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Giant Orbital Cysts After Strabismus Surgery - University of Colorado ...

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more, immunostaining analysis was remarkable for the<br />

cytokeratin-7 marker, suggesting an epithelial-derived tumor.<br />

Systemic evaluation was negative for a primary<br />

malignancy. The patient’s right eye became prephthisical<br />

and painful. Because the potential for rehabilitation was<br />

considered extremely poor, the right eye was enucleated.<br />

Histopathology <strong>of</strong> the globe revealed an ocular surface<br />

squamous neoplasm that appeared to invade the eye<br />

through the cataract incision site and remain contiguous<br />

with epithelial cells on the corneal endothelium and the<br />

iris epithelial tumor (Figure 3). A prominent inflammatory<br />

infiltrate was observed in the region <strong>of</strong> the iris mass<br />

(Figure 3). These findings were consistent with metaplastic<br />

epithelial downgrowth <strong>of</strong> a limbal squamous cell carcinoma<br />

with secondary intraocular inflammation. This phenomenon<br />

should be included in the differential diagnosis<br />

<strong>of</strong> chronic inflammation after intraocular surgery.<br />

REFERENCES<br />

1. Vargas LG, Vroman DT, Soloman KD, et al. Epithelial<br />

downgrowth after clear cornea phacoemulsification: report <strong>of</strong><br />

two cases and review <strong>of</strong> the literature. Ophthalmology 2002;<br />

109:2331–2335.<br />

2. Giaconi JA, Coleman AL, Aldave AJ. Epithelial downgrowth<br />

following surgery for congenital glaucoma. Am J Ophthalmol<br />

2004;138:1075–1077.<br />

3. Kim SK, Ibarra MS, Syed NA, Sulewski ME, Orlin SE.<br />

Development <strong>of</strong> epithelial downgrowth several decades after<br />

intraocular surgery. Cornea 2005;24:108–109.<br />

<strong>Giant</strong> <strong>Orbital</strong> <strong>Cysts</strong> <strong>After</strong> <strong>Strabismus</strong><br />

<strong>Surgery</strong><br />

Theodore H. Curtis, MD, Ann U. Stout, MD,<br />

Arlene V. Drack, MD,<br />

and Vikram D. Durairaj, MD<br />

PURPOSE: To describe a rarely reported complication <strong>of</strong><br />

strabismus surgery.<br />

DESIGN: Observational case series.<br />

METHODS: A review <strong>of</strong> four eyes in three patients with<br />

orbital cysts following strabismus surgery.<br />

RESULTS: Each patient had either a symptomatic strabismus<br />

or visible mass that brought them to medical attention<br />

many years, <strong>of</strong>ten decades after surgery (mean 34<br />

years). All had some degree <strong>of</strong> incomitancy. During<br />

surgery, all cysts were found to be associated with the<br />

involved rectus muscle.<br />

Accepted for publication Apr 25, 2006.<br />

From the Rocky Mountain Lions Eye Institute, <strong>University</strong> <strong>of</strong> <strong>Colorado</strong>,<br />

Aurora, <strong>Colorado</strong> (T.H.C., A.V.D., V.D.D.); and the Casey Eye Institute,<br />

Oregon Health and Sciences <strong>University</strong>, Portland, Oregon (A.U.S.).<br />

Inquiries to Vikram D. Durairaj, MD, 1675 N Ursula St, Box 6510,<br />

Aurora, CO 80045; e-mail: vikram.durairaj@uchsc.edu<br />

FIGURE 1. An intraoperative photo <strong>of</strong> a giant orbital cyst,<br />

which developed after strabismus surgery. The inferior portion<br />

<strong>of</strong> the medial rectus is shown under the muscle hook and the<br />

collapsed cyst is drawn superiorly on the suture.<br />

CONCLUSIONS: <strong>Orbital</strong> cysts are a rarely recognized complication<br />

