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PURPOSE: To describe the use of anti-osteoclastic medications (i.e., bisphosphonates and receptor activator of nuclear factor kappa beta (RANK) ligand inhibitors) in treating choroidal osteoma. METHODS: A 42-year-old male with bilateral posterior choroidal osteomas showed extensive outer retinal atrophy in the right eye secondary to decalcification of a choroidal osteoma and progressive extension of retinal pigment epithelial atrophy in the left eye encroaching on the macula with subtle changes in the choroidal osteoma. Bisphosphonates (i.e., oral alendronate 70 mg weekly) or RANK ligand inhibitors (i.e., subcutaneous denosumab 60 mg every six months) were initiated to prevent calcium resorption. RESULTS: After the introduction of more directed therapy, there was stabilization of the patients condition over the next nine years while under bisphosphonate treatment, followed by a switch to RANK ligand inhibitor therapy and later bisphosphonate treatment again. This was made evident through stability in fundus photography, enhanced depth imaging optical coherence tomography, and B-scan ultrasonography regarding outer retinal layers thickness, and choroidal osteoma thickness and calcification. CONCLUSION: Anti-osteoclastic therapy using bisphosphonates or RANK ligand inhibitors could be a new treatment paradigm to prevent resorption of choroidal osteomas and prevent the subsequent atrophy of outer retinal layers. SUMMARY STATEMENT: A patient with bilateral posterior choroidal osteomas was treated with bisphosphonates and RANK ligand inhibitors to prevent osteoma resorption in the better-seeing left eye. This has led to stability over six years of outer retinal layers thickness, choroidal osteoma thickness, and choroidal osteoma calcification.

Dafne Fernandes Machado,1 Stela Souza Peña,1 Mariana Carneiro Alves,1 Giovana Capecci Siqueira,1 Leonardo Amarante Pereira,1 Samantha Sanches de Carvalho,2 Thais Moura Gascon,2 Beatriz da Costa Aguiar Alves,2 Glaucia Luciano da Veiga,1,2 Fernando Luiz Affonso Fonseca,1– 3 Vagner Loduca Lima,1 Priscilla Ballalai1 1Ophthalmology Discipline, Centro Universitário Saúde ABC/ Faculdade de Medicina Do ABC, Santo André, Brasil; 2Clinical Analysis Laboratory, Centro Universitário Saúde ABC/ Faculdade de Medicina Do ABC, Santo André, Brasil; 3Department of Pharmaceutical Sciences, Universidade Federal de São Paulo, Diadema, BrasilCorrespondence: Dafne Fernandes Machado, Ophthalmology Discipline, Centro Universitário Saúde ABC/ Faculdade de Medicina do ABC, Av. Lauro Gomes, 2.000, Santo André, São Paulo, 09060-870, Brasil, Tel +55-114993-5400, Email dafnefm13@gmail.comAbstract: Choroidal osteoma is a rare, benign, osseous choristoma presenting as an orange-yellow, well-defined fundus mass. It presents unilaterally in most cases, has a predilection for the female sex, and favor a juxtapapillary location, becoming clinically manifest when it involves the macula. Almost 60% of eyes with osteoma may suffer significant visual loss. Choroidal osteomas can easily be mistaken for other conditions with similar presentations. We herein report two rare cases presentation of choroidal osteoma: a 74-year-old male with 2-year blurred vision in the right eye (RE) initially misdiagnosed with age-related macular degeneration received intravitreal injections and after a multimodal evaluation, following cataract surgery in RE, confirmed to be choroidal osteoma and a 19-year-old female with a history of choroidal hemangioma presented with blurred vision in her left eye (LE), with examination revealed an irregular orange-yellow lesion along the superotemporal arcade with serous macular detachment, later diagnosed as choroidal osteoma.Keywords: choroidal osteoma, osseous choristoma, visual loss, case reports

Purpose: To describe the use of anti-osteoclastic medications (i.e., bisphosphonates and receptor activator of nuclear factor kappa beta (RANK) ligand inhibitors) in treating choroidal osteoma. Methods: A 42-year-old male with bilateral posterior choroidal osteomas showed extensive outer retinal atrophy in the right eye secondary to decalcification of a choroidal osteoma and progressive extension of retinal pigment epithelial atrophy in the left eye encroaching on the macula with subtle changes in the choroidal osteoma. Bisphosphonates (i.e., oral alendronate 70 mg weekly) or RANK ligand inhibitors (i.e., subcutaneous denosumab 60 mg every six months) were initiated to prevent calcium resorption. Results: After the introduction of more directed therapy, there was stabilization of the patient's condition over the next nine years while under bisphosphonate treatment, followed by a switch to RANK ligand inhibitor therapy and later bisphosphonate treatment again. This was made evident through stability in fundus photography, enhanced depth imaging optical coherence tomography, and B-scan ultrasonography regarding outer retinal layers thickness, and choroidal osteoma thickness and calcification. Conclusion: Anti-osteoclastic therapy using bisphosphonates or RANK ligand inhibitors could be a new treatment paradigm to prevent resorption of choroidal osteomas and prevent the subsequent atrophy of outer retinal layers. Summary statement: A patient with bilateral posterior choroidal osteomas was treated with bisphosphonates and RANK ligand inhibitors to prevent osteoma resorption in the better-seeing left eye. This has led to stability over six years of outer retinal layers thickness, choroidal osteoma thickness, and choroidal osteoma calcification.

