Purpose: To report a case of choroidal osteoma associated with reactivation of choroidal neovascularization (CNV) and development of focal choroidal excavation (FCE).
Case Report: A 34-year-old woman with choroidal osteoma complicated by CNV in the right eye for two years presented with deterioration of visual acuity in her right eye. A small retinal hemorrhage accompanied by subretinal fluid (SRF) was seen in the macular area of the right eye. Optical coherence tomography (OCT) showed that the inner retina was intact, and the outer retinal layers had outward displacement. SRF and a wedge-shaped choroidal depression were also seen. This choroidal excavation was not present on previous OCT images. The integrity of the inner retinal layers was maintained, and an optically clear space was present between the neurosensory retina and the retinal pigment epithelium.
Conclusion: Choroidal osteoma can be complicated by CNV and FCE could occur as a consequence. Again, FCE can lead to CNV development. This cascade can deteriorate vision and sometime lead to permanent visual loss

Azimizadeh, Mahdieh

Babaei, Esmaeil

Maryam Hosseini, Seyedeh

English

Azimizadeh, M. (Mahdieh)

Hosseini, S. M. (Seyedeh)

Babaei, E. (Esmaeil)

Neliti

Focal Choroidal Excavation in A Case of Choroidal Osteoma Associated with Choroidal Neovascularization

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Focal Choroidal Excavation in a Case of Choroidal Osteoma Associated with Choroidal Neovascularization

