acute exudative polymorphous vitelliform maculopathy probably indicates shadowing by the dense subretinal deposit in the initial stages of the disorder. Resolution of the hyporeflective space precedes resolution of the macular lesions and improvement in vision. Multiple perimacular lesions, lack of family history, sudden onset, and rapid resolution make adult-onset foveomacular vitelliform dystrophy unlikely, despite the similarity of OCT findings. Best macular dystrophy, adult-onset foveomacular vitelliform dystrophy, and acute exudative polymorphous vitelliform maculopathy may each share retinal pigment epithelium dysfunction initiated by different stimuli. Mutations in the VMD2 gene are associated with Best macular dystrophy,5 and novel mutations occur in maculopathies other than Best macular dystrophy, including adult-onset foveomacular vitelliform dystrophy.5,6 In Best macular dystrophy, accumulation of lipofuscin in dysfunctional retinal pigment epithelium cells is the probable cause of the abnormal electrooculogram and the accumulation of subretinal fluid. In acute exudative polymorphous vitelliform maculopathy, acquired retinal pigment epithelium dysfunction may cause deposition of subretinal material, as shown by OCT, that is similar to the pseudovitelliform material of adult-onset foveomacular vitelliform dystrophy. Interestingly, no recovery of the electrooculogram occurred in our patients despite improvement in vision, fundus, and OCT findings. Acute exudative polymorphous vitelliform maculopathy may occur in predisposed patients with abnormal retinal pigment epithelium after an unknown trigger.
REFERENCES
1. Gass JDM, Chuang EL, Granek H. Acute exudative polymorphous vitelliform maculopathy. Trans Am Ophthalmol Soc 1988;86:354 –366. 2. Gass JDM. Stereoscopic atlas of macular diseases: diagnosis and treatment, 4th ed. St Louis: CV Mosby, 1997:168 – 169,312–313,696 – 697. 3. Benhamou N, Souied EH, Zolf R, Coscas F, Coscas G, Soubrane G. Adult-onset foveomacular vitelliform dystrophy: a study by optical coherence tomography. Am J Ophthalmol 2003;135:362–367. 4. Pierro L, Tremolada G, Introini U, Calori G, Brancato R. Optical coherence tomography findings in adult-onset foveomacular vitelliform dystrophy. Am J Ophthalmol 2002;134: 675–680. 5. Kramer F, White K, Pauleikhoff D, et al. Mutations in the VMD2 gene are associated with juvenile-onset vitelliform macular dystrophy (Best disease) and adult vitelliform macular dystrophy but not age-related macular degeneration. Eur J Hum Genet 2000;8:286 –292. 6. Seddon JM, Afshari MA, Sharma S, et al. Assessment of mutations in the Best macular dystrophy (VMD2) gene in patients with adult-onset foveomacular vitelliform dystrophy, age-related maculopathy, and bull’s-eye maculopathy. Ophthalmology 2001;108:2060 –2067.
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Epiretinal Membrane Formation With Internal Limiting Membrane Wrinkling After Nd:YAG Laser Membranotomy in Valsalva Retinopathy Alvin K. H. Kwok, FRCS, Timothy Y. Y. Lai, MRCS, and Nongnart R. Chan, MD
PURPOSE: To report the location of premacular hemorrhage in Valsalva retinopathy and epiretinal membrane formation with internal limiting membrane (ILM) wrinkling after neodymium;yttrium-aluminum-garnet (Nd: YAG) laser membranotomy. DESIGN: Interventional case report. METHODS: A 35-year-old man with a massive premacular hemorrhage due to Valsalva retinopathy underwent Nd: YAG laser membranotomy. After membranotomy, his visual acuity improved from 20/200 to 20/25. Ten months later, he developed metamorphopsia with visual acuity of 20/30. A semiopacified membrane with radiating striae was found, and pars plana vitrectomy was performed for membrane removal. RESULTS: Histologic examination of the membrane showed hemosiderin deposits within macrophages on the retinal side of the ILM. One year later, the patient was asymptomatic with visual acuity of 20/20. CONCLUSIONS: To our knowledge, histopathologic confirmation of the sub-ILM hemorrhage in Valsalva retinopathy and epiretinal membrane formation with ILM wrinkling as a complication after Nd:YAG laser membranotomy have not been previously reported. (Am J Ophthalmol 2003;136:763–766. © 2003 by Elsevier Inc. All rights reserved.)
