Bilateral choroidal osteoma: long-term follow-up of secondary choroidal neovascularization in a child using antiangiogenic therapy.

Choroidal osteoma is a rare condition, and its treatment is not well established, especially in the pediatric population, where use of antiangiogenics for choroidal neovascularization is poorly studied. Few studies have reported the long-term follow-up of pediatric patients with bilateral choroidal osteomas. We report the case of a girl who was diagnosed at the age of 3, with the appearance of bilateral secondary choroidal neovascularization, and has been under strict observation for 12 years. The effectiveness of antiangiogenic agents as a long-term therapeutic option for secondary choroidal neovascularization in pediatric patients with symptomatic choroidal osteomas is discussed.

Predicting Visual Outcomes in Choroidal Osteoma Treated with Anti-vascular Endothelial Growth Factor.

PURPOSE: To determine potential factors associated with the long-term visual prognoses in patients with choroidal osteoma undergoing anti-vascular endothelial growth factor (VEGF) treatment. DESIGN: Retrospective case series. METHODS: Patients diagnosed at tertiary high-volume hospitals between January 2000 and December 2020 were evaluated. The primary outcome measure was visual acuity at 5-year follow-up. The secondary outcome measures included factors associated with favorable vision, defined as loss of <1 line and >20/200 vision. RESULTS: Of 38 eyes from 36 patients (22 female, 61%; mean age 38 years) with choroidal osteoma, 23 eyes (61%) received anti-VEGF treatment (bevacizumab 1.25mg/0.05 cc, monthly or treat-and-extend) and 65% completed the 5 years of follow-up. All treated eyes had associated chorioretinal comorbidities (subretinal fluid 91%; choroidal neovascularization 74%; subretinal hemorrhage 30%). Although there was significant vision loss by 5 years (P = .002), 12 eyes (44%) had favorable outcomes. Only tumor thickness was significantly associated with unfavorable visual outcomes (OR 917.1, 95% CI 1.0-5687.7; P = .049). The optimal cut-off point predictive of visual outcomes was 1.4 mm, and tumor thickness ≥ 1.4 mm was associated with unfavorable vision (OR 27.0, 95% CI 2.0-368.4; P = .013). CONCLUSIONS: Among patients with choroidal osteoma undergoing anti-VEGF therapy, a particular patient subset presented with divergent outcomes with very poor vision. Tumor thickness appeared to be the differentiating factor and is thus a potential prognostic indicator for long-term visual prognoses.

DEVELOPMENT OF CHOROIDAL NEOVASCULARIZATION AFTER TREATMENT WITH PHOTODYNAMIC THERAPY IN A 5-YEAR-OLD FEMALE WITH CHOROIDAL OSTEOMA.

PURPOSE: To describe a patient with a choroidal osteoma treated with photodynamic therapy to prevent tumor growth in whom choroidal neovascularization (CNV) developed after being treated with photodynamic therapy. METHODS: Case report. RESULTS: A 5-year-old Hispanic woman presented with an asymptomatic choroidal osteoma, temporal to the macula of her right eye. According to the patient's mother, her medical, surgical, and family history was unremarkable. At examination, best-corrected visual acuity was 20/30 in both eyes. After 11 months of follow-up, signs of tumor growth toward the fovea without any signs of CNV was noted. Photodynamic therapy was performed to prevent invasion of the foveola. Two months thereafter, the patient developed CNV in the macula region in the right eye, decreasing visual acuity to 20/200. The patient was treated with four total intravitreal injections of 1.25 mg of bevacizumab over 24 weeks, which resulted in inactivation of the CNV and improved visual acuity to 20/20. Choroidal neovascularization had been never reported in her past history and her follow-up visits over 7 years. In addition, no evidence of recurrent neovascular activity or tumor growth was reported. CONCLUSION: Choroidal osteoma is a benign tumor that can result in vision-threatening complications, caused by tumor growth and tumor decalcification. Photodynamic therapy is an effective modality in inducing choroidal osteoma decalcification and stabilization; however, CNV due to reperfusion following photodynamic therapy can be seen in the retina.

Photodynamic therapy for macular choroidal osteoma.

PURPOSE: To report a case of macular choroidal osteoma treated with photodynamic therapy. OBSERVATIONS: A 34-years old woman with decreased visual acuity in her left eye came to our observation for assessment of an amelanotic choroidal tumor in the left eye. On the basis of ophthalmoscopic and echographic features the tumor was diagnosed as choroidal osteoma. Imaging examination revealed subretinal fluid involving the foveal area associated with alterations of outer neuroepithelial layers and photoreceptors without evidence of choroidal neovascularization. Foveal sparing standard fluence rate photodynamic therapy was performed. After treatment, subretinal fluid reabsorption and visual acuity recovery was noted with progressive restoration of foveal architecture. Due to the relapse of fluid and visual impairment, 1 year after treatment, a second PDT session was made using the same parameters and protocol of treatment. Despite a complete subretinal fluid reabsorption and visual acuity recovery the second treatment was complicated by the development of subretinal fibrosis. CONCLUSIONS: PDT is effective to induce subretinal fluid reabsorption and visual recovery in choroidal osteoma located in the macular area. However, the risk of possible complications related to the treatment have to be considered.

