Case Report Acute Zonal Occult Outer Retinopathy with Atypical Findings

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1 Cse Reports in Medicine Volume 24, Article ID 29696, 7 pges Cse Report Acute Zonl Occult Outer Retinopthy with Atypicl Findings Dimitrios Krginnis, Georgios A. Kontdkis, Artemios S. Kndrkis, Nikolos Mrkomichelkis, 2 Ilis Georgls, 2 Efstrtios A. Prikkis, nd Stmtin A. Knrou 3 Ophthlmitreio Eye Hospitl of Athens, Eleftheriou Venizelou 26, 6 72 Athens, Greece 2 Deprtment of Ophthlmology, Generl Hospitl of Athens, Leoforos Mesogion 54, 5 27 Athens, Greece 3 Ophthlmology Deprtment, Red Cross Hospitl, Erythrou Stvrou, 5 26 Athens, Greece Correspondence should e ddressed to Georgios A. Kontdkis; Received 22 Jnury 24; Revised June 24; Accepted 2 June 24; Pulished 2 July 24 Acdemic Editor: Aron S. Dumont Copyright 24 Dimitrios Krginnis et l. This is n open ccess rticle distriuted under the Cretive Commons Attriution License, which permits unrestricted use, distriution, nd reproduction in ny medium, provided the originl work is properly cited. Bckground. To report cse of cute zonl occult outer retinopthy (AZOOR) with typicl electrophysiology findings. Cse Presenttion. A 23-yer-old-femle presented with visul cuity deteriortion in her right eye ccompnied y photopsi ilterlly. Corrected distnce visul cuity t presenttion ws 2/5 in the right eye nd 2/2 in the left eye. Fundus exmintion ws unremrkle. Visul field (VF) testing reveled lrge scotom. Pttern nd full-field electroretinogrms (PERG nd ERG) reveled mculr involvement ssocited with generlized retinl dysfunction. Electrooculogrm (EOG) light rise nd the Arden rtio were within norml limits ilterlly. The ptient ws dignosed with AZOOR due to clinicl findings, visul field defect, nd ERG findings. Conclusion. This is cse of AZOOR with chrcteristic VF defects nd clinicl symptoms presenting with typicl EOG findings.. Introduction Acute zonl occult outer retinopthy (AZOOR) is rre retinl disorder first descried in the literture y Gss in 992 [, 2]. Ptients with AZOOR typiclly present with photopsi nd scotoms. Fundoscopy in ptients t initil presenttion my e norml nd dignosis is confirmed with the norml findings in electrophysiology [, 2]. According to Gss, AZOOR elongs to white dot syndromes, which is wide spectrum of idiopthic inflmmtory retinl disorders [, 2]. Acute zonl occult outer retinopthy ffectsmostlywomen,withnvergegetpresenttion of ll ptients descried in the literture out 37 yers. Typicl chrcteristic of the disese is the presenttion with vision deteriortion in n re of the visul field ccompnied y photopsi. Most of the ptients present with unilterl disese, ut involvement of the contrlterl eye ultimtely ppers in the mjority of the cses ccording to the literture []. In electrophysiology, most of the ptients descried in the literture hve norml electroretinogrm (ERG) in t lest one eye (the most severely ffected in symmetric disese) [, 2]. The electrooculogrm (EOG) ws lso tested in mny of the cses descried in the literture, nd so fr ll of them hd norml results [, 3]. The purpose of our study is to descrie cse of AZOOR with typicl clinicl findings nd norml ERG tht presented with norml EOG, in contrst to the up till now pulished reports. 2. Cse Presenttion A 23-yer-old-femle ptient presented in the Outptients Deprtment of the Ophthlmitreio Eye Hospitl of Athens due to vision deteriortion in her right eye nd photopsis ilterlly. The ptient underwent complete evlution with detiled ophthlmic nd systemic history, ssessment ofcorrecteddistncevisulcuity(cdva),ndslitlmp

2 2 Cse Reports in Medicine Single field nlysis Nme: ID: Centrl 3-2 threshold test Fixtion monitor: off Fixtion trget: centrl Fixtion losses: / Flse POS errors: % Flse NEG errors: 66% Test durtion: 9:22 Stimulus: III, white Bckground: 3.5 ASB Strtegy: SITA stndrd 4 4 Pupil dimeter: Visul cuity: RX: DS DC X Eye: right DOB: Dte: Time: 4:42 Age: 23 Fove: off Pttern devition not shown for severely depressed fields. Refer to totl devition. GHT Outside norml limits VFI 24% MD 4.47 db P <.5% PSD 9.89 db P <.5% Totl devition Pttern devition Pttern devition not shown for severely depressed fields. Refer to totl devition. <%.5% Ophthlmitrio Athinon Glukom Deprtment, 26 Pnepistimiou st. Tel Crl Zeiss Meditec HFA II /4.2.2 Figure : Visul field of the ptient s right eye demonstrting generlized depression.

