Capsulorrhexis in Capsular Delamination

CLINICOPATHOLOGIC REPORTS, CASE REPORTS, AND SMALL CASE SERIES

SECTION EDITOR: W. RICHARD GREEN, MD

Separation of the anterior layer of the lens capsule, also known as true exfoliation and capsular delamination, has become an increasingly rare clinical finding. Although its pathogenesis is not precisely known, the condition has been associated with age, trauma, and exposure to toxins and/or to thermal radiation.1 An association with occupational infrared radiation exposure was ac- cepted after Elschnig2 described the classic clinical findings in 2 glass- blowers and Kubik3 and others4,5 noted the condition in black- smiths, puddlers, chainmakers, and steelworkers. With improved safety Figure 1. Preoperative appearance of a diaphanous membrane (arrows) in the anterior chamber that is standards, the condition is now re- attached to the lens. ported less frequently in association with occupational hazards. Cap- pigmentation of the angle, and no sular delamination remains of phacodonesis. Results of tonom- interest particularly because there etry, fundus, and optic nerve exami- may be mild subclinical forms of the nations were all normal. condition,6 and because modern The patient underwent bilat- cataract surgery is dependent on suc- eral cataract extraction with a 2-month cessful anterior capsule removal. interval between procedures. The phacoemulsification technique was Report of a Case. An 81-year-old man standard except for a larger than usual who under treatment for a cardiac ar- capsulorrhexis and the submission of rhythmia and hypertension reported the capsule specimens for histopatho- blur and glare in both eyes. There was logic study. The diaphanous mem- a family history of cataracts and glau- brane was gently teased to the side and coma. His career involved more than the deeper capsular layer was dis- 20 years in a steel mill. During 4 of sected with a bent 30-gauge needle. these years, he experienced frequent There were no complications in either and intense prolonged exposure to operation. Two years postoperatively, the heat of the blast furnaces. the uncorrected vision was 20/25 OU On ophthalmic examination, his with a mild astigmatism with the rule best-corrected visual acuity was 20/70 notedonrefraction.Theposteriorcap- OU. The patient had hyperopia of 4 sules remained clear and the anteri- Figure 2. Electron microscopy ultrastructural diopters. Bilateral cataracts were pres- or capsular edges appeared normal. appearance of a 3-µm-thick layer (asterisk) that is split from the anterior lens capsule. Note ent, having combined cortical and Findings from the histopatho- surface-parallel vacuolization of the anterior nuclear elements. Within the cen- logic examination of the specimens capsule extending beyond the split (original tral anterior chamber in both eyes was revealed delamination of the lens magnification ϫ12000). a folded cellophane-like membrane capsule that was best illustrated by fixed to the anterior lens capsule’s transmission electron microscopy Comment. Multiple reports exist of surface, unassociated with any evi- (Figure 2). The capsule was mod- capsulardelaminationspecimensfrom dence of inflammation (Figure 1). erately electron dense with a lami- successful intracapsular and extracap- The degree of delamination was ap- nated granular appearance. The sular cataract surgery.4,7,8 In this case, proximately symmetrical. There were splitting of the capsule was docu- the curvilinear capsulorrhexis tech- no other abnormal deposits on the mented with the anterior layer thin- nique was successful. With the excep- lens capsule or the iris, no unusual ner than the posterior layer. tion of the manipulation required to