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Case ReportFocal Choroidal Excavation in a Case of ChoroidalOsteoma Associated with ChoroidalNeovascularizationMahdieh Azimizadeh1; MD; Seyedeh Maryam Hosseini2, MD; Esmaeil Babaei1,3, MD1Eye Research Center, Mashhad University of Medical Sciences, Mashhad, Iran2Retina Research Center, Mashhad University of Medical Sciences, Mashhad, Iran3Eye Research Center, Yazd University of Medical Sciences, Yazd, IranORCID:Mahdieh Azimizadeh: https://orcid.org/0000-0001-9467-4249Esmaeil Babaei: https://orcid.org/0000-0001-8490-3589AbstractPurpose: To report a case of choroidal osteoma associated with reactivation of choroidalneovascularization (CNV) and development of focal choroidal excavation (FCE).Case Report: A 34-year-old woman with choroidal osteoma complicated by CNV in theright eye for two years presented with deterioration of visual acuity in her right eye. Asmall retinal hemorrhage accompanied by subretinal fluid (SRF) was seen in the maculararea of the right eye. Optical coherence tomography (OCT) showed that the inner retinawas intact, and the outer retinal layers had outward displacement. SRF and a wedge-shaped choroidal depression were also seen. This choroidal excavation was not presenton previous OCT images. The integrity of the inner retinal layers was maintained, andan optically clear space was present between the neurosensory retina and the retinalpigment epithelium.Conclusion: Choroidal osteoma can be complicated by CNV and FCE could occur as aconsequence. Again, FCE can lead to CNV development. This cascade can deterioratevision and sometime lead to permanent visual loss.Keywords: Bevacizumab; Choroidal Excavation; Choroidal Neovascularization; ChoroidalOsteoma; MultifocalJ Ophthalmic Vis Res 2020; 15 (3): 419–423Correspondence to:Esmaeil Babaei, MD. Retina Research Center, KhatamAl Anbia Eye Hospital, Mashhad, Aboutaleb Junction,Gharanei BLVD, Mashahad University of MedicalSciences, Mashhad 917789, Iran.Email: esmaeil.babaei@yahoo.comReceived: 10-12-2018 Accepted: 29-02-2020Access this article onlineWebsite: https://knepublishing.com/index.php/JOVRDOI: 10.18502/jovr.v15i3.7461INTRODUCTIONChoroidal osteoma is a rare, ossifying, benigntumor of the choroid. It occurs predominantly inThis is an open access journal, and articles are distributed under the terms ofthe Creative Commons Attribution-NonCommercial-ShareAlike 4.0 License, whichallows others to remix, tweak, and build upon the work non-commercially, aslong as appropriate credit is given and the new creations are licensed under theidentical terms.How to cite this article: Azimizadeh M, Hosseini SM, Babaei E. FocalChoroidal Excavation in a Case of Choroidal Osteoma Associated withChoroidal Neovascularization. J Ophthalmic Vis Res 2020;15:419–423.© 2020 JOURNAL OF OPHTHALMIC AND VISION RESEARCH | PUBLISHED BY KNOWLEDGE E 419Choroidal Osteoma with Excavation; Azimizadeh et alFigure 1. Color fundus photograph of the right eye.young healthy women in the second and thirddecades of life and is unilateral in 80% of cases.[1]Although it has been recognized as a benigntumor, it can grow in 51% of patients by 10years and can be complicated by enlargement,decalcification, and choroidal neovascularization(CNV).[2]Focal choroidal excavation (FCE) refers to focaldepression of the choroid that has been recentlydescribed thanks to the development of spectral-domain optical coherence tomography (SD-OCT).It may be solitary or occur in association with otherchorioretinal disorders such as central serouschorioretinopathy (CSC), CNV, and polypoidalchoroidal vasculopathy (PCV).[3] It has alsobeen reported in eyes with choroidal osteomaassociated with CNV.[4, 5]Here, we report an interesting case involvinga 34-year-old woman with bilateral choroidalosteoma complicated by CNV in the right eye, withdevelopment of FCE following reactivation of theCNV.CASE REPORTA 34-year-old healthy woman with bilateralchoroidal osteoma complicated by CNV in theright eye for the two years presented with acutedeterioration of visual acuity (VA) in the right eye.The findings of multimodal imaging for this casewere reported and published in 2015, and therewas no FCE in the right eye at that time.[6]Best corrected visual acuity (BCVA) of the righteye had decreased from 20/40 to 20/100, whilethat of the left eye was 20/20. Fundus examinationof both eyes showed a yellow–orange lesion withsharp borders at the level of the choroid (Figure1). A small retinal hemorrhage accompanied bysubretinal fluid (SRF) was seen in the maculararea of the right eye. Enhanced depth imagingOCT (EDI-OCT) (Spectralis HRA + OCT, HeidelbergEngineering, Heidelberg, Germany) showed thatthe inner retina was intact, although the outerretinal layers had outward displacement dueto active CNV along with SRF and a wedge-shaped choroidal depression in the right eye.The choroidal excavation involved the retinalpigment epithelium (RPE) and outer retinal layersup to the outer plexiform layer. The integrity ofthe inner retinal layers was maintained, and anoptically clear space was present between theneurosensory retina and RPE. EDI-OCT showedthinning of the choriocapillaris and large choroidalvessels and a sponge-like choroidal lesion (Figure420 JOURNAL OF OPHTHALMIC AND VISION RESEARCH VOLUME 15, ISSUE 3, JULY-SEPTEMBER 2020Choroidal Osteoma with Excavation; Azimizadeh et alFigure 2. SD- OCT of the right eye: (A) Spectral-domain OCT (SD-OCT) of the right eye performed in 2014 revealed subretinalfluid (SRF) associated with choroidal osteoma complicated by choroidal neovascularization (CNV). (B) OCT of the right eye (2016)clearly shows focal choroidal excavation and SRF due to CNV development. (C) OCT of the right eye clearly shows FCE andcomplete resolution of SRF after two consecutive intravitreal bevacizumab injections.2). Fluorescein angiography (FAG) demonstratedmild hyperfluorescence in the choroidal osteomadue to window defect, as well as juxtafovealhyperfluorescence corresponding to active CNVin the right eye (Figure 3), which was betterdelineated on OCT angiography (Figure 