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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 1  |  Page : 80-82

Reverse masquerade: Benign sebaceous tumors mimicking sebaceous gland carcinoma of the eyelid


1 Orbit, Oculoplasty, Reconstructive and Aesthetic Services, Sankara Nethralaya, Medical Research Foundation, Chennai, Tamil Nadu, India
2 Larsen and Toubro Department of Ophthalmic Pathology, Sankara Nethralaya, Medical Research Foundation, Chennai, Tamil Nadu, India

Date of Submission31-Mar-2020
Date of Acceptance03-Aug-2020
Date of Web Publication31-Dec-2020

Correspondence Address:
Dr. Bipasha Mukherjee
Orbit, Oculoplasty, Reconstructive and Aesthetic Services Sankara Nethralaya, Medical Research Foundation, 18, College Road, Chennai - 600 006, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_758_20

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  Abstract 


Sebaceous lesions encompass a spectrum from benign lesions to disfiguring malignant lesions. Eyelid sebaceomas are extremely rare and only two definite cases are described in the English literature, whereas sebaceous adenomas occur in the eyelids more commonly and can be distinguished easily from sebaceous gland carcinoma by its clinical and histological appearance. We describe two cases of eyelid lesions that clinically resembled a possible malignant eyelid tumor. In both the cases, frozen section report was suggestive of a well-differentiated sebaceous gland carcinoma. However, histopathology examination of the permanent section was consistent with the diagnosis of sebaceoma and sebaceous adenoma respectively.

Keywords: Reverse masquerade, sebaceoma, sebaceous adenoma, sebaceous gland carcinoma


How to cite this article:
Barh A, Mukherjee B, Poonam NS, Krishnakumar S. Reverse masquerade: Benign sebaceous tumors mimicking sebaceous gland carcinoma of the eyelid. Indian J Ophthalmol Case Rep 2021;1:80-2

How to cite this URL:
Barh A, Mukherjee B, Poonam NS, Krishnakumar S. Reverse masquerade: Benign sebaceous tumors mimicking sebaceous gland carcinoma of the eyelid. Indian J Ophthalmol Case Rep [serial online] 2021 [cited 2021 Feb 25];1:80-2. Available from: https://www.ijoreports.in/text.asp?2021/1/1/80/305537



The histologic spectrum of eyelid sebaceous neoplasia includes sebaceous adenoma, sebaceoma, and sebaceous gland carcinoma (SGC). They can present as isolated skin lesions or as a part of Muir–Torre syndrome (MTS).[1]

Sebaceous adenomas are relatively common with distinctive clinical and histopathological features that differentiate them from the SGCs.

In contrast, sebaceomas are relatively rare benign tumors most commonly arising in the head and neck region and share features of both sebaceous adenoma and SGC. Diagnosis is essentially based on histopathology. Sebaceoma typically shows predominant basaloid cells (>50%) over the mature sebocytes which distinguish it from sebaceous adenoma where mature sebocytes are more predominant.[1] Unlike SGC, sebaceomas do not show atypical mitosis, nucleolar atypia, and/or necrosis.[2] Eyelid sebaceomas, per se, are very rare. We could find only two cases published in the English literature.[3],[4] In this report, we describe the atypical presentations of an eyelid sebaceoma and a sebaceous adenoma mimicking as SGC.