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©2002 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/28/2021 take the surgical specimen, the cases has also been suggested that capsu- most commonly in neonates, and were routine and without complica- lar protein abnormalities may play a they are frequently located in the sa- tion. To our knowledge, no series to role.10 In this case, however, a volun- crococcygeal and presacral areas.1 Al- date has reported a rate of complica- teered history of prolonged expo- though intracranial teratomas are tion in cataract extraction with cap- sure to the heat of a blast furnace pro- rare, they are the most common brain sular delamination, but the true inci- vided the most likely etiologic factor tumors of the neonatal period,2 and denceofcomplicationsassociatedwith related to both the cataract and de- most frequently involve the pineal re- this finding will be difficult to estab- lamination of the anterior capsule. gion, suprasellar region, hypothal- lish because of its rarity. Clinically, diaphanous transparent mus, cerebellar vermis, and ven- While capsular delamination is membranes were similar to those re- tricles.1-3 Teratomas of the optic nerve rare, mild and subclinical forms of the ported as glassblower’s cataracts. For- are exceedingly rare, with few re- condition may be more prevalent than tunately, occupational safety stan- ports in the English literature.4 currently recognized. In a series of 10 dards and protective engineering have We report a neonate with an cases, Wollensak and Wollensak6 re- made true exfoliation from infrared optic nerve teratoma, an odonto- ported the appearance of a double exposure rare. genic orbital dermoid cyst, and per- contour visible at the capsulorrhexis James S. Kelley, MD sistent fetal vasculature (PFV). We edge. Pathologic analysis of the cap- Tony Tsai, MD are not aware of any previous case sulorrhexis specimens by light and Mary B. Kansora, MD in which these entities have coex- electron microscopy revealed the W. Richard Green, MD isted, and, to our knowledge, this is double contour to result from a char- Baltimore, Md the first report in which any 2 of the acteristic step formation at the cap- 3 have been documented as occur- sulorrhexis edge. In 7 of 10 of these The authors have no financial inter- ring in the same patient. cases, these authors also noted surface- est in this article. parallel splits in the outer third of the Corresponding author: James S. Report of a Case. A healthy 2-day- capsule. They postulated that the Kelley, MD, 6565 N Charles St, Suite old white boy was referred for man- double contour and microscopically 302, Baltimore, MD 21204 (e-mail: agement of PFV of the right eye. The evident surface-parallel splits may rep- [email protected]). patient was carried to term and had resent a subclinical form of true ex- no family history of any unusual eye 1. Duke-Elder S. System of Ophthalmology: Dis- foliation that results from zonular trac- eases of the Lens and Vitreous; Glaucoma and Hy- disease. On examination under an- tion on the superficial capsule over less potony. St Louis, Mo: CV Mosby; 1969. esthesia, the patient appeared enoph- elastic deeper layers in older pa- 2. Elschnig A. Abhlo¨sung der Zonulalamelle bei thalmic on the right side, and had right Glasblasern. Klin Monatsbl Augenheilkd. 1922; tients. These findings suggest that true 69:732-734. microcornea (corneal diameters: 6.0 exfoliation may represent one ex- 3. Kubik J. Ablo¨sung der zonulalamelle bei Glasbla- mm OD, 10.0 mm OS). Intraocular treme of a continuum representing sern. Klin Monatsbl Augenheilkd. 1923;70. pressures were 5 mm Hg OD and 10 4. Burde RM, Bresnick G, Uhrhammer J. True different degrees of capsular delami- exfoliation of the lens capsule: an electron mm Hg OS. Biomicroscopy of the right nation. Although Wollensak and Wol- microscopic study. Arch Ophthalmol. 1969;82: eye (Figure 1) revealed patches of 651-653. lensak reported anecdotally that the 5. Holloway TB, Cowan A. Concerning lamellar band keratopathy at the 4-o’clock and incidence of radial capsular tears ap- membranes of the anterior surface of the lens. 8-o’clock positions, with peripheral peared lower when a double contour Am J Ophthalmol. 1931;14:189-195. corneal neovascularization extend- 6. Wollensak G, Wollensak J. Double contour of was seen, no evidence currently ex- the lens capsule edges after continuous curvi- ing 360°. Dense fibrotic membranes ists regarding the relative strength or linear capsulorhexis. Graefes Arch Clin Exp Oph- extended from the pupillary margin weakness of the capsulorrhexis with thalmol. 1997;235:204-207. and anterior iris stroma into the an- 7. Fukuo Y, Takeda N, Hirata H et al Histologi- the double contour. Likely, the find- cal findings of capsular delamination of the lens. terior chamber angle. A yellow mush- ing goes unnoticed in most cases. No Jpn J Ophthalmol. 1994;38:87-91. room-shaped mass protruded through 8. Kuchle M, Iliff WJ, Green WR. Kombinierte double contour was observed after Feuerlamelle und Pseudoexfoliation der vor- the pupil. B-scan ultrasonography re- curvilinear capsulorrhexis in our case. deren Linsenkapsel. Klin Monatsbl Augen- vealed a funnel-shaped stalk that ex- Perhaps this is because the delami- heilkd. 1996;208:127-129. tended from an area surrounding the 9. Cashwell LF Jr, Holleman IL, Weaver RG, van nation did not extend to the capsu- Rens GH. Idiopathic true exfoliation of the lens optic nerve to the retrolenticular re- lorrhexis edge, although evidence for capsule. Ophthalmology. 1989;96:348-351. gion, as is consistent with PFV. Axial shearing of the capsular layers be- 10. Anderson IL, van Bockxmeer FM. True exfolia- lengths were 15.8 mm OD and 22.9 tion of the lens capsule: a clinicopathological re- yond the edge of true exfoliation is port. Aust N Z J Ophthalmol. 1985;13:343-347. mm OS. Examination results from the suggested by the vacuolization of the left eye were normal. capsule seen ultrastructurally be- At age 1 month, the patient un- yond the split (Figure 2). Optic Nerve Teratoma and derwent attempted anterior segment Although associations with Odontogenic Dermoid Cyst reconstruction, but the dense fi- trauma, toxins, inflammation, and in a Neonate With brotic membranes precluded lensec- heat are well recognized, the under- Persistent Fetal Vasculature tomy. At age 3 months, the patient de- lying etiology of true exfoliation of the veloped right lower-lid ectropion, and lens capsule remains uncertain. Small Teratomas are embryonal tumors that a cystic mass was palpated in the an- case series of patients without a his- are derived from all 3 germinal lay- terior inferior orbit. Computed to- tory of trauma or heat exposure sug- ers (ectoderm, mesoderm, and mography demonstrated a hypoplas- gest aging may be a major factor.9 It endoderm). These neoplasms occur tic right orbit containing an extraconal

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©2002 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/28/2021 infraorbital cyst (10ϫ23ϫ13 mm) cm). The resected cystic lesion mea- ductular structures with focal squa- with calcifications. Magnetic reso- sured 2.3ϫ1.5ϫ1.0 cm, with a frag- mous metaplasia, glands with gas- nance imaging (Figure 2) depicted ment of smooth white tissue tric specialized type cells, fibrous tis- a second discrete mass (10ϫ10ϫ13 (0.5ϫ0.4ϫ0.3 cm) attached to it. sue, and focal chronic inflammatory mm) that traversed the right optic ca- Histology revealed a dermoid cyst infiltrate (Figure 5). nal into the suprasellar cistern. This (Figure 4) and separate soft tissue, The patient underwent diag- second lesion compressed the right including dental papillae and odon- nostic workup for other germ cell tu- optic nerve at the orbital apex and ex- togenic rests. The 2 well-developed mors of the mediastinum, abdo- tended along the length of the optic teeth were not submitted for histo- men, sacrum, and testes, with no nerve to the anterior optic chiasm. An logic examination. further evidence of teratomas. orbitotomy was performed on the in- At age 6 months, the patient fant at age 3 months, with removal of underwent a right pterional crani- Comment. Teratomas and orbital the cystic lesion from the right in- otomy for excision of the orbital apex dermoid cysts typically contain tis- feromedial orbit. During the proce- lesion. The mass was fibrous and ad- sues that are foreign to their ana- dure, the lesion was isolated above the herent to the ophthalmic segment of tomic location.5 Many authorities periosteum along the inferior orbital the internal carotid artery as well as believe that teratomas arise from wall and was noted to be multilocu- the right optic nerve. The intracra- pleuripotential primordial germ cells lated, with 3 distinct cystic bulges nial portion of the nerve was tran- that undergo erroneous migration, (Figure 3). While dissecting the le- sected, and part of the lesion re- later acquiring oncogenic proper- sion at its base, we found a uni- mained within the orbit. Microscopic ties.6 Similarly, dermoid cysts are cusped tooth (0.5ϫ 0.5 cm) be- analysis of the excised lesion re- derived from epithelial cells that, tween the cyst and orbital wall, as well vealed a mature teratoma com- during embryogenesis, become im- as a second toothlike structure that re- posed of cartilage, nerve fascicles, properly sequestered within the sembled a hollow crown (0.7ϫ0.6 smooth muscle, pancreatic islets, and spaces between the sutures of orbital bones.7 While not anatomi- cally joined, the association and close proximity of the teratoma and the dermoid cyst in our patient suggests a common origin or process in the early intrauterine period. Imaging studies revealed no evidence of a connection between the optic nerve teratoma and the odontogenic dermoid cyst, indicating that the 2 lesions were distinct entities. We have classified the orbital lesion as an odontogenic dermoid cyst rather than a teratoma because this terminology more accurately describes the pathologic findings and does not mandate that the lesion arose from a neoplas- tic process. The presence of well- Figure 1. External and anterior segment of the right eye. The patient was enophthalmic on the right side, developed teeth in intimate associa- with lower eyelid ectropion. A cystic mass is present behind the right lower eyelid. tion with the dermoid cyst suggests

