resemble the cells of the cutaneous nevi. It is important to differentiate them from metastatic carcinoma or malignant melanoma. The characteristic features of nevus cell inclusions are presence of nevus cells within the capsule and supporting stroma, without presence in the marginal sinus of the lymph node, and the absence of cytological atypia or mitosis of nevus cells. We report two cases of nevus cell inclusions, in the axillary lymph nodes in a patient with breast carcinoma and in an enlarged inguinal lymph node in a patient without a malignant tumor.

Squamous Cell Carcinoma Developed in Nevus Sebaceus: A Case Report.: Hyun Joo Choi, Jinyoung Yoo, Lee So Maeng, Seok Jin Kang, Chang Suk Kang; Korean J Pathol. 2004;38(1):60-63.

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Abstract PDF: We report a case of squamous cell carcinoma developed within the nevus sebaceus (NS) and review the literature. An 82-year-old woman presented with a 3-month history of pain within the lifelong skin lesion on her right cheek. Examination revealed a 1.0 x 1.0 cm poorly marginated, slightly raised yellow-brown nodule. Microscopically, the skin nodule revealed the typical findings of NS. Squamous cell carcinoma developed contiguously within the keratin-filled infundibulocyst of NS disconnected from the epidermis. We suggest that squamous cell carcinoma is derived from the embryonal stratum germinativum de-differentiated from the primary epithelial germ cells in the infundibulocyst of NS. Most secondary carcinomas associated with NS grow slowly over a period of years. In contrast, squamous cell carcinoma developed in NS often grow rapidly with aggressive behavior. Thus, the accurate pathologic diagnosis is necessary.

Malignant Melanoma Arising in Giant Congenital Melanocytic Nevus: A case report.: Jung Sun Kim, Sang Yong Song, Kye Yong Song, Je G Chi; Korean J Pathol. 1993;27(6):650-655.

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Abstract PDF: Giant congenital melanocytic nevus is found in 0.1% of live born infants. If present, this lesion has a 6.3% chance to develop malignant melanoma. We report such a case in a 22-year-old woman who had multiple pigmented skin lesions since birth. Rapidly growing masses were recently detected in the 19 cm-sized occipital pigmented lesion. Removed scalp lesion revealed yellowish white lobulated soft nodules in the background of pigmented nevus. Microscopically, the nodules consisted of epithelioid cells with prominent nucleoli, and pleomorphic cells including signetring cells. These cells seldom contained melanin pigment. There were metastatic aggregates of tumor cells in the cervical lymph node, which were reminiscent of germinal centers of lymph nodes. S-100 protein immunostaining was helpful to distinguish them. Incidentally, focally scattered pigmented spindle cells were seen in the capsule of a lymph node

Cellular Blue Nevus: A case report.: Na Hye Myong, Je G Chi, Byung Kyu Cho, Kye Yong Song; Korean J Pathol. 1993;27(5):542-545.

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Abstract PDF: Cellular blue nevus is an benign variant of blue nevus group, characterized by cellular islands composed of closely aggregated spindle cells with little or no melanin. The other variants include atypical and malignant blue nevus which often had the difficulties in the differential diagnosis from the malignant melanoma. A 4-year-old boy presented with hemispheric scalp nodule, which has been slowly grown since birth, measuring 3.5x3x1.5 cm. The surface showed geographic black pigmentation without hairs, hemorrhage, and ulceration. The excised mass disclosed homogeneoulsy dark black, glistening, and muddy cut surface. Histological examination revealed exuberant melanocytic proliferation with both spindle and dendritic components. Heavily pigment ed spindle cells, melanophages, focal necrosis, and vacuolated epithelioid cells were unusually noted in our case and diagnosed as cellular blue nevus. Electron microscopy disclosed largely mature melanosomes in tumor cells and melanophages. During about 16 months since operation, he has been relatively well with no evidence of disease.

Five Korean Cases of Dysplastic Melanocytic Neves.: Kyoung Me Kim, Seok Jin Kang, Sang In Shim, Sun Moo Kim, Baik Kee Cho; Korean J Pathol. 1993;27(4):411-417.