Complications, treatments, and visual prognosis of choroidal osteomas.

PURPOSE: This study aims to report complications, treatments, and visual prognosis of choroidal osteoma. METHODS: We retrospectively reviewed electronic medical records and multimodal images of 41 patients with choroidal osteoma. RESULTS: Visually significant complications included choroidal neovascularization (CNV) in 21 (47.7%) eyes and subretinal fluid (SRF) without CNV in 14 (31.8%) eyes. The most common treatment was intravitreal anti-vascular endothelial growth factor (VEGF) injection: 13 (61.9%) eyes with CNV received an average of 6.3 injections, and 6 (42.9%) eyes with SRF but without CNV received 1.8 injections. As the first-line treatment, intravitreal anti-VEGF injection induced complete or partial remission in 93.4% of eyes with CNV and 57.1% of eyes with SRF. The probability of legally low vision estimated at 3 and at 5 years was 29.1% and 34.2%, respectively. The presence of CNV and outer retinal tubulation (ORT) was independent risk factors for vision loss (adjusted odds ratio, 8.08 and 6.94, respectively). CONCLUSIONS: The development of CNV and ORT was strong risk factors for visual impairment. Due to the frequent recurrence of complications and poor visual prognosis, regular check-ups and appropriate treatment choices are warranted.

Secondary choroidal neovascularization due to choroidal osteoma after 9 years follow-up.

BACKGROUND: Choroidal osteoma is a benign intraocular tumor that can increase risk of developing choroidal neovascularization. The visual prognosis is influenced by the tumor location, decalcification status, overlying RPE atrophy, presence of choroidal neovascularization, persistence of subretinal fluid and occurrence of subretinal hemorrhages. CASE PRESENTATION: The authors present a 40-year-old woman diagnosed with choroidal osteoma of the right eye. Her best corrected visual acuity was 12/20 but decreased to 5/20 due to secondary choroidal neovascularization after 8 years follow up. Fundus examination revealed an enlarged choroidal osteoma in most margins at posterior pole with schistose hemorrhage beside macula. Optical coherence tomography angiography revealed unique features in the vascular changes of choroidal neovascularization in choroidal osteoma in the outer retinal layer and choroid capillary layers, and subretinal neovascularization. Indocyanine green fluorescence angiography showed there was hypo-fluorescence at the peripapillary with faint hyper-fluorescence at the macular, corresponding to the location on the fundus photograph. The patient received 3 injections of intravitreal ranibizumab. After 1 year follow up, her visual acuity of the right eye was 18/20 and the CNV had regressed. CONCLUSIONS: We present the findings and treatment of a case of choroidal osteoma with secondary choroidal neovascularization. Optical coherence tomography angiography combined with FFA and ICGA is used to analysis the characteristics of secondary choroidal neovascularization. Optical coherence tomography angiography can reveal some unique characteristics in the vascular changes compared to fundus fluorescein angiography.

Choroidal Osteoma with Choroidal Neovascular Membrane treated with Anti- vascular Endothelial Growth Factor: A Rare Case with Review of Literatures.

INTRODUCTION: Choroidal osteoma, usually unilateral, is a rare benign tumor of choroid. It is predominantly seen in young females. CASE: We report a case of a 35-year-old female with unilateral choroidal osteoma associated with choroidal neovascular membrane (CNVM). The diagnosis was confirmed with clinical examination, ocular ultrasonography, fundus fluorescein angiography and macular optical coherence tomography. Choroidal neovascular membrane improved with 3 doses of bevacizumab, an anti-vascular endothelial growth factors (Anti-VEGF). CONCLUSION: Anti-VEGF monotherapy can be a cost effective primary modality of treatment for choroidal neovascular membrane secondary to choroidal osteoma.

Focal choroidal excavation and pitchfork sign in choroidal neovascularisation associated with choroidal osteoma.

