Ophthal Plast Reconstr Surg, Vol. 31, No. 2, 2015

Case Reports

Treatment recommended for glial heterotopia is a complete excision of the mass even though there is no malignant or metastatic potential.4–9 There is a controversy in the literature regarding the growth potential of these masses, but many believe that conservative management risks a possible continued growth with its attendant complications depending on the site. However, incomplete excisions are noted to have a recurrence rate of around 10%.6–8 The beneficial effects of radiotherapy are unproven and questionable.6 In conclusion, although extremely rare, glial heterotopic nodule should be kept in the differential diagnosis of a dacryocystocele. The presence of the bony defect in this case gives evidence to the theory of mechanical isolation of glial tissues from the brain by the developing cranial bones.

aerodigestive tract. Herein, the authors present a ­77-yearold woman with a basaloid squamous cell carcinoma over her limbal conjunctiva in the OD.

REFERENCES

A 77-year-old Taiwanese woman had a painless conjunctival mass in the OD that increased in size gradually for many years. On ophthalmic examinations, an elastic, pedunculated mass measuring 9 × 6 × 5 mm in size originating from the limbus with corneal involvement was found (Fig. 1). Other ophthalmic findings were not remarkable. Orbital CT scan demonstrated a soft tissue mass over medial portion of conjunctiva in the OD without orbital involvement or localized lymphadenopathy. Superficial keratectomy was performed to dissect the corneal and limbal component of the lesion; the conjunctival component along with a cuff of normal conjunctival tissue was removed. The histopathologic study (Fig. 2) showed small and crowded basaloid cells with scant cytoplasm and hyperchromatic nuclei. The tumor cells clustered in variably sized lobules and nests with prominent peripheral palisading. There were stromal hyalinosis and some small intercellular cystic spaces with mucin-like materials. Some foci of conventional SCC adjacent to the basaloid component were also seen, which was compatible with BSCC. The additional immunohistochemistry staining for human papillomavirus (HPV) diagnosis were p53 positive and p16 ­negative (Fig. 3A,B), which indicates HPV-negative. Meanwhile, positive staining of p63 and Ki-67 occurred diffusely throughout the tumor mass (Fig. 3C, D), and the ­ ­pattern is suggestive of a rapid growing malignancy. Although further excision and adjuvant mitomycin-C application was suggested, the patient was reluctant to receive

1. MacEwen CJ, Young JD. Epiphora during the first year of life. Eye (Lond) 1991;5(Pt 5):596–600. 2. Mansour AM, Cheng KP, Mumma JV, et al. Congenital dacryocele. A collaborative review. Ophthalmology 1991;98:1744–51. 3. Shashy RG, Durairaj VD, Durairaj V, et al. Congenital dacryocystocele associated with intranasal cysts: diagnosis and management. Laryngoscope 2003;113:37–40. 4. Tambay MC, Rodriguez IZ, Gil YR, et al. Heterotopic neuroglial tissue as a congenital laterocervical mass: a case report. Int J Oral Maxillofac Surg 2009;38:382–4. 5. Aanaes K, Hasselby JP, Bilde A, et al. Heterotopic neuroglial tissue: two cases involving the tongue and the buccal region. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:e22–9. 6. Newman NJ, Miller NR, Green WR. Ectopic brain in the orbit. Ophthalmology 1986;93:268–72. 7. Bajaj MS, Kashyap S, Wagh VB, et al. Glial heterotopia of the orbit and extranasal region: an unusual entity. Clin Experiment Ophthalmol 2005;33:513–5. 8. Ghose S, Balasubramaniam ST, Mahindrakar A, et al. Orbital ectopic glial tissue in relation to medial rectus: a rare entity. Clin Experiment Ophthalmol 2005;33:67–9. 9. Kiratli H, Sekeroğlu MA, Tezel GG. Orbital heterotopic glial tissue presenting as exotropia. Orbit 2008;27:165–8. 10. Skelton HG, Smith KJ. Glial heterotopia in the subcutaneous tissue overlying T-12. J Cutan Pathol 1999;26:523–7.

Basaloid Squamous Cell Carcinoma of the Conjunctiva

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asaloid squamous cell carcinoma (BSCC), which commonly arises in the upper aerodigestive tract, is an aggressive variant of squamous cell carcinoma (SCC).1,2 To date, only 1 case of BSCC of the conjunctiva has been reported in the literature.3 Our report presents a patient with conjunctival BSCC, which was recognized and further confirmed with histopathological studies. The patient had no disease recurrence during the 6-month follow-up period.

