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PubMed2005

[Group 2A idiopathic parafoveal telangiectasis].

CASE REPORT: Two cases of group 2A idiopathic parafoveal telangiectasis associated with abnormal glucose metabolism are reported with the typical sings of this disease as well as a foveal vitelliform lesion in one patient, an infrequent association. DISCUSSION: Group 2A idiopathic parafoveal telangiectasis are a disease with well characterized clinical signs, being some very infrequent such as a vitelliform maculopathy. Its pathogenesis seems to be linked to some alterations in the parafoveal capillary network endothelial cells. These alterations are similar to those that appear in the beginning of the diabetic retinopathy.

Aged↗
PubMed1999

[Temporal lobe epilepsy associated with old intracerebral hemorrhage due to capillary telangiectasis in the temporal lobe: case report].

A case of 45-year-old female, who presented with temporal lobe epilepsy was reported. The patient was found to have old intracerebral hemorrhage due to capillary telangiectasis in the temporal lobe. On the intraoperative electrocorticography, frequent paroxysmal activities were recorded independently both on the medial and lateral aspects of the temporal lobe. Even after resection of the lateral temporal lobe, frequent paroxysmal discharges were noted on the hippocampus. Histologically, there are astrogliosis and hemosiderin deposits in the white matter around the telangiectasis and the old hematoma. It is postulated that the hippocampus gained secondary epileptogenicity.

Adult↗
PubMed2001

Idiopathic juxtafoveal retinal telangiectasis: case report and literature review.

BACKGROUND: Idiopathic juxtafoveal retinal telangiectasis (IJRT) is an uncommon cause of vision loss. It is characterized by irregularly dilated capillaries in the macula, often found temporal to the fovea and straddling the horizontal raphe. These telangiectatic vessels appear similar to microaneurysms and can be difficult to detect without fluorescein angiography. Varying degrees of vessel leakage and exudation are also common features. Three distinct types of IJRT have been described on the basis of clinical features. Within the spectrum of disease encompassed by IJRT, it can affect either gender--present unilaterally or bilaterally--and occur at any age. CASE REPORT AND REVIEW: Presented here is a case with type 1A idiopathic juxtafoveal retinal telangiectasis and a review of all types.

Diagnosis, Differential↗
PubMed1975

[Variable pseudoerythroplasic telangiectasis balanitis].

This is a special case of balanitis, that authors separate from entities clinically established such as Erthroplasie of Queyrat, Balanitis of Zoon, Liquenoide Balantis with Plasmocytes and the Balantis of Sulsberger and Garbe's illness. The V. P. T. B. is clinically characterized by the presence of telangiectasies, ertroplasiform aspect, without any infiltration, non purpure, the V. P. T. B. goes through a first period truly esythematous and a second one in which these is also desquamation. That cycle is completed in a month. At the histopathologic level, the most important characteristics are: epidermis with its Malpighian layer in a normal state, the basal layers showing hidropic degeneration. The repper dermis shows a lichenoid picture that, in certain places affects the basal layer. The infiltrate is composed of: lymphocytes, monocytes and plasmocytes. Numerous telangiectasies are also observed.

Adult↗
PubMed1982

Cerebral capillary telangiectasis in an infant. Coincidental Association with hypsarrhythmia.

We studied a case of cerebral capillary telangiectasis in infancy. The patient, who had received treatment with intramuscular corticotropin for hypsarrhythmia, suffered disseminated intravascular coagulation and died on the 66th day of life. Neuropathological examination disclosed the presence of two fresh hemorrhages of moderate size within the cerebral hemispheres, one of which seemed to have originated from a capillary telangiectasis within the left frontoorbital white matter.

Brain↗
PubMed1980

Parafoveal retinal telangiectasis. Light and electron microscopy studies.

The clinical, histopathological, and ultrastructural features of a case of parafoveal telangiectasis are reported. No telangiectasis of retinal vessels was found. Instead, there was thickening of the wall of the retinal capillaries and narrowing of the calibre of the lumen. Thickening was due to marked proliferation of basement membrane in a multilayered configuration, Cellular debris from degenerated endothelial cells and pericytes, and multimembranous lamellar lipid material were entrapped between layers of basement membrane. Extensive degeneration of pericytes was observed, and occasional areas showed degenerated endothelial cells. These changes were not restricted to the clinical-affected parafoveal area but were also seen to a lesser degree throughout the retina. The changes are interpreted as being due to primary endothelial cell degeneration and regeneration, with successive waves of basement membrane production and secondary degeneration of pericytes.

