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A cystic epithelial downgrowth mimics an intraocular tumour following penetrating eye trauma
CORRESPONDENCE A cystic epithelial downgrowth mimics an intraocular tumour following penetrating eye trauma Epithelial ingrowth, also known as epithelial downgrowth (ED), is a potentially blinding complication of intraocular surgery or penetrating eye trauma.1 It is characterized by the proliferation of corneal or conjunctival epithelium in the anterior chamber.1 ED can be cystic or diffuse.2 The cystic type has a more benign course and is defined by its well-circumscribed margins.2 The diffuse type has a sheetlike morphology and tends to follow an aggressive, recurring course.3 In the literature, approximately 82% of affected individuals will present for care within 1 year after a penetrating ocular event.4 These patients typically complain of decreasing visual acuity, ocular injection, and pain.5 Clinical signs such as a visible iris cyst or a retrocorneal membrane may be observed.6 Pupil irregularities, an abnormal iris surface, and corneal edema could also indicate a potential ED.5 Early identification of an ED is important, because the proliferation of epithelial cells in the anterior chamber can lead to secondary glaucoma and phthisis.4
In this article, we report a case of a cystic ED that was diagnosed on an eye enucleated 43 years after penetrating eye trauma. This case highlights the potential of mucinsecreting goblet cells, incorporated in the ED, to complicate the diagnosis. This case also represents one of the longest reported time intervals between eye trauma and enucleation. A 61-year-old female with a blind left eye presented with a pearly-white lesion in the anterior chamber. At age 20 years, she had been involved in a car accident in which shards of glass penetrated her left eye, resulting in blindness. Apart from a history of recurrent intraocular inflammation within recent years, this patient had minimal to no ocular complaints in the left eye after the trauma. The clinician decided that she should be followed without intervention to observe the growth of the lesion. After 8 months, there had been minimal growth of the pearly white lesion. However, after 1 year, noticeable growth of the lesion warranted further investigation. Unfortunately, this patient was lost to follow-up for a subsequent year and, therefore, did not complete the necessary investigations. When she presented again, 2 years after her initial presentation, slit-lamp examination revealed a significantly enlarged lesion and a high
Fig. 1 — Clinical pictures of cystic epithelial downgrowth as it progressed over 2 years. A, Initial presentation. B, Eight months later. C, One year later. D, Two years later. CAN J OPHTHALMOL — VOL. ], NO. ], ] 2014
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Correspondence intraocular pressure (IOP) (36 mm Hg). Ultrasound and fine-needle aspiration were conducted immediately. Cells from the fine-needle aspiration biopsy stained negative for HMB-45, indicating that this lesion was likely not a melanoma. However, the ocular ultrasound revealed a seemingly solid mass. Given the rapid growth rate and ultrasound findings, this lesion was interpreted as a possible malignant tumour of the iris. The left eye was enucleated. Subsequent histopathologic evaluation of the left eye revealed that the lesion was not a malignant tumour, but rather a conjunctival ED cyst filled with a mucinous material. Immunohistochemistry confirmed negative HMB-45 staining. Cellular features of malignancy such as prominent nucleoli, pleomorphism, abnormal mitotic figures, and a high nuclear-to-cytoplasmic ratio were not observed. Staining for cytokeratin 8/18 confirmed the epithelial nature of the lining of the cyst. Furthermore, the presence of goblet cells in the epithelium helped distinguish the origin of the cyst as conjunctival. Signs of inflammation such as multinucleated giant cells, as well as the remnants of the damaged lens, were features that fit with the history of ocular trauma. Finally, obstruction of
the anterior chamber angle by the ED cyst helped explain the cause for the raised IOP (Figs 1 and 2). In this patient, conjunctival epithelium was translocated from the surface of the eye into the anterior chamber after penetrating eye trauma. Secretions from goblet cells, which were interspersed in the epithelium, led to expansion of the cystic space. This gave the impression of malignant growth. Furthermore, because the cyst was filled with mucin, there was a tendency to believe this was a solid mass and not a typical serous ED cyst. This atypical feature of the cyst skewed the interpretation of the ocular ultrasound. Essentially, the clinical diagnosis of a malignant intraocular tumour was inaccurate. However, this patient would likely have required an eventual enucleation because of recurrent inflammation and a high IOP.7 In conclusion, ED should always be considered in the differential diagnosis of an anterior segment lesion. A history of intraocular surgery or trauma should not be disregarded on the basis of how much time has elapsed, because an ED has the propensity to remain dormant for many years. Although small, stable, and asymptomatic ED cysts may be observed, treatment is indicated for rapidly growing cysts to prevent iridocyclitis, glaucoma, and vision loss.8
Fig. 2 — Ocular ultrasound and histopathology images of the cystic epithelial downgrowth (ED). A, Ocular ultrasound. B, Enucleation specimen shows ED cyst lined by conjunctival epithelium and filled with mucinous material. The remnant of the damaged lens is also observed. C, Anterior chamber angle obstruction. D, Goblet cells (a) and squamous conjunctival cells (b) inside the ED cyst.
