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Birdshot Chorioretinopathy: Clinical Characteristics and Evolution*

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Birdshot Chorioretinopathy: Clinical Characteristics and Evolution* Br J Ophthalmol: first published as 10.1136/bjo.72.9.646 on 1 September 1988. Downloaded from British Journal of Ophthalmology, 1988, 72, 646-659 Birdshot chorioretinopathy: clinical characteristics and evolution* HILDE A PRIEM'2 AND JENDO A OOSTERHUIS2 From the 'Eye Clinic, University ofGent, Belgium, and the 2Eye Clinic, University ofLeiden, The Netherlands SUMMARY During the period 1980-6 102 patients from 14 European eye clinics were diagnosed as having birdshot chorioretinopathy (BSCR). All were Caucasian, and the series consisted of 47 men and 55 women, with a mean age of 52*5 years. The major findings in this rare disorder concern the ocular fundus. Most marked are the patterned distribution of depigmented spots without hyperpigmentation, radiation from the optic disc in association with vitritis, retinal vasculopathy with frequent cystoid macular oedema, and involvement of the optic nerve head. The distribution and appearance of the lesions suggest that they are related to the major choroidal veins. Complications of the disease were epiretinal membranes, retinal neovascularisation, recurrent vitreous haemorrhage, subretinal neovascular membranes occurring both in the juxtapapillary and macular regions, and optic atrophy. The medical history was not contributory. HLA testing showed very strong disease association with HLA A29 (95.8%). The evidence suggests that it is a single disease a entity rather than group of disorders because of the remarkable similarity in the copyright. ophthalmological appearance and the clinical course, combined with the exceptionally high association with HLA A29. In 1980 a rare ocular disease was described by Ryan and Maumenee.' In the absence of a known aetiology, they chose the descriptive name 'birdshot http://bjo.bmj.com/ retinochoroidopathy' inspired by the unusual picture, which showed multiple small, cream coloured lesions, scattered mainly around the optic disc and radiating towards the equator, without hyperpigmentation at the level of the retinal pigment _ * epithelium. Because of the presence of vitritis, : retinal vasculitis, and cystoid macular oedema they <7i on September 29, 2021 by guest. Protected likened the disease to pars planitis, but emphasised the absence of snowbanking over the pars plana. Other descriptive names have been used for this *This paper is the result of a collaboration by the following members of the European Fluorescein Angiography Club: P Amalric (Albi, France); M Spitznas (Bonn, FRG); C Orsini Dupont (Caen, France); G Coscas, A Gaudric, G Soubrane (Creteil, France); JJ de Laey, H A Priem (Gent, Belgium); J Hooymans, Z Timmerman (Groningen, The Netherlands); J A Oosterhuis, A H Renger-van _ Louvain,aitnDijkLoviBlim;ABrRBah(Leiden,Belgium);The ANetherlands);Bird, R Blach,AZ Leys.Gregor,rgnB ASnyersM Hamilton, D \. _ Sharp, J Shilling (London, England); A F Deutman (Leuven-UThe (Nijmegen, Netherlands); J A Bernard, G Quentel (Paris, France); G S Fig. 1 PatientofFranceschettiand Babel with systemic Baarsma, P T V M de Jong, B C P Polak (Rotterdam, The sarcoidosis. Whitefleckspredominantly in nasalperiphery Netherlands). and round optic disc with radialpattern and tendency toform Correspondence to Dr H A Priem, Dienst Oogheelkunde, lines towardsperiphery. (Bypermission ofthe Fondation Academisch Ziekenhuis, De Pintelaan 185, 9000 Gent, Belgium. ProfessorA Franceschetti.) 646 Br J Ophthalmol: first published as 10.1136/bjo.72.9.646 on 1 September 1988. Downloaded from Birdshotchorioretinopathy: clinical characteristics and evolution 647 entity, such as 'vitiliginous chorioretinitis' by Gass,2 (average 54*4 years). All were Caucasian. They were who stressed the similarity of the fundus patches to examined both clinically and by colour photography cutaneous vitiligo, 'salmon-patch choroidopathy' by and fluorescein angiography (Table 1). The clinical Aaberg,3 and 'chorioretinopathie en grains de riz' by files and the photographic documentation were Amalric and Cuq,4 who saw a similarity to the rice studied by one of us (HAP). grain pattern. The first set of data included the general history, Probably the first description of birdshot chorio- with specific reference to skin disease, allergy, and retinopathy (BSCR) in the early literature occurred hearing loss; and the ocular history, which covered in 1949 in a case report by Franceschetti and Babel.' previous eye disease, surgery, and family history. They described a 63-year-old woman with a condition The second set of data concerned the clinical examin- they named 'chorioretinopathie en taches de bougie' ation, including visual acuity, findings on slit-lamp (candlewax spots), who had systemic features of