  Case Reports Top


Case 1

A 59-year-old gentleman presented with painless progressive right upper eyelid swelling for 3 months. On examination, a reddish mass was noted in the inner surface of the right upper lid with localized loss of eyelashes. On everting the eyelid, a lobulated, nodular, reddish-orange colored mass was found attached to the underlying tarsal plate with congestion of surrounding palpebral conjunctiva [Figure 1]A and [Figure 1]B. The rest of his ocular examination was normal. In view of a possible SGC, he underwent complete excision of the mass under the frozen section, which showed numerous sebaceous glands in the dermis with multiple tumor lobules composed of sebocytes and basaloid cells [[Figure 1]C1]. Mitosis and nuclear atypia were seen in the tumor lobules, suggestive of a well-differentiated SGC. Primary reconstruction was done after margin clearance with direct closure. However, subsequently, the permanent section of the mass showed a lobular arrangement of sebaceous gland-derived tumor composed of peripheral basaloid cells (>50%) and central sebaceous glands [[Figure 1]C2]. Numerous intermediate cells, sebaceous ducts, and cystic spaces were seen. Peripheral basaloid cells showed occasional mitotic activity (6–8 per 10 HPF). Nucleolar atypia, atypical mitosis, and necrosis were not found. These findings were suggestive of a sebaceoma. Immunohistochemistry was positive for epithelial membrane antigen (EMA) and cytokeratin (AE1/AE), but negative for carcinoembryonic antigen (CEA). The patient was screened for MTS. His colonoscopy was normal. At 18-month follow-up, he showed good cosmetic outcomes without any recurrence [Figure 1]D and [Figure 1]E.
Figure 1: (A and B) Clinical picture of case1 showing a lobulated, nodular, reddish-orange mass attached to the inner surface of the right upper eyelid; C1. A frozen section (400×) showing numerous sebaceous glands in the dermis with multiple tumor lobules composed of sebocytes (black arrow) and basaloid cells (white arrow); C2. Permanent section (100×) showing a lobular arrangement of sebaceous gland derived tumor composed of peripheral basaloid cells (>50%, white arrow) and central sebaceous glands (black arrow); (d and e) Clinical picture showing good cosmesis without any recurrence at follow-up

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Case 2

A 62-year-old gentleman presented with painless progressive left lower eyelid swelling for 6months. On examination, a nodular, non-tender, reddish-pink colored lesion (7 × 6 mm) was noted in the middle third of the left lower eyelid with the involvement of the lid margin and tarsal conjunctiva. There was a localized loss of eyelashes [Figure 2]a. The rest of his ocular examination was normal. He underwent excision biopsy under the frozen section which revealed the lobular arrangement of the tumor cells with extensive sebaceous differentiation suggestive of a possible well-differentiated SGC. Primary repair was done by direct closure after margin clearance. Histopathology of the permanent section showed a well-circumscribed tumor composed of lobules of tumor cells. Tumor lobules were composed of sebaceous glands (>50%) and less peripheral basaloid cells [Figure 2]b. There was no atypia or mitotic activity. All features were suggestive of a sebaceous adenoma. Colonoscopy was normal. The patient did not show any recurrence at 6-month follow-up.
Figure 2: (a) Clinical picture of the Second patient at presentation showing nodular, reddish-pink colored lesion (size 7 mm × 6 mm) involving the middle third of the left lower eyelid with the involvement of the lid margin. There was a localized loss of eyelashes and the lesion involved the left lower tarsal conjunctiva. (b) Histopathology microphotograph of permanent section (hematoxylin and eosin staining, original magnification ×200) showing well-circumscribed lid tumor composed of lobules of tumor cells. More than 50% of the lobules are composed of sebaceous glands (arrow) and less peripheral basaloid cells. There was no atypia and no mitotic activity; features were suggestive of a sebaceous adenoma

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  Discussion Top


In the spectrum of sebaceous tumors, sebaceoma is the most uncommon benign neoplasm. These lesions are usually located in the dermis but can extend into the overlying epidermis.[1],[2] The basaloid components are more prominent (>50%) than sebocytes.[1] Though mitoses are not a characteristic feature of sebaceoma, conspicuous mitoses have been documented in previous literature, which may give rise to the suspicion of possible SGC.[4] Nonetheless, the absence of nucleolar atypia and necrosis within the tumor lobules usually rules out an SGC [Table 1].[3]
Table 1: Histopathologic features of eyelid sebaceous neoplasms

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Immunohistochemistry cannot differentiate amongst various sebaceous tumors. Intracytoplasmic lipid contents of sebaceous components are positive for oil red-O stain. Sebaceous cells are positive for EMA but negative for CEA. Basaloid cells are usually positive for cytokeratin.[1]

