Journal of Pathology and Translational Medicine

Case Studies

Unilateral Pigmented Extramammary Paget's Disease of the Axilla Associated with a Benign Mole: A Case Study and a Review of Literature: Aleem Ladak, Maria Bramley, Sami Titi; Korean J Pathol. 2014;48(4):292-296. Published online August 26, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.4.292

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Pigmented extramammary Paget's disease (PEMPD) is an uncommon intraepithelial adenocarcinoma and a rare variant of Paget's disease affecting skin that is rich in apocrine sweat glands such as the axilla, perianal region and vulva. It most commonly occurs in postmenopausal women and presents as a superficial pigmented scaly macule, mimicking a melanocytic lesion. The histological presentation is adenocarcinoma in situ with an increased number of melanocytes scattered between the Paget's cells. Therefore, PEMPD may be misdiagnosed as a melanocytic tumour both clinically and histologically. The tumour cells are usually positive for cytokeratin 7, epithelial membrane antigen, Cam 5.2, HER2, and mucicarmine stain while S100 and human melanoma black-45 highlight the processes of reactive dendritic cells. The association between Paget's cells and intratumoural reactive melanocytes is still unclear. We report our first case of PEMPD associated with an intradermal naevus involving the axilla in a 63-year-old woman.

Citations

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Atypical cells on reflectance confocal microscopy may not represent melanoma: A case of axillary pigmented extramammary Paget disease
Nadiya Chuchvara, Catherine Reilly, Attiya Haroon, Cindy Wassef, Amin Maghari, Babar Rao
Journal of Cutaneous Pathology.2020; 47(12): 1170. CrossRef
“Pigmented Extramammary Paget Disease”—A Potential Mimicker of Malignant Melanoma and a Pitfall in Diagnosis: A Case Report and Review of the Literature
Katrin Kiavash, Steve Kim, Andrew D. Thompson
The American Journal of Dermatopathology.2019; 41(1): 45. CrossRef
Vulvar cancer: a review for dermatologists
Anastasiya Atanasova Chokoeva, Georgi Tchernev, Elena Castelli, Elisabetta Orlando, Shyam B. Verma, Markus Grebe, Uwe Wollina
Wiener Medizinische Wochenschrift.2015; 165(7-8): 164. CrossRef

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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Abstract PDF