3 Cse Reports in Medicine 3 OD OS () () Figure 2: Opticl coherence tomogrphy of the mcul without ny norml findings. 2.5 μv/div Right eye Left eye P5 Norml N35 P5 N95 N35 N95 2 ms/div () 25 μv/div N N N 2 ms/div () 25 μv/div N 2 ms/div (c) μv/div P P P N N N 2 ms/div (d) Figure 3: () Pttern electroretinogrm (PERG) showing reduction of the P5 mplitude in the right eye. () Bright flsh drk dpted ERGs (DA.) demonstrting negtive ERG in the right eye, s -wve is of lower mplitude thn -wve. (c) Photopic single flsh ERGs (LA 3.) responses were sunorml in the right eye. (d) Light dpted 3 Hz flicker ERG (LA 3 Hz) demonstrting implicit time dely in the right eye.

4 4 Cse Reports in Medicine Right Drk (5 ) Light (5 ) Left Drk (5 ) Light (5 ) Pek Pek Trough Trough /div /div () () Figure 4: EOG recordings demonstrting norml light rise nd Arden rtio ilterlly. inspection of nterior nd posterior segments. The ptients CDVA ws 2/5 in the right eye nd 2/2 in the left eye. Introculr pressure ws 4 mmhg in the right eye nd 5 mmhg in the left eye. The ptient hd no history of other ophthlmic or systemic diseses. Anterior nd posterior segments exmintion ws unremrkle. The ptient underwent visul field testing tht reveled lrge centrl scotom in the right eye nd ws norml in the left eye (Figure ). Fluoresceinngiogrphyndindocyninegreenngiogrphy were unremrkle ilterlly. Opticl coherence tomogrphy (OCT) (Spectrlis SD-OCT, Heidelerg Engineering, Inc., Germny) lso reveled no mculr pthology (Figure 2). Additionlly, the ptient underwent PERG nd ERG electroretinogrms ccording to the protocols recommended y the Interntionl Society for Clinicl Electrophysiology of Vision (ISCEV) [4, 5]. Stimulus.6 log units stronger thn the ISCEV mximum stndrd flsh ws lso used etter to demonstrte the drk dpted -wve (DA.). Visul evoked potentils to pttern stimulus (PVEP) nd (EOG) were lso recorded ccording to ISCEV stndrds [6, 7]. According to the results, PERG P5 mplitude s n index of mculr function ws reduced in the right eye ut ws within norml limits in the left eye indicting right eye mculr dysfunction (Figure 3()). The PVEP P component ws of reduced mplitude nd norml pek time in the right eye (possily secondry to mculr dysfunction rther thn reflecting primry optic nerve disese) while responses in the left eye were recorded within norml rnge. Rod specific ERG (DA.) ws of reduced mplitude in the right eye only. Bright flsh drk dpted ERGs (DA.) were of norml -wve mplitude ilterlly, while -wve ws of reduced mplitude in the right eye nd sunorml in the left eye, indicting negtive ERG in the right eye (wve mplitudes: 32 μv nd 35μV in the right nd left eye, resp.; -wve mplitudes: 276 μv nd 345 μv in the right nd left eye, resp.) (Figure 3()). Photopic single flsh ERGs (LA 3.) responses were sunorml in the right eye only (-wve mplitudes: 25 μv nd 38.5 μv in the right nd left eye, resp.; -wve mplitudes: 5 μv nd2μv in the right nd left eye (Figure 3(c)). Lightdpted3HzflickerERG (LA3Hz) ws delyed in the right eye ut within norml limits in the left eye (implicit time of 3 msec nd 26 msec in the right nd left eyes, resp.) (Figure 3(d)). EOG light rise nd the Arden rtio were within norml limits ilterlly (Arden rtio: 4.2 nd 3.9 in the right nd left eyes, resp.) (Figure 4). The electrophysiologicl findings thus indicted generlized retinl dysfunction (ffecting oth cone nd rod systems nd inner retin) ssocited with mculr involvement in the right eye only. The ptient ws dignosed with AZOOR due to typicl clinicl, visul field nd ERG findings despite the norml EOG findings. Two months fter first visit, ptient evlution ws repeted. Vision in her right eye hd deteriorted to 2/63, whileinthelefteyeitwsstlet2/2.visulfieldtesting ws lso repeted nd reveled further depression of sensitivityintherighteyendinvolvementofthelefteye(figure 5). The ptient ws followed up for 8 dditionl months nd her symptoms nd clinicl findings were stilized ilterlly. In the course of follow-up she lso underwent mgnetic resonnce imging of hed nd domen without findings. 3. Discussion Acute zonl occult outer retinopthy is rre disese of unknown etiology ffecting mostly young femle dults []. There re only few studies nd cse reports in the literture of this condition descriing electroretinogrphy s the criticl test for confirmtion of dignosis in ddition to the clinicl findings nd the visul field defect [ 3]. To the est of our knowledge, this cse is the first descried in the literture with nontypicl electrophysiology due to the norml findings in EOG. Our cse presented with vision deteriortion in one eye nd photopsis ilterlly. Photopsis re descried s symptomtpresenttioninthevstmjorityofthecses of AZOOR []. Visul cuity is not lwys ffected, since most of eyes retin visul cuity of 2/2. Still, there re

