Ocular Sebaceous Carcinoma In Situ With Biphenotypic Differentiation: A Reappraisal of the Alternative Origin of the Tumor

Abstract

Ocular sebaceous carcinoma is an uncommon, aggressive tumor arising from the Meibomian gland, Zeis gland, or sebaceous glands in the caruncle or eyelashes. We described a rare case of sebaceous carcinoma in situ in a 51-year-old female that was characterized by intraepithelial growth of sebaceous carcinoma cells with no invasive carcinoma in the underlying sebaceous glands. Early stromal invasion was identified that featured 2 distinctive but focally intermixed populations of sebaceous carcinoma cells and squamous carcinoma cells. The 2 populations of cells exhibited their respective distinct immunophenotype but both showed strong overexpression of p53. This case provides evidence to support the hypothesis that some ocular sebaceous carcinoma may arise from intraepithelial pluripotent stem cells.

Keywords

sebaceous carcinoma ocular sebaceous carcinoma in situ pluripotent stem cell squamous cell carcinoma

Get full access to this article

View all access options for this article.

References

Dubovy

Eberhart

Vemuganti

. Sebaceous carcinoma of the conjunctiva and caruncle. In: Grossniklaus

Eberhart

Kivela

, eds. WHO Classification of Tumours of the Eye. Lyon, France: IARC Press; 2018:25.

Jakobiec

. Sebaceous tumors of the ocular adnexa. In: Albert

Jakobiec

, eds. Principles and Practice of Ophthalmology: Clinical Practice. Vol 3. Philadelphia, PA: WB Saunders; 1994:1745-1770.

Honavar

Shields

Maus

Shields

Demirci

Eagle

Jr.

Primary intraepithelial sebaceous gland carcinoma of the palpebral conjunctiva. Arch Ophthalmol. 2001;119:764-767.

Margo

Grossniklaus

HE.

Intraepithelial sebaceous neoplasia without underlying invasive carcinoma. Surv Ophthalmol. 1995;39:293-301.

Currie

Plaza

Harris

GJ.

Intraepithelial sebaceous carcinoma: a case report of an unusual occurrence. Am J Dermatopathol. 2014;36:673-676.

Margo

Lessner

Stern

GA.

Intraepithelial sebaceous carcinoma of the conjunctiva and skin of the eyelid. Ophthalmology. 1992;99:227-231.

Loeffler

Perlman

JI.

Diffuse intraepithelial sebaceous carcinoma of the conjunctiva. Br J Ophthalmol. 1997;81:168.

Arits

van Marion

Thissen

Kloos

Kelleners-Smeets

NW.

Development and progression of a periorbital sebaceous gland carcinoma in situ. Acta Derm Venereol. 2010;90:529-530.

Leibovitch

Selva

Huilgol

Davis

Dodd

James

CL.

Intraepithelial sebaceous carcinoma of the eyelid misdiagnosed as Bowen’s disease. J Cutan Pathol. 2006;33:303-308.

10.

Margo

CE.

Periocular intraepithelial sebaceous neoplasia: critical appraisal of nomenclature and prognostic importance. J Clin Pathol. 2020;73:172-175.

11.

Rao

Hidayat

McLean

Zimmerman

LE.

Sebaceous carcinomas of the ocular adnexa: a clinicopathologic study of 104 cases, with five-year follow-up data. Hum Pathol. 1982;13:113-122.

12.

Kramer

Chen

Sebaceous carcinoma in situ. Am J Dermatopathol. 2010;32:854-855.

13.

Kazakov

Kutzner

Spagnolo

Rutten

Mukensnabl

Michal

What is extraocular cutaneous sebaceous carcinoma in situ?

Am J Dermatopathol. 2010;32:857-858.

14.

Komforti

Asgari

Chen

Sebaceous carcinoma in situ as a concept and diagnostic entity. Dermatol Pract Concept. 2017;7:27-31.

15.

Foster

Allansmith

MR.

Chronic unilateral blepharoconjunctivitis caused by sebaceous carcinoma. Am J Ophthalmol. 1978;86:218-220.

16.

Freeman

Iliff

Green

WR.

Extramammary Paget’s disease/pagetoid change of the conjunctiva without underlying sebaceous carcinoma (abstract). Invest Ophthalmol Vis Sci. 1988;29(suppl):321.

17.

Fulling

Strayer

Cruz

DJS

. Adnexal metaplasia in carcinoma in situ of the skin. J Cutan Pathol. 1981;8:79-88.

18.

Oka

Katsumata

Intraepidermal sebaceous carcinoma: case report. Dermatologica. 1990;180:181-185.

19.

Ansai

Mihara

Sebaceous carcinoma arising on actinic keratosis. Eur J Dermatol. 2000;10:385-388.

20.

Ishida

Okabe

Intraepidermal sebaceous carcinoma occurring concurrently with actinic keratosis. J Cutan Pathol. 2012;39:731-732.

21.

Aung

Batrani

Mirzabeigi

Goldberg

LJ.

Extraocular sebaceous carcinoma in situ: report of three cases and review of the literature. J Cutan Pathol. 2014;41:592-596.

22.

Jacobs

Sandles

Leboit

PE.

Sebaceous carcinoma arising from Bowen’s disease of the vulva. Arch Dermatol. 1986;122:1191-1193.

23.

Nakashima

Adachi

Yamasaki

Yamada

Yoshida

Yamamoto

Sebaceous carcinoma with actinic keratosis. Acta Derm Venereol. 2010;90:196-198.

24.

Ishida

Iwai

Yoshida

Kagotani

Okabe

Sebaceous carcinoma associated with Bowen’s disease: a case report with emphasis on the pathogenesis of sebaceous carcinoma. Int J Clin Exp Pathol. 2013;6:3029-3032.

25.

Misago

Kuwashiro

Tsuruta

Narisawa

Sebaceous carcinoma in association with actinic keratosis: a report of two cases with an immunohistochemical study. J Dermatol. 2015;42:616-620.

26.

Namiki

Miura

Yokozeki

Ansai

SI.

Bowen disease with sebaceous differentiation: a case report and immunohistochemical analysis of adipophilin and cytokeratin 1. Am J Dermatopathol. 2018;40:841-845.

27.

Omori

Tanito

Ito

, et al. Bowen’s carcinoma of the penis with sebaceous differentiation associated with human papillomavirus type 16. Int J Dermatol. 2014;53:e531-532.