To date, only two cases of well-documented eyelid sebaceomas have been reported in the English literature.[3],[4] In these reports, the tumors presented as a well-defined, small, reddish nodule, involving the lid margin. Occasional mitosis was noted in one case, but cytological atypia was not seen in any of the cases [Table 2]. In our patient, the lesion was comparatively larger, ill-defined, and attached to the underlying tarsus.
Table 2: Details of all reports describing eyelid sebaceoma

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SGC is the most common eyelid malignancy in the Asian-Indian population.[5] Since the frozen section showed occasional mitosis with nuclear atypia, it was initially considered as a well-differentiated SGC.

Another differential of sebaceoma is basal cell carcinoma (BCC) which can be distinguished from sebaceoma by histopathology and immunohistochemistry. On histopathology, BCC usually shows distinct peripheral basal cells palisading, loose fibromucinous stroma, and tumor-stroma separation (crack) artifacts in formalin-fixed sections. The BCCs typically show positive staining for BerEP4 and negative for EMA, whereas the sebaceomas show exactly the opposite pattern.[1]

Sebaceous adenoma usually presents as a tan to yellow colored nodular lesion <5 mm in the greatest dimension.[1],[2] Clinical evidences of a malignant lesion include involvement of the lid margin, loss of the lashes, and tumor extension towards the palpebral conjunctiva. In case 2, though the clinical presentation and frozen section findings were not very conclusive, histopathology of the permanent section clearly ruled out SGC.

Benign sebaceous tumors, located above the neck, are usually not associated with MTS. However, a routine colonoscopy is advisable in all such patients.

The diagnostic accuracy of frozen sections for eyelid tumors is quite high. However, it is always inferior to the paraffin-based permanent sections. Though the maximum discordancy is due to false-negative results, a false positive result is not uncommon, as we experienced in both these cases. The factors commonly affecting the accuracy of the frozen sections include technical faults while preparing the sample or cutting the sections, poor staining quality, and an error of judgment by the pathologist.[6] The rarity of the disease might impart a false impression as seen in the first case.


  Conclusion Top


To conclude, the sebaceoma of the eyelid is extremely rare. Hence, diagnosis is difficult and because of overlapping features with SGC, it can be easily misdiagnosed. On the other hand, though common, the sebaceous adenoma can present atypically like a malignant lesion. To avoid unnecessary over-treatment, one should preferably wait for the permanent histopathology reports before proceeding with definitive management.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Shalin SC, Lyle S, Calonje E, Lazar AJ. Sebaceous neoplasia and the Muir-Torre syndrome: Important connections with clinical implications. Histopathology. 2010;56:133-47.  Back to cited text no. 1
    
2.
Misago N, Mihara I, Ansai S, Narisawa Y. Sebaceoma and related neoplasms with sebaceous differentiation: A clinicopathologic study of 30 cases. Am J Dermatopathol 2002;24:294-304.  Back to cited text no. 2
    
3.
Mittal R, Tripathy D. Sebaceoma of the eyelid: A rare entity. Can J Ophthalmol 2014;49:e78-80.  Back to cited text no. 3
    
4.
Yonekawa Y, Jakobiec FA, Zakka FR, Fay A. Sebaceoma of the eyelid. Ophthalmology 2012;119:2645.e1-4.  Back to cited text no. 4
    
5.
Kaliki S, Ayyar A, Dave TV, Ali MJ, Mishra DK, Naik MN. Sebaceous gland carcinoma of the eyelid: Clinicopathological features and outcome in Asian Indians. Eye (Lond) 2015;29:958-63.  Back to cited text no. 5
    
6.
Alam MS, Tongbram A, Krishnakumar S, Biswas J, Mukherjee B. Sensitivity and specificity of frozen section diagnosis in orbital and adnexal malignancies. Indian J Ophthalmol 2019;67:1988-92.  Back to cited text no. 6
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