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HE EXACT ANATOMIC LOCATION OF PREMACULAR
hemorrhage in Valsalva retinopathy, either subinternal limiting membrane (ILM) or subhyaloid, or a combination of both, is not yet defined.1 We treated a patient
Accepted for publication April 11, 2003. From the Department of and Ophthalmology, The Hong Kong Sanatorium and Hospital, Hong Kong, People’s Republic of China (A.K.H.K.); Department of Ophthalmology and Visual Sciences, The Chinese University of Hong Kong, Hong Kong Eye Hospital, Hong Kong, People’s Republic of China (A.K.H.K., T.Y.Y.L., N.R.C.). Inquiries to Alvin K. H. Kwok, FRCS, Department of Ophthalmology, Hong Kong Sanatorium and Hospital, 2 Village Road, Happy Valley, Hong Kong, China; fax: (⫹852) 2835 8008; e-mail:
[email protected]
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FIGURE 1. Right eye: (A) Fundus photo on presentation showing a massive well circumscribed fresh premacular hemorrhage of 5-disk-diameter underneath a transparent membrane. There was a fluid level within the hematoma and a small reddish clot over the superotemporal arcade suggestive of the bleeding site. (B) Fundus photo 1 week after laser membranotomy showing resolution of the macular hemorrhage with a transparent membrane left behind trapping a small amount of blood inferiorly. (C) Fundus photo 10 months after laser membranotomy showing wrinkled membrane at the foveal region with radiating striae. There were scattered rusty orange-brown granular deposits (iridescent spots) under the semiopaque part of the membrane. (D) Internal limiting membrane (ILM) was stained by indocyanine green dye intraoperatively. The membrane (arrowheads) was held by an intraocular forceps during removal. Long arrows indicated the margin of retina devoid of ILM.
with secondary epiretinal membrane (ERM) after neodymium:yttrium-aluminum-garnet (Nd:YAG) laser membranotomy for premacular hemorrhage due to Valsalva retinopathy by removing the ERM with the underlying ILM. To our knowledge, histologic confirmation of the sub-ILM hemorrhage and ERM formation as a complication of Nd:YAG laser membranotomy for Valsalva retinopathy have not been previously reported. A 35-year-old healthy Chinese man presented with sudden right eye visual loss after nose blowing with the nostrils and mouth closed by his hands. On examination, his visual acuity was 20/200 right eye (OD) and 20/20 left eye (OS). Fundus examination of the OD showed a massive well-circumscribed premacular hemorrhage under764
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neath a transparent membrane (Figure 1A). The diagnosis was premacular hemorrhage due to Valsalva maneuver. Laboratory investigations showed normal complete blood count, prothrombin time, and activated partial thromboplastin time. Blood pressure and urinalysis were normal. Management options like observation, laser membranotomy, or primary vitrectomy were discussed. The patient preferred Nd:YAG laser membranotomy that was performed with a single 4.0 mJ Q-switched pulse targeting on the membrane at the dependent position over the hemorrhage. This caused immediate drainage of the hemorrhage into the vitreous cavity. One week later, his vision improved to 20/25 OD, and fundus examination showed resolution of the premacular hemorrhage, with a transparOF
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FIGURE 2. (A) Histologic section showing continuous folded periodic acid–Schiff-positive membrane consistent with the internal limiting membrane (periodic acid–Schiff stain, original magnification ⴛ 20). (B) Internal limiting membrane shows adherent clump of pigmented macrophages containing intracellular iron deposition over the irregular retinal side of internal limiting membrane. The presence of hemosiderin in the pigmented macrophage is confirmed by iron staining (periodic acid–Schiff and Prussian blue stain, original magnification ⴛ 40). (C) Histologic section showing nuclei of a fine glial nonvascularized membrane (arrow) on the surface of internal limiting membrane suggestive of epiretinal membrane (hematoxylin and eosin stain, original magnification ⴛ 10).
ent membrane demarcating the previous location of the hemorrhage (Figure 1B). Ten months after laser membranotomy, the patient complained of metamorphopsia in the OD. Visual acuity was 20/30 OD and 20/20 OS. Examination revealed an ERM with radiating striae at the right macula (Figure 1C). The patient underwent pars plana vitrectomy for membrane removal. After induction of posterior vitreous detachment and posterior hyaloid removal, the wrinkled membrane remained undisturbed on the macula, suggesting it was not located at the level of the posterior hyaloid. Indocyanine green dye was then injected with the ERM unstained, whereas other parts of the membrane were stained green. The loosely adherent membrane was removed using myringo-vitreal-retinal blade and intraocular forceps (Figure 1D ). One year after surgery, the patient VOL. 136, NO. 4
was metamorphopsia free with visual acuity of 20/20 OD, and the macula appeared unremarkable. Histologic examination of the membrane revealed continuous folded periodic acid–Schiff-positive membranes consistent with ILM (Figure 2A). Prussian blue staining confirmed the presence of hemosiderin within macrophages over the retinal side of ILM (Figure 2B); hematoxylin and eosin staining showed nuclei of a fine glial ERM with underlying ILM (Figure 2C). Previous studies have shown that indocyanine green selectively stained ILM but not the overlying ERM.2,3 From the histology, the ERM observed correlated with the unstained foveal part of the membrane, which was surrounded by the stained ILM. The presence of hemosiderin in pigmented macrophage located at the retinal side of the ILM confirmed the sub-ILM location of hemorrhage in
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Valsalva retinopathy. As seen in this case, ERM formation with ILM wrinkling may occur as a late complication of Nd:YAG laser membranotomy. This potential complication has not been reported previously, even in long-term studies using multiple pulses of Nd:YAG laser with energy up to 11.5mJ.4,5 REFERENCES
1. Gass JDM. A Stereoscopic atlas of macular diseases: diagnosis and treatment. St. Louis: C. V. Mosby, 1987. 2. Gandorfer A, Messmer EM, Ulbig MW, Kampik A. Indocyanine green selectively stains the internal limiting membrane. Am J Ophthalmol 2001;131:387–388. 3. Kwok AKH, Li WW, Pang CP, et al. Indocyanine green staining and removal of internal limiting membrane in macular hole surgery: histology and outcome. Am J Ophthalmol 2001;132:178 –183. 4. Raymond LA. Neodymium: YAG laser treatment for hemorrhages under the internal limiting membrane and posterior hyaloid face in the macula. Ophthalmology 1995;102:406 – 411. 5. Ulbig MW, Mangouritsas G, Rothba¨ cher H, et al. Long-term results after drainage of premacular subhyaloid hemorrhage into the vitreous with a pulsed Nd:YAG laser. Arch Ophthalmol 1998;116:1465–1469.
