Ceying Shen

CL Shields

DJ Browning

GW Aylward

H Heimann

Hong Zhao

J Nemiroff

JD Gass

JH Song

JT Szelog

Ling Shao

LS Carol

Min Du

Shu Yan

SV Azad

Yibo Hu

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CASE REPORTAssessment of choroidal osteoma complicating choroidalneovascularization by optical coherence tomographyangiographyCeying Shen . Shu Yan . Min Du . Hong Zhao . Ling Shao . Yibo HuReceived: 8 October 2016 / Accepted: 23 March 2017 / Published online: 31 March 2017 The Author(s) 2017. This article is an open access publicationAbstractPurpose Choroidal osteoma (CO) frequently leadsto progressive visual loss due to complications ofsecondary choroidal neovascularization (CNV).Wereport herein the function of optical coherencetomography angiography (OCTA) in observation ofCO complicating CNV.Methods A 25-year-old female presented to ourhospital with chief complaint of sudden unilateralvisual acuity decrease for one week, with metamor-phopsia in the left eye. Her best corrected visual acuitywas 0.12 in the left eye. Then complete ophthalmo-logical examinations including fundus photography,B-scan ultrasound, fundus fluorescent angiography,and spectral-domain optical coherence tomography(SD-OCT) were performed. She was diagnosed as COon the basis of these results. But the diagnosis could notexplain the sudden visual loss and submacular hyper-reflective lesion by SD-OCT. Furthermore, she under-went OCTA and indocyanine green angiography.Results A diagnosis of classic juxtafoveal CNVsecondary to CO was made eventually. Then she wastreated with an intravitreous injection of ranibizumabtwice. The visual acuity got better and better duringthe treatment, and the efficacy was stable, giving riseto both subjective and anatomic improvement.Conclusions Optical coherence tomography angiog-raphy has the advantage of varying the segmentationand scrolling through the different retinal layers, andlayer-specific observation of blood flow in each layer.In addition, OCTA can measure the vessel area changeof CNV and provide a better appreciation of CNV,observing the efficacy more elaboratively and quan-tizedly. OCTA makes promising noninvasive identi-fication of the CO-related CNV.Keywords Choroid osteoma  Choroidalneovascularization  Optical coherence tomographyangiography  Fundus fluorescent angiographyAbbreviationsCO Choroidal osteomaCNV Choroidal neovascularizationC. Shen (&)  S. Yan  M. Du  H. Zhao L. Shao  Y. HuDepartment of Zhengzhou Second People Hospital,Ophthalmology, Zhengzhou Eye Hospital, ZhengzhouOphthalmic Institution, Zhengzhou Hanghai Middle RoadNo. 90, Zhengzhou 450000, Chinae-mail: 516045610@qq.comS. Yane-mail: yanshu8302@126.comM. Due-mail: 15890075057@163.comH. Zhaoe-mail: zhaohong120@hotmail.comL. Shaoe-mail: shao_95@163.comY. Hue-mail: yiyi019@126.com123Int Ophthalmol (2018) 38:787–792https://doi.org/10.1007/s10792-017-0503-9OCTA Optical coherence tomographyangiographyFFA Fundus fluorescent angiographySD-OCTSpectral-domain optical coherencetomographyRAPD Relative afferent pupil defectICGA Indocyanine green angiographyRPE Retinal pigment epitheliumBackgroundChoroidal osteoma (CO) is a benign choroidal tumorwhich contains mature bone [1]. It is most present inyoung healthy females in the second decade of life [2].Although most cases are asymptomatic, metamor-phopsia, blurred vision, and visual field defects may beinitial symptoms [3, 4].The prognosis of the tumor isdeteriorated by the presence of choroidal neovascu-larization (CNV). In this study, we report herein a caseof CO complicating CNV that was specifically diag-nosed by optical coherence tomography angiography(OCTA).Case presentationA 25-year-old female presented to our hospital withchief complaint of sudden unilateral visual acuitydecrease for one week, with metamorphopsia in theleft eye. Her best corrected visual acuity was 1.0 in theright eye and 0.12 in the left eye. Intraocular pressurewas 14 mmHg in the right eye and 16 mmHg in theleft eye. The grade of relative afferent pupil defect(RAPD) was 3? in the left eye. There was no otherrelevant past ocular, medical or family history. Sheunderwent complete ophthalmological examinationsincluding fundus photography, B-scan ultrasound,fundus fluorescent angiography (FFA), and spectral-domain optical coherence tomography (SD-OCT).Fundus photography showed peripapillary subretinalorange and yellow mass, without involvement offovea, but accompanied with submacular blood(Fig. 1a). B-scan ultrasound delineated a high reflec-tive echo at the posterior pole with shadowing of theretrobulbar tissues (Fig. 1b). FFA demonstrated peri-papillary mottled fluorescence, and macularhypofluorescence, without late staining indicatingthe absence of neovascularization (Fig. 1c). SD-OCTrevealed submacular hyperreflective lesion, bulge ofretinal pigment epithelium (RPE), and nasal choroidalhyperreflectivity (Fig. 1d). She was diagnosed as COon the basis of these results. But the results could notexplain the sudden visual loss and submacular hyper-reflective lesion. In addition, to further confirm, sheunderwent indocyanine green angiography (ICGA)and OCTA. ICGA showed there was a