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PubMed1994

Acquired peripheral retinal telangiectasia after retinal surgery.

BACKGROUND: Acquired peripheral retinal telangiectasia (APRT) is a well-recognized clinical entity that can be mistaken for conditions such as choroidal melanoma or the angiomata of von Hippel-Lindau disease. Although most cases are idiopathic, some have been shown to be associated with conditions such as uveitis and retinal detachment. METHODS: The clinical histories of four patients in whom APRT developed after retinal surgery are presented. RESULTS: For each patient studied, no retinal vascular abnormality was observed at the time of initial surgery. All patients were myopic, and three had clinical features compatible with Sticklers syndrome. APRT developed either in untreated retina remote from the area of detachment, in undetached retina treated with cryotherapy, in reattached retina in the region of subretinal fluid release, or in reattached retina over a scleral buckle. CONCLUSIONS: Possible risk factors for the development of APRT include retinal ischemia after longstanding retinal detachment, retinal cryotherapy or scleral buckling, and damage to Bruch's membrane at the time of scleral suture placement or release of subretinal fluid. This report provides further evidence of an association between APRT and retinal detachment, and that APRT may be more specifically associated with retinal detachments in patients with Sticklers syndrome.

Adult↗
PubMed1979

[Vascular anastomoses at the posterior pole of the eye (author's transl)].

Anastomoses between vessels of the fundus may occur as congenital malformations or secondary to vascular diseases affecting the eye. Congenital anastomoses can be of the racemose hemangioma type, with severe malformation of the retinal vessels, ocular complications, and neurologic manifestations, or as simple A-V shunts with visual impairment but limited tendency to show other ocular or neurologic involvement. These anastomoses must be distinguished from other vascular malformations like telangiectasis (Leber, Coats) or the angiomatous lesions of von Hippel's disease. Secondary anastomoses may be confined to the retinal vessels or involve chorioretinal connections. Retinal anastomoses occur in vascular disorders like retinal vein occlusions, diabetes, periphlebitis and hemoglobinopathies. They are often amenable to treatment by light coagulation. Chorioretinal anastomoses occur whenever Bruch's membrane is destroyed by traumatic, inflammatory, degenerative or physical (light coagulation) influences. Light coagulation may be the appropriate treatment, although complications are not rare.

Adolescent↗
PubMed2005

Coats' disease diagnosed in adulthood.

PURPOSE: To investigate the manifestations and clinical course of Coats' disease diagnosed in adulthood and to describe the characteristics of patients who develop it. DESIGN: Retrospective observational case series. PARTICIPANTS: Thirteen eyes of 13 patients with Coats' disease diagnosed after age 35 years. METHODS: All patients examined in the authors' referral practices for Coats' disease diagnosed after the age of 35 years were identified. Patients with a history of radiation exposure, intraocular inflammation, retinal vascular occlusion, or Coats' response were excluded. A review of their historical, clinical, and fluorescein angiographic features and demographics was performed. RESULTS: There were 13 patients with a mean age at diagnosis of 50 years (range, 36-79) and a mean follow-up of 5.8 years (range, 0-17). These patients manifested many findings typical of Coats' disease, including the unilateral nature of the disease (13/13 patients), male predominance (12/13), vascular telangiectasis, lipid exudation, macular edema, and areas of capillary nonperfusion with adjacent webs of filigreelike capillaries. Disease was limited to < or =6 clock hours in 10 of 13 patients (76.9%), did not generally involve the retinal vasculature anterior to the equator, and showed limited potential to expand during the follow-up period. A localized hemorrhage was noted in 10 of 13 patients (76.9%), almost always near larger aneurysmal dilatations. Limited exudative detachment of the retina was seen in 4 eyes, 3 of which responded to localized laser photocoagulation. One patient had subretinal fluid seen on ocular coherence tomography that did not require treatment. On average, patients lost 2.1 lines of visual acuity during the follow-up period; 6 patients had a decline in vision, 2 patients had improved vision, and 3 patients had stable vision. No patient had end-stage findings of Coats' disease, such as iris neovascularization or total exudative detachment. CONCLUSIONS: Coats' disease can first be diagnosed in adulthood with retinal vascular abnormalities similar to those seen in younger patients. There are a number of important differences in disease manifestation in adults, including limited area of involvement, slower apparent progression of disease, and hemorrhage near larger vascular dilatations.

