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CASE REPORTS AND SMALL CASE SERIES the right eye. Her intraocular pres- thors noted that nevi or freckles did Evidence of Early Change sure was maintained in the high not increase in size and the likeli- in Iris Color With teens to low 20s using this treat- hood of change was greatest in those Latanoprost Use ment regimen until she was seen in patients who had heterogeneous pig- 1993 with evidence of progression mentation at baseline. The adverse Latanoprost, a 17-phenyl–substi- of her visual field defect. In 1994, the effect is likely related to an increase medical therapy was changed to 1% in melanin production in the mela- tuted analog of PGF2 a, has been shown to effectively lower intraocu- pilocarpine hydrochloride 4 times nocytes of the iris stroma. lar pressure in clinical trials and daily. During a 3-year period, the op- In the case presented herein, darken the irides in both subhu- tic nerve of her right eye pro- the patient was unilaterally treated, man primates and humans.1 The re- gressed with evidence of vertical thus the slightest change in iris color ported time of onset of the change elongation and a superior rim de- could be detected early. At base- in iris color has been noted to be as fect. In 1996, after a course of dor- line, the patient had an iris color with early as 3 months. To our knowl- zolamide hydrochloride was given mixed pigmentation, placing her at edge, this case represents the earli- in the right eye 3 times daily with high risk for the development of this est reported change in iris color minimum improvement, the pa- adverse effect. This report differs following the initiation of latano- tient was then given latanoprost in from previous reports in that the on- prost use. the right eye only for a 4-week pe- set of change occurred after 4 weeks riod. The iris color in the right eye of treatment. Previous reports2 in Report of a Case. A 78-year-old was the same as the left eye at that subhuman primates noted the white woman was first seen in 1986 time. During the 4-week period, the change after at least 6 weeks of treat- complaining of worsening vision, patient’s iris color changed from ment. We noted no change in the which was found to be secondary to blue-green to brown-green. Use of lashes, which may be related to the nuclear sclerosis involving her right the medication was subsequently short exposure to the drug.3 eye. She underwent an uncompli- discontinued (Figure). cated extracapsular cataract extrac- Regine M. Pappas, MD tion with intraocular lens implan- Comment. Alm et al1 summarized Sharon Pusin, MD tation in December 1988. Prior to the results of 198 patients who pre- Eve J. Higginbotham, MD surgery, she had intraocular pres- viously participated in the original Baltimore, Md sure in the midteens and small sym- phase 3 clinical trials that assessed metric cups with healthy neuroreti- the safety and efficacy of latano- nal rims. In 1992, she had elevated prost. Photographs of the iris were Dr Higginbotham was an investiga- intraocular pressure in the right eye not taken prior to 2 months after the tor in the phase 3 clinical trial and is in the high 20s to low 30s. On vi- initiation of treatment. Darkening of currently an investigator in the com- sual field testing, she demonstrated the iris occurred in 14 patients (7%) bined latanoprost-timolol study, which a nasal step. The patient was then at 6 months and in 24 patients (12%) is sponsored by Pharmacia-Upjohn, given timolol maleate twice daily in after 1 year of treatment. The au- Kalamazoo, Mich. Her expenses for Magnified view of the affected right eye (left) and unaffected left eye (right). ARCH OPHTHALMOL / VOL 116, AUG 1998 1115 ©1998 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/02/2021 travel to various meetings have also maculopathy, and fluorescein angi- sis of a patient with bull’s-eye macu- been reimbursed by Pharmacia- ography showed a retinal pigment lopathy. Certainly, a history of chlo- Upjohn. epithelial window defect in the roquine or hydroxychloroquine use Reprints: Eve J. Higginbotham, macula surrounding a central area would lead the clinician to suspect MD, Department of Ophthalmology, of normal fluorescence (Figure 1, toxicity from systemic medica- University of Maryland School of right). tions. Patients with cone dystro- Medicine, 22 S Greene St, Baltimore, Case 2. A 60-year-old man phy or Stargardt disease generally MD 21201. noted central distortion of vision in have symptoms of visual loss within his left eye after he covered his right the first 2 decades of life and may re- 1. Alm A, Camras C, Watson P. Phase III latano- prost studies in Scandinavia, the United King- eye in 1978. Examination of the left port a family history of ocular dis- dom and the United States. Surv Ophthalmol. eye disclosed a stage 2 macular hole ease.2 In contrast, patients with id- 1997;41(suppl 2):S105-S110. Figure 2 iopathic macular holes generally 2. Selen G, Stjernschantz J, Resul B. Prostaglandin- ( , left) and a visual acuity induced iridial pigmentation in primates. Surv of 20/50. Twenty years later, best- have normal vision until the sixth Ophthalmol. 