A B

Figure 2. Magnetic resonance imaging (MRI) scans of optic nerve teratoma and an odontogenic orbital dermoid cyst. A, T2-weighted MRI scan depicting optic nerve teratoma (arrows) involving the right optic nerve and anterior optic chiasm. B, An extraconal infraorbital cyst with calcifications (arrows) surrounds the microphthalmic right eye (arrowheads).

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Figure 3. Gross photograph of the resected multiloculated cystic lesion (orbital dermoid cyst) and 2 teeth.

Figure 5. Some of the diverse elements within the mature teratoma included islands of cartilage (A) (arrows), pancreatic islets (B) (arrow), and gastric-type glands with adjacent B smooth muscle (C) (arrow) (hematoxylin-eosin, original magnification ϫ200).

The congenital defects described in our patient represent unique developmental anomalies involving the eye and brain. While systemic findings associated with PFV are uncommon, the present case underscores the need to be vigilant for signs of concurrent orbital and intracranial abnormalities. Medhat F. Guirgis, MD Figure 4. Portion of dermoid cyst wall containing epidermis with hair follicle (A) (arrow) and sebaceous Frances V. White, MD (B) (arrow) structures (hematoxylin-eosin, original magnification ϫ100). Jennifer A. Dunbar, MD that both components of the orbital rant cellular migration. However, a Morton E. Smith, MD lesion were derived from tissue dis- retrolenticular plaque containing el- Gregg T. Lueder, MD placed from the skin and oral cavity ements such as adipose, smooth St Louis, Mo during embryogenesis.8 The pres- muscle, and cartilage may be found Presented at the Association for Re- ence of dental structures within the in some cases, which represent ecto- search in Vision and Ophthalmology orbital teratomas and orbital dermoids pic mesenchymal tissue.10 No intra- Meeting, May 11, 1997, Ft Lauder- has previously been reported,9 but re- ocular specimens were extracted from dale, Fla. mains exceedingly rare. our patient; therefore, the presence of Corresponding author: Gregg T. Persistent fetal vasculature re- such mesenchymal tissues could not Lueder, MD, St Louis Children’s Hos- sults from improper development of be determined. The findings of mi- pital, Division of Pediatric Ophthal- secondary vitreous and an incom- crocornea and dense fibrotic mem- mology, Suite 2 South 89, One Chil- plete regression of the primary vitre- branes extending from the pupillary dren’s Place, St Louis, MO 63110 (e- ous during the ninth week of gesta- margin into the anterior chamber mail: [email protected]). tion.10 This malformation primarily angle may also indicate a compo- 1. Azizkhan RG, Caty MG. Teratomas in child- occurs from improper intrauterine nent of anterior segment dysgenesis hood. Curr Opin Pediatr. 1996;8:287-292. ocular development rather than er- in this severely malformed eye. 2. Hunt SJ, Johnson PC, Coons SW, Pittman HW.

