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Journal Français d'Ophtalmologie
Volume 41, n° 1
pages e35-e38 (janvier 2018)
Doi : 10.1016/j.jfo.2017.03.025
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Four-year SD-OCT follow-up of a treated bilateral choroidal osteoma complicated by choroidal neovascularisation
Suivi en SD-OCT pendant 4 ans d’un ostéome choroïdien traité compliqué de néovascularisation choroïdienne
 

M. Bouladi , R. Bouraoui, N. Zerei, L. El Matri, F. Nafaa
 Service B, institut Hédi Rais d’ophtalmologie, faculté de médecine, université de Tunis El Manar, Tunis, Tunisia 

Corresponding author.
Introduction

Choroidal osteoma is a benign tumour of the choroid composed of mature bone. It was first defined by Gass et al. in 1978 [1]. Young healthy women are typically diagnosed with choroidal osteoma. However, it can occur in all ages and the juxtapapillary area is often concerned by this tumour with variable extension to the macula [2, 3]. Choroidal neovascularisation (CNV) occurs in 31% of cases by 5 years and 46 to 56% by 20 years, leading to poor visual acuity [3]. The treatment is not well-established but intravitreal injection of antivascular endothelial growth factor (anti-VEGF) seems to represent an effective therapy option [4, 5].

We report the 4-year follow-up on SD-OCT of a case of bilateral choroidal macular osteoma in a 52 year-old woman associated with bilateral CNV and treated with intravitreal bevacizumab.

Case report

A 52 year-old woman presented with the complaints of decreased vision in the left eye for seven days. Her past ocular, medical and family histories were unremarkable. On examination, best corrected visual acuity of 20/200 in the right eye and 20/30 in the left eye. The anterior segment examination was normal. The fundus examination revealed, in the right eye, large elevated yellow-white lesion located in the macula, of approximately six disc diameters with distinct geographic borders associated with subfoveal choroidal neovascularisation surrounded by subretinal pigmentation and haemorrhage (Figure 1a). The left eye showed a same lesion of about four disc diameters without haemorrhage (Figure 1b). The diagnosis of bilateral choroidal osteoma complicated by choroidal neovascularisation of the right eye was made and confirmed by ultrasound B-Scan of both eyes which revealed focal subretinal calcification as a high reflective echo with shadowing at the posterior pole of both eyes (Figure 2).



Figure 1


Figure 1. 

a: evolution of the right eye based on fundus photography and spectral domain optical coherence tomography showing the course of choroidal neovascularisation, the decrease of choroidal thickness and the growth of the tumor (A) at baseline, choroidal neovascularisation (white arrow) and subretinal fluid (B) at 4 months after 4 intravitreal bevacizumab injection (C) at 3 years, absence of serous retinal detachment, growth of the tumor in temporal (white arrows) and decrease in choroidal thickness; b: evolution of the left eye based on fundus photography and spectral domain optical coherence tomography (A) calcified osteoma (B) serous retinal detachment (C) central decalcification with posterior hyperreflectivity (white arrow) (D) choroidal neovascularisation (E) recurrence of CNV (F) successful bevacizumab treatment.

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Figure 2


Figure 2. 

Ultrasound B-scan of both eyes revealed focal subretinal calcification as a high reflective echo with shadowing at the posterior pole of both eyes.

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Spectral domain optical coherence tomography (SD-OCT) of the right eye showed serous retinal detachment, central retinal thickness was 340μm and tumour thickness was 586μm. SD-OCT of the left eye showed tumour thickness of 680μm without serous retinal detachment.

She was treated with 1.25mg/0.05mL intravitreal injections of bevacizumab (Avastin; Genentech, South San Francisco, CA, USA). Retreatment was performed every month if persistence of subretinal fluid determined by SD-OCT.

A total of 4 injections were performed in the right eye leading to improvement of visual acuity 20/80 and disappearance of serous retinal detachment on SD-OCT (Figure 1a). However, little subfoveal serous retinal detachment appeared in the left eye for which we indicate observation (Figure 1b). Twenty-four months later, she returned with blurred vision in her left eye, visual acuity decreased to 20/50 and fundus examination showed tumour growth along the margins with subfoveal haemorrhage, SD-OCT demonstrated subfoveal decalcification and serous retinal detachment, which progressed rapidly to subfoveal choroidal neovascularisation. She also underwent monthly intravitreal bevacizumab with a total of 8 injections (Figure 1b).

At 4 years’ follow-up, visual acuity was maintained to 20/80 in the right eye, fundus photography showed further tumour growth and subfoveal atrophy with a decrease of choroidal osteoma thickness to 380μm. In the left eye, visual acuity was 20/200, choroidal osteoma thickness was still at 680μm with presence of intraretinal fluid (Figure 3).



