Intraretinal Calcification and Osseous Metaplasia in Coats Disease

Miller, Daniel M.; Benz, Matthew S.; Murray, Timothy G.; Dubovy, Sander R.

doi:10.1001/archopht.122.11.1710

Cited by 18 publications

(7 citation statements)

References 7 publications

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“…Although previous studies mostly reported that RPE hyperplasia and ossification take place at subretinal or intraretinal level [ 5 , 6 ], no retinal tissue but gliosis was found in our specimen. In other words, the ossification process might take place above the retina in company with proliferative vitreoretinopathy change.…”

Section: Discussioncontrasting

confidence: 69%

Intraocular osseous metaplasia presenting as a solid mass in chronic retinal detachment: a case report

2021

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Background Intraocular osseous metaplasia is a rare histological finding associated with benign cellular transformation. Its development requires inflammatory cytokines and the process takes many years. Previous case reports of intraocular ossification manifested as linear calcification or white plaques. In contrast, our case presented with a tumor-like solid mass, in which a long-standing chronic inflammatory stimulation may contribute to the stunning appearance. Case presentation This is a 48-year-old woman with past history of advanced Coat’s-like retinopathy and chronic retinal detachment in the left eye for 12 years. She underwent vitreoretinal surgery to prevent phthisis bulbi. During the operation, a 9 mm solid mass was found embedded within the proliferative tissue above the retina and was removed. Pathological findings revealed bone formation in the center of the mass surrounded by fibrous metaplasia and focal gliotic changes. Layers of cohesive cells were found lining on the external side of the mass, and further immuno-histochemical study suggested them retinal pigment epithelial cells. Postoperatively, the retina was attached with stable visual acuity and normal intraocular pressure. Conclusion To our knowledge, the appearance of a tumor-like mass representing intraocular osseous metaplasia in eyes with chronic inflammation or retinal detachment has not been reported in previous case reports. This case emphasizes the importance of considering osseous metaplasia as one of the differential diagnoses of an unknown intraocular mass, especially in eyes with great severity of chronic inflammation. Also, our immuno-histochemical study provided more evidence on the pathological role of retinal pigment epithelial cells in developing ossification.

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Section: Discussioncontrasting

confidence: 69%

Intraocular osseous metaplasia presenting as a solid mass in chronic retinal detachment: a case report

2021

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“…There are a number of mechanisms by which bone and/or cartilage form within ocular structures; these include (i) congenital ectopic (choristomatous) bone/cartilage formation, (ii) osseous metaplasia as a pathologic response (e.g., post‐traumatic or intratumoral), (iii) an adaptive response to physiologic stressors within an individual's environment, or (iv) as a normal developmental finding within all or the vast majority of individuals within a species. Given the high prevalence of affected animals, and the lack of other ectopic tissues, it seems unlikely that our findings represented choristomatous cartilage or bone formation.…”

Section: Discussionmentioning

confidence: 99%

Gross, histologic, and computed tomographic characterization of nonpathological intrascleral cartilage and bone in the domestic goat (Capra aegagrus hircus)

Tusler

Good

Maggs

et al. 2016

Veterinary Ophthalmology

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Objective To characterize grossly, histologically, and via computed tomography (CT) the appearance of intrascleral cartilage, bone, or both in domestic goats with otherwise normal eyes and to correlate this with age, sex, and breed. Animals studied Sixty-eight domestic goats (89 eyes). Procedures Forty-nine formalin-fixed globes from 38 goats underwent high-resolution CT, and gross and light microscopic examination. An additional 40 eyes from 30 goats underwent light microscopy only. Age, breed, and sex of affected goats were retrieved from medical records.Results Considering all methods of evaluation collectively, cartilage was detected in 42% of eyes (44% of goats) and bone in 11% of eyes (12% of goats); bone was never seen without cartilage. Goats in which bone, cartilage, or both were detected ranged from 0.25 to 13 (median = 3.5) years of age, represented 11 of 12 breeds of the study population, and had a male:female ratio of 11:19. Bone was detected in the eyes of significantly more males (n = 8) than females (n = 2). No sex predilection was noted for cartilage alone. Histology revealed intrascleral chondrocyte-like cells, hyaline cartilage, and islands of lamellar bone. Some regions of bone had central, adipose-rich, marrowlike cavities. CT localized mineralized tissue as adjacent to or partially surrounding the optic nerve head. Conclusions This is the first report of intrascleral bone or cartilage in a normal goat and of intrascleral bone in an otherwise normal mammal. The high prevalence of intrascleral cartilage and bone in this study suggests that this finding is normal and likely represents an adaptation in goats.

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“…44 Calcifications of the retina, retinal vessels, and the presence of osseous metaplasia in the retina have also been noted in patients with renal failure, Coats' disease, tuberous sclerosis, and retinocytomas. 45,46 Whether calcification in these lesions is associated with angiogenesis has not been determined.…”

Section: Ectopic Calcification In Other Tissuesmentioning

confidence: 99%

Angiogenesis and Pericytes in the Initiation of Ectopic Calcification

Collett

Canfield

2005

Circulation Research

231

174

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Abstract-Ectopic calcification of blood vessels, heart valves, and skeletal muscle is a major clinical problem. There is now good evidence that angiogenesis is associated with ectopic calcification in these tissues and that it is necessary, but not sufficient, for calcification to occur. Angiogenesis may regulate ectopic calcification in several ways. First, many angiogenic factors are now known to exert both direct and indirect effects on bone and cartilage formation. Second, cytokines released by endothelial cells can induce the differentiation of osteoprogenitor cells. Third, the new blood vessels provide oxygen and nutrients to support the growing bone. Finally, the new blood vessels can serve as a conduit for osteoprogenitor cells. These osteoprogenitor cells may be derived from the circulation or from pericytes that are present in the neovessels themselves. Indeed, there is now compelling evidence that pericytes can differentiate into osteoblasts and chondrocytes both in vitro and in vivo. Other vascular cells, including adventitial myofibroblasts, calcifying vascular cells, smooth muscle cells, and valvular interstitial cells, have also been shown to exhibit multilineage potential in vitro. Although these cells share many properties with pericytes, the precise relationship between them is not known. Furthermore, it still remains to be determined whether all or some of these cells contribute to the ectopic calcification observed in vivo. A better understanding of the underlying mechanisms that link angiogenesis, pericytes, and ectopic calcification should provide a basis for development of therapeutic strategies to treat or arrest this clinically significant condition.

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Intraretinal Calcification and Osseous Metaplasia in Coats Disease

Cited by 18 publications

References 7 publications

Intraocular osseous metaplasia presenting as a solid mass in chronic retinal detachment: a case report

Intraocular osseous metaplasia presenting as a solid mass in chronic retinal detachment: a case report

Gross, histologic, and computed tomographic characterization of nonpathological intrascleral cartilage and bone in the domestic goat (Capra aegagrus hircus)

Angiogenesis and Pericytes in the Initiation of Ectopic Calcification

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