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CAN J OPHTHALMOL — VOL. ], NO. ], ] 2014
Correspondence Lisa G. Jagan, Gerardo Discepola, Eman Al-Sharif, Vasco Bravo-Filho, Miguel N. Burnier, Jr. Henry C. Whitelson Ocular Pathology Laboratory, McGill University, Montreal, Que. Correspondence to: Lisa G. Jagan, BHSc: [email protected] REFERENCES 1. Nemi A, Bahadur RP, Randleman JB. Traumatic epithelial downgrowth after radial keratotomy. J Cataract Refract Surg. 2008;34:327-9. 2. Vargas LG, Vroman DT, Solomon KD, et al. Epithelial downgrowth after clear cornea phacoemulsification: report of two cases and review of the literature. Ophthalmology. 2002;109:2331-5. 3. Levy J, Lifshitz T. Diffuse benign epithelial downgrowth after cataract surgery. Can J Ophthalmol. 2006;41:67-70.
4. Kim SK, Ibarra MS, Syed NA, et al. Development of epithelial downgrowth several decades after intraocular surgery. Cornea. 2005;24:108-9. 5. Jadav DS, Rylander NR, Vold SD, et al. Endoscopic photocoagulation in the management of epithelial downgrowth. Cornea. 2008;27:601-4. 6. Lenhart PD, Randleman B, Grossniklaus HE, Stulting RD. Confocal microscopic diagnosis of epithelial downgrowth. Cornea. 2008;27:1138-41. 7. Weiner MJ, Trentacoste J, Pon DM, Albert DM. Epithelial downgrowth: a 30-year clinicopathological review. Br J Ophthalmol. 1989;73:6-11. 8. Ganesh A, Al-Fadhil N, Wali U, Bialasiewicz AA. En bloc excision of intraocular epithelial cystic downgrowth using syngeneic auricular cartilage. Eye. 2005;19:97-100. Can J Ophthalmol 2014;]:]]]–]]] 0008-4182/14/$-see front matter & 2014 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2014.04.015
CAN J OPHTHALMOL — VOL. ], NO. ], ] 2014
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In this article, we report a case of a cystic ED that was diagnosed on an eye enucleated 43 years after penetrating eye trauma. This case highlights the potential of mucinsecreting goblet cells, incorporated in the ED, to complicate the diagnosis. This case also represents one of the longest reported time intervals between eye trauma and enucleation. A 61-year-old female with a blind left eye presented with a pearly-white lesion in the anterior chamber. At age 20 years, she had been involved in a car accident in which shards of glass penetrated her left eye, resulting in blindness. Apart from a history of recurrent intraocular inflammation within recent years, this patient had minimal to no ocular complaints in the left eye after the trauma. The clinician decided that she should be followed without intervention to observe the growth of the lesion. After 8 months, there had been minimal growth of the pearly white lesion. However, after 1 year, noticeable growth of the lesion warranted further investigation. Unfortunately, this patient was lost to follow-up for a subsequent year and, therefore, did not complete the necessary investigations. When she presented again, 2 years after her initial presentation, slit-lamp examination revealed a significantly enlarged lesion and a high
Fig. 1 — Clinical pictures of cystic epithelial downgrowth as it progressed over 2 years. A, Initial presentation. B, Eight months later. C, One year later. D, Two years later. CAN J OPHTHALMOL — VOL. ], NO. ], ] 2014
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Correspondence intraocular pressure (IOP) (36 mm Hg). Ultrasound and fine-needle aspiration were conducted immediately. Cells from the fine-needle aspiration biopsy stained negative for HMB-45, indicating that this lesion was likely not a melanoma. However, the ocular ultrasound revealed a seemingly solid mass. Given the rapid growth rate and ultrasound findings, this lesion was interpreted as a possible malignant tumour of the iris. The left eye was enucleated. Subsequent histopathologic evaluation of the left eye revealed that the lesion was not a malignant tumour, but rather a conjunctival ED cyst filled with a mucinous material. Immunohistochemistry confirmed negative HMB-45 staining. Cellular features of malignancy such as prominent nucleoli, pleomorphism, abnormal mitotic figures, and a high nuclear-to-cytoplasmic ratio were not observed. Staining for cytokeratin 8/18 confirmed the epithelial nature of the lining of the cyst. Furthermore, the presence of goblet cells in the epithelium helped distinguish the origin of the cyst as conjunctival. Signs of inflammation such as multinucleated giant cells, as well as the remnants of the damaged lens, were features that fit with the history of ocular trauma. Finally, obstruction of