examination, tonometry, and ophthalmoscopy. The sarcoidosis. On ocular examination she had minimal third described the fluorescein angiographic findings. involvement of the anterior segment with only The fourth dealt with additional investigations such minute keratic precipitates (KP) but had dense as colour vision tests, perimetry, dark adaptation, vitreous opacity. The authors described a pale optic electro-oculogram (EOG), electroretinogram disc, very narrow retinal vessels, and multiple white (ERG), visually evoked potentials (VEP), and HLA 'retinal and chorioretinal' spots, and stressed the typing. Finally, long term behaviour and the absence of pigmentation of the spots (Fig. 1). Later influence of treatment were assessed. they saw areas of choroidal atrophy appear in the inferonasal quadrant. All these features are characteristic of BSCR. Table 1 Clinicalandfunctional investigations Other authors have highlighted special character- NumberofBSCRpatients istics of BSCR such as distinctive electrophysiologi- involved cal abnormalities,6 its association with the HLA A29 copyright. gene,7 and the occurrence of complications such as Total number in study 102 Fundus pictures 102 subretinal neovascular membranes'9 and optic Fluorescein angiography 102 atrophy.'2 The association with a rhegmatogenous Medical history 71 retinal detachment has also been reported.'6""' Ocular history 79 More case reports followed""'7 dealing with small Slit-lamp 68 Tonometry 60 numbers of patients only, giving the overall impres- Visual acuity 94 sion that BSCR is a rare disease. Colourvision tests 31 In this study we analyse in a large series of patients Perimetry 43 http://bjo.bmj.com/ the entity known as birdshot chorioretinopathy Dark adaptation tests 24 (BSCR) as defined by the EOG and ERG 42 accepted clinical picture. In VEP 30 particular, we sought to determine whether or not HLA typing 48 (in 5 patients A29 only) this is a single clinical entity or simply a collection of exotic cases with similar appearance. Table 2 Medicalhistory in 71 BSCRpatients Subjects and methods No. ofpatients on September 29, 2021 by guest. Protected The patients were collected from 14 ophthalmologi- Arterial hypertension 16 cal in Cardiac disease 5 departments five European countries. The Cerebrovascular accident 2 minimum criteria used for patient selection were Rheumatic disease, arthritis 6 based on the clinical manifestations as described by Low backache 2 Ryan and Maumenee.' Each patient was required to Gout I have the cream spots a Allergy (penicillin, fluorescein) 6 typical coloured birdshot with Eczema, psoriasis 4 symmetrical appearance in both eyes (if asymmetri- Hirsutism I cal, then one eye should at least be very characteristic Vitiligo (only in summer season) 1 of BSCR in number, size, and distribution of the Hearing loss 3 lesions), together with one of the following features: Meniere's disease 1 Migraine I evidence of retinal vasculopathy, evidence of disc Infectious disease: oedema or optic atrophy, or presence of vitreous tuberculosis 2 cells. viral hepatitis 2 The patients consisted of 47 men aged 23-79 years pericarditis I sarcoidosis 1 (average 50-7 years) and 55 women aged 32-74 years Br J Ophthalmol: first published as 10.1136/bjo.72.9.646 on 1 September 1988. Downloaded from 648 Hilde A Priem andJendo A Oosterhuis Results Table 3 Ocular history in 79patients with BSCR The study comprised 203 eyes of 102 BSCR patients; Eyes one eye with an extensive corneal leucoma since Open angle glaucoma 16 (1 eye Scheie operation) childhood preventing fundus examination was not Secondary glaucoma 2 (2 trabeculectomies) included. Full information on the clinical and Cataract extraction 5 (3 ECCE with IOL, 2 ICCE) Rhegmatogenous retinal 2 functional investigations were not always available at detachment the time of collection of the material (Table 1). Aphakic retinal detachment 1 Peripheral retinal breaks 1 MEDICAL AND OCULAR HISTORY Central retinal vein occlusion 1 Branch vein occlusion 2 A full medical history was recorded for 71 patients, Chloroquine keratopathy 1 but in most cases no relevant general disorder was Corneal leucoma (unknown 1 identified (Table 2). In one patient with hilar aetiology) lymphadenopathy the diagnosis of sarcoidosis was confirmed by biopsy and a positive Kveim test. Table 4 Oculat Sarcoidosis was specifically sought in 38 other patients symptoms at onset (81 BSCRpatients) but was negative in all. The incidence of arterial No. ofpatients hypertension in 16 patients, coronary artery disease in Blurred vision 58 5, cerebrovascular accident in 2, and retinal vein Floaters 30 obstruction in 3 was higher than one would expect in Night blindness 8 a similar age group. Distorted vision 4 With the exception of a rather high incidence of Photopsia 4 open angle glaucoma there were no apparent ocular Irritation 4
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