1997;41(suppl 2):S125-S128. corrected visual acuity was 20/200 through eighth decades of life and 3. Johnstone M. Hypertrichosis and increased pig- mentation of eyelashes and adjacent hair in the OS. Fundus photography revealed a have no family history of macular region of the ipsilateral eyelids of patients treated characteristic bull’s-eye maculopa- disease. The bull’s-eye maculopa- with unilateral topical latanoprost. Am J Oph- thy (Figure 2, right). thy associated with chronic macu- thalmol. 1997;124:544-547. lar holes generally has very sharp Comment. The term bull’s-eye macu- borders between the depigmented lopathy refers to the ophthalmo- macular area and the surrounding Bull’s-eye Maculopathy scopic appearance of a central area normal retinal pigment epithe- Associated With Chronic of retinal pigment epithelial depig- Macular Hole mentation surrounded by rela- tively normal retinal pigment epi- Differential Diagnosis Bull’s-eye maculopathy has been as- thelium giving a “bull’s-eye” of Bull’s-eye Maculopathy* sociated with various macular dis- appearance to the macula. This ap- eases, most notably chloroquine and pearance is shared by a relatively Age-related macular degeneration hydroxychloroquine maculopathy, large group of unrelated condi- Bardet-Biedl syndrome 1 Benign concentric annular macular cone dystrophy, and Stargardt dis- tions. The 2 patients described dystrophy 1-4 ease (Table). To our knowledge, herein demonstrate that chronic Chloroquine and hydroxychloroquine bull’s-eye maculopathy associated macular holes should be included in maculopathy with chronic macular holes has not the differential diagnosis of bull’s- Clofazimine retinopathy been previously reported. eye maculopathy. Selective depig- Cone dystrophy Cone-rod dystrophy mentation of the retinal pigment epi- Fenestrated sheen macular dystrophy Report of Cases. Case 1. A 71-year- thelium may occur under the cuff of Hallervorden-Spatz syndrome old woman developed progressive subretinal fluid that surrounds a Idiopathic central serous visual loss in her left eye in 1990. Ex- chronic macular hole. The retinal chorioretinopathy amination of the left eye disclosed pigment epithelial pigmentation un- Leber congenital amaurosis a macular hole and a visual acuity derlying the macular hole is usu- Lipofuscinosis Sorsby central areolar choroidal dystrophy of 20/200. Seven years later, best- ally preserved and corresponds to the Stargardt disease corrected visual acuity was 20/ center of the bull’s-eye pattern. 200 OS (Figure 1, left). Fundus Several historical and clinical *Differential diagnosis of bull’s-eye photography revealed a bull’s-eye features aid in the specific diagno- maculopathy listed in alphabetical order.1 Figure 1. Case 1. Left, Macular hole in the left eye associated with bull’s-eye maculopathy. Right, Fluorescein angiogram demonstrates retinal pigment epithelial window defect surrounding a circular area of intact retinal pigment epithelium in the central macula. ARCH OPHTHALMOL / VOL 116, AUG 1998 1116 ©1998 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/02/2021 Figure 2. Case 2. Left, Macular hole in the left eye (in 1978). Right, Macular hole (in 1998) associated with bull’s-eye maculopathy. lium, in contrast to the rather indis- 2nd ed. St Louis, Mo: Mosby–Year Book Inc; (right eye), resulting in bilateral 1994:xxxiv. tinct borders of the bull’s-eye macu- 4. Berkow JW, Flower RW, Orth DH, Kelley JS. aphakia. In 1993, the patient first lopathy associated with toxic Fluorescein and Indocyanine Green Angiogra- was seen by his ophthalmologist maculopathies, cone dystrophy, or phy: Technique and Interpretation. 2nd ed. San with redness, discharge, and ocular Francisco, Calif: Palace Press; 1997:108-112. Stargardt disease. The configura- American Academy of Ophthalmology mono- irritation in the left eye. At that time, tion of the bull’s-eye associated with graph. he had an exposed area of conjunc- chronic macular hole appears more tiva superiorly over his scleral rounded in comparison to the more buckle. The buckle was still thought oval configuration in most other to be well positioned, and the pa- bull’s-eye maculopathies. On con- Acute Macular Edema tient was treated using topical cipro- tact lens biomicroscopy, visualiza- Associated With an floxacin. He was seen periodically tion of a cuff of subretinal fluid usu- Infected Scleral Buckle and was thought to be doing well ally allows the clinician to confirm with vision of 20/25 OS with con- the diagnosis of bull’s-eye macu- Exposure or infection of a scleral servative management.

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