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PubMed2006

Optical coherence tomography findings in idiopathic juxtafoveal retinal telangiectasis.

PURPOSE: To describe optical coherence tomography (OCT) findings in patients with juxtafoveal retinal telangiectasis (JRT). METHODS: Fourteen consecutive patients (28 eyes) with JRT (12 patients with JRT type II, one with JRT type I and one with JRT type III) were examined using fluorescein angiography (FA) and OCT. RESULTS: Despite prominent leakage in FA, macular oedema was absent in all 26 eyes with type II JRT. In contrast to that, in patients with type I and type III JRT, cystoid macular oedema was evident. In 14 of 28 eyes (all with type II JRT), a single foveal cyst was found in OCT. It varied significantly in size and was associated with visual acuity decrease. An intraretinal hyperreflective lesion was seen in eight of 28 eyes and flattening of the fovea in three eyes. CONCLUSIONS: Foveal cyst, absent macular oedema, intraretinal hyperreflective lesions and foveal flattening were the most common OCT findings in patients with JRT type II. These may represent progressive loss of retinal tissue, possibly due to Müller cells degeneration, and provide additional diagnostic criteria for JRT.

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PubMed2007

Optical coherence tomography findings in nonproliferative group 2a idiopathic juxtafoveal retinal telangiectasis.

PURPOSE: To determine the optical coherence tomography (OCT) findings in eyes with group 2a idiopathic juxtafoveal retinal telangiectasis (IJRT). METHODS: Forty-one eyes of 22 patients with nonproliferative group 2a IJRT were examined. OCT testing including retinal topographic mapping and analysis, and horizontal and vertical line scans, was obtained on each eye. RESULTS: None of the 41 eyes had a thickened foveal center. The average center foveal thickness was 166 microm (31-264 microm). Stage 1 eyes (n = 2) were normal fellow eyes in patients with contralateral group 2a IJRT. Stage 2 eyes (n = 11) all had parafoveolar temporal graying and intraretinal temporal fluorescein leakage, but rarely had photoreceptor disruption (18%) on OCT testing. Stage 3 eyes (n = 14) all had clinical and fluorescein findings similar to or more pronounced than stage 2 eyes. All stage 3 eyes also had one or more foveal cysts at various retinal depths on OCT. Most of these eyes (86%) had photoreceptor disruption and outer retinal atrophy on OCT. Stage 4 eyes (n = 14) all had a black foveal or parafoveolar pigment plaque and intraretinal temporal fluorescein leakage. All stage 4 eyes had a hyper-reflective plaque with shadowing on OCT corresponding to the pigment plaque. Most of these eyes had one or more foveal cysts (64%) and all of these eyes had photoreceptor disruption and outer retinal atrophy. CONCLUSION: OCT helps in the staging of group 2a IJRT and reveals multiple retinal structural abnormalities.

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PubMed2006

Morphologic features of group 2A idiopathic juxtafoveolar retinal telangiectasis in three-dimensional optical coherence tomography.

PURPOSE: To investigate morphologic features of group 2A idiopathic juxtafoveolar retinal telangiectasis (IJRT) with three-dimensional optical coherence tomography (OCT). DESIGN: Observational case series. METHODS: We performed OCT ophthalmoscopy on six eyes of three consecutive patients with group 2A IJRT. RESULTS: Thin cystic low-intensity space within inner and outer retinal layers was detected with the longitudinal B-scan through the fovea in five eyes. Partial loss of highly reflective line considered as the boundary between photoreceptor inner segments and outer segments was also detected. The transverse C-scan provided en-face images of the low-intensity space shown with the B-scan, if not entirely. These morphologic alterations appeared not to correspond with the staining observed in the late phases of fluorescein angiography. CONCLUSIONS: The OCT ophthalmoscope could visualize morphologic alterations indicating degeneration or atrophy of neurosensory retina, including photoreceptor layers. These alterations may play a role in the pathogenesis of group 2A IJRT.

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PubMed2006

Intravitreal bevacizumab (Avastin) associated with the regression of subretinal neovascularization in idiopathic juxtafoveolar retinal telangiectasis.