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

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Tumor of follicular infundibulum – reappraisal in a series of 28 patients with critical review of the literature
Michael Wilk, Bettina G. Zelger, Bernhard Zelger
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Tumor des follikulären Infundibulums – Neubewertung in einer Serie von 28 Patienten mit kritischer Analyse der Literatur
Michael Wilk, Bettina G. Zelger, Bernhard Zelger
JDDG: Journal der Deutschen Dermatologischen Gesellschaft.2024; 22(2): 223. CrossRef
Adnexal neoplasms of the eye
Roman Drozdowski, Jane M. Grant-Kels, Madina Falcone, Campbell L. Stewart
Clinics in Dermatology.2024;[Epub] CrossRef
Melanotrichoblastoma: sixth case report in the literature
Juliana Polizel Ocanha-Xavier, José Cândido Caldeira Xavier-Júnior
Anais Brasileiros de Dermatologia.2023; 98(6): 871. CrossRef
Multiple secondary neoplasms in nevus sebaceus excision
Travis S. Dowdle, David A. Mehegran, Dylan Maldonado, Cort D. McCaughey
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Development of seven secondary neoplasms in a nevus sebaceous: a case report and literature review
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Multiple rare neoplasms arising from the nevus sebaceous of the scalp: A case report
Deepthi Shetty, Anilkumar Desai, Niranjan Kumar, Dinesh U.S., Aditya Agnihotri, Saurav Bhaduri
Gulhane Medical Journal.2022; 64(2): 197. CrossRef
Syringocystadenoma Papilliferum and Basal Cell Carcinoma Arising in Nevus Sebaceous
Jingjing Jiang, Yujuan Chen, Qi He, Jiao Yang, Zhengzhong Zhang, Hao Yang, Huan Zhang, Chuan Yang
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Eyelid trichoblastoma – A case series
Gunja Chowdhury, Meghana Tanwar, Usha Kim, ShanthiR Krishnan
Journal of Clinical Ophthalmology and Research.2021; 9(3): 123. CrossRef
Trilogy revisited: A case report of co-sexisting nevus sebaceous with syringocystadenoma papilliferum and tubular apocrine adenoma
Anand Bardia, Debajyoti Chatterjee, Keshavamurthy Vinay
Indian Dermatology Online Journal.2021; 12(4): 577. CrossRef
Trichilemmoma coexisting with sebaceous nevus
AngooriG Rao, VangaliS Reddy, M Tejal, M Divya
Indian Dermatology Online Journal.2020; 11(2): 253. CrossRef
Syndromic sebaceous nevus: current findings
Oumama El Ezzi, Anthony S. de Buys Roessingh, Michèle Bigorre, Guillaume Captier
International Journal of Dermatology.2018; 57(5): 599. CrossRef
Syringocystadenoma papilliferum and trichoblastoma arising in the nevus sebaceous
Feifei Wang, Yatong Wu, Zhancai Zheng, Yanping Bai
Indian Journal of Pathology and Microbiology.2018; 61(1): 106. CrossRef
Dermoscopic Analysis of Nevus Sebaceus of Jadassohn: A Study of 13 Cases
Awatef Kelati, Hanane Baybay, Salim Gallouj, Fatima Zahra Mernissi
Skin Appendage Disorders.2017; 3(2): 83. CrossRef
Secondary neoplasms arising from nevus sebaceus: A retrospective study of 450 cases in Taiwan
Ming‐Chun Hsu, Jau‐Yu Liau, Jin‐Liern Hong, Yin Cheng, Yi‐Hua Liao, Jau‐Shiuh Chen, Yi‐Shuan Sheen, Jin‐Bon Hong
The Journal of Dermatology.2016; 43(2): 175. CrossRef
A Histological Snapshot of Hypothetical Multistep Progression From Nevus Sebaceus to Invasive Syringocystadenocarcinoma Papilliferum
Vishwas Parekh, Cesar E. Guerrero, Charles F. Knapp, Craig A. Elmets, Kristopher M. McKay
The American Journal of Dermatopathology.2016; 38(1): 56. CrossRef
Trichoblastoma, syringocystadenoma papilliferum, desmoplastic trichilemmoma and tumor of the follicular infundibulum with signet‐ring cells, all arising in nevus sebaceus
Emilie Dore, Megan H. Noe, Brian L. Swick
Journal of Cutaneous Pathology.2015; 42(9): 645. CrossRef
Ceruminous adenoma (ceruminoma) arising in a nevus sebaceus of Jadassohn within the external auditory canal of a 3 year-old boy – A case report
Elżbieta Niemczyk, Kazimierz Niemczyk, Jadwiga Małdyk, Lidia Zawadzka-Głos
International Journal of Pediatric Otorhinolaryngology.2015; 79(11): 1932. CrossRef
Fehlbildungen und Nävi des behaarten Kopfes
V. Behle, H. Hamm
Der Hautarzt.2014; 65(12): 1022. CrossRef

Case Reports

Angiomyomatous Hamartoma of Popliteal Lymph Nodes Occurring in Association with Diffuse Pigmented Villonodular Synovitis of Knee.: Hyun Soo Kim, Ki Yong Na, Jae Hoon Lee, Nam Su Cho, Gou Young Kim, Sung Jig Lim; Korean J Pathol. 2011;45:S58-S61.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S58

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Abstract PDF: We report the first case of an angiomyomatous hamartoma (AH) of the popliteal lymph nodes (LNs) occurring in association with diffuse pigmented villonodular synovitis (PVNS) of the knee. AH is a rare benign vascular disease with a predisposition for the LNs of the inguinal region. Twenty-five cases of AH have been reported to date; however, the precise pathogenesis is still undetermined. In the present case, an open synovectomy revealed two of three popliteal LNs in close proximity to the extra-articular component of diffuse PVNS. These LNs demonstrated irregularly distributed thick-walled blood vessels in the hilum. These vessels extended into the medulla and cortex and were associated with haphazardly arranged smooth muscle cells in the sclerotic stroma. These findings are compatible with an AH. Our observations raise the possibility that AH of the popliteal LNs may represent an abnormal proliferative reaction against the inflammatory process caused by PVNS of the knee.

Pigmented Squamous Cell Carcinoma Arising from Pigmented Actinic Keratosis.: Hyun Joo Choi, Gyeong Sin Park, Seok Jin Kang, Yeong Jin Choi, Byung Kee Kim, Sun Moo Kim, Sang In Shim; Korean J Pathol. 1998;32(1):76-79.