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©2002 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/28/2021 Neonatal intracranial tumors. Surg Neurol. 1990; junctival graft was sloughing off and 34:336-342. 3. Tekeuchi J, Mori K, Moritake K, Tani F, Waga then disappeared, exposing bare S, Handa H. Teratomas in the suprasellar re- sclera. One week later, a dark area gion: report of five cases. Surg Neurol. 1975;3: consistent with scleral melting was 247-255. 4. Samii M, Ramina R, Koch G, Reusche E. Ma- noted at the 9-o’clock position. Di- lignant teratoma of the optic nerve: case re- clofenac was discontinued and a lu- port. Neurosurgery. 1985;16:696-700. 5. Sreenan C, Johnson R, Russell L, Bhargava R, bricating gel was prescribed to be ap- Osiovich H. Congenital orbital teratoma. Am plied every 2 hours. The patient was J Perinatol. 1999;16:251-255. then referred to the cornea service at 6. Sano K. Pathogenesis of intracranial germ cell tumors reconsidered. J Neurosurg. 1999;90: Wills Eye Hospital (Philadelphia, Pa). 258-264. On initial examination, the pa- 7. Jakobiec FA, Font RL. Orbit. In: Spencer WH, ed. Ophthalmic Pathology: An Atlas and Text- tient reported photophobia, pain, and Slitlamp photograph of the left eye. Bare book. Philadelphia, Pa: WB Saunders; 1986: tearing in his left eye. His best- necrotic sclera is observed between the 5- and 2488-2496. corrected visual acuity was 20/25 OD 9-o’clock positions. The sclera was mildly 8. El-Sayed Y. Sinonasal teeth. J Otolaryngol. 1995; thinned and stained with fluorescein. 24:180-183. and 20/50 OS. Slitlamp examination 9. Carter JB. Transconjunctival resection of an or- of the right eye revealed a nasal pte- bital dermoid tumor. J Maxillofac Surg. 1985; rygium measuring 1.5 mm onto the pterygia. Different routes of admin- 13:239-242. 10. Traboulsi EI. Colobomatous microphthalmia, cornea and 5 mm vertically at the lim- istration, including instillation of anophthalmia, and associated malformation bus. The left eye had a large area of drops after surgery and intraopera- syndromes. In: Traboulsi EI, ed. Genetic Dis- eases of the Eye. New York, NY: Oxford Uni- porcelain white sclera extending 7 to tive application of a sponge soaked versity Press; 1998:64-66. 8 mm posterior from the limbus be- with MMC on the scleral bed of ex- tween the 5- and 9-o’clock positions. cision, have been used.1 A single The whitened sclera was thinned and 0.1-mL injection of 0.1 mg/mL of stained with fluorescein but there was MMC into the pterygium head in 6 Scleral Ulceration After no uveal show (Figure). The con- patients, followed by pterygium ex- Preoperative Injection junctiva was injected superiorly, and cision 4 weeks later, was described of Mitomycin C in a papillary reaction was noted in the by Donnenfeld et al.4 This is the only the Pterygium Head inferior tarsus. The anterior cham- report, to our knowledge, to sup- ber was deep and quiet and the lens port the use of subconjunctival A pterygium is a common degenera- was clear. Examination of the lower MMC before surgery to prevent re- tive corneal disorder. Adjunctive treat- puncta revealed that the right side was currence of pterygia. In this case, a ments, including radiation, antime- closed and the left was open. The in- higher volume of the same concen- tabolites, and conjunctival and limbal traocular pressure was 14 mm Hg OU. tration was injected, with serious ad- grafts, are used to decrease the rate of The patient was treated conserva- verse effects. recurrence after simple excision.1-2 tively, with bacitracin-polymyxin B The use of topical MMC eye Mitomycin C (MMC) is an alkylat- ointment every 2 hours, a lubricat- drops after pterygium excision has ing agent that is commonly used in ing ointment twice per day, and 25 been associated with severe discom- glaucoma filtration surgery to treat mg/d of oral rofecoxib. fort and vision-threatening compli- conjunctival neoplasia and also to pre- At 8 weeks, the examination re- cations,includingglaucoma,cataract, vent recurrence of pterygia.3 sults were consistent with advanc- corneal edema, corneal perforation, ing conjunctival epithelium from the and scleral calcification.5,6 Rubinfeld Report of a Case. A 55-year-old man limbal area. The epithelial defect was et al5 recommended avoiding MMC was referred for a nonhealing con- also noted to be smaller. The patient in patients who had other conditions junctival epithelial defect and scleral was still symptomatic and showed a associated with poor wound healing, necrosis. The patient had dry eyes and slow healing process. An amniotic such as keratoconjunctivitis sicca and pterygia in both eyes. He had punc- membrane graft was offered, but the Sjo¨gren syndrome. tal cautery of both lower puncta. He patient refused further surgery. He In our patient, multiple fac- received a 0.15-mL subconjunctival was followed up closely every few tors may have predisposed to poor injection of 0.1 mg/mL of MMC in the weeks. Eight months after the pte- wound healing. We suspect that the pterygium head in his left eye. One rygium surgery, he still complained MMC accumulated inferiorly, caus- month after the MMC injection, he of light sensitivity. Ocular examina- ing damage to the inferior sclera and underwent pterygium excision com- tion revealed a best-corrected visual inferior conjunctival tissue used for bined with a free conjunctival auto- acuity of 20/30; the sclera was still the conjunctival graft. Dry eye syn- graft taken from the inferior bulbar mildly thinned. The large conjuncti- drome and long-term postopera- conjunctiva. Postoperatively the pa- val epithelial defect had improved, al- tive treatment with topical nonste- tient was prescribed topical 0.3% though 2 epithelial defects measur- roidal anti-inflammatory drugs and ciprofloxacin for 3 days, followed by ing 4.5ϫ2.5 mm and 1.0ϫ0.5 mm steroids are additional factors that a combination of 0.3% tobramycin remained. There was no evidence of could have interfered with the heal- and 0.1% dexamethasone drops 4 recurrent pterygium. ing process. Although MMC has times per day and 0.1% diclofenac been demonstrated to decrease the twice per day. Eleven weeks after sur- Comment. Mitomycin C has been rate of recurrences after pterygium gery, the patient noticed that the con- used to treat primary and recurrent excision, a conventional route of ad-