PURPOSE: To describe the clinical, angiographic, and optical coherence tomography findings of a patient with subretinal neovascularization (SRN) in idiopathic juxtafoveolar retinal telangiectasis (IJRT) treated with intravitreal bevacizumab injection. METHODS: In the setting of a tertiary referral center, a patient with bilateral acquired IJRT, complicated with juxtafoveal SRN in one eye, was treated with a single intravitreal injection of 1.5 mg of bevacizumab and then prospectively followed for 24 weeks. RESULTS: Vision improved from 20/40(-1) to 20/20(-2) by 24 weeks of follow-up. Fluorescein angiography demonstrated the absence of leakage from a previously active SRN. Optical coherence tomography revealed gradual neovascular lesion contraction and the resolution of subretinal fluid over the 24-week follow-up period. No obvious adverse events were noted. CONCLUSIONS: In the short-term, intravitreal bevacizumab treatment resulted in partial restoration of the macular architecture and vision improvement by means of neovascular lesion regression and the resolution of associated serohemorrhagic complications.

Angiogenesis Inhibitors↗
PubMed1992

[Parafoveal retinal telangiectasis and changes of the capillary free zone in non-diabetic patients].

Microaneurysms, hard exudates, hemorrhages and/or edema in the macular region were observed in 10 eyes of 10 non-diabetic patients. By fluorescein angiography, parafoveal retinal telangiectasis was found in all cases. Deformation, enlargement and boundary disruption of the foveolar avascular zone were discernible in most cases, as were unevenness of capillary spacing, small areas of non-perfusion and leakage of dye in some cases. Analysis and calculations of the foveolar avascular zone were done with the Model-970 Quantmet computerized image analysis system. The data showed that the values of circumference, vertical diameter and roundness of the avascular zone differed significantly from those of the 10 normal control eyes.

Adult↗
PubMed1985

Retinal telangiectasis in facioscapulohumeral muscular dystrophy with deafness.

A 22-year-old patient with newly diagnosed facioscapulohumeral (FSH) muscular dystrophy had a macular lesion in her right eye and poor central vision, which had been present since early childhood. Fluorescein angiographic examination revealed bilateral peripheral vessel closure, peripheral retinal telangiectasis, and hyperfluorescence in both foveae. This widespread vascular abnormality was deemed responsible for her macular disease. Her mother, brother, and sister, all of whom are affected by varying degrees of FSH muscular dystrophy and clinical deafness, also have abnormal retinal vasculature, as determined by fluorescein angiography. However, none had related visual symptoms and two showed no ophthalmoscopic evidence of vascular abnormalities. In young patients with unexplained retinal vascular lesions, the diagnosis of FSH muscular dystrophy should be considered. Similarly, young patients with FSH muscular dystrophy should be examined for sight-threatening and potentially treatable vascular retinopathy.

Adult↗
PubMed2005

Idiopathic juxtafoveal retinal telangiectasis: new findings by ultrahigh-resolution optical coherence tomography.

OBJECTIVE: To investigate the capabilities of ultrahigh-resolution optical coherence tomography (UHR OCT); to compare with the commercially available OCT standard-resolution system, StratusOCT, for imaging of idiopathic juxtafoveal retinal telangiectasis (IJT); and to demonstrate that UHR OCT provides additional information on disease morphology, pathogenesis, and management. DESIGN: Retrospective, observational, interventional case series. PARTICIPANTS: Nineteen eyes of 10 patients diagnosed with IJT in at least one eye. METHOD: All patients were imaged with UHR OCT and StratusOCT at the same visit. A subset of patients was also imaged before and after treatment of IJT. MAIN OUTCOME MEASURES: Ultrahigh- and standard-resolution cross-sectional tomograms of IJT pathology. RESULTS: Using both standard- and ultrahigh-resolution OCT, we identified the following features of IJT: (1) a lack of correlation between retinal thickening on OCT and leakage on fluorescein angiography, (2) loss and disruption of the photoreceptor layer, (3) cystlike structures in the foveola and within internal retinal layers such as the inner nuclear or ganglion cell layers, (4) a unique internal limiting membrane draping across the foveola related to an underlying loss of tissue, (5) intraretinal neovascularization near the fovea, and (6) central intraretinal deposits and plaques. In 63% of cases, the presence of abnormal vessels and a discontinuity of the photoreceptor layer correlated with visual acuity. CONCLUSIONS: Ultrahigh-resolution OCT improves visualization of the retinal pathology associated with IJT and allows identification of new features associated with it. Some of these features, such as discontinuity of the photoreceptor layer, are revealed only by UHR OCT.

Adult↗
PubMed2000

Idiopathic juxtafoveolar retinal telangiectasis: clinical pattern in 19 cases.