Figure 3


Figure 3. 

Fundus photography and spectral domain optical coherence tomography at four years showing in the right eye (A) tumor growth and subfoveal atrophy, SD-OCT showed no serous retinal detachment, the retinal structure of foveal area was conserved with decrease in choroidal osteoma thickness to 380μm. In the left eye (B), choroidal osteoma thickness was 680μm with presence of intraretinal fluid.

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Discussion

Choroidal osteoma is a rare condition. It is often unilateral (80%), and occurs mainly in young females [2]. Bilateral choroidal osteoma is rarely seen in 20% of patients. Natural course over 10 years was studied by Shields and al. in 74 eyes. It showed tumour growth in 51% of eyes, tumour decalcification in 46% of eyes and choroidal neovascularisation in 31% of eyes leading to visual acuity loss in 45%, and poor visual acuity in 56% [3].

The strongest predictive factors for CNV associated with choroidal osteoma are overlying haemorrhage and irregular surface [3]. Moreover, CNV were found within the decalcified portion of the osteoma in 66% of cases [3, 4], as was the case for our patient. The association between CNV and decalcification had been explained by a secondary disruption of the retinal pigment epithelium allowing growth of underlying choroidal new vessels [3]. Recently, Yoshikawa and Takahashi reported that CNV may be also implicated in subretinal pigmentation [4] and indeed, we noted this association in both eyes of our patient. Choroidal neovascularisation was bilateral in our patient and the delay for development of CNV between the two eyes was 24 months.

We also noticed a growth of the tumour over 2 years along calcified margins after the onset of the decalcification. The decalcification generally occurs gradually over many months, it may be with a decrease in tumour thickness and leads to a bed of atrophy [3]. The absence of retinal pigment epithelium changes overlying the tumour was the only predictive factor of tumour growth [3].

Many treatments have been proposed for choroidal osteoma associated with choroidal neovascularisation including laser photocoagulation, surgical removal of CNV, transpupillary thermotherapy, and photodynamic therapy (PDT) with limited success. Few studies demonstrates efficacy of intravitreal bevacizumab [4].

Bevacizumab treatment was successful in treating CNV in our patient. Recently, Yoshikawa and Takahashi reported also bevacizumab efficacy at long-term in 3 patients, their initial visual acuity varied from 0.7 to 0.9 and improved over a mean follow-up of 56.0±38.4 months to 0.1 to 0.7 [4].

Our patient received a total of 4 injections in the right eye and 8 injections in the left eye, which was important compared to a reported number of injections varying from 1 to 3 with a mean of 1.8 injections at a mean follow-up of 56 months [4]. However, Khan et al. reported in 8 treated eyes a number of 12.3 injections at a mean of 32 months follow-up [5].

The thickness of the choroidal osteoma was measured with SD-OCT in this case and showed that tumour thickness decreased from 580μm to 380μm at a mean of 48 months of follow-up. Yoshikawa and Takahashi reported that mean tumour thickness decreased from 709μm to 608μm at a mean of 41.0 months of follow-up using ultrasonography and EDI-OCT [4].

Conclusion

This case illustrates the SD-OCT evolution over 4 years, of a bilateral macular choroidal osteoma showing decrease of choroidal osteoma thickness over time, sequential occurrence of choroidal neovascularisation after onset of decalcification and successful treatment of CNV with bevacizumab which seems to represent an effective therapy option.

Disclosure of interest

The authors declare that they have no competing interest.

References

Gass J.D., Guerry R.K., Jack R.L., Harris G. Choroidal osteoma Arch Ophthalmol 1978 ;  96 : 428-435 [cross-ref]
Shields C.L., Sun H., Demirci H., Shields J.A. Factors predictive of tumor growth, tumor decalcification, choroidal neovascularization, and visual outcome in 74 eyes with choroidal osteoma Arch Ophthalmol 2005 ;  123 : 1658-1666 [cross-ref]
Shields C.L., Shields J.A., Augsburger J.J. Choroidal osteoma Surv Ophthalmol 1988 ;  33 : 17-27 [cross-ref]
Yoshikawa T., Takahashi K. Long-term outcomes of intravitreal injection of bevacizumab for choroidal neovascularization associated with choroidal osteoma Clin Ophthalmol 2015 ;  9 : 429-437 [cross-ref]
Khan M.A., De Croos F.C., Storey P.P., Shields J.A., Garg S.J., Shields C.L. Outcomes of anti-vascular endothelial growth factor therapy in the management of choroidal neovascularization associated with choroidal osteoma Retina 2014 ;  34 : 1750-1756 [cross-ref]



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