Adult↗
PubMed2001

Classification and management of Coats disease: the 2000 Proctor Lecture.

PURPOSE: To review the methods and results of management in a large series of patients with Coats disease, to determine risk factors for poor visual outcome and enucleation, and to propose a practical classification of Coats disease. METHODS: In a retrospective consecutive series in 150 patients, Coats disease was defined as idiopathic retinal telangiectasia with intraretinal or subretinal exudation without appreciable signs of retinal or vitreal traction. We reviewed our experience with management, including observation, laser photocoagulation, cryotherapy, and various techniques of retinal detachment surgery and enucleation. The anatomic outcome, complications of treatment, visual results, and reasons for enucleation were tabulated. Factors predictive of poor visual outcome (20/200 or worse) and enucleation were determined using Cox proportional hazards regression models. Based on these observations, a staging classification of Coats disease, applicable to treatment selection and ocular prognosis, is proposed. RESULTS: In 117 patients (124 eyes) with a mean follow up of 55 months (range, 6 months to 25 years) primary management was observation in 22 eyes (18%), cryotherapy in 52 (42%), laser photocoagulation in 16 (13%), various methods of retinal detachment surgery in 20 (17%), and enucleation in 14 (11%). Anatomic improvement or stability was achieved in 76% of eyes, and final visual acuity was 20/50 or better in 17 eyes (14%), 20/60 to 20/100 in eight (6%), 20/200 to finger counting in 30 (24%), and hand motion to no light perception in 49 (40%) Enucleation was ultimately necessary in 20 eyes (16%). Risk factors predictive of poor visual outcome (20/200 or worse) included postequatorial (P =.01), diffuse (P =.01), or superior (P =.04) location of the telangiectasias and exudation, failed resolution of subretinal fluid after treatment (P =.02), and presence of retinal macrocysts (P =.02). The main risk factors for enucleation were elevated intraocular pressure (greater than 22 mm Hg; P less than or equal to.001) and iris neovascularization (P less than or equal to.001). Coats disease was classified into stage 1, telangiectasia only; stage 2, telangiectasia and exudation (2A, extrafoveal exudation; 2B, foveal exudation) stage 3, exudative retinal detachment (3A, subtotal; 3B, total); stage 4, total detachment and secondary glaucoma; and stage 5, advanced end-stage disease. Poor visual outcome (20/200 or worse) was found in 0% of eyes with stage 1, 53% with stage 2, 74% with stage 3, and 100% of stages 4 and 5 Coats disease. Enucleation was ultimately necessary in 0% of stages 1 and 2, 7% of stage 3, 78% of stage 4, and 0% of stage 5 disease. CONCLUSIONS: Carefully selected treatment can anatomically stabilize or improve the eye with Coats disease in 76% of eyes. However, poor visual outcome of 20/200 or worse commonly results. Patients who present with stages 1 to 3 Coats disease have the best visual prognosis, and patients with stages 4 and 5 have a poor visual prognosis.

Cryosurgery↗
PubMedDate not supplied

Optical coherence tomography evaluation of macular edema after intravitreal triamcinolone acetonide in patients with parafoveal telangiectasis.