CASE REPORT

Yu Chang Liu*, Nancy Chen, M.D.†, Yung-Hsiang Hsu, M.D., M.S.‡, Min-Muh Sheu, M.D., M.T.M.†§, and Rong Kung Tsai, M.D., Ph.D.†§ Abstract: Basaloid squamous cell carcinoma is a distinct variant of squamous cell carcinoma, and it is more aggressive and has a poorer prognosis than conventional squamous cell carcinoma. Basaloid squamous cell carcinoma has been reported to arise from many organs, mainly in the upper Accepted for publication September 24, 2013. *Department of Medicine, College of Medicine, Tzu Chi University; Departments of †Ophthalmology and ‡Pathology, Buddhist Tzu Chi General Hospital; and §Department of Ophthalmology and Visual Science, Tzu Chi University, Hualien, Taiwan The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Rong Kung Tsai, M.D., Ph.D., Department of Ophthalmology, Buddhist Tzu Chi General Hospital and Department of Ophthalmology and Visual Science, Tzu Chi University, 707 Sec. 3, Chung Yung Road, Hualien 970, Taiwan. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000056

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FIG. 1.  An elastic, pedunculated mass originated from the limbus with corneal invasion in OD.

© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

Ophthal Plast Reconstr Surg, Vol. 31, No. 2, 2015

Case Reports

FIG. 2.  A, Small and crowded basaloid cells arranged in lobular or nesting pattern with peripheral palisading and small intercellular cystic spaces (arrows) (hematoxylin-eosin, ×100). B, Basaloid cells with scant cytoplasm and hyperchromatic nuclei, numerous mitotic figures (arrows) and stromal hyalinosis (arrowheads) were found (hematoxylin-eosin, ×400). C, Some foci of conventional squamous cell carcinoma (asterisk) were adjacent to the basaloid component (hematoxylin-eosin, ×40). D, Squamous differentiation was found in the central area of some basaloid cell nests (hematoxylin-eosin, ×400).

FIG. 3.  The results of immunohistochemistry staining: Very strong expression of p53 (A) in tumor cells (×100) and negative staining of p16 (B) (×400) indicated no HPV infection. Diffuse staining of p63 (C) and Ki-67 (D) in tumor cells (×100) revealed a rapid growing malignancy.

© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

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Ophthal Plast Reconstr Surg, Vol. 31, No. 2, 2015

Case Reports

more advance treatments. At 6 months after the surgery, the tumor had no progression on the ocular surface, and there was no lymphadenopathy found.

DISCUSSION The most common affected site of BSCC is the upper aerodigestive tract, including base of tongue, hypopharynx, and supraglottic larynx.1,2 Although BSCC has been reported in mouth, palate, tonsil, paranasal sinus, lung, esophagus, anus, and penis,3–8 BSCC of the conjunctiva is rare.3 Common amelanotic ocular surface tumors originating from limbus include dermoid, pyogenic granuloma, squamous papilloma, conjunctival intraepithelial neoplasia, SCC, and sometimes amelanotic melanoma.9 Conjunctival dermoid is a congenital, well-circumscribed, and yellow-white solid tumor; pyogenic granuloma is mostly found on the patient with previous tissue insult. The tumor of this patient presented at old age and was pinkish and pedunculated; meanwhile, this patient did not have eye trauma or surgery previously. However, it is difficult to differentiate basaloid SCC from squamous papilloma, other squamous neoplasms, and amelanotic melanoma solely by the clinical picture. Characteristic histopathologic findings demonstrated that the tumor comprises 2 different components. The basaloid component is characterized by solid growths of cells in a lobular pattern with peripheral palisading, small and crowded cells with scant cytoplasm, hyperchromatic nuclei without nucleoli, and small cystic spaces containing mucinlike materials.1 Accessory findings, including coagulative necrosis within the central areas of the tumor lobules and stromal hyalinosis, may be found in some cases.1 The squamous component of the tumor, such as SCC, dysplasia, or focal squamous differentiation, is always beside the basaloid component.1 The pathologic study of this patient is compatible with the above descriptions. The histopathologic features of BSCC are similar to those of adenoid cystic carcinoma, small cell neuroendocrine carcinoma, conventional SCC, basal cell carcinoma, and mucoepidermoid carcinoma, especially in a small biopsy specimen.1–3 However, the presence of both basaloid and squamous components could differentiate BSCC from those tumors. Another entity, basosquamous carcinoma, which is a variant of basal cell carcinoma, is considered transition between basal cell and SCC,10 whereas BSCC was defined as a variant of SCC. In this case, no basal cell carcinoma component is noted. Therefore, the diagnosis of BSCC was made. Vasudev et al.3 reported a 64-year-old man with an eyelid mass arising from the palpebral conjunctiva. The histopathologic diagnosis of the biopsy specimen was basal cell carcinoma initially. However, the following tumor wide excision revealed typical features of BSCC with both basaloid and squamous components. After tumor wide excision, the patient did not receive adjunvant therapy as this patient. BSCC occurs predominantly in older men with a mean age of 63 years.2 Tobacco and alcohol usage have been associated with the occurrences of BSCC.4,11 Recently, HPV infection is regarded as an important risk factor.12,13 The HPV-positive BSCC tends to occur in the oropharynx and has a favorable outcome.12,13 This patient, with p16-negative and p53-positive immunohistochemistry staining, was in the HPV-negative category.13 BSCC had a more aggressive behavior than conventional