PURPOSE: To describe a complex case of choroidal osteoma complicated by choroidal neovascularisation and focal choroidal excavation and presence of pitchfork sign. METHODS: A young female patient with confirmed diagnosis of choroidal osteoma was followed up for 5 years. Multimodal imaging including optical coherence tomography and fluorescein angiography (Spectralis HRA + OCT; Heidelberg Engineering, Heidelberg, Germany) was performed. RESULTS: Optical coherence tomography showed the presence of a unilateral choroidal osteoma in the first visit which was later followed by the appearance of focal choroidal excavation. After 3 years, choroidal neovascularisation developed in choroidal osteoma area, with subretinal fluid, subretinal hyper-reflective material, hyper-reflective foci and pitchfork sign. Decalcification of the tumour was not noted in this patient. Treatment with one intravitreal injection of Aflibercept resulted in complete disappearance of subretinal fluid, subretinal hyper-reflective material, pitchfork sign and decreased hyper-reflective foci. CONCLUSION: Inflammatory response can be a propulsive element in the development of focal choroidal excavation and choroidal neovascularisation in choroidal osteoma patients.

Choroidal osteoma: the varied response of subretinal fluid to anti-VEGF agents.

The present case shows a remarkable long-term response of the choroidal neovascular membrane and serous retinal detachment associated with choroidal osteoma to two injections of ranibizumab which is an anti-vascular endothelial growth factor (anti-VEGF) agent.

PHOTODYNAMIC THERAPY FOR EXTRAFOVEOLAR CHOROIDAL OSTEOMA.

PURPOSE: To evaluate the outcome of photodynamic therapy (PDT) in the management of extrafoveolar choroidal osteoma. METHODS: The authors performed a retrospective chart review of all patients with choroidal osteoma that did not involve the foveola and were treated with standard-fluence PDT. RESULTS: Nine eyes with extrafoveolar choroidal osteoma were studied. Mean logarithm of the minimum angle of resolution best-corrected visual acuity at initial examination was 0.07 (Snellen ∼20/25). The osteoma was treated with 1 (8/9) or 2 (1/9) PDT sessions using 50 J/cm. After a mean follow-up of 49 months, the treated area of osteoma demonstrated complete (4/9) or partial (5/9) regression, with a mean of 73% regression in the PDT-treated areas. Tumor growth in the region of PDT was noted in 3 cases (3/9) (one tumor toward the foveola and two tumors at the margin away from the foveola), but in no case did the tumor reach the foveola. Therefore, PDT controlled tumor growth in 8 of 9 cases with only 1 of 9 cases showing growth through the PDT scar into foveola. Mean logarithm of the minimum angle of resolution visual acuity at last follow-up was 0.04 (Snellen ∼20/20) (P = 0.59). CONCLUSION: Photodynamic therapy is an effective modality for the management of extrafoveolar choroidal osteoma, minimizing tumor growth toward the foveola and preserving visual acuity.

Sequential imaging of a case of choroidal osteoma using swept-source OCT and optical coherence tomography angiography: A 4-year follow-up study.

A 33-year-old gentleman was presented with metamorphopsia in the left eye due to choroidal osteoma (CO) complicated by choroidal neovascular membrane (CNVM). Optical coherence tomography angiography (OCTA) proved to be a valuable, noninvasive tool in monitoring treatment response of CNVM. The tumor subsequently underwent decalcification over a period of 4 years. In addition, SS-OCT scans were instrumental in documenting the natural course of the tumor and focal choroidal excavations (FCE), which were found in correspondence with tumor decalcification. Close follow-up is warranted in FCE, secondary to decalcification of CO, as CNVM has been documented to occur on the slope or bottom of eyes with FCE.

Choroidal neovascularization due to choroidal osteoma treated with anti-vascular endothelial growth factor therapy: An optical coherence tomography angiography study.

PURPOSE: To evaluate the response to anti-vascular endothelial growth factor therapy for choroidal neovascularization secondary to choroidal osteoma using optical coherence tomography angiography. METHODS: This retrospective study included four eyes of four females with choroidal osteoma complicated by choroidal neovascularization, treated with ranibizumab. All patients underwent full ophthalmologic examination, including ocular ultrasound, retinography, fluorescein angiography, spectral-domain or swept-source optical coherence tomography, and optical coherence tomography angiography. These images were analyzed to measure choroidal osteoma and to study choroidal neovascularization changes after intravitreal anti-vascular endothelial growth factor. RESULTS: In all cases, fluorescein angiography revealed the presence the choroidal neovascularization, as an early hyperfluorescence area increasing during the exam. Optical coherence tomography showed both the choroidal osteoma and choroidal neovascularization and intra- or subretinal fluid as activity sign. In optical coherence tomography angiography, choroidal osteoma vessels were valuable in outer retina and choroidal slabs, and were irregular and did not change after ranibizumab injection; neovascular network correlating with choroidal neovascularization showed a hyperflow tangled vessels in outer retina, decreasing in density after anti-vascular endothelial growth factor therapy. CONCLUSION: Optical coherence tomography angiography seems to be a useful tool in visualizing and distinguishing vascular networks of choroidal osteoma and of choroidal neovascularization secondary to choroidal osteoma better than fluorescein angiography.