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©2002 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/28/2021 ministration, careful dosing, and pa- within 3 months of birth. The ex- mal tear drainage due to lacrimal tient selection are recommended. cess tears were noted to run down gland secretion through a lacrimal the lateral aspect of the right eye. Ab- ductule misdirected to the external Marı´a A. Carrasco, MD normal tearing of the left eye had not eyelid. Excessive tearing was most Christopher J. Rapuano, MD been observed. Epiphora was fre- severe in association with ocular ir- Elisabeth J. Cohen, MD quent, and when most severe, was ritants, when reflex tearing would be Peter R. Laibson, MD associated with environmental irri- maximal. There was no evidence of Philadelphia, Pa tants, such as dust or sand. There additional lacrimal system abnor- was no additional complaint of ocu- malities. The patient had a healthy lar irritation or other symptom. cornea, normal tear lake, and nor- This research was supported in part On examination, several hairs, mal measured tear production. by XV Congreso Argentino de Oftal- identical in appearance to adjacent This most likely represents a de- mologı´a, Mendoza, Argentina. eyelashes, were present 3 mm above velopmental abnormality. Other pos- The authors have no proprietary the lateral edge of the lash line of the sible lesions that might contain glan- interest in any of the products or tech- right upper eyelid. With applied cor- dular tissue, such as a teratoma or niques discussed in this article. neal irritation, tears were noted to dermoid tumor, are unlikely in the Corresponding author and re- originate from the same cutaneous absence of a palpable mass and do prints: Christopher J. Rapuano, MD, ostium as the ectopic lashes and to not produce tears. Although not as- Cornea Service, Wills Eye Hospital, run down the patient’s cheek sociated with epiphora, cases have 900 Walnut St, Philadelphia, PA (Figure). No baseline secretion from been reported of ectopic lacrimal 19107 (e-mail: [email protected]). the aberrant opening was observed glandular tissue and duct cysts.1-4 1. Hoffman RS, Power WJ. Current options in pte- during examination. Ophthalmic ex- Moreover, misdirected lacrimal rygium management. Int Ophthalmol Clin. 1999; amination results were otherwise gland secretion through a congeni- 39:15-26. 2. Figueiredo RS, Cohen EJ, Gomes JA, Rapuano CJ, within normal limits, with a best- tal aberrant lacrimal gland ductule Laibson PR. Conjunctival autograft for pte- corrected visual acuity of 20/20 OU, is supported by the presence of the rygium surgery: how well does it prevent recur- normal corneal examination re- observed neural link with lacrimal rence? Ophthalmic Surg Lasers. 1997;28:99-104. 3. Hardten DR, Samuelson TW. Ocular toxicity of sults, normal and equal tear lakes, no gland secretion, evidenced by an as- mitomycin-C. Int Ophthalmol Clin. 1999;39:79- palpable palpebral or orbital lacri- sociation of secretion with ocular ir- 90. 4. Donnenfeld ED, Perry HD, Kornstein H, D’Aversa mal gland mass, no exophthalmos, ritation and the lack of secretion in G, Fromer S. Subconjunctival mitomycin C in the and normal ocular motility. Ocular its absence. In short, secretion from management of recurrent pterygia. Paper pre- tear production, estimated with fil- an ectopic lacrimal gland ductule sented at: the Castroviejo Cornea Society Annual Meeting; November 7, 1998; New Orleans, La. ter paper saturation, was normal and should be considered when evalu- 5. Rubinfeld RS, Pfister RR, Stein RM, et al. Seri- equal in both eyes. The lacrimal ating patients with congenital ous complications of topical mitomycin-C after drainage system was patent; dye dis- epiphora. pterygium surgery. Ophthalmology. 1992;99: 1647-1654. appearance testing and irrigation 6. DunnJP,SeamoneCD,OstlerHB,NickelBL,Beallo were normal and equal in both eyes. Timothy J. McCulley, MD A. Development of scleral ulceration and calcification after pterygium excision and mitomycin On general physical examination, the Irvine, Calif therapy. Am J Ophthalmol. 1991;112:343-344. patient was healthy, with no addi- Chee-Chew Yip, MD tional congenital abnormalities. The Robert C. Kersten, MD patient declined surgical excision for Dwight R. Kulwin, MD control of symptoms. Cincinnati, Ohio An Ectopic Site of Lacrimal Gland Secretion Comment. To our knowledge, this The authors do not have any commer- Mimicking Epiphora is the first reported case of abnor- cial or proprietary interest in any ma-

Epiphora, starting at or shortly after birth, is most commonly associated with obstruction of the lacrimal drainage system. It may also be due to reflex hypersecretion secondary to ocular irritation, such as that caused by glaucoma, trichiasis, and infectious or chemical conjunctivi- tis. We present an additional cause of congenital tearing, an ectopic lacrimal gland ductule opening on the external upper eyelid at the lateral canthus, mimicking epiphora.

Report of a Case. A healthy 8-year- old white male was evaluated for Ectopic eyelashes and lacrimal gland secretion site (arrow) with tear directed lateral to the right eye tearing of the right eye, which started (arrowheads).

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©2002 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/28/2021 terials or techniques mentioned in this Her family history was notable for thickness and no foveal depression article. poor vision, nystagmus, and early was detectable (Figure 2D and E). Corresponding author and re- cataracts in her paternal grand- Instead, there was continuity of mul- prints: Timothy J. McCulley, MD, De- mother, father, and sister. tiple retinal layers through the area partment of Ophthalmology, Univer- The patient exhibited no signs where the foveal center was ex- sityofCalifornia–Irvine,118MedSurge of cutaneous albinism. Her best- pected to be located. I, Irvine, CA 92697-4375 (e-mail: corrected visual acuity was 20/80 OU [email protected]). and her pupillary responses were Comment. Foveal hypoplasia is an normal. Mild horizontal nystag- ocular abnormality that may be seen 1. Boccato P, Blandamura S, Midena E, Carollo C. Orbital ectopic lacrimal gland tissue simulating mus and a 15 prism diopter exotro- in isolation or in association with a neoplasm: report of a case with fine needle as- pia were present. A 1-mm corneal other ocular or systemic signs. piration biopsy diagnosis. Acta Cytol. 1992;36: pannus encompassed the superior Typical associated findings include 737-743. 2. Kao SC, Yeh LK, Tsai CC, Hsu WM. Ectopic lac- 270° of each eye. The fundi were nystagmus, aniridia or iris transil- rimal gland cyst of the orbit. Chung Hua I Hsueh lightly pigmented. The optic nerves lumination, cataract, and skin hy- Tsa Chih (Taipei). 2000;63:334-338. 3. Rush A, Leone CR Jr. Ectopic lacrimal gland cyst were normal. In each eye, the reti- popigmentation. Visual acuity is in 1 of the orbit. Am J Ophthalmol. 1981;92:198- nal vessels were slightly dragged the range of 20/50 to 20/200. An au- 201. temporally, and foveal reflexes were tosomal dominant syndrome of con- 4. Von Domarus H. A lacrimal gland cyst in the orbit. J Craniomaxillofac Surg. 1987;15:106- absent (Figure 1). genital nystagmus, foveal hypopla- 109. Fluorescein angiography re- sia, corneal pannus, and presenile vealed an irregular capillary-free cataracts has been reported,2 and our zone in each eye. In the right eye, patient’s phenotype and family his- several vessels traversed the periph- tory are most compatible with this Optical Coherence eral aspects of the capillary-free zone. syndrome. In cases with only mod- Tomography in the No choroidal neovascularization was erate reduction in vision or with- Diagnosis of Foveal seen in either eye. Electroretino- out associated signs, the diagnosis Hypoplasia graphic amplitudes were within nor- of foveal hypoplasia may not be mal limits. straightforward. The differential di- It is frequently difficult to diagnose Following pupillary dilation, a agnosis includes high myopia, early ocular albinism or foveal hypopla- commercial OCT unit (Humphrey retinal degeneration, and retinopa- sia in patients who have darkly pig- Instruments, Zeiss-Humphrey, San thy of prematurity. mented hair, skin, or irides. Fun- Leandro, Calif) was used to obtain Optical coherence tomography dus findings may be subtle and most 5.92-mm radial sections through the allows detailed examination of macu- commonly include the absence of fo- entire macular area of each eye lar anatomy. It can thus provide in- veal pigmentation or the circumfo- (Figure 2A and B). Standard OCT sight into the foveal architecture in pa- veal light reflex.1 Traditionally in software was used to generate reti- tients with foveal abnormalities or such cases, the diagnosis has been nal topographic measurements. In all visual impairment as well as confir- based on the detection of nystag- sections, the retina was of normal mation of clinical diagnosis, per- mus coupled with fluorescein angiographic demonstration of a poorly formed capillary-free zone. Since these findings may be difficult to de- tect, especially in the setting of nystagmus, foveal hypoplasia may be more common than is generally be- lieved. Optical coherence tomography (OCT) is a noninvasive imaging modality that produces high- resolution, 2-dimensional images of ocular structures. We report the use of OCT to establish a diagnosis of foveal hypoplasia in a patient with decreased vision, in whom the diagnosis had been suspected clinically.