4).Thus, the diagnosis was reactivation of CNVassociated with the development of FCE inthe right eye in a known case of choroidalosteoma. The patient received two consecutiveintravitreal bevacizumab (Avastin, Genentech Inc.,San Francisco, CA) injections, which resulted inimprovement of BCVA to 20/40. OCT showeddisappearance of SRF and resolution of thedisturbances in the outer retinal layers. FCE wasmore clearly visible and associated with severechoroidal thinning after quiescence of the CNV(Figure 2c). BCVA was maintained in both eyesduring a follow-up period of one year.DISCUSSIONWe report development of FCE followingreactivation of CNV after two years of inactivity inan eye with choroidal osteoma.Initially, FCEwas defined as choroidal excavationin the submacular area without posteriorstaphyloma, any positive history of trauma,or any chorioretinal disease; however, somechorioretinal disorders such as CSC, PCV, CNV,and choroidal osteoma have been reported inassociation with FCE.[7] Although the inner retina iscommonly intact in eyes with FCE, the outer retinalJOURNAL OF OPHTHALMIC AND VISION RESEARCH VOLUME 15, ISSUE 3, JULY-SEPTEMBER 2020 421Choroidal Osteoma with Excavation; Azimizadeh et alFigure 3. Fluorescein angiography (FAG) showing choroidal neovascularization in the right eye.Figure 4. OCTA images of the right eyes illustrate high-flow vessels above the RPE in the outer retina as well as in thechoriocapillaris.layers, RPE, choriocapillaris, and choroid can beinvolved.[8]FCE can be detected by SD-OCT. However, incases with bilateral involvement or multiple lesionsin one eye, foveal SD-OCT which is confinedto the posterior pole may not detect all lesions.Therefore, multiple imaging modalities such asfundus photography, fundus autofluorescence,indocyanine green angiography, and FAG areconsidered helpful.[9]In the present case, FCE developed adjacent toa previous CNV scar and the decalcified part of thetumor as seen on previous OCT images, and it wasdetected two years after CNV occurrence. Thus,we can conclude that FCE was a consequenceof the CNV scar and/or tumor decalcification that422 JOURNAL OF OPHTHALMIC AND VISION RESEARCH VOLUME 15, ISSUE 3, JULY-SEPTEMBER 2020Choroidal Osteoma with Excavation; Azimizadeh et alresulted in tissue loss, contraction of fibrosis, andoutward displacement and excavation of the outerretina and choroid. On the other hand, FCE mayhave been involved in reactivation of CNV,[4, 5]as choroidal excavation has been reported to beassociated with the development or reactivation ofCNV.[10] There are multiple options for treating CNVin eyes with choroidal osteoma,[11–14] although thebest response has been achieved by intravitrealinjection of anti-vascular endothelial growth factoragents such as bevacizumab.[15]This is a documented development of FCEin an eye with CNV associated with choroidalosteoma. FCE could occur as a consequence ofCNV, tumor decalcification, or both. Close follow-up and prompt treatment are necessary to achievethe best results.Financial Support and SponsorshipNil.Conflicts of InterestThere are no conflicts of interest.REFERENCES1. Erol MK, Coban DT, Ceran BB, Bulut M. Enhanceddepth imaging optical coherence tomography and fundusautofluorescence findings in bilateral choroidal osteoma:a case report. Arq Bras Oftalmol 2013; 76:189-91.2. Shields CL, Perez B, Materin MA, Mehta S, Shields JA.Optical coherence tomography of choroidal osteoma in22 cases: evidence for photoreceptor atrophy over thedecalcified portion of the tumor. Ophthalmology 2007;114:e53-83. Cebeci Z, Bayraktar Ş, Oray M, K𝚤r N. Focal choroidalexcavation. Turk J Ophthalmol 2016; 46:296.4. Pierro L, Marchese A, Gagliardi M, Introini U, ParodiMB, Casalino G, et al. Choroidal excavation in choroidalosteoma complicated by choroidal neovascularization.Eye (Lond). 2017;31:1740-1743.5. Wakabayashi Y, Nishimura A, Higashide T, Ijiri S, SugiyamaK. Unilateral choroidal excavation in the macula detectedby spectral-domain optical coherence tomography. ActaOphthalmol 2010;88:e87-91.6. MirNaghi M, Nasser S, SeyedehMaryam H, Ali ST.Bilateral multifocal choroidal osteoma with cho-roidal neovascularization. Case Rep Ophthalmol Med2015;2015:346415.7. Navarro VC, Hernández JM, Palop CN, Fort PP, TauletEC. Focal choroidal excavation: Clinical findings andcomplications. Arch Soc Esp Oftalmol 2016;91:34-9.8. Liu GH, Lin B, Sun XQ, He ZF, Li JR, Zhou R, et al. Focalchoroidal excavation: a preliminary interpreta-tion basedon clinic and review. Int J Ophthalmol 2015;8:513-21.9. Chung H, Byeon SH, Freund KB. Focal choroidalexcavation and its association with pachychoroidspectrum disorders. Retina 2017;37:199-221.10. Xu H, Zeng F, Shi D, Sun X, Chen X, Bai Y. Focal choroidalexcavation complicated by choroidal neovascularization.Ophthalmology 2014;121:246-250.11. Mansour AM, Arevalo JF, Al Kahtani E, ZegarraH, Abboud E, Anand R, et al. Role of intravitrealantivascular endothelial growth factor injections forchoroidal neovascularization due to choroidal osteoma. JOphthalmol 2014;2014:210458.12. Shields CL, Sun H, Demirci H, Shields JA. Factorspredictive of tumor growth, tumor decalcification,choroidal neovascularization, and visual outcome in74 eyes with choroidal osteoma. Arch Ophthalmol2005;123:1658-66.13. Browning D. J, Choroidal osteoma: observations from acommunity setting, Ophthalmology 2003;110:1327-34.14. Shields CL, Perez B, Materin MA, Mehta S, Shields JA.Optical coherence tomography of choroidal osteomain 22 cases: evidence for photoreceptor atrophy overthe decalcified portion of the tumor. Ophthalmology2007;114:e53-8.15. Ahmadieh H, Vafi N. Dramatic response of choroidalneovascularization associated with choroidal osteoma tothe intravitreal injection of bevacizumab (Avastin). GraefesArch Clin Exp Ophthalmol 2007;245:1731-3.JOURNAL OF OPHTHALMIC AND VISION RESEARCH VOLUME 15, ISSUE 3, JULY-SEPTEMBER 2020 423

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Focal Choroidal Excavation in a Case of Choroidal Osteoma Associated with Choroidal Neovascularization

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