Osteoma de coroides con excavación coroidea y membrana neovascular asociada: estudio mediante angiografía-TCO / Choroidal osteoma with choroidal excavation and associated neovascular membrane: An OCT-angiography study

CASO CLÍNICO: Presentamos un caso de osteoma de coroides unilateral con excavación coroidea y membrana neovascular asociada estudiado mediante angiografía-TCO. En TCO y angiografía-TCO se objetivó excavación coroidea y una membrana neovascular coroidea activa. Se administraron tres dosis de aflibercept estabilizando la neovascularización coroidea. DISCUSIÓN: La angiografía-TCO permitió el análisis morfológico de la red vascular superficial del osteoma así como el control evolutivo de la membrana neovascular

CLINICAL CASE: A case is presented of a unilateral choroidal osteoma with choroidal excavation and associated neovascular membrane that was studied using OCT-angiography. The OCT and OCT-angiography revealed an area of choroidal excavation and an active neovascular membrane. Three doses of aflibercept were administered to the patient, which stabilised the neovascularisation. DISCUSSION: OCT-angiography was used to analyse the morphology of the superficial vascular network of the osteoma, as well as to monitor the evolution of the neovascular membrane

Osteoma frontoetmoidal con extensión intraorbitaria. A propósito de un caso / Frontoethmoidal osteoma with orbital extension. A case report

CASO CLÍNICO: Presentamos el caso de un osteoma frontoetmoidal derecho con invasión intraorbitaria en un varón de 43 años que comenzó como un cuadro de dolor retroocular con proptosis, inflamación peripalpebral y diplopía binocular por limitación del recorrido muscular. Se realizó cirugía abierta mediante abordaje subcraneal anterior, con extirpación de varios fragmentos de osteoma, cuyo análisis histológico confirmó el diagnóstico. DISCUSIÓN: Los osteomas son tumores benignos formadores de hueso de crecimiento lento. Habitualmente son asintomáticos, hasta que producen complicaciones por efecto masa en el cerebro o en la órbita, o a nivel local, hasta que obstruyen el drenaje del seno donde asientan, generando mucoceles

CASE REPORT: A case is presented of a right frontoethmoidal osteoma with intraorbital invasion in a 43-year-old male, who was seen in the clinic with a proptosis, retro-orbital pain, peri-palpebral inflammation, and binocular diplopia due to muscular route limitation. Open surgery was performed using a subcranial approach, with removal of several fragments of osteoma. Histological analysis confirmed the diagnosis. DISCUSSION: Osteomas are benign bone-forming tumours with slow growth. They are usually asymptomatic until mass effect complications occur in the brain or in the orbit, or locally, generating mucoceles due to sinus drainage obstruction

Baixa visão secundária a osteoma de coroide / Reduced vision caused by choroid osteoma

RESUMO Relatamos um caso de osteoma de coroide em uma paciente feminina de 25 anos. Apresentava uma lesão amarelada e elevada no polo posterior, característica da lesão tumoral. O diagnóstico foi confirmado com a ultrassonografia ocular. A paciente apresentava baixa acuidade visual e edema de mácula, que melhorou após injeção intravítrea de bevacizumab.

ABSTRACT The authors present a case of choroidal osteoma diagnosed in a 25-year-old female patient. A well-defined and slightly elevated yellow lesion located in the posterior pole of the right eye was suspected to be a tumor. Confirmation of diagnosis was obtained with A and B ecography. The patient had low vision and macular edema, which improved after intraocular injection of bevacizumab .

Optical Coherence Tomography Angiography for Detecting Choroidal Neovascularization Secondary to Choroidal Osteoma.

Choroidal osteoma is an ossifying tumor that is found predominantly in the peripapillary and macular areas. It typically affects otherwise healthy females. Vision loss may occur secondary to the development of choroidal neovascularization (CNV). Fluorescein angiography (FA) remains the gold standard for diagnosing CNV; however, the use of optical coherence tomography angiography (OCTA) as an adjunct to FA is growing. In this report, a 16-year-old female with a large, unilateral peripapillary choroidal osteoma presented with blurred vision. Exam revealed scattered intraretinal hemorrhage, but FA was unable to detect CNV overlying the tumor. OCTA detected abnormal flow in the outer retina corresponding to a type 2 CNV. Following intravitreal anti-vascular endothelial growth factor therapy, the CNV regressed, the hemorrhage resolved, and there was less fluid. OCTA may be helpful in detecting CNV noninvasively in eyes in which FA is equivocal, such as those with choroidal osteoma.