BACKGROUND: Idiopathic juxtafoveolar telangiectasis (IJT) is a rare retinal vasculopathy of adulthood that may cause central loss of visual acuity. The purpose of this paper is to evaluate the type and distribution of fundus changes in patients with this disorder. METHODS: Medical records of 19 patients with IJT using the Gass & Blodi classification were studied. Eight patients in group 1A (42%) and 11 in group 2A (58%) were analyzed. By projecting color photographs we divided the macula into four quadrants and studied the fundus changes in each one. RESULTS: All patients in group 1A presented unilateral disease with easily seen telangiectasis, lipid deposits and cystic macular edema. The mean age of this group was 55.8 years. Five (63%) patients were male and 3 (37%) were female. All patients in group 2A, 2 males (18%) and 9 females (82%), had bilateral disease evidenced by fluorescein angiography. The mean age of this group was 54.5 years. Fundoscopic findings included: intraretinal pigment plaques, grayish coloring of the macular retina, superficial crystalline deposits, right-angle venules, subretinal and intraretinal neovascularization. CONCLUSIONS: All patients in this survey were adults. In group 1A, men were affected 2 times more often than women, and the main affected macular quadrant was the inferior temporal, followed by the inferior nasal. In group 2A, women were affected 4 times more often than men and all eyes had inferior and superior temporal macular quadrant involvement.

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PubMed1982

Idiopathic juxtafoveolar retinal telangiectasis.

Twenty-seven healthy adult patients had visual loss in one or both eyes because of exudation from juxtafoveolar retinal capillary telangiectasis of uncertain cause. These patients were subdivided as follows: group 1, men with uniocular involvement, intraretinal lipid exudation, and telangiectasis largely confined to the temporal half of the juxtafoveolar area; group 2, mostly men with symmetric areas of telangiectasis affecting the temporal half of the juxtafoveolar areas and minimal intraretinal exudation; group 3, both sexes with symmetric involvement of all of the parafoveolar capillary bed an minimal exudation; and group 4, one case of telangiectasis with occlusive perifoveolar capillary changes and familial optic disc pallor. The visual acuity prognosis in groups 1 through 3 is relatively good. Photocoagulation may be of some value in the treatment of patients in group 1.

Adult↗
PubMed1983

Peripheral retinal telangiectasis in adults simulating a vascular tumor or melanoma.

In ten adult patients a localized peripheral retinal vascular lesion is described, which is characterized by retinal telangiectasia, formation of aneurysms, deposition of lipoidal material and exudative detachment. All patients were of middle or older age and men and women were affected equally. Small lesions had a definite association with the most peripheral vessels, whereas larger ones appeared more as a tumor-like mass causing exudative detachment with deposition of lipoidal material. Additional neovascularization was present in at least two of the ten patients. The lesion is of clinical importance because it is often the cause of loss of central vision and is often confused with a vascular tumor or a choroidal melanoma.

Adult↗
PubMed1989

[Asteroid hyalitis: an uncommon complication of retinal telangiectasis].

The authors report a case of asteroid hyalosis which required a vitrectomy because of the decreased vision. The vitrectomy places in a prominent position an epi-macular membrane which has been peeling during the procedure and retinal vascular malformations like "telangiectasies" which are the starting point of other complications.

Adult↗
PubMed2004

Photodynamic therapy of subfoveal neovascular membrane in type 2A idiopathic juxtafoveolar retinal telangiectasis.

PURPOSE: To evaluate the effect of photodynamic therapy on subfoveal neovascular membrane related to type 2A idiopathic juxtafoveolar retinal telangiectasia. DESIGN: Interventional case series. METHODS: Retrospective review of four eyes of four patients who underwent photodynamic therapy for subfoveal neovascular membrane secondary to idiopathic juxtafoveolar retinal telangiectasia. Ocular photodynamic therapy with verteporfin was performed in all cases using standard protocols. Results are given in terms of final visual acuity and neovascular membrane activity based on clinical examination, fluorescein and indocyanin green angiography, and, in two cases, optical coherence tomography. RESULTS: Baseline visual acuity of 20/30 and 20/40 (x2) was maintained in three patients after one, two, and three sessions of photodynamic therapy respectively, and a follow-up of 23, 21, and 9 months. Leakage specific to the subfoveal neovascular membrane ceased on the fluorescein angiography. In the other patient, the final vision decreased from 20/50 to 20/200 after four sessions of photodynamic therapy and a follow-up of 14 months. Although there was still mild persistent leakage on the fluorescein angiography, neovascular membrane size was unchanged, and no subretinal fluid was demonstrated on optical coherence tomography. CONCLUSIONS: Data from this case series suggest that photodynamic therapy may be effective in managing subfoveal neovascular membrane associated with idiopathic juxtafoveolar retinal telangiectasia, which usually carries a poor visual prognosis. Prospective study is required to confirm the beneficial effect of this treatment.

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