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Abstract PDF: Pigmented squamous cell carcinoma is a very rare malignant, pigmented, epidermal tumor. The rarity of pigmented squamous cell carcinomas may reflect in part their misdiagnosis as other pigmented neoplasms, particularly malignant melanoma. To our knowledge, only five cases have been reported in literature. We recently experienced a case of pigmented squamous cell carcinoma arising from pigmented actinic keratosis in a 77 years old female. Physical examination showed a 0.8 0.6 cm, smooth, dark brown pigmented patch with irregular but sharply defined borders located on the upper left chest. The biopsy specimen showed histologic findings of pigmented actinic keratosis with abundant melanin pigments, which became pigmented squamous cell carcinoma. Most of pigments in the squamous cell carcinoma were contained within the melanocytes along with the neoplastic squamous cells.

Papillary Adenocarcinoma of Nonpigmented Ciliary Epithelium of the Eye.: Hyun Joo Choi, Yeong Jin Choi, Youn Soo Lee, Eun Jung Lee, Seok Jin Kang, Byung Kee Kim, Sang In Shim; Korean J Pathol. 1998;32(12):1104-1107.

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Abstract: Adenocarcinoma of the ciliary epithelium is a rare tumor, usually occuring in elderly patients who have a history of severe ocular trauma or chronic inflammation. We report an adenocarcinoma of the nonpigmented ciliary epithelium found within the phthisical globe of a 36-year-old female whose eye had been loss of vision since infancy. The mass, measured 4.0 x 4.0 cm, was relatively limited by sclera but had invasion to posterior portion. Histologically, the tumor was a compact mass which consisted of tubular and papillary structures with foci of the pleomorphic area. Strands of cells and individual cells were invested with thick basement membrane that have positivity for periodic acid-Schiff stain. Immunohistochemical staining showed strong reactivity for cytokeratin and epithelial membrane antigen, and focal for neuron-specific enolase and S-100 protein.

Fine Needle Aspiration Cytology of Pigmented Villonodular Synovitis: A Case Report .: Joon Hyuk Choi, Young Kyung Bae, Young Ran Shim, Mi Jin Kim, Won Hee Choi; Korean J Cytopathol. 1999;10(2):191-195.

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Abstract PDF: Pigmented villonodular synovitis is a destructive, fibrohistiocytic proliferation producing innumerable villous and nodular synovial protrusions. Its common locations are knee, ankle, foot, and hip. Although histologic feature of this tumor is well known, there have been few reports on the fine needle aspiration cytology findings. We report the cytologic features of a biopsy-proven case of pigmented villonodular synovitis. The patient was a 21-year-old male with a mass of the right knee for 2 years. On fine needle aspiration cytology, the aspirates was composed of abundant mononuclear histiocytic cells, singly and in clusters, multinucleated giant cells, and hemosiderin pigments.

Pigmented(melanotic) Schwannoma of the Cervical Spinal Canal: A case report.: Hyunee Yim, Jai Hyang Go, Chang Soo Ahn, Sun Woon Hong, Woo Hee Jung; Korean J Pathol. 1995;29(2):256-262.

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Abstract PDF: Pigmented(melanotic) schwannoma is a very rare variant of schwannoma that characteristically has massive cytoplasmic melanin. Since it was described in 1946 by Bjorneboe, about 44 cases have been reported in the English literature. It has a relatively benign clinical course, but the cases arising in the cranial nerve and sympathetic chain show aggressive behavior with malignant potential. We herein report a typical case of pigmented schwannoma with light microscopic findings. The results of immunohistochemical and electronmicroscopic study are also presented. The patient was a 30 year-old Korean male who had a mass in his posterior neck for 10 years that recently.began to induce neurologic manifestations. The tumor, which was mainly in the extramedullary intradural space of the cervical canal extending to the extradural space and soft tissues of the neck, was relatively well defined and composed of black solid tissue. Microscopic, densely pigmented spindle cells forming fascicles, nuclear palisading, whorling and polygonal cells with vacuolated or clear cytoplasm were characteristic. Immunohistochemically, the tumor cells were positive for vimentin, S-100 protein, and HMB-45. Electron microscopic study revealed that the tumor cells have interdigitating cytoplasmic processes containing varying stages of melanosomes with a very focal basal lamina and Luse body like collagen bundles.

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.

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