<strong>of</strong> strabismus surgery. However, it should be<br />

considered in the differential <strong>of</strong> orbital cysts after strabismus<br />

surgery because <strong>of</strong> the risk <strong>of</strong> muscle damage<br />

during surgical excision. (Am J Ophthalmol 2006;<br />

142:697–699. © 2006 by Elsevier Inc. All rights reserved.)<br />

ORBITAL CYSTS ARE UNCOMMON LONG-TERM COMPLIcations<br />

<strong>of</strong> strabismus surgery. Herein, we report on<br />

three patients with giant orbital cysts after strabismus<br />

surgery, including one with bilateral cysts. A retrospective<br />

review <strong>of</strong> three patients with four orbital cysts after<br />

strabismus surgery was initiated after obtaining approval<br />

from the institutional review board.<br />

● CASE 1: A 52-year-old male presented with an enlarging<br />

left orbital mass for two years. His history was remarkable<br />

for esotropia with bilateral strabismus surgery at age 10,<br />

forty-two years before presentation, without subsequent<br />

eye surgeries or injuries. The motility examination showed<br />

an adduction deficit <strong>of</strong> the left eye with a left exotropia<br />

at distance. Slit-lamp examination showed a blue-hued,<br />

mildly vascular cystic mass superonasally. An orbital CT<br />

scan revealed a well-defined anterior-medial mass associated<br />

with the left medial rectus. The globe and bony orbit<br />

were otherwise normal. During surgery, the superior portion<br />

<strong>of</strong> the medial rectus was found attached to the<br />

posterior aspect <strong>of</strong> the cyst; the inferior portion <strong>of</strong> the<br />

medial rectus was attached to sclera posterior to it’s<br />

original insertion (Figure 1). Histopathology revealed a<br />

VOL. 142, NO. 4 BRIEF REPORTS<br />

697


FIGURE 2. External view <strong>of</strong> a giant orbital cyst, which developed<br />

decades after strabismus surgery in Case 3. The cyst is<br />

located over the insertion <strong>of</strong> the right lateral rectus.<br />

sudoriferous cyst with an attached stump <strong>of</strong> striated<br />

muscle.<br />

● CASE 2: A 31-year-old male presented with unsightly<br />

eye misalignment. He had strabismus surgery at age 20,<br />

eleven years before presentation. On examination, he had<br />

a large-angle exotropia, greater in right gaze. Slit-lamp<br />

examination showed bilateral conjunctival scars. No adduction<br />

deficit was appreciated. During surgery, a 10-mm<br />

bluish mass was noted attached to the left medial rectus<br />

and connected to the insertion by fibrous tissue. The cyst<br />

and the underlying muscle were resected. Histopathology<br />

revealed an epithelial inclusion cyst partially surrounded<br />

by striated muscle.<br />

● CASE 3: A 53-year-old woman presented with a fivemonth<br />

history <strong>of</strong> a right orbital mass. She had strabismus<br />

surgery as an infant, almost fifty years previously. Her<br />

motility examination showed an alternating exotropia,<br />

worse in left gaze. Slit-lamp examination revealed a large,<br />

bluish cystic lesion over the insertion <strong>of</strong> the right lateral<br />

rectus (Figure 2). The left eye was unremarkable. Preoperative<br />

magnetic resonance imaging showed a 13 11-mm<br />

cystic lesion involving the right lateral rectus; a similar 9<br />

7-mm lesion was seen within the left medial rectus<br />

(Figure 3). During surgery, the cyst was found to have<br />

intimate adhesions to the lateral rectus muscle. Histopathology<br />

showed a cyst lined by stratified and simple<br />

columnar epithelium.<br />

<strong>Giant</strong> orbital cysts after strabismus surgery are a rarely<br />

described complication. Patients can present either with<br />

the complaint <strong>of</strong> a mass or with the clinical picture <strong>of</strong> a<br />

slipped or lost muscle. 1–4 The cystic mass maybe undetectable,<br />

even on careful slit-lamp examination. One <strong>of</strong> our<br />

cases had no signs or symptoms to suggest a cyst other than<br />

FIGURE 3. T-1 weighted MRI image without contrast (axial<br />

cut) <strong>of</strong> Case 3, showing a giant orbital cyst intimately involved<br />

with both the right lateral rectus and left medial rectus at their<br />

insertions. The patient had bilateral strabismus surgery as an<br />

infant.<br />

incomitant, recurrent strabismus. The two who presented<br />

with a mass were found to have an incomitant strabismus.<br />

Incomitant strabismus as a late finding after strabismus<br />

surgery may suggest the presence <strong>of</strong> an orbital cyst. Finally,<br />

our last patient had evidence <strong>of</strong> bilateral orbital cysts. To<br />