Report of a Case. A 45-year-old white woman was referred for evalu- ation of bilaterally decreased vision. She related a history of nystagmus, high myopia, and poor vision since childhood. She was not born prema- Figure 1. Representative color photographs of the left fundus reveals an ill-defined capillary-free zone. turely and was systemically healthy. The foveal light reflex is absent.

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∗ NFL ∗ NFL IPL IPL OPL OPL Photoreceptors Photoreceptors RPE RPE

Choroid Choroid

C D D + 1.00 2.22 3.45 mm Maximum Display Diameter 3.45 mm NFL 257

∗ IPL 273 OPL Photoreceptors 257 263 268 271 266 RPE 273 Choroid 269

150 200 250 300 350 400 450 500 µm

E D + 1.00 2.22 3.45 mm F D + 1.00 2.22 3.45 mm Maximum Display Diameter 3.45 mm 259 289 Maximum Display Diameter 3.45 mm

257 300

276 267 248 237 226 294 285 208 268 285

259 287

236 285

150 200 250 300 350 400 450 500 µm 150 200 250 300 350 400 450 500 µm

Figure 2. NFL indicates nerve fiber layer; IPL, inner plexiform layer; OPL, outer plexiform layer; and RPE, retinal pigment epithelium. A and B, Representative optical coherence tomographic (OCT) section through the center of the macular area of the right (A) and left (B) eyes of our patient. The asterisk denotes the expected location of the fovea. The posterior hyaloid is attached, and multiple inner retinal layers are preserved. C, OCT of a normal adult macula provided for comparison demonstrates a normal foveal depression. D and E, Retinal topography of the right (D) and left (E) eyes of our patient shows a uniform thickness of approximately 260 µm, which is compatible with normal retinal thickness found in a perifoveal location. F, Retinal topography of a normal adult macula is provided for comparison.

haps even obviating the need for elec- nosis of patients with unexplained troretinography or angiography. visual loss. Apparent Disappearance The OCT findings in this case of Choroidal offer insight into the microana- Franco M. Recchia, MD Neovascularization After tomy of foveal hypoplasia. Nor- Cynthia A. Carvalho-Recchia, MD Initial Photodynamic mally, the foveal declivity is evi- Michael T. Trese, MD denced angiographically by a Royal Oak, Mich Therapy With Verteporfin termination of capillaries. Histologi- cally, this area consists of cone pho- Support for this research was pro- Photodynamic therapy with verte- toreceptors and lacks several of the vided by the Heed Ophthalmic Foun- porfin (Visudyne; Novartis AG, Swit- inner retinal layers as well as retinal zerland), also termed verteporfin 3 dation, Cleveland, Ohio (Dr Recchia). vasculature. None of the authors has any pro- therapy, can reduce the risk of In histologic specimens of fo- prietary or financial interest in any moderate and severe vision loss in se- veal hypoplasia, by contrast, the lected patients with choroidal material or instrumentation pre- 1-4 structure of the central macula re- sented herein. neovascularization (CNV). The sembles that of the peripheral Corresponding author and re- Japanese Age-Related Macular De- macula, with persistence of gan- generation Trial (JAT), a photody- 4 prints: Michael T. Trese, MD, Asso- glion cells and nuclear layers. These ciated Retinal Consultants, PC, 3535 namic therapy trial started in May histologic findings are recapitu- W 13 Mile Rd, Suite 632, Royal Oak, 2000, was designed to evaluate the lated in our patient’s OCT data, MI 48073 (e-mail: [email protected]). safety and fluorescein angiographic ef- which show preservation of mul- fects of verteporfin therapy. This re- tiple inner retinal layers when there 1. Oliver MD, Dotan SA, Chemke J, Abraham FA. port describes an unusual case from should be none. We are aware of no Isolated foveal hypoplasia. Br J Ophthalmol. 1987; this trial in which subfoveal CNV was 71:926-930. other conditions that may produce 2. O’Donnell FE Jr, Pappas HR. Autosomal domi- no longer apparent on fluorescein an- similar OCT findings. Perhaps a nant foveal hypoplasia and presenile cataracts: giography after an initial application a new syndrome. Arch Ophthalmol. 1982;100: more accurate term for this condi- 279-281. of photodynamic therapy with verte- tion, then, is foveal dysgenesis.In 3. Fine BS, Yanoff M. Ocular Histology: A Text and porfin. There was no obvious fluo- summary, OCT provided a defini- Atlas. Hagerstown, Md: Harper & Rowe; 1979: rescence from fibrosis or atrophy of 111-118. tive diagnosis of foveal hypoplasia 4. Duke-Elder S. System of Ophthalmology. St Louis, the retinal pigment epithelium within and may prove helpful in the diag- Mo: Mosby; 1963:3:652-653. the area initially occupied by CNV.