5 Cse Reports in Medicine 5 Single field nlysis Eye: right Nme: ID: DOB: Centrl 3-2 threshold test Fixtion monitor: lind spot Fixtion trget: centrl Fixtion losses: /7 Flse POS errors: % Flse NEG errors: N/A Test durtion: 9: Stimulus: III, white Bckground: 3.5 ASB Strtegy: SITA stndrd Pupil dimeter: Visul cuity: RX: DS DC X Dte: Time: 3:7 Age: 23 Fove: off GHT Outside norml limits Totl devition Pttern devition MD PSD 2.4 db P <.5% 4.48 db P <.5% <%.5% <%.5% Humphrey Systems HFA II /3.2 () Figure 5: Continued.

6 6 Cse Reports in Medicine Single field nlysis Eye: left Nme: ID: DOB: Centrl 3-2 threshold test Fixtion monitor: off Fixtion trget: centrl Fixtion losses: / Flse POS errors: % Flse NEG errors: 6% Test durtion: 6:59 Stimulus: III, white Bckground: 3.5 ASB Strtegy: SITA stndrd Pupil dimeter: Visul cuity: RX: DS DC X Dte: Time: 3:9 Age: 23 Fove: off GHT Outside norml limits MD 7.7 db P <.5% PSD.77 db P <.5% Totl devition Pttern devition <%.5% <%.5% Humphrey Systems HFA II /3.2 () Figure 5: Visul field of right eye () nd left eye () demonstrting deteriortion of the right eye nd involvement of the left.