our knowledge, this is the first reported bilateral case in the<br />

literature.<br />

The prevailing hypothesis on the etiology <strong>of</strong> these<br />

cysts is that conjunctival cells are captured by the suture<br />

material and deposited while securing the muscle or<br />

while making the scleral pass. 1 This is supported by<br />

the pathology <strong>of</strong> our cases, where muscle fibers were<br />

involved with the cystic mass. The slow growth <strong>of</strong><br />

these lesions can be inferred by the lag time between<br />

surgery and presentation. Previously, cysts were noted<br />

as long as 35 years after strabismus operations. 1,5 Our<br />

mean time between strabismus surgery and presentation<br />

was 34 years, with two presenting four decades after<br />

surgery.<br />

Although these cysts are benign, they can encroach on<br />

other orbital structures. Special care is required during<br />

surgery because <strong>of</strong> the involvement <strong>of</strong> the affected rectus<br />

muscle. Extensive dissection may be necessary. <strong>Orbital</strong><br />

cysts should be considered in the differential <strong>of</strong> orbital<br />

masses in all patients who have a history <strong>of</strong> strabismus<br />

surgery, no matter how remote.<br />

698 AMERICAN JOURNAL OF OPHTHALMOLOGY<br />

OCTOBER 2006


REFERENCES<br />

1. Kushner BJ. Subconjuctival cysts as a complication <strong>of</strong> strabismus<br />

surgery. Arch Ophthalmol 1992;110:1243–1245.<br />

2. Cibis CW, Waelterman JM. Muscle inclusion cyst as a<br />

complication <strong>of</strong> strabismus surgery. Am J Ophthalmol 1985;<br />

100:740–741.<br />

3. Basar E, Pazarli H, Ozdemir H, Kaner G. Subconjunctival cyst<br />

extending into the orbit. Int Ophthalmol 1998;22:341–343.<br />

4. Metz HS, Searl S, Rosenberg P, Sterns G. <strong>Giant</strong> orbital cyst<br />

after strabismus surgery. J AAPOS 1999;3:185–187.<br />

5. Shields JA, Shields CL. <strong>Orbital</strong> cysts <strong>of</strong> childhood-classification,<br />

clinical features, and management. Surv Ophthalmol<br />

2004;49:281–299.<br />

Real Depth Vs Randot Stereotests<br />

David A. Leske, BS, Eileen E. Birch, PhD,<br />

and Jonathan M. Holmes, BM, BCh<br />

PURPOSE: To compare the performance <strong>of</strong> real depth and<br />

Randot stereotests in strabismic and nonstrabismic patients.<br />

DESIGN: Observational case series.<br />

METHODS: Stereoacuity was tested in 182 patients with a<br />

variety <strong>of</strong> strabismic conditions, using the Frisby-Davis 2<br />

(FD2) distance stereotest, the near Frisby (nF) (both real<br />

depth tests), the Preschool Randot (nR), and Distance<br />

Randot (dR) tests (both based on Polaroid vectographs).<br />

RESULTS: Patients appreciated finer disparities with the<br />

nF test than the nR test at near and with the FD2 test<br />

than the dR test at distance.<br />

CONCLUSIONS: The type <strong>of</strong> stereotest influences measurable<br />

thresholds, and the results from different tests are<br />

not interchangeable. The choice <strong>of</strong> test should depend on<br />

the question being asked; nF and FD2 would be appropriate<br />

for determining presence or absence <strong>of</strong> stereopsis<br />

and best measurable stereopsis. The more rigorous Randot<br />

tests would be appropriate for determining subtle<br />

changes. (Am J Ophthalmol 2006;142:699–701. ©<br />

2006 by Elsevier Inc. All rights reserved.)<br />

TWO COMMONLY USED CATEGORIES OF STEREOTESTS<br />

are “real depth” tests, such as the near Frisby (nF) 1 and<br />

the distance Frisby-Davis 2 (FD2), 2,3 and Polaroid vecto-<br />

Accepted for publication Apr 28, 2006.<br />

From the Department <strong>of</strong> Ophthalmology, Mayo Clinic College <strong>of</strong><br />

Medicine, Rochester, Minnesota (D.A.L., J.M.H.); Retina Foundation <strong>of</strong><br />

the Southwest, Dallas, Texas (E.E.B.); and Department <strong>of</strong> Ophthalmology,<br />

<strong>University</strong> <strong>of</strong> Texas Southwestern Medical Center, Dallas, Texas<br />