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C D

E F

A, A color fundus photograph shows subretinal hemorrhage surrounding a subfoveal lesion. B, A late-phase frame fluorescein angiogram shows leakage from choroidal neovascularization (CNV) under the center macula with additional slight hyperfluorescence without leakage just superior to the lesion. C, A color fundus photograph taken 3 months after photodynamic therapy with verteporfin shows resolution of the hemorrhage and no fibrosis or significant retinal pigment epithelial abnormalities within the region previously occupied by CNV. D, A late-phase frame fluorescein angiogram shows no leakage or staining within the area previously occupied by CNV. Fluorescent staining, without leakage, is unchanged just superior to the central macula. E, A color fundus photograph taken 6 months after photodynamic therapy with verteporfin shows no fibrosis and only slightly increased pigmentation within the region previously occupied by CNV. F, A late-phase frame fluorescein angiogram shows no leakage or staining within the area previously occupied by CNV.

Report of a Case. A 79-year-old Japa- equivalent) was 20/126 OD and 20/ nantly classic lesion1) with a great- nese woman, who had visual distur- 200 OS. Ophthalmoscopic examina- est linear dimension of 3400 µm (Fig- bance in her left eye for 2 years be- tion showed a subfoveal lesion with ure, B). After reviewing the risks and fore seeking treatment, subsequently subretinal hemorrhage in the right benefits of participating in the JAT, developed decreased vision in her eye (Figure, A) and atrophy in the the patient signed a consent form pre- right eye. She was referred to Osaka macular area of the left eye. Fluores- viously approved by the local insti- University Hospital, Osaka, Japan, in cein angiography showed leakage tutional review board and enrolled in August 2000. Her best-corrected vi- from CNV in a pattern composed of the JAT in October 2000. Vertepor- sual acuity (approximate Snellen at least 50% classic CNV (a predomi- fin therapy was performed without

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©2002 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/28/2021 any complications, following the pro- ing of fibrovascular tissue in the re- 4. Verteporfin in Photodynamic Therapy Study Group. Verteporfin therapy of subfoveal cho- tocol used in the Treatment of Age- maining cases (N.M.B., unpub- roidal neovascularization in age-related macu- Related Macular Degeneration With lished observations, 2001). The lar degeneration: two-year results of a random- Photodynamic Therapy (TAP) inves- absence of fluorescence following ized clinical trial including lesions with occult with no classic choroidal neovascularization: VIP tigation. verteporfin therapy may be due to report 2. Am J Ophthalmol. 2001;131:541-560. One week after treatment, the isofluorescence within the area of 5. Miller JW, Schmidt-Erfurth U, Sickenberg M, et al. Photodynamic therapy with verteporfin for patient’s best-corrected visual acu- treatment, wherein the pigmenta- choroidal neovascularization caused by age- ity (approximate Snellen equiva- tion in the macular area was suffi- related macular degeneration: results of a single lent) was 20/160 OD. Ophthalmo- cient to obscure any fluorescence treatment in a phase 1 and 2 study. Arch Oph- thalmol. 1999;117:1161-1173. scopic examination of her right eye that might otherwise cause stain- 6. Schmidt-Erfurth U, Miller JW, Sickenberg M, et showed no change to the subreti- ing of the choroidal neovascular le- al. Photodynamic therapy with verteporfin for sion. As this is the first verteporfin choroidal neovascularization caused by age- nal hemorrhage. Fluorescein angi- related macular degeneration: results of retreat- ography showed no leakage from therapy trial exclusively in a Japa- ments in a phase 1 and 2 study. Arch Ophthal- CNV and no fluorescein staining in nese population, the fluorescein mol. 1999;117:1177-1187. the macular area previously occu- findings may be related specifically pied by CNV. to this population, although such At 3 and 6 months after treat- findings have not been reported in Chronic Subdural ment, best-corrected visual acuity any of the few Asian participants in (approximate Snellen equivalent) a previous verteporfin therapy trial.3 Hematoma: An Unusual improved to 20/100 OD and 20/80 Longer-term follow-up continues. Sequela of Laser OD, respectively. No CNV was de- Photocoagulation of the tected in her right eye on ophthal- Miki Sawa, MD Retina at the Slitlamp moscopic examination at either of Masahito Ohji, MD these visits. Fluorescein angiogra- Fumi Gomi, MD Report of a Case. A 61-year-old phy at the 3-month (Figure, C and Shunji Kusaka, MD healthy patient was seen for a sec- D) and 6-month (Figure, E and F) Yasuo Tano, MD ond opinion regarding whether ad- examinations showed no abnormal Osaka, Japan ditional laser treatment was needed fluorescence within the region origi- Neil M. Bressler, MD to treat a retinal break in his left eye. nally occupied by CNV, although Baltimore, Md He had been seen elsewhere for some fluorescence superior to the monitoring of retinoschisis, where macula was noted. a superiorly located horseshoe tear, This study was supported by Novartis unassociated with the schisis cav- Comment. This case from the JAT Ophthalmics, Bu¨lach, Switzerland, and ity, and a smaller break were found demonstrates an apparent disappear- QLT Inc, Vancouver, British Colum- in his left eye. Laser treatment was ance of CNV for at least 6 months bia. Dr Bressler has been paid as a con- recommended. At that time, a con- on fluorescein angiography follow- sultant by QLT Inc and Novartis Oph- tact lens was placed on the left eye ing a single application of photody- thalmics. The terms of this agreement under topical anesthesia, and ap- namic therapy with verteporfin. are being managed by Johns Hopkins proximately 100 moderate-inten- There was no fluorescein staining of University in accordance with its con- sity argon laser lesions were ap- fibrosis or atrophy of the retinal pig- flict of interest policies. plied at the slitlamp, some of which ment epithelium within the area Corresponding author and re- were uncomfortable. Treatment re- originally occupied by CNV. The prints: Miki Sawa, MD, Department quired him to remain motionless, new area of fluorescence noted su- of Ophthalmology, Osaka University and after about 10 minutes, he told perior to the treated area cannot be Medical School, Room E7, 2-2 Yama- his laser surgeon that he felt light- explained at this time. This out- daoka Suita, Osaka 565-0871, headed. The surgeon stopped treat- come has not been seen by any of us Japan (e-mail: sawamiki@ophthal ment but the patient lost conscious- before, including one of us (N.M.B.) .med.osaka-u.ac.jp). ness, fell off his stool, and struck his who served as an investigator at the 1. Treatment of Age-Related Macular Degenera- head against the floor. The physi- Photograph Reading Center, Johns tion With Photodynamic Therapy (TAP) Study cian immediately went to summon Group. Photodynamic therapy of subfoveal cho- Hopkins University, Baltimore, Md, roidal neovascularization in age-related macu- assistance. On return with ancil- and who reviewed 1-week, 4-week, lar degeneration with verteporfin: one-year re- lary medical personnel, the patient and 12-week posttreatment fluores- sults of 2 randomized clinical trials: TAP report was found on the linoleum floor, 1. Arch Ophthalmol. 1999;117:1329-1345. cein angiograms from phase 1 and 2. Treatment of Age-Related Macular Degenera- semiconscious. 2 studies and angiograms from phase tion With Photodynamic Therapy (TAP) Study He was examined in the emer- 3 trials1-4 evaluating verteporfin Group. Photodynamic therapy of subfoveal cho- gency department, where x-rays and 5,6 roidal neovascularization in age-related macu- therapy. Although complete ab- lar degeneration with verteporfin: two-year re- a computed tomographic scan of his sence of fluorescein leakage from sults of 2 randomized clinical trials: TAP report head were obtained. As all findings 2. Arch Ophthalmol. 2001;119:198-207. CNV 1 week after photodynamic 3. Verteporfin in Photodynamic Therapy Study were negative except for an ecchy- 5 therapy has been reported, leak- Group. Photodynamic therapy of subfoveal cho- mosis on his scalp, the patient was age usually reappears within 12 roidal neovascularization in pathologic myopia released with the diagnosis of a va- with verteporfin: one-year results of a random- weeks in approximately 90% of ized clinical trial: VIP report 1. Ophthalmology. sovagal event and returned home. treated cases,1 with fluorescein stain- 2001;108:841-852. During the ensuing weeks, his wife