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Abstract PDF: The dysplastic nevus is a rare form of melanocytic nevus in Korean people. During the past 5 years the authors experienced 5 cases in the St. Mary's Hospital and Kang Nam St. Mary's Hospital. The criteria and the histologic findings of dysplastic nevi are debated in the western literature. Clinically, all cases were greater than 5 mm in size with ill defined irregular borders and irregularly distributed pigmentation. Histopathologically, all cases showed numerous discrete individual nests of melanocytes located in the dermoepidermal junction and the papillary dermis and were characterized by extension of dermoepidermal component of the nevus at the "shoulder" beyond the dermal component of the nevus. There was no cytologic atypia of nevus cells in all cases. Our cases lacked personal or family history of malignant melanoma. Therefore, we believe our cases are nonfamilial, sporadic and benign nevi with no prognostic significance. A brief review of the literature and a discussion on the term "dysplastic" and clinicopathologic correlation are made.

Meningeal Melanocytoma Associated with Ota's Nevus: Report of a case.: Woo Sung Moon, Joo Heon Kim, Dong Geun Lee, Ho Yeul Choi, Sang Ho Kim; Korean J Pathol. 1992;26(6):605-609.

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Abstract PDF: Primary meningeal melanocytoma of the central nervous system is extremely rare. We report a case of meningeal melanocytoma associated with Ota's nevus as a recurrent form in a 53-year old male. The meningeal melanocytoma was removed from right parietooccipital lobe 4 years ago and recurred in right parietal, occipital and left frontal lobes. Ultrastructurally, the tumor cells were characterized by the presence of numerous melanosomes and premelanosomes in their cytoplasm. Moreover, the tumor was lacking in histologic and ultrastructural features of pigmented meningioma, melanotic schwannoma and prolonged clinical course was different from primary meningeal melanoma or metastatic malignant melanoma.

Original Article

A Study on Immunohistochemical Stain for S-100 Protein, HMB 45 and Proliferating Cell Nuclear Antigen(PCNA) of Spitz Nevus Compared with Benign Nevus and Malignant Melanoma.: Mee Yon Cho, Kwang Gil Lee, Myung Wook Kim; Korean J Pathol. 1992;26(6):552-560.

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Abstract PDF: The differential diagnosis between Spitz nevus and malignant melanoma is difficult due to similar histologic findings. To obtain the useful aids for the differential diagnosis between two diseases, we studied 13 cases of Spitz nevus, 8 benign nevi of compound and intradermal, and 9 melanomas of primary and metastatic, using the immunohistochemical stain for S-100 protein, HMB.45 and proliferating cell nuclear antigen(PCNA). The staining pattern and intensity of S-100 protein showed homogenously strong positive reactivity in all cases. The frequency of HMB.45 positive cell in Spitz nevus was significantly lower than that in melanoma. When compared with the usual compound and intradermal nevi, Spitz nevi showed more significantly positive reaction in the dermal component of nevus cells. The expression of PCNA was higher in melanoma than in Spitz nevus. The immunohistochemical stains for HMB.45 and PCNA are considered as a useful methods for differentiation between Spitz nevus and melanoma, while stain for S-100 protein is not helpful.

Case Report

Blue Nevus of the Uterine Endocervix : Report of three cases.: Moon Hyang Park, Eun Kyung Hong; Korean J Pathol. 1991;25(5):471-475.

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Abstract: The clinical, gross, light microscopic and immunohistochemical findings of three cases of blue nevus of the uterine endocervix were described. All three cases were incidentally found in hysterectomy specimens from middle-aged women, 45 to 48 years of age. The lesions were small and measured 1 to 4 mm in the greatest diameter. The presence of elongated, somewhat wavy and dendritic melanin-containing cells, in clusters or scattered deep in the subepithelial stroma and between the endocervical glands, was the distinct feature. The cytoplasmic granules appeared black with Grimelius and Fontana-Masson stains. The cells showed strong positive reaction with S-100 protein in perinuclear cytoplasm, in addition to the diffusely dispersed melanin granules. The demonstration of S-100 protein in the blue nevus, along with the histochemical findings, supports combined melanocytic and schwannian differentiation of the blue nevus cells.