PURPOSE: Parafoveal telangiectasis (PT) is a developmental or acquired microvascular abnormality of the macula. Leakage of the abnormal parafoveal capillaries leads to macular edema with subsequent decrease in visual acuity. Intravitreal triamcinolone acetonide is recently widely used in the management of intraocular proliferative, edematous, and neovascular diseases. METHODS: This report presents the evaluation of three cases with PT in whom intravitreal triamcinolone acetonide (IVTA) injection was performed. Fundus fluorescein angiography (FA) and optical coherence tomography (OCT) were used in follow-up of the patients. RESULTS: Following pars plana intravitreal injection of 4 mg of triamcinolone acetonide, the patients had angiographic improvement of the macular edema and minimal decrease in retinal thickness on OCT, accompanied by improvement in visual acuity and subjective visual assessment. CONCLUSIONS: The results of the present study on parafoveal capillary telangiectasis suggest that the intravitreal injection of triamcinolone acetonide may be a therapeutic option to increase visual acuity and decrease vascular leakage on FA. Following IVTA procedure, follow-up of these patients with both OCT and FA is important for correct clinical evaluation. Future studies on this method seem to be warranted.

Aged↗
PubMed1999

Radiation exposure: a new risk factor for idiopathic perifoveal telangiectasis.

OBJECTIVE: To examine the association between previous radiation exposure and idiopathic perifoveal telangiectasis (IPT). DESIGN: A multicentered, individually matched, case-control study design was used. PARTICIPANTS/CONTROLS: Sixty-five case subjects were matched with 175 control subjects. Individuals with unequivocal evidence of angiographically confirmed IPT were included as cases. Control subjects were matched for center, age, and gender. MAIN OUTCOME MEASURE: The main exposures of interest were a history of therapeutic head or neck irradiation and environmental radiation exposure. METHODS: A standardized questionnaire was administered to case and control subjects. Data were collected for the main exposures of interest as well as pertinent covariates. Conditional logistic regression was used to evaluate therapeutic and environmental radiation as risks for IPT. RESULTS: On univariate analysis, head or neck irradiation was associated with IPT (odds ratios [OR] = 4.15, 95% confidence interval [CI] = 1.30-13.24). While controlling for diabetes and family history of diabetes, IPT was found to be associated with both head or neck irradiation (OR = 4.06, 95% CI = 1.20-13.76) and with environmental irradiation (OR = 6.73, 95% CI = 1.06-42.74). CONCLUSIONS: This study presents a previously unreported association between prior radiation exposure and IPT.

Aged↗
PubMed1989

[Clinical aspects of Coats].

Coats' disease associates on exudative retinopathy to vascular telangiectasis. The authors analyse the different factors which take part to many clinical aspects of the Coats' exudation: the age of the patients and the vascular trouble which causes the exudative retinopathy.

Adult↗
PubMed2000

Bilateral juxtafoveolar telangiectasis in monozygotic twins.

PURPOSE: To report the clinical and angiographic features of two monozygotic twins affected by bilateral group 2 idiopathic juxtafoveolar telangiectasis. METHOD: Case reports. RESULTS: Two 64-year-old women, who were identical twins, were suffering from visual loss. One twin had suffered from visual loss for 1 year and had a visual acuity of 20/25 in both eyes, and the other twin had suffered for 2 years and had a visual acuity of 20/30 in both eyes. Fluorescein angiography disclosed similar fundus features. An analogous area of capillary telangiectasis and leakage was observed in the right macula, where no intraretinal pigment was seen; the left macula showed a similar amount of intraretinal pigment associated with tiny right-angle venules and minimal leakage. CONCLUSION: This observation raises the issue of genetic influences in the pathogenesis of this disease.

Diseases in Twins↗
PubMed2004

[Ciliary body melanomas. Survival and prognostic aspects after brachytherapy or proton therapy].

