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PubMedDate not supplied

Coats'-type retinitis pigmentosa.

Coats'-like changes (i.e., retinal telangiectasia and/or exudative detachment) have been reported in as many as 1.2 to 3.6 percent of patients with retinitis pigmentosa. In severe cases this disorder may progress to total retinal detachment and visual loss in the context of longstanding retinitis pigmentosa. Forty-six cases of Coats'-type retinitis pigmentosa gathered from the literature are reviewed. Historical and epidemiological features, hereditary factors, clinical features, histopathological findings, pathogenesis, differential diagnosis, prognosis and possible treatment are discussed.

Choroid↗
PubMed2005

Group 2A idiopathic juxtafoveolar retinal telangiectasia in monozygotic twins.

PURPOSE: To report the clinical and fluorescein angiographic characteristics of monozygotic twins affected by group 2A idiopathic juxtafoveolar retinal telangiectasia. DESIGN: Observational case report. METHODS: Both eyes of identical twins were examined in a clinical practice setting. RESULTS: Two 68-year-old Caucasian, monozygotic, female twins were examined. In one twin, visual acuity was 20/50 in the right eye (OD) and 20/40 in the left (OS). Ophthalmoscopy demonstrated right-angle venules without edema in both eyes (OU). Fluorescein angiography demonstrated areas of leakage temporal to the fovea OU with some nasal leakage OS. In her identical twin sister, with type II diabetes, visual acuity was 20/60 OD and 20/25 OS. Right-angle venules OU, refractile deposits OS, and no macular edema OU were present. Fluorescein angiography demonstrated areas of parafoveal leakage temporal and nasal OD and temporal OS. CONCLUSION: This is the second set of female monozygotic twins with group 2A idiopathic juxtafoveolar retinal telangiectasia reported in the literature. This finding suggests a genetic component in the pathogenesis of this retinal vascular disease.

Aged↗
PubMed2006

A novel RPGR exon ORF15 mutation in a family with X-linked retinitis pigmentosa and Coats'-like exudative vasculopathy.

PURPOSE: To describe the ophthalmic and genetic findings in a family with X-linked retinitis pigmentosa (RP) and Coats'-like exudative vasculopathy. DESIGN: Observational case series. METHODS: Family members underwent comprehensive ophthalmologic examination. Leukocyte genomic DNA samples were obtained and screened for RPGR (RP3) mutations by direct polymerase chain reaction sequencing. RESULTS: The proband had RP with bilateral Coats'-like vasculopathy and was treated with fluorescein-potentiated argon laser therapy. The findings in two other affected male patients and three obligate carrier female patients were within the clinical spectrum of a typical X-linked-recessive RP. A novel nonsense RPGR exon ORF15 mutation (912G>T) was found to segregate with RP in this family. CONCLUSIONS: This report expands the clinical heterogeneity spectrum caused by RPGR mutations and our knowledge concerning the molecular pathologic condition that pertains to Coats'-like RP. Consistent with the literature, Coats' response was not observed in all family members who were affected by RP, which suggests the involvement of other genetic and/or environmental factors.

Adolescent↗
PubMed1997

Coats-type retinitis pigmentosa in a 4-year-old child.

PURPOSE: To describe the occurrence of Coats-like exudative retinopathy secondary to underlying retinitis pigmentosa in a 4-year-old child. METHOD: Case report. RESULTS: A 4-year-old girl had bilateral exudative retinal telangiectasia requiring photocoagulation. She subsequently developed progressive nyctalopia, photophobia, and reduced peripheral vision. Electroretinography and dark adaptometry at age 8 years confirmed the diagnosis of retinitis pigmentosa. CONCLUSIONS: Coats-like exudative retinopathy secondary to retinitis pigmentosa can manifest as early as age 4 years and can precede the diagnosis of the underlying retinal dystrophy.

Child, Preschool↗
PubMed1981

Tractional venous loops in diabetic retinopathy.

A venous loop developed in a patient with proliferative diabetic retinopathy. Fluorescein angiography showed staining of the vessel wall and nonperfusion of the surrounding capillary bed. Light microscopy and scanning and transmission electron microscopy showed the loop to consist of a telangiectatic retinal vein that had passed through a discontinuity in the internal limiting membrane. The thin-walled vessel loop was lined by an attenuated endothelium and occasional pericytes. The vitreous was detached posteriorly except at the venous loop and a few other points at which there was tenting of the internal limiting membrane. These findings suggested that vitreous traction plays a role in the pathogenesis of these venous loops.

Aged↗
PubMed2003

Medical imaging in pediatric ophthalmology.

