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Primary Intraepithelial Sebaceous Gland Carcinoma of the Palpebral Conjunctiva

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Primary Intraepithelial Sebaceous Gland Carcinoma of the Palpebral Conjunctiva CLINICOPATHOLOGIC REPORT SECTION EDITOR: W. RICHARD GREEN, MD Primary Intraepithelial Sebaceous Gland Carcinoma of the Palpebral Conjunctiva Santosh G. Honavar, MD; Carol L. Shields, MD; Marlon Maus, MD; Jerry A. Shields, MD; Hakan Demirci, MD; Ralph C. Eagle, Jr, MD ebaceous gland carcinoma usually arises from meibomian or Zeis glands deep within the eyelid, but it can rarely arise within the conjunctival epithelium without a deep com- ponent. We describe a woman with a history of chronic blepharoconjunctivitis unre- sponsive to topical medications. Examination disclosed confluent papillary hypertro- phyS of the upper palpebral conjunctiva and deposits of white flaky material. Tarsoconjunctival punch biopsy revealed intraepithelial sebaceous gland carcinoma. Management consisted of frozen section– controlled complete tumor excision with removal of the entire posterior lamella of the right upper eyelid, cryotherapy to the margins, and reconstruction. Histopathologic analysis confirmed pri- mary sebaceous gland carcinoma localized to the conjunctival epithelium without involvement of underlying meibomian or Zeis glands or the caruncle. Patients with unexplained chronic unilat- eral blepharoconjunctivitis or papillary hypertrophy of the palpebral conjunctiva should be con- sidered for biopsy to rule out neoplasia, even when there is no sign of an underlying eyelid mass. Arch Ophthalmol. 2001;119:764-767 Sebaceous gland carcinoma of the ocular fined to and presumably arising primar- adnexa is a relatively rare tumor that arises ily within the conjunctival epithelium from the meibomian glands, Zeis glands, without underlying glandular or invasive or sebaceous glands in the caruncle or eye- component is uncommon.8-10 Herein, we brow.1-7 It is estimated that this cancer rep- describe one such case with supportive resents 1% to 6% of all eyelid malignant histopathologic findings. neoplasms.4 The clinical presentation of sebaceous gland carcinoma depends on its site of origin. Sebaceous gland carcinoma REPORT OF A CASE of meibomian gland origin usually pre- sents as a slowly enlarging, deep tarsal mass A 33-year-old white woman developed that may simulate a chalazion.4 The Zeis contact lens intolerance, conjunctival hy- gland tumor appears as a well-circum- peremia, and irritation in her right eye for scribed mass near the eyelid margin.3 Some 1 year. She had been treated with topical patients have unilateral chronic blepha- medications for chronic blepharoconjunc- roconjunctivitis before the tumor is clini- tivitis without relief. Her only medical cally obvious, leading to delay in diag- problem was idiopathic thrombocytope- nosis.8 Patients with this presentation nic purpura, treated with varying doses of generally manifest conjunctival intraepi- oral corticosteroids for 14 years. There was thelial invasion of sebaceous gland carci- no history of radiotherapy to the face or noma (pagetoid spread) from a primary fo- systemic malignancy. On examination, her cus of tumor in the meibomian or Zeis visual acuity was 20/20 OU. The left eye glands.4,8 Sebaceous gland carcinoma con- was unremarkable. The right eye re- vealed diffuse hyperemic papillary hyper- From the Oncology Service (Drs Honavar, C. L. Shields, J. A. Shields, and Demirci), trophy of the entire upper palpebral con- Oculoplastics Service (Dr Maus), and Department of Pathology (Dr Eagle), Wills Eye junctiva and scattered deposits of white Hospital, Thomas Jefferson University, Philadelphia, Pa; and Oncology Service, flaky material (Figure 1A). There was no L.V. Prasad Eye Institute, Hyderabad, India (Dr Honavar). evident eyelid mass or madarosis. Mini- (REPRINTED) ARCH OPHTHALMOL / VOL 119, MAY 2001 WWW.ARCHOPHTHALMOL.COM 764 ©2001 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/26/2021 A B Figure 1. A, Conjunctival primary intraepithelial sebaceous gland carcinoma manifesting as diffuse papillary hypertrophy of the upper palpebral conjunctiva. B, An intraoperative photograph showing the entire tarsus and the palpebral conjunctiva up to the superior fornix being excised. Margins were uninvolved by frozen section biopsy examination. mal bulbar conjunctival conges- and hyperchromatic nuclei, focally Conjunctival intraepithelial se- tion and diffuse superficial punc- vacuolated cytoplasm, and promi- baceous gland carcinoma typically tate keratopathy were noted. There nent mitotic activity (Figure 2A-B). arises from an underlying primary was no obvious involvement of the The cells stained positively with oil- meibomian or Zeis gland carci- bulbar or inferior palpebral conjunc- red-O confirming the presence of