7 Cse Reports in Medicine 7 percentge of ptients with impired visul cuity. In series of ptients presented y Gss et l. CDVA ws 2/4 or more in 76% of ptients t presenttion nd finl visul cuity ws 2/4 or more in 68% of ptients [2]. Slit lmp iomicroscopy nd fundus imging exmintion (FA nd ICG) were unremrkle in our cse, which ccording to thelitertureiscommonconditionindignosedazoor cses, since 5% of pulished cses present with no relted findings in FA []. Opticl coherence tomogrphy (OCT) of themculwsnorml.accordingtotheliterture,oct my show disruptions in the inner segment/outer segment junction line nd in the outer segment tip lines of the photoreceptors, especilly in the lte phse of the disese [8]. Visulfielddefectislsooneoftheminfindingsincses of AZOOR. Severl types of visul field defects hve een oserved, such s lind spot enlrgement, constriction of the peripherl visul field, nd centrl scotoms []. In our cse the ptient presented with full scotom of the centrl visul field in the initilly ffected eye which ws not relted to ny other optic nerve pthology. The course of our ptient (deteriortion of the most ffected eye in the first months nd involvement of the contrlterl eye) ws lso typicl of AZOOR. Electrophysiology is the criticl testing for confirmtion of dignosis in cses of AZOOR [ 3]. Gss et l. [2]reported norml findings in ERG of ll 5 ptients in their series, nd most of the pulished cses in the literture report norml ERGs []. Common findings re depressed scotopic nd/or photopic mplitudes in one eye (the most severely ffected) or oth eyes [2]. A negtive mplitude is not common ut wslsoreportedinthelitertureinonecseygssetl. [2] ndinonecseypioetl.,wheredysfunctionofthe inner retin, s well s the outer retin, ws proposed in such cses [9]. Our ptient lso presented with negtive ERG in his right eye. Mculr involvement ws lso demonstrted in tht eye. In retrospective series of ptients Frncis et l. [3] identified pttern of electrophysiologicl nomlies in AZOOR cses where ll ffected eyes demonstrted delyed implicit time of the 3 Hz cone flicker ERG. Our cse lso displyed delyed implicit time in his right eye which is in ccordnce with these criteri. In the forementioned cse series [3] lloftheptientshdmrkedreductioninthe EOG light rise, indicting generlized dysfunction of the retinl pigment epithelium. Accordingly, cse reports in the pulished literture tht include EOG testing of the ffected eyes report norml results of EOG []. In our cse EOG light rise nd the Arden rtio were within norml limits ilterlly, despite the fct tht the rest of the test results were suggestive of AZOOR. In ll reported cses nd series of ptients in the literture there is no definite description of the disese. Even the pthophysiology of AZOOR is not cler since it is either considered prt of the white dot disese spectrum or triggered y inflmmtory disorders such s punctute inner choroiditis nd multifocl inner choroiditis [3]. Differentil dignosis of cses of AZOOR with such findings t presenttion s in our cse includes other conditions such s cncer ssocited retinopthy nd melnom ssocited retinopthy []. Our ptient hd cler medicl history nd ws referred to for complete evlution for possile occult mlignncy including clinicl exmintion, lood testing, nd chest nd dominl MRIwhichwerenotsuggestiveofsuchcondition. 4. Conclusion In conclusion, our ptients hd typicl clinicl findings of AZOORwithphotopsindvisulfielddefect.However, it represents n unusul cse of AZOOR s she presented with negtive ERG, indicting n norml inner nd outer retin dysfunction, nd with norml EOG recordings. The ltter my indicte tht involvement of retinl pigment epithelium, s demonstrted y EOG normlity, is not impertive in the course of AZOOR. Conflict of Interests The uthors declre tht there is no conflict of interests regrding the puliction of this pper. References [] D. M. Monson nd J. R. Smith, Acute zonl occult outer retinopthy, Survey of Ophthlmology, vol. 56,no., pp.23 35, 2. [2] J.D.Gss,A.Agrwl,ndI.U.Scott, Acutezonloccultouter retinopthy: long-term follow-up study, Americn Journl of Ophthlmology, vol. 34, no. 3, pp , 22. [3] P.J.Frncis,A.Mrinescu,F.W.Fitzke,A.C.Bird,ndG.E. Holder, Acute zonl occult outer retinopthy: towrds set of dignostic criteri, The British Journl of Ophthlmology, vol. 89, no., pp. 7 73, 25. [4] M. Bch, M. G. Brigell, M. Hwlin et l., ISCEV stndrd for clinicl pttern electroretinogrphy (PERG): 22 updte, Document Ophthlmologic,vol.26,no.,pp. 7,23. [5]M.F.Mrmor,A.B.Fulton,G.E.Holder,Y.Miyke,M. Brigell, nd M. Bch, ISCEV Stndrd for full-field clinicl electroretinogrphy ( updte), Document Ophthlmologic,vol.8,no.,pp.69 77,29. [6] J. V. Odom, M. Bch, M. Brigell, G. E. Holder, D. L. McCulloch, nd A. P. Tormene, ISCEV stndrd for clinicl visul evoked potentils (29 updte), Document Ophthlmologic, vol. 2,no.,pp. 9,2. [7] M. F. Mrmor, M. G. Brigell, D. L. McCulloch, C. A. Westll, nd M. Bch, ISCEV stndrd for clinicl electro-oculogrphy (2 updte), Document Ophthlmologic,vol.22,no.,pp. 7, 2. [8] T. Wkzono, S. Ooto, M. Hngi, nd N. Yoshimur, Photoreceptor outer segment normlities nd retinl sensitivity in cute zonl occult outer retinopthy, Retin, vol. 33, no. 3, pp ,23. [9] C.Pio,M.Kondo,S.Ishikw,S.Okinmi,ndH.Terski, A cse of unusul retinopthy showing fetures similr to cute zonl occult outer retinopthy ssocited with negtive electroretinogrms, Jpnese Journl of Ophthlmology, vol. 5, no., pp. 69 7, 27.

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