dark area at theperipapillary with faint hyperfluorescence at themacular, indicating the presence of CNV (Fig. 1e).The instrument used in the OCTA was to obtainamplitude-decorrelated angiography images (softwareversion: 2015.1.0.90; Optovue, Inc., Fremont, CA,USA). OCTA (3 mm 9 3 mm) exhibited that therewas abnormal irregular vascular signal in the outerretina layer and the choriocapillaris layer, and peri-papillary hyperreflectivity was present in the chorio-capillaris layer. The osteoma showed no flow in thechoriocapillaris layer. There was vascular morphologyin the RPE bulge on the B-scan OCTA imagescorresponding to the abnormal vascular signal in theangiography (Fig. 1f). A diagnosis of CO complicat-ing CNV was confirmed eventually. CNV detected inthe region was responsible for the subretinal fluid. Andthe flow area of CNV was 0.024 mm2 visible onOCTA. Then she was treated with an intravitreousinjection of 0.5 mg ranibizumab (Lucentis;cFig. 1 Ophthalmological examinations of a 25-year-old femalewith choroid osteoma complicating choroidal neovasculariza-tion. Fundus photography a showed peripapillary subretinalorange and yellowmass (yellow arrow), without involvement offovea, but accompanied with submacular blood. B-scanultrasound b delineated a high reflective echo at the posteriorpole with shadowing of the retrobulbar tissues. FFA c demon-strated peripapillary mottled fluorescence (yellow arrow), andmacular hypofluorescence, without late staining indicating theabsence of neovascularization. SD-OCT d revealed submacularhyperreflectivity, bulge of RPE (yellow arrowhead), and nasalchoroidal hyperreflectivity (yellow arrow). ICGA e showedthere was a dark area at the peripapillary with faint hyperflu-orescence at the macular (yellow arrowhead). OCTA images of3 mm 9 3 mm, f exhibited that there was abnormal irregularvascular signal in the outer retina layer and the choriocapillarislayer (yellow arrowhead), and peripapillary hyperreflectivitywas present in the choriocapillaris layer. The osteoma showedno flow in the choriocapillaris layer. There was vascularmorphology in the RPE bulge on the B-scan OCTA imagescorresponding to the abnormal vascular signal in theangiography788 Int Ophthalmol (2018) 38:787–792123Genentech, South San Francisco, CA). The risks andbenefits of the intravitreal injection of ranibizumabwere explained to the patient, and written consent wasobtained.Two weeks after intravitreous injection, she cameback to recheck, and the visual acuity got improved.Her best corrected visual acuity increased to 0.3, witha reduction of metamorphopsia in the left eye. SD-Int Ophthalmol (2018) 38:787–792 789123OCT revealed that submacular hyperreflectivitydecreased, bulge of RPE disappeared, but nasalchoroidal hyperreflectivity sustained. Abnormal vas-cular signal was absent in the outer retina layer and thechoriocapillaris layer by using OCTA. This patientwas sensitive to ranibizumab. The surgery yieldedfavorable outcomes. One month after operation, herbest corrected visual acuity was 0.3 in the left eye. SD-OCT revealed the absence of submacular hyperreflec-tivity and discontinuous ellipsoid zone and interdig-itation zone, without bulge of RPE. OCTA showedthat abnormal vascular signal was absent in the outerretina layer and the choriocapillaris layer. She wasthen performed the second intravitreous injection of0.5 mg ranibizumab. One month after the secondinjection, the patient complained of an evident reduc-tion of metamorphopsia and the best corrected visualacuity increased to 0.4 in the left eye. SD-OCTrevealed that discontinuous ellipsoid zone andinterdigitation zone were in regression, without bulgeof RPE. Abnormal vascular signal was not detected byOCTA. The visual acuity got better and better duringthe treatment, and the efficacy was stable, giving riseto both subjective and anatomic improvement (Fig. 2).DiscussionIn 1978 Gass et al. first described the disease of CO infour healthy young women with characteristic oph-thalmoscopy appearance of a slightly elevated, juxta-papillary, yellowish orange, choroidal lesion withwell-defined margins [5]. CO frequently causes pro-gressive visual decline as a result of pigment epithelialdecompensation and atrophy of the fovea, or as a resultof complications of secondary CNV [6]. CNV devel-ops in 47% of CO patients by 10 years and 56% by20 years, and it is a major cause of visual impairmentFig. 