in- noma that secondarily invades the tiva. The clinical differential diag- tracytoplasmic lipid (Figure 2C). Im- conjunctival epithelium by a cen- nosis included squamous or seba- munohistochemical analysis showed tripetal or radial migration of tu- ceous neoplasia of the conjunctiva that the cells were strongly immu- mor cells.8 This has been correlated as well as atypical conjunctival in- noreactive for BRST-2, focally posi- with poor ocular and life progno- fection. A deep tarsoconjunctival tive for cytokeratin marker CAM 5.2, sis.6,7 Three histopathologic pat- punch biopsy revealed purely in- and minimally immunoreactive for terns are exhibited by intraepithe- traepithelial sebaceous gland carci- epithelial membrane antigen, con- lial conjunctival sebaceous gland noma without an underlying tarsal sistent with the diagnosis of seba- carcinoma—the bowenoid, paget- component. Map biopsy specimens ceous gland carcinoma. Step sec- oid, and papillary types.6-8 The from 9 other sites on the bulbar and tions showed that the tumor was bowenoid type is characterized by lower palpebral conjunctiva were confined to the palpebral conjunc- full-thickness replacement of the negative for malignancy. The tu- tiva without deep involvement of the epithelium by tumor cells that are mor management involved com- meibomian glands or Zeis glands. large and pleomorphic and exhibit plete excision of all sebaceous units The margins of the excised area pro- prominent mitotic activity. The pag- of the upper eyelid using frozen sec- cessed for frozen sections were nega- etoid type is characterized by scat- tion–controlled excision of the en- tive for tumor. The final diagnosis tered individual tumor cells or nests tire posterior lamella including the was primary intraepithelial seba- of tumor cells within the epithe- eyelid margin (Figure 1B) and cryo- ceous gland carcinoma of the pal- lium that are devoid of intercellular therapy to the margins. The defect pebral conjunctiva. bridges. The papillary pattern is rare was reconstructed with a free tarso- and manifests with intraepithelial conjunctival graft from the oppo- COMMENT confluent cells resembling carci- site upper eyelid. Healing was ex- noma in situ.8 In our case, the pat- cellent and all permanent margins In 1967, Theodore11 and Irvine12 de- tern of conjunctival involvement was were free of tumor. The patient was scribed a “masquerade syndrome” of the bowenoid type. free of tumor recurrence 1 year af- characterized by chronic blepharo- It has been estimated that ap- ter the surgery. conjunctivitis due to an unsus- proximately 5% of patients with con- The surgical specimen was re- pected conjunctival intraepithelial junctival intraepithelial sebaceous ceived as a single piece of fresh un- squamous cell or sebaceous gland gland carcinoma show no detect- fixed tissue. A segment of the speci- carcinoma. Although the originally able eyelid nodule at initial presen- men was submitted for frozen described neoplasms were squa- tation.8 However, this has not been sections and staining for the lipid mous cell carcinomas, in retro- histopathologically proven with step with oil-red-O stain. The remain- spect, many of the tumors produc- or serial sections in most cases.8-10 der of the specimen was fixed in for- ing this clinical picture may have Freeman and associates15 described malin, sectioned perpendicular to been sebaceous gland carcino- 2 such cases but the histopathologic the lid margin in a bread-loaf fash- ma.13,14 Others have also empha- evidence was inadequate. Margo and ion, and submitted for routine light sized that tarsoconjunctival inflam- associates9 reported one case of a 65- microscopy. The palpebral conjunc- mation is common in patients with year-old woman whose exentera- tiva was thickened and replaced by intraepithelial sebaceous gland car- tion specimen showed intraepithe- tumor cells that had pleomorphic cinoma of the conjunctiva.8-10 lial sebaceous gland carcinoma (REPRINTED) ARCH OPHTHALMOL / VOL 119, MAY 2001 WWW.ARCHOPHTHALMOL.COM 765 ©2001 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/26/2021 orbital exenteration. The meibo- A mian and Zeis glands showed no car- cinoma but were completely re- placed by lipogranulomatous inflammation. Our case was unique in several respects. The tumor oc- curred in a young patient aged 33 years who had been receiving oral corticosteroids. Immunosuppres- sion may have contributed to the young age at onset of sebaceous neo- plasia, as the patient had no other known predisposition, such as radia- tion exposure.16 In addition, there was no histopathologic evidence of inflammation, scarring, or tumor in the underlying sebaceous glands. The lack of underlying tarsal tumor on B histopathologic examination is ex- tremely unusual and raises specula- tion as to the source of the malig- nant
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