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SCC with poor survival rate and a high incidence of cervical lymphadenopathy and distal metastasis at initial presentation.2,14 The diffuse p63 and ki-67 pattern is also suggestive of a ­rapid-growing malignancy.15 Although the prognosis of conjunctival BSCC is unknown, the authors should at least treat it as conventional conjunctival SCC. Herein, the authors reported a case of conjunctival BSCC, which has a typical feature of basaloid cells in lobular pattern with peripheral palisading and along with some foci of SCC beside. The risk factors on this patient are old age, tobacco, and alcohol usage. BSCC tends to be more aggressive than convention SCC, and the HPV-negative ones have poorer prognosis than HPV-positive ones. However, this patient had no clinical evidence of progression at 6 months after the en bloc excision.

REFERENCES 1. Wain SL, Kier R, Vollmer RT, et al. Basaloid-squamous carcinoma of the tongue, hypopharynx, and larynx: report of 10 cases. Hum Pathol 1986;17:1158–66. 2. Raslan WF, Barnes L, Krause JR, et al. Basaloid squamous cell carcinoma of the head and neck: a clinicopathologic and flow cytometric study of 10 new cases with review of the English literature. Am J Otolaryngol 1994;15:204–11. 3. Vasudev P, Boutross-Tadross O, Radhi J. Basaloid squamous cell carcinoma: two case reports. Cases J 2009;2:9351. 4. Ereño C, Gaafar A, Garmendia M, et al. Basaloid squamous cell carcinoma of the head and neck: a clinicopathological and ­follow-up study of 40 cases and review of the literature. Head Neck Pathol 2008;2:83–91. 5. Brambilla E, Moro D, Veale D, et al. Basal cell (basaloid) carcinoma of the lung: a new morphologic and phenotypic entity with separate prognostic significance. Hum Pathol 1992;23: 993–1003. 6. Sarbia M, Verreet P, Bittinger F, et al. Basaloid squamous cell carcinoma of the esophagus: diagnosis and prognosis. Cancer 1997;79:1871–8. 7. Chetty R, Serra S, Hsieh E. Basaloid squamous carcinoma of the anal canal with an adenoid cystic pattern: histologic and immunohistochemical reappraisal of an unusual variant. Am J Surg Pathol 2005;29:1668–72. 8. Cubilla AL, Reuter VE, Gregoire L, et al. Basaloid squamous cell carcinoma: a distinctive human papilloma virus-related penile neoplasm: a report of 20 cases. Am J Surg Pathol 1998;22:755–61. 9. Shields CL, Shields JA. Tumors of the conjunctiva and cornea. Surv Ophthalmol 2004;49:3–24. 10. de Faria J. Basal cell carcinoma of the skin with areas of squamous cell carcinoma: a basosquamous cell carcinoma? J Clin Pathol 1985;38:1273–7. 11. Banks ER, Frierson HF, Jr, Mills SE, et al. Basaloid squa mous cell carcinoma of the head and neck. A clinicopathologic and immunohistochemical study of 40 cases. Am J Surg Pathol 1992;16:939–46. 12. Begum S, Westra WH. Basaloid squamous cell carcinoma of the head and neck is a mixed variant that can be further resolved by HPV status. Am J Surg Pathol 2008;32:1044–50. 13. Chernock RD, Lewis JS Jr, Zhang Q, et al. Human papillomavirus-positive basaloid squamous cell carcinomas of ­ the upper aerodigestive tract: a distinct clinicopathologic and molecular subtype of basaloid squamous cell carcinoma. Hum Pathol 2010;41:1016–23. 14. Soriano E, Faure C, Lantuejoul S, et al. Course and prognosis of basaloid squamous cell carcinoma of the head and neck: a c­ ase-control study of 62 patients. Eur J Cancer 2008;44:244–50. 15. Winters R, Naud S, Evans MF, et al. Ber-EP4, CK1, CK7 and CK14 are useful markers for basaloid squamous carcinoma: a study of 45 cases. Head Neck Pathol 2008;2:265–71.

© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

Basaloid squamous cell carcinoma of the conjunctiva.

Basaloid squamous cell carcinoma is a distinct variant of squamous cell carcinoma, and it is more aggressive and has a poorer prognosis than conventio...
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