The use of CT and MR imaging has improved the understanding of many congenital and acquired conditions in pediatric ophthalmology, resulting in more appropriate therapeutic intervention and giving insight into the pathogenesis of these conditions.

Child↗
PubMed1998

Visual prognosis of Coats' disease.

PURPOSE: Our purpose was to determine the visual prognosis of retinal telangiectasia (Coats' disease). METHODS: We performed a retrospective review of 35 patients with Coats' disease seen at the Hospital for Sick Children, Toronto, Canada between 1987 and 1996. Ten patients were excluded because of incomplete records. Treatment modalities consisted of no treatment, cryotherapy with and without 532 nm laser through the indirect ophthalmoscope, and enucleation. Visual outcome was determined where possible. RESULTS: Median follow-up was 4.5 years. Deterioration in visual acuity was associated with the presence of greater than 5 clock hours of involved retina and retinal detachment at diagnosis. Final visual acuity did not correlate with age of onset of disease. No eye treated with cryotherapy progressed to retinal detachment. CONCLUSIONS: Aggressive treatment of Coats' disease with cryotherapy with or without 532 nm laser, before retinal detachment, is likely to stabilize vision and decrease the risk of future total retinal detachment.

Adolescent↗
PubMed2002

A case of Coats' disease with a peeling of premacular fibrosis after photocoagulation.

We report a 26-year-old man with Coats' disease associated with premacular fibrosis. As an initial treatment, the peripheral exduative area was treated with argon laser photocoagulation. Six weeks later, the premacular fibrosis was peeled off and the posterior vitreous membrane was also detached. The patient's visual acuity improved to 20/20. We also observed a change of the vitreous component before and after the treatment that was similar to posterior vitreous detachment (PVD). This is the first reported case in which a distinct vitreous change was observed after premacular fibrosis peeling in Coat's disease.

Adult↗
PubMed2002

Bilateral Coats' disease: long-term follow up.

PURPOSE: To report on the long-term follow-up of a female patient with bilateral Coats' disease, who showed marked asymmetry between the two eyes. METHODS: A five year old girl presented in 1978 with leukocoria in a blind right eye. A total exudative retinal detachment and extensive retinal telangiectasiae were noted. In the other eye, there was a localized area of retinal exudation and vascular abnormality in the supero-temporal periphery. Ultrasonography showed no evidence of intraocular tumour in the right eye and a clinical diagnosis of bilateral Coats' disease was made. RESULTS: In 1995, the area or retinal exudation in the left eye increased and laser photocoagulation was applied successfully. To date, no disease recurrences have occurred. CONCLUSION: Although Coats' disease is usually unilateral, bilateral, asymmetrical involvement may occur on rare occasions. Long-term follow-up of the least affected eye is necessary so that late complications can be identified early and treated adequately to prevent visual loss.

Child, Preschool↗
PubMed1994

Inherited venous beading.

Inherited retinal venous beading is a rare autosomal disorder. We describe three affected members in three generations of a single family. It is evident that there may be gross asymmetry of affection between the two eyes, and highly variable expressivity such that the diagnosis may not be evident without a family survey. One patient had remarkable spontaneous recovery of vision.

Adolescent↗
PubMed2000

[Retinal vasculopathy associated with Berger's IgA nephropathy].

AIM: Ophthalmological complications associated with Berger's IgA nephropathy comprise scleritis, episcleritis, keratoconjunctivitis as well as anterior uveitis. We present a new association of IgA nephropathy with a retinal vasculopathy. METHODS: Presentation of two clinical cases. RESULTS: Two patients presented with hematuria and epistaxis associated with a retinal vasculopathy characterised by teleangiectasies, capillary occlusion with retinal hemorrhages, neovascularisations and macular edema with decreased visual acuity. Fluorescein angiography showed zones of non-perfusion as well as vasculitic changes. A general medical exam revealed a normal arterial pressure but a slightly elevated creatinine. Immunological investigations for the presence of antibodies showed no positive results. Renal biopsy demonstrated mesangial proliferations with diffuse deposits of IgA. Over the course of a 2 year follow-up some of the retinal changes regressed under treatment with cortisone and visual acuity returned to normal. The teleangiectasies showed no progression. CONCLUSION: Berger's IgA nephropathy can be associated with a retinal vasculopathy which may be due to local deposition of IgA immune complexes in the retinal vessels.

Adult↗
PubMed1988

[Traction detachment in Coats disease].

Immunocytochemical examination of a subretinal membrane removed during vitrectomy from a patient suffering from Coats' disease revealed macrophages, fibronectin, and vimentin. Vimentin is considered to be a marker of mesenchymal cells. Fibronectin may represent a guide rail and scaffold for the cellular organization of subretinal exudates.

Fibronectins↗