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Reticular Tapeto-Retinal Dystrophy As a Possible Late Stage of Sjogren's Reticular Dystrophy

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Reticular Tapeto-Retinal Dystrophy As a Possible Late Stage of Sjogren's Reticular Dystrophy Brit. Ophthal. (1970) 6o, 35 Y. Br J Ophthalmol: first published as 10.1136/bjo.60.1.35 on 1 January 1976. Downloaded from Reticular tapeto-retinal dystrophy As a possible late stage of Sjogren's reticular dystrophy GERALD A. FISHMAN, MICHAEL B. WOOLF, MORTON F. GOLDBERG, AND BRUCE BUSSE From the Department of Ophthalmology, University of Illinois Eye and Ear Infirmary, Chicago, Illinois Two patients with a reticular dystrophy of the formed polygonal reticular units analogous to the retina were noted to have fundus changes reminis- meshes of a fishnet. Hyperpigmented 'knobs' were cent of those reported in patients with Sjogren's noted at the junction of individual polygonal units as Our were older than in a net. Within each unit, extensive hypopigmentation reticular dystrophy. subjects of the retinal pigment epithelium and minimal atrophy previously reported cases of Sjogren's dystrophy of the choriocapillaris could be demonstrated by fluo- and manifested more extensive retinal abnormali- rescein angiography (Fig. 2). Fig. 2 also shows linear or ties involving both the retinal pigment epithelium sickle-shaped dark areas of hypofluorescence which and photoreceptors. Similarities were noted both correspond to zones of hyperpigmentation. Lesions in ophthalmoscopically and on fluorescein angi- the posterior pole appeared more confluent and less ography. Reduced central visual acuity, abnormal hyperpigmented, no longer maintaining a reticular findings on electroretinography, ophthalmoscopi- appearance as they approached the optic disc. Within copyright. cally evident hypopigmentation, and probable the maculae were accumulations of moderately dense of the retinal pigment epithelium were black clumps of retinal pigment epithelium (Fig. 3). atrophy The retinal vessels and optic discs were normal. Elec- specific to the more advanced condition. troretinographic (ERG) responses were nondetectable in both eyes. Because of poor co-operation and a marked Case reports reduction in visual acuity, it was impossible to obtain reproducible visual fields, electro-oculogram (EOG), Case I, a 72-year-old white man, had a 6-year history or dark-adaptation studies. The plasma omithine level of bilateral decrease in central vision and a slowly was normal (Simmell and Takki, http://bjo.bmj.com/ progressive loss of night vision over the past io years. I973). The patient could not recall having been examined by an ophthalmologist before the onset of these symptoms. His father allegedly had had similar difficulties with both central and night vision which had begun when he was in his forties, although further details of his eye disease were not available. Other family members, including seven children, were said to be not as yet on September 26, 2021 by guest. Protected affected. The patient was unaware of either Swedish or Dutch ancestors. When he was seen in September I974, the patient's best corrected vision was 3/200 in the right eye and Io/2oo in the left, with correction + iD sph. in the right eye and + I*25D sph. in the left. His general health was good. External and motility examinations were normal, both pupils reacted normally to direct and con- sensual light stimuli, slit-lamp examination of the anterior segments was normal, as was the intraocular pressure of 14 mm Hg in the right eye and i2 in the left. There was no increase in cellularity of the vitreous. The patient hadl to 2+ bilateral nuclear sclerotic and anterior cortical changes in both lenses. Fundus exami- FIG. 2 Case i. Late-stage fluorescein angiogram of nation showed bilateral, midperipheral 360° reticular- peripheral lesions. Hypopigmentation of retinal pigment appearing lesions (Fig. i). Alternate areas of pigment epithelium is demonstrated by hyperfluorescence within epithelial hypopigmentation and hyperpigrnentation polygonal units (large arrows). Patchy choriocapillaris atrophy is demonstrated by retained visibility of large Address for reprints: Gerald A. Fishman, MD, Eye and Ear choroidal vessels within some polygonal units (arrows). Infirmary, I855 West Taylor Street, Chicago, Illinois 6o6I2, USA Also seen are sickle-shaped areas of hypofluorescence 36 British 7ournal of Ophthalmology Br J Ophthalmol: first published as 10.1136/bjo.60.1.35 on 1 January 1976. Downloaded from Case 2, a 63-year-old white woman, had a history of fields measured on a Goldmann perimeter revealed a I 5° progressively failing central vision during the preceding relative central scotoma bilaterally. Peripheral fields were decade and diminishing night vision of approximately irregularly constricted within 30° to 2 isoptres (II-4e and I2 years' duration. Visual