(E.E.B.).<br />

Supported by National Institutes <strong>of</strong> Health Grants EY015799 and<br />

EY011751 (J.M.H.) and EY005236 (E.E.B.), Research to Prevent Blindness,<br />

New York, New York (J.M.H. as Olga Keith Weiss Scholar and an<br />

unrestricted grant to the Department <strong>of</strong> Ophthalmology, Mayo Clinic<br />

College <strong>of</strong> Medicine).<br />

Inquiries to Jonathan M. Holmes, BM, BCh, Ophthalmology E7, Mayo<br />

Clinic College <strong>of</strong> Medicine, Rochester, MN 55905; e-mail: holmes.<br />

jonathan@mayo.edu<br />

graph “randot” tests, such as the near Preschool Randot<br />

(nR) 4 and the new Distance Randot (dR). 5 To determine<br />

whether there are systematic differences between these<br />

categories <strong>of</strong> tests, we evaluated their performance in a<br />

large cohort <strong>of</strong> patients.<br />

One hundred and eighty-two patients in a strabismus<br />

practice, ages 4 to 84 years, with visual acuity <strong>of</strong> 20/40 or<br />

better in each eye, up to 70 prism diopters (pd) <strong>of</strong><br />

esotropia, 55 pd exotropia, or 30 pd <strong>of</strong> hypertropia completed<br />

the nF, FD2, nR, and dR stereotests as part <strong>of</strong><br />

routine evaluation. Criterion <strong>of</strong> 20/40 in each eye was used<br />

to avoid bias against Randot tests because <strong>of</strong> dot resolution.<br />

All four tests were administered using previously<br />

described presentation protocols. 3–6 The study was approved<br />

by respective Institutional Review Boards and<br />

conducted in a Health Insurance Portability and Accountability<br />

Act (HIPAA)–compliant manner. To eliminate<br />

potential bias <strong>of</strong> comparing tests with different ranges <strong>of</strong><br />

stereopsis, results were rescored as “fine” (20 to 60 seconds<br />

<strong>of</strong> arc [sec arc]), “moderate” (75 to 200 sec arc), or<br />

“coarse/nil” (400 sec arc to nil) stereo (Table 1). We<br />

compared these rescored stereopsis levels between tests and<br />

determined the frequency <strong>of</strong> one test indicating a finer or<br />

measurable threshold compared with the alternative test.<br />

At near, patients appreciated finer disparities with the<br />

nF than the nR test (median “moderate” vs “coarse/nil,” P<br />

.0001). At distance, patients appreciated finer disparities<br />

on the FD2 than the dR test (median “moderate” vs<br />

“coarse/nil,” P .0001). Only 4% <strong>of</strong> patients had better<br />

stereoacuity with nR than nF, and no patient had better<br />

stereoacuity with dR than FD2. When one test indicated<br />

nil stereopsis, the patient was much more likely to have<br />

measurable stereopsis with a real depth test (nF or FD2)<br />

than a Randot test (Table 2).<br />

One possible explanation for different median thresholds<br />

is that one or more tests were more prone to false<br />

positives. We therefore analyzed patients with 20 pd <strong>of</strong><br />

constant deviation. Only one <strong>of</strong> 21 (5%) patients with<br />

20 pd at near had measurable stereopsis with the nF and<br />

one <strong>of</strong> 25 (4%) with 20 pd at distance had measurable<br />

stereopsis the FD2 (none with the Randot tests), and that<br />

individual patient had Duane’s syndrome with marked<br />

incomitance. It is possible that, despite attempting to<br />

measure the stereoacuity in primary position, this patient<br />

adopted a subtle face turn during stereoacuity testing,<br />

reducing his angle <strong>of</strong> deviation. Previous studies have<br />

suggested that our standardized presentation protocols 3,7<br />

reduce the impact <strong>of</strong> monocular cues to a negligible level.<br />

There are at least three possible explanations why the<br />

real depth tests might demonstrate measurable stereopsis<br />

when the Randot tests demonstrate no stereopsis: (1)<br />

patients had intermittent tropias that were better controlled<br />

when viewing real world targets, (2) there was<br />

greater dissociation under conditions <strong>of</strong> Randot testing<br />

(when the deviation by alternate cover test (ACT) <br />

simultaneous prism cover test [SPCT]), and (3) the tests<br />

VOL. 142, NO. 4 BRIEF REPORTS<br />

699

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