the anterior chamber angle by the ED cyst helped explain the cause for the raised IOP (Figs 1 and 2). In this patient, conjunctival epithelium was translocated from the surface of the eye into the anterior chamber after penetrating eye trauma. Secretions from goblet cells, which were interspersed in the epithelium, led to expansion of the cystic space. This gave the impression of malignant growth. Furthermore, because the cyst was filled with mucin, there was a tendency to believe this was a solid mass and not a typical serous ED cyst. This atypical feature of the cyst skewed the interpretation of the ocular ultrasound. Essentially, the clinical diagnosis of a malignant intraocular tumour was inaccurate. However, this patient would likely have required an eventual enucleation because of recurrent inflammation and a high IOP.7 In conclusion, ED should always be considered in the differential diagnosis of an anterior segment lesion. A history of intraocular surgery or trauma should not be disregarded on the basis of how much time has elapsed, because an ED has the propensity to remain dormant for many years. Although small, stable, and asymptomatic ED cysts may be observed, treatment is indicated for rapidly growing cysts to prevent iridocyclitis, glaucoma, and vision loss.8
Fig. 2 — Ocular ultrasound and histopathology images of the cystic epithelial downgrowth (ED). A, Ocular ultrasound. B, Enucleation specimen shows ED cyst lined by conjunctival epithelium and filled with mucinous material. The remnant of the damaged lens is also observed. C, Anterior chamber angle obstruction. D, Goblet cells (a) and squamous conjunctival cells (b) inside the ED cyst.
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CAN J OPHTHALMOL — VOL. ], NO. ], ] 2014
Correspondence Lisa G. Jagan, Gerardo Discepola, Eman Al-Sharif, Vasco Bravo-Filho, Miguel N. Burnier, Jr. Henry C. Whitelson Ocular Pathology Laboratory, McGill University, Montreal, Que. Correspondence to: Lisa G. Jagan, BHSc: [email protected] REFERENCES 1. Nemi A, Bahadur RP, Randleman JB. Traumatic epithelial downgrowth after radial keratotomy. J Cataract Refract Surg. 2008;34:327-9. 2. Vargas LG, Vroman DT, Solomon KD, et al. Epithelial downgrowth after clear cornea phacoemulsification: report of two cases and review of the literature. Ophthalmology. 2002;109:2331-5. 3. Levy J, Lifshitz T. Diffuse benign epithelial downgrowth after cataract surgery. Can J Ophthalmol. 2006;41:67-70.
4. Kim SK, Ibarra MS, Syed NA, et al. Development of epithelial downgrowth several decades after intraocular surgery. Cornea. 2005;24:108-9. 5. Jadav DS, Rylander NR, Vold SD, et al. Endoscopic photocoagulation in the management of epithelial downgrowth. Cornea. 2008;27:601-4. 6. Lenhart PD, Randleman B, Grossniklaus HE, Stulting RD. Confocal microscopic diagnosis of epithelial downgrowth. Cornea. 2008;27:1138-41. 7. Weiner MJ, Trentacoste J, Pon DM, Albert DM. Epithelial downgrowth: a 30-year clinicopathological review. Br J Ophthalmol. 1989;73:6-11. 8. Ganesh A, Al-Fadhil N, Wali U, Bialasiewicz AA. En bloc excision of intraocular epithelial cystic downgrowth using syngeneic auricular cartilage. Eye. 2005;19:97-100. Can J Ophthalmol 2014;]:]]]–]]] 0008-4182/14/$-see front matter & 2014 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2014.04.015
CAN J OPHTHALMOL — VOL. ], NO. ], ] 2014
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