acuity, documented else- IV-4e). An ERG revealed markedly subnormal photopic where in 1972, was 20/20 in the right eye and 20/30 and scotopic responses in both eyes. The scotopic b-wave in the left. At that time, the visual fields were noted to amplitude was only 50 LV in the right eye and 75 ptV in be constricted. Because of a fundus abnormality, her the left after 20 min of dark adaptation. (Scotopic b-wave physicians considered the diagnosis of tapeto-retinal values for her age would normally be greater than degeneration or gyrate atrophy of the choroid and 275 to 300 [tV in our laboratory.) An EOG showed retina. light-peak to dark-trough ratios of I-50 bilaterally. The patient's father was alleged to have had extremely These ratios are significantly below I-70, the lowest limit poor eyesight at the time of his death at the age of 85 of normal for the patient's age group. Dark-adaptation years. His ocular problems began at about the age of studies were unsuccessful because of the patient's 55 years with night blindness and decreasing central marked loss of central vision. A quantitative urine- vision. He was told by an ophthalmologist that he had a analysis and plasma determination for amino-acids 'pigment dispersal'. The patient's elder brother also revealed a normal electrophoretic pattern with no had markedly reduced vision at the time of his death at increase in ornithine. the age of 78 years. However, he was a diabetic, and The patient's only child, a 41-year-old son, was details of his eye disease were not available. Other examined ophthalmoscopically and found to be normal. members of the family, including six other siblings, Findings on ERG, EOG, and fluorescein angiography were not similarly affected. Although her specific were also normal. recollections were uncertain, the patient stated that she did have Dutch ancestors. When first examined by us in May I973 the patient's Discussion visual acuity was hand movements at I m with correction Previously reported diseases with reticular pig- +3 D sph. in both eyes. Extemal and motility examina- mentary changes of the retina include Sjogren's tions were unremarkable. Both pupils reacted briskly to direct and consensual light stimuli. Slit-lamp bio- reticular dystrophy, Mesker's macroreticular dys-copyright. microscopy of the anterior segments was normal. Early cortical and nuclear sclerotic changes were seen in both lenses. The vitreous was normal bilaterally. Ophthalmoscopic examination showed that the appear- FIG. I Case i. Reticular-appearing lesions in ance of both eyes was virtually identical (Fig. 4). Both midperiphery of right eye. Numerous polygonal units discs were normal, with a cup-disc ratio of 0o4. The are formed by alternate areas of pigment epithelial retinal vessels appeared to be normal in calibre and hypopigmentation and hyperpigmentation. Note was a knob-like at polygonal interfaces (arrows) course. There scattered mottling of the retinal formations http://bjo.bmj.com/ pigment epithelium within the macular region, espe- cially in the right eye (Fig. 5). The sensory retinal tissue FIG. 3 Case i. Posterior pole of right eye. Moderately in this region appeared thin on biomicroscopic exami- dense clumps of retinal pigment are seen within macula. nation. Approximately 4 disc diameters temporal to the Also demonstrated are areas of retinal pigment epithelial fovea and i disc diameter nasal to the disc, discrete hypopigmentation. Disc and retinal vessels are normal reticular pigmentary changes began which extended anteriorly to the equator for 3600. This pattern became FIG. 4 Case 2. Montage from right eye depicting confluent and therefore indistinct at the posterior pole. mosaic of polygonal units formed by areas of retinal on September 26, 2021 by guest. Protected As in Case I, the reticular changes were formed by pigment epithelial hypopigmentation alternate areas of decreased and increased density of the retinal pigment epithelium. Areas of increased pig- FIG. 5 Case 2. Pigment mottling within right macula. ment density circumscribed zones of hypopigmentation Optic disc and retinal vessels were normal for patient's creating a polygonal mosaic pattern. These areas were age. Note areas of retinal pigment epithelial hypopig- approximately one-half disc diameter in size (Fig. 6). mentation in perimacular regions (arrows) Occasional scattered clumps of pigment were seen in the sensory retina. Fluorescein angiography demon- FIG. 6 Case 2. Reticular changes in midperiphery. strated that choriocapillaris vessels were present in As in Case i, both areas of pigment epithelial hypopig- areas of clinically evident retinal pigment epithelial mentation (larger arrows) and hyperpigmentation hypopigmentation (Fig. 7). Even within the posterior (smaller arrows) are seen. Although less evident than in pole, where the reticular pattern was clinically less Case i, knob-like formations
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