Choroidal osteoma (CO) is a rare benign tumor usually unilateral and have a female preponderance. We report a case of a 34-year-old healthy male diagnosed with unilateral Choroidal Osteoma with an associated Choroidal Neo Vascular Membrane (CNVM). The diagnosis was confirmed based on clinical examination, typical findings on B-scan, Fundus fluorescein angiography, Indocyanine green angiography and Optical coherence tomography. The CNVM was treated with intravitreal Bevacizumab injections and showed a good response initially. There was a recurrence of CNVM over two years when patient was lost to follow-up. Treatment with intravitreal Ranibizumab bio similar injection helped to stabilize his vision. Regular follow-up is essential for timely treatment of recurrence of CNVM and optimum visual outcomes. [ABSTRACT FROM AUTHOR]

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Purpose: To describe the use of anti-osteoclastic medications (i.e., bisphosphonates and receptor activator of nuclear factor kappa beta (RANK) ligand inhibitors) in treating choroidal osteoma., Methods: A 42-year-old male with bilateral posterior choroidal osteomas showed extensive outer retinal atrophy in the right eye secondary to decalcification of a choroidal osteoma and progressive extension of retinal pigment epithelial atrophy in the left eye encroaching on the macula with subtle changes in the choroidal osteoma. Bisphosphonates (i.e., oral alendronate 70 mg weekly) or RANK ligand inhibitors (i.e., subcutaneous denosumab 60 mg every six months) were initiated to prevent calcium resorption., Results: After the introduction of more directed therapy, there was stabilization of the patient's condition over the next nine years while under bisphosphonate treatment, followed by a switch to RANK ligand inhibitor therapy and later bisphosphonate treatment again. This was made evident through stability in fundus photography, enhanced depth imaging optical coherence tomography, and B-scan ultrasonography regarding outer retinal layers thickness, and choroidal osteoma thickness and calcification., Conclusion: Anti-osteoclastic therapy using bisphosphonates or RANK ligand inhibitors could be a new treatment paradigm to prevent resorption of choroidal osteomas and prevent the subsequent atrophy of outer retinal layers., Summary Statement: A patient with bilateral posterior choroidal osteomas was treated with bisphosphonates and RANK ligand inhibitors to prevent osteoma resorption in the better-seeing left eye. This has led to stability over six years of outer retinal layers thickness, choroidal osteoma thickness, and choroidal osteoma calcification., Competing Interests: The authors have no conflict of interest., (© 2024 The Authors. Published by Elsevier Inc.)

Purpose: To report a very rare and atypical case of an elderly Caucasian female patient who developed perilesional multiple polypoidal choroidal vasculopathy (PCV) as a probable complication of choroidal osteoma (CO), associated to preretinal neovascular membrane overlying the lesion., Methods: Observational case report., Case Observation: A 60-year-old Caucasian woman presented with blurred vision in her right eye (RE). Fundus examination revealed a round white-yellowish calcified deep lesion in the juxta-papillary superior area, measuring 4 disc-diameters, with well-defined scalloped margins and an irregular surface. B-scan ultrasonography and orbital tomography confirmed the diagnosis of choroidal osteoma (CO). Further investigation with multimodal imaging including infracyanine green angiography, fluorescein angiography, swept source optical coherence tomography and angiography highlighted the presence of multiple aneurysmal choroidal dilations around the CO, corresponding to PCV. We also noted the presence of a preretinal neovascular membrane overlying the CO. The patient was monitored with regular follow-up since no signs of activity were detected on multimodal imaging., Conclusion: Our case report represents an exceptional and atypical association between pre-retinal neovascularization, PCV and choroidal osteoma. While the mechanisms underlying the development of PCV and pre-retinal neovascularization in the setting of CO are not well understood, it is imperative for ophthalmologists to recognize this association as a potential cause of sudden vision loss in patients with CO, and to consider appropriate diagnostic and management strategies., Competing Interests: Declaration of conflicting interestsThe authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.