2 Optical coherence tomography angiography and SD-OCT for the patient during the follow-up. Two weeks afterintravitreous injection, abnormal vascular signal was absent inthe outer retina layer and the choriocapillaris layer (yellowarrowhead) by using OCTA. a SD-OCT, d revealed thatsubmacular hyperreflectivity decreased, bulge of RPE disap-peared (yellow arrowhead), but nasal choroidal hyperreflectiv-ity sustained. One month after operation, OCTA showed thatabnormal vascular signal was absent in the outer retina layer andthe choriocapillaris layer (yellow arrowhead). b SD-OCT,e revealed the absence of submacular hyperreflectivity, anddiscontinuous ellipsoid zone and interdigitation zone, withoutbulge of RPE. One month after the second injection, abnormalvascular signal was not detected (yellow arrowhead) by OCTA.c SD-OCT, f revealed that discontinuous ellipsoid zone andinterdigitation zone were in regression, without bulge of RPE(yellow arrowhead)790 Int Ophthalmol (2018) 38:787–792123[2]. When serous retinal detachment is complicated byCO, it is a recognized association that triggers a searchfor possible underlying subretinal neovascularization[3]. If it was not detected by FFA or OCT, ICGAshould be performed to exclude the subretinal neo-vascularization. Now we have had a new noninvasivediagnosis examination to preclude CNV, which isOCTA.Optical coherence tomography angiography pro-vides a high-resolution image of the microvasculatureof the retina at various levels in the retina and choroidsuch as superficial capillary plexus, deep capillaryplexus, outer retina, and choriocapillaris in the para-and peri-foveal areas [7, 8]. The advantage of varyingthe segmentation and scrolling through the differentretinal layers, and layer-specific observation of bloodflow in each layer makes OCTA widely used inclinical. In our patient, the secondary CNV was notdetected during the follow-up. So the change of vesselarea was not observed. We set the layer to thechoriocapillaris level and then operated the moving ofthe scan line to detect CNV rigorously. CNV wasdetected with OCTA in areas where it was unde-tectable on the FFA due to the diffuse hyperfluores-cence. Of note, there have been publicationsdescribing CNV secondary to CO detected by OCTA[9, 10]. In our study, the CNV showed regression oflesion size after treatment, same as Szelog et al.reported [9]. Otherwise, we measured the vessel areachange of CNV, observing the efficacy more elabo-ratively and quantizedly. Above all, the detection ofCNV secondary to CO is facilitated with OCTA, as theneovascularization could be covered by osteoma indye-based angiography.The common choroidal tumors misdiagnosed asCO include choroidal hemangiomas, choroidal mela-noma, and choroidal metastases. Choroidal haeman-gioma is a benign vascular tumor, orange in color andclassically located in the macular or paramacularregion of the eye [11]. Associated clinical features areexudative retinal detachment, macular edema, pig-mentary changes within the RPE, subretinal fibrosisover the surface of the lesion, and orange pigment[12]. Ultrasonography helps to differentiate choroidalhemangiomas from other choroidal tumors [12].Choroidal melanoma is suggestive of melanoma suchas greater tumor thickness, the presence of subretinalfluid and overlying orange pigment, tumor marginnear the optic disk, and the absence of drusen and halo[13]. OCT generally shows gentle domed shaped,smooth surface topography with relatively freshsubretinal fluid demonstrating shaggy photoreceptors[14]. Choroidal metastases appear as a yellow massdeep to the retina, often with overlying subretinal fluid[15].ConclusionsIn the present study, we demonstrate OCTA findingsin patients with CO complicating CNV. It is necessaryto perform noninvasive examinations during thefollow-up to observe the apostasis, replacing the useof FFA or ICGA. Meanwhile, OCTA can detect thearea change of CNV and provide a better appreciationof CNV. OCTAmay serve as a marker for patients at arisk for CNV secondary to CO. And it makespromising noninvasive identification of the CO-re-lated CNV. Further study with more number ofpatients is still warranted.Availability of data and materials All the data supportingour findings are contained within the manuscript.Compliance with ethical standardsConflict of interest The authors declare that there is no con-flict of interest regarding the publication of this paper.Ethics approval and consent to participate The study par-ticipant gave oral informed consent for OCTA imaging andsigned informed consent for FFA/ICGA imaging. The study wasapproved by the Institutional Review Board of ZhengzhouSecond People Hospital and carried out in accordance with thetenets of the Declaration of Helsinki.Open Access This article is distributed under the terms of theCreative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unre-stricted use, distribution, and reproduction in any medium,provided you give appropriate credit to the originalauthor(s) and the source, provide a link to the Creative Com-mons license, and indicate if changes were made.References1. Song JH, Bae JH, Rho MI, Lee SC (2010) Intravitrealbevacizumab in the management of subretinal fluid asso-ciated with choroidal osteoma. Retina 30:945–9512. Aylward GW, Chang TS, Pautler SE, Gass JD (1998) Along-term follow-up of choroidal osteoma. Arch Ophthal-mol 116:1337–1341Int Ophthalmol (2018) 38:787–792 7911233. Shields CL, Shields JA, Augsburger JJ (1988) Choroidalosteoma. Surv Ophthalmol 33:17–274. 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Assessment of choroidal osteoma complicating choroidal neovascularization by optical coherence tomography angiography

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Assessment of choroidal osteoma complicating choroidal neovascularization by optical coherence tomography angiography

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