CONCLUSIONS: Internal limiting membrane defects may facilitate the entry of silicone oil into the retina, leading to accumulation of oil vacuoles. The use of silicone oil in macular hole surgery with ILM peeling may complicate the postoperative outcome. (Am J Ophthalmol 2003; 136:766 –767. © 2003 by Elsevier Inc. All rights reserved.)
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Intraretinal Silicone Oil Vacuoles After Macular Hole Surgery With Internal Limiting Membrane Peeling Juliet Chung, BS, and Richard Spaide, MD To report the detection of intraretinal silicone oil vacuoles after the use of a silicone oil tamponade for macular hole surgery with internal limiting membrane (ILM) peeling. DESIGN: Observational case report. METHODS: A 57-year-old woman with a recurrent macular hole in the left eye underwent macular hole surgery with ILM peeling and silicone oil tamponade. After early silicone oil emulsification was detected, the silicone oil was removed. RESULTS: Follow-up ophthalmoscopic examination and optical coherence tomography imaging revealed intraretinal silicone oil vacuoles in the area of ILM peeling. PURPOSE:
Accepted for publication April 15, 2003. From the Vitreous-Retina-Macula-Consultants of New York, NY LuEsther T. Mertz Retinal Research Center, Manhattan Eye, Ear and Throat Hospital, New York, New York. Inquiries to Richard Spaide, MD, Vitreous-Retina-Macula Consultants of New York, 519 E 72nd St, Suite 203, New York, NY 10021; fax: (212) 628-0698; e-mail:
[email protected]
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ILICONE OIL INJECTION HAS BEEN SUGGESTED AS A
means to tamponade macular holes to obviate the need for rigorous postoperative positioning or to allow highaltitude travel in the immediate postoperative period.1 There are several problems inherent with silicone use, however. Silicone oil has a lower surface tension than gas, and eventually it can emulsify. Also, silicone oil injection necessitates its removal, obligating the patient to another operative procedure. Silicone oil is associated with a number of unique complications, including what is described in this case—silicone oil droplets within the retina after macular hole surgery using internal limiting membrane (ILM) peeling. A 57-year-old woman with a recurrent macular hole in the left eye was unable to maintain face-down positioning after her previous surgery and did not wish to undergo any restrictive positioning after a repeat surgery. Her bestcorrected visual acuity was 20/400. She underwent pars plana vitrectomy with indocyanine green dye-assisted ILM peeling followed by silicone oil (5,000 centistoke) injection. The macular hole closed by the first postoperative day, but 2 months later emulsification of the silicone oil was observed. The silicone oil was removed and replaced with fluid. During the silicone oil removal surgery, the pars plicata was observed with scleral depression and small bubbles of silicone oil were dislodged with gentle tapping. A fluid-gas exchange was repeated twice to remove all visible remnants of silicone oil. Postoperatively, the macular hole was closed, but the patient had small clear vacuoles within the thickness of the retina in the macular region where the ILM peeling had been performed (Figure 1). These vacuoles were found to be intraretinal cystoid spaces by optical coherence tomography imaging. Follow-up fundoscopic examinations showed a decrease in intraretinal silicone oil vacuoles over a period of 18 months, and the final best-corrected visual acuity was 20/60. The patient never had evidence of clinically evident intraocular inflammation. Silicone oil droplets have been seen in the retina, either in enucleated eyes or in eyes with recurrent retinal detachments and having concurrent retinal biopsy.2 Eckardt and associates2 found single intraretinal macrophages containing silicone in eyes injected with silicone oil for 2 years and showed that the retina had multiple defects in the ILM. Ohira and coworkers3 showed that emulsified silicone oil injected into rabbit eyes appeared in the inner retinal layers as early as 1 week after the injection. Our patient had early emulsification of her
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