INTRODUCTION: Our main goals were to study the survival of patients with uveal melanomas involving the ciliary body 5 and 10 years after treatment and to review prognosis. MATERIAL AND METHODS: This study investigated 106 tumors (27 ciliary body melanomas and 79 choroidal-ciliary melanomas) of patients treated between June 1983 and April 1998. Seventy-two patients were treated with 106-ruthenium applicators and 34 were treated with proton therapy. Some large tumors or recurrences required a second treatment. The mean follow-up period was 91 months. RESULTS: The mean tumor sizes before treatment were 6,6mm for melanoma thickness and 10.3mm for mean largest basal melanoma diameters (LTD). Of the patients studied, 71% were still alive at 5 years, 35% had metastasis and 27.7% developed recurrences. Multivariate analysis showed that the risk factors for melanoma-related death were LTD greater than 13mm, presence of exudative retinal detachment, macroscopic iris root involvement at the time of diagnosis, and choroidal-ciliary location. DISCUSSION AND CONCLUSION: With 71% of patients still alive at 5 years, this review shows that melanomas involving the ciliary body do not seem less severe than more posterior uveal tumors. Because of size differences between ciliary body melanomas and choroidal-ciliary melanomas, we cannot come to the conclusion that choroidal-ciliary melanomas have a poorer prognosis than ciliary body melanomas. Largest tumor diameter over 13mm, presence of exudative retinal detachment, and macroscopic iris root involvement at the time of diagnosis are important risk factors for melanoma-related death, as shown by the multivariate analysis.

Adolescent↗
PubMedDate not supplied

Cutis marmorata telangiectatica congenita with cerebral and ophthalmic anomalies: report of one case.

A term male neonate had an uncommon congenital vascular disorder of the skin covering the whole body and extremities. These skin lesions were characterized by mottling and persistent telangiectasia. A skin biopsy showed nevus vascularis reticularis which was recognized as cutis marmorata telangiectatica congenita. In addition to the cutaneous involvement, the patient also had fetal ascites, pleural effusion, patent ductus arteriosus, glaucoma, retinal detachment in the left eye, and telangiectasia in the right eye. Neonatal focal seizure developed and expired on the 34th day of life. The cerebral image studies suggested vascular anomalies with secondary cerebral parenchymal damage. Cutis marmorata telangiectatica congenita may be solitary but is frequently associated with other developmental defects. However, the specific findings of cerebral and ophthalmic lesions in our case were rather unusual.

Abnormalities, Multiple↗
PubMed1997

Cutis marmorata telangiectatica congenita associated with an elevated maternal serum human chorionic gonadotrophin level and transitory isolated fetal ascites.

A 34-year-old woman with an abnormal maternal serum screening result and a Down syndrome risk of 1:60 calculated from a maternal serum alpha-fetoprotein (AFP) value of 1.4 multiples of the median (MoM) and a human chorionic gonadotrophin (hCG) level of 4.32 MoM at 18 weeks' gestation was found to have isolated fetal ascites at 23 weeks' gestation. Spontaneous resolution occurred 10 weeks after the initial presentation. After birth, the neonate had generalized cutis marmorata telangiectatica congenita (CMTC), large vascular plaques on the scalp with superficial ulceration and crusts, a small atrial septal defect, a patent ductus arteriosus, hepatomegaly, micrognathia, seizures, an abnormal electroencephalogram, congenital retinal detachment, glaucoma and widely spaced toes. Our patient illustrates that CMTC in utero may be associated with a markedly elevated maternal serum hCG level as well as transitory isolated fetal ascites. However, such associations can be coincidental and further collaborative studies and cases will be necessary before it can be determined that a disproportionately elevated hCG level and transitory isolated fetal ascites are predictive of CMTC in utero.

Abnormalities, Multiple↗
PubMed2002

Atypical choroidal neovascularization in radiation retinopathy.

PURPOSE: To report atypical choroidal neovascularization associated with radiation retinopathy. DESIGN: Case report. METHODS: The patient was examined with ophthalmoscopy, fluorescein angiography, and indocyanine green angiography. RESULTS: A 32-year-old woman presented with an accelerated course of radiation retinopathy with macular edema, capillary telangiectasis, subretinal fluid, and lipid during and after pregnancy. Indocyanine green, but not fluorescein angiography, demonstrated an unusual area of choroidal neovascularization with prominent saccular dilations adjacent to a large zone of poor choroidal perfusion. Laser photocoagulation of the choroidal neovascularization resulted in an angiographic closure with absorption of the subretinal fluid and improvement of visual acuity. CONCLUSIONS: Choroidal neovascularization may be associated with radiation retinopathy and, when present, may require indocyanine green angiography for its diagnosis.

Adult↗