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©2002 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/28/2021 noted some short-term memory loss, choroidal neovascularization, the de- and he complained of a fullness in velopment of subdural hematoma the head. On his return from a busi- precipitated by discomfort occur- ness trip, he was reunited with a phy- ring during laser treatment is dis- sician friend of his who had not seen tinctly unusual. him for some time. She noted a Many laser surgeons are cur- change in her friend’s affect. Both she rently exploring the use of several and another physician questioned newer laser strategies for the treat- the patient and were concerned that ment of macular degeneration, such he might have experienced a sub- as photodynamic therapy2 and dural hematoma from the fall. A re- transpupillary thermal therapy.3 peated computed tomographic scan Such treatment, along with the ap- was ordered, and a marked shift (30 plication of many lesions during mm) of the falx cerebri was found, conventional panretinal photoco- confirming the diagnosis (Figure). agulation, requires that the patient The patient then underwent a remain motionless, often for ex- craniotomy, 8 weeks after the pho- tended periods of time. In this case, Computed tomographic image taken 2 months tocoagulation session. A second cra- the production of strong chorioreti- after an aborted laser treatment to a retinal break, niotomy was necessary 1 month later nal lesions for retinopexy necessi- showing a radiolucent hygroma (small white because he developed paresis of his tated the use of moderately intense arrows) and subdural hemorrhage (black arrows) that have grossly shifted the falx cerebri from left hand, secondary to a reaccumu- laser spots. These conditions and right to left (large white arrow). Evacuation of the lation of the hematoma. His paresis perhaps pressure on the eye from the hemorrhage required 2 serial craniotomies. eventually resolved. contact lens ultimately precipi- Four months after the acci- tated a vasovagal response in this laser surgeons should have the ap- dent, the patient was seen at our cen- case, which in turn resulted in se- propriate medical training to deal ter. The retinal break had been in- vere consequences. with such issues. completely treated, and additional The head-mounted binocular photocoagulation was necessary. indirect ophthalmoscope laser deliv- Thomas R. Friberg, MD, MS Pittsburgh, Pa This time, an indirect laser delivery ery system is preferred by many sur- system was suggested to allow him geons for panretinal photocoagula- Silvia Pignatto, MD to lie in a more comfortable, su- tion and for the treatment of retinal Verona, Italy pine position during treatment and breaks in the outpatient setting. With The authors do not have any propri- to avoid the necessity of placing a this delivery system, patients can be etary interest relevant to this study. contact lens on his eye. He re- treated while lying down in a re- Corresponding author: Thomas turned 2 months and then 8 months laxed, supine position, which mini- R. Friberg, MD, MS, Department of later and has remained stable and mizes their discomfort and elimi- Ophthalmology, 203 Lothrop St, Suite free from neurologic and visual nates their chance of falling backward 824, Pittsburgh, PA 15213 (e-mail: symptoms. during photocoagulation.4 Such an [email protected]). option should be considered for the Comment. Laser photocoagulation initial treatment of a patient with a 1. Rice TA. Photocoagulation. In: Rice TA, Mich- els RG, Stark WJ, eds. Ophthalmic Surgery.4th is customarily a safe outpatient pro- peripheral lesion, and especially in a ed. St Louis, Mo: CV Mosby; 1984:311-350. cedure associated with few compli- patient for whom discomfort and in- 2. Treatment of Age-Related Macular Degenera- cations.1 However, laser surgery to stability of positioning have been ap- tion with Photodynamic Therapy (TAP) Study Group. Photodynamic therapy of subfoveal cho- an eye is never trivial. Proper facili- parent during previous slitlamp la- roidal neovascularization in age-related macu- ties, equipment, and extensive train- ser delivery. lar degeneration with verteporfin: two-year results of 2 randomized clinical trials: TAP report ing are necessary to safely perform Regardless of the technique 2. Arch Ophthalmol. 2001;119:198-207. it and to manage potential compli- used, this case underscores the fact 3. Ip M, Kroll A, Reichel E. Transpupillary thermal cations. While treatment with ex- that very serious and unexpected se- therapy. Semin Ophthalmol. 1999;14:11-18. 4. Friberg TR. Laser photocoagulation using bin- cessive powers can cause chorioreti- quelae can develop secondary to la- ocular indirect ophthalmoscope laser delivery sys- nal hemorrhage and subsequent ser treatment of the human eye. All tems. Ophthalmic Surg Lasers. 1995; 26:549-599.

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