Original Article

Nevus Sebaceous with Special Reference on Its Aging Effect.: Jin Seok Seo, Mi Kyung Kim, Mikyung Kim, Kye Yong Song, Yun Lim Seo, Je G Chi; Korean J Pathol. 1990;24(4):436-445.

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Abstract PDF: A histopathological study was performed on nevus sebaceus to observe its aging effect based on 75 cases of neuvs sebaceus those were collected during the past 10 years from three university hospitals in Seoul. The results are as follows: 1) Clinical findings The incidence was most frequent in the teenage group. The 75 cases consisted of 41 males and 34 females with a sex ratio of 1.2:1. Most of cases developed in the head and neck areas with 62.7% on the scalp and 29.3% on the face. 2) Histopathologic findings. The epidermal changes such as acanthosis(40%), papillomatosis(73%), hypergranulosis(44%) were most remarkable in the second decade and gradually decreased with aging. The apparent proliferation of sebaceous gland was observed in 73% and it was most prominent in the second decade. Apocrine glands were absent before the first decade but apparently increased after then. Proliferation of eccrine gland was not significant in all the age groups. Mild increase of immature hair follicles were noted in 49% of our cases with gradually decreasing tendency in the older age. The dermal inflammatory infiltrates were noted from the 2nd decade(28%) and thereafter gradually increased. Associated neoplasms were one apocrine adenoma, one sebaceous adenoma, two trichilemmomas and two arteriovenous hemangiomas. The majority of tumors occured in the third decade. Therefore, it is observed that neuvs sebaceous undergoes dynamic histopathologic changes according to the age of patient and later develop various secondary neoplastic changes. The pathogenesis of the nevus sebaceus is suggested to be closely related with developmental anomalies of primitive hair germ units in fetal stage.

Case Reports

Blue Nevus in Uterine Cervix: A case report.: Hyo Min Kim, Eui Keun Ham, Hyo Pyo Lee; Korean J Pathol. 1988;22(1):88-91.

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Abstract PDF: Blue nevi commonly occur on the skin of head, neck, and arms, and in occasional instances they have been observed in the mucosa of oral cavity, vagina, hard palate, and even breast, and in very rarity observed in the uterine cervix. We have experienced a case of blue nevi on the uterine cervix of a 45 year old famale who was operated under the diagnosis of uterine myoma. In gross findings, besides two well circumscribed uterine myomas measuring 3.5 cm and 0.6 cm in diameter in the anterior myometrium, multiple pin-point sized grayish blue pigments measuring 2-3 mm in diameter aggregated in the submucosa of the uterine cervix. Microscopically the blue nevi showed greatly elongated, slender often slightly wavy melanocytes with long, occasionally branching dendritic processes lie grouped in irregular bundles in the submucosa of the uterine endocervix. The pigments showed positive response to the Fontana-Masson stain in the cytoplasm and the extracellular area.

Spindle and Epithelioid Cell Nevus: Report of four cases.: Hye Ju An, Kyo Young Lee, Sang In Shim, Sun Moo Kim, Jun Young Lee, Baik Kee Cho; Korean J Pathol. 1987;21(4):317-322.

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Abstract PDF: The spindle and epithelioid cell nevus, is known also as Spitz's nevus or benign juvenile melanoma. The histologic picture of the lesion often closely resembles that of a malignant melanoma, but spindle and epithelioid cell nevus hehaves benignly. Criteria for accurate histological diagnosis of this nevus now exist. However, a difficult lesion occasionally brings one to the worrisome situation even now. We experienced four cases (three children and one adult) of spindle and epithelioid cell neveus with nodular or macular skin lesions. In children (7/F, 9/F, 4/M), all of the lesion show a solitary pinkish red or light brown nodular firm masses, 0.7 cm to 1.0 cm in greatest dimension, which are located at the left posterior thigh, posterior neck and right anterior neck region, respectively. In adult (36/F), the lesion shows a light brown, erythematous macule, 0.8 cm in greatest diameter, in the skin of the back. Microscopically, both spindle and epithelioid cells are found in all the four cases, but epithelioid cells predominate in children and spindle cells predominate in adult.

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