Basement Membrane↗
PubMed1993

Familial parafoveal telangiectasis.

We report two cases of parafoveal telangiectasis encountered in the same family. The patients are father and son. Both patients are healthy. Both presented with small parafoveal telangiectatic vessels, with minimal associated subretinal fluid. There were no other associated conditions. To the best of our knowledge these cases represent the first reported cases of father-to-son transmission of parafoveal telangiectasis.

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↗
PubMed2002

Neovascular membranes associated with idiopathic juxtafoveolar telangiectasis.

OBJECTIVE: To report the visual outcome in patients with a neovascular membrane (NVM) associated with idiopathic juxtafoveolar telangiectasis (IJFT). METHODS: We performed a retrospective, noncomparative analysis of 26 eyes of 16 patients with an NVM associated with bilateral IJFT (Gass classification group 2A). Eyes were divided into 2 groups: group WO (n = 11) included eyes with IJFT without evidence of an NVM on initial examination; eyes in group W (n = 15) had an NVM at the initial diagnosis of IJFT. In group WO, the initial visual acuity and the time between the initial examination to the diagnosis of an NVM were evaluated. Characteristic fundus findings, including the presence or absence of a chorioretinal anastomosis, intraretinal pigmentary plaques, and crystalline deposits, as well as the final visual acuity were reviewed for both groups. RESULTS: The initial visual acuity for eyes in group WO ranged from 20/20 to 20/70 (median, 20/30); in group W, from 20/20 to 4/200 (median, 20/70). The average time from initial diagnosis of IJFT to the development of an NVM was 73 months (range, 5-142 months). In group WO, chorioretinal anastomosis and concurrent perivascular retinal pigment epithelial hyperplasia were observed before the development of an NVM. The final visual acuity for all eyes ranged from 20/40 to 2/200 (median, 20/200). Eighty-one percent of eyes (21/26) had a final visual acuity of 20/200 or worse. CONCLUSIONS: The stable final visual acuity in patients with an NVM associated with IJFT is generally poor, with 80% of eyes in this series having a final visual acuity of l20/200 or worse. In patients with IJFT, the presence of a chorioretinal anastomosis and retinal pigment epithelial hyperplastic plaques always preceded the development of an NVM.

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↗
PubMed1977

Capillary telangiectasis of the brain in chimpanzee.

The occurrence of capillary telangiectasis of the brain in a chimpanzee is reported. The telangiectases were multiple and diffusely scattered throughout the brain although the cerebral and cerebellar cortex were particulary affected. Hemorrhage into surrounding gliotic parenchyma was present. The were associated neuroligic sings, most significantly paralysis and convulsions, and the outcome was fatal. A Case of this nature has not previously been reported.

Animals↗
PubMed1995

Ruptured bronchial artery aneurysm associated with pleural telangiectasis and tortuous portal obstruction: report of a case.

A 25-year-old woman presenting with an emergent condition of massive hemothorax due to a ruptured bronchial artery aneurysm was successfully treated by transcatheter arterial embolization. She had previously undergone portosystemic shunt splenopneumopexy for hepatic portal hypertension at 6 years of age. When undergoing right thoracotomy for the removal of a clot, a prominent telangiectasis on the pleural surface was noted. The lesion appeared to be a rare systemic vascular abnormality although this could not be confirmed.

Adult↗
PubMed2006

Subretinal neovascular membrane associated with type 2a idiopathic juxtafoveolar telangiectasis in pseudoxanthoma elasticum.

BACKGROUND: The objective was to report a case of pseudoxanthoma elasticum associated with type 2A idiopathic juxtafoveolar telangiectasis (T2AIJT) complicated by bilateral subretinal neovascular membranes (SRNVM). METHODS: The right eye with juxtafoveal SRNVM and the left eye with subfoveal SRNVM underwent laser photocoagulation and photodynamic therapy respectively. The follow-up duration was 48 months. RESULTS: Best corrected visual acuity was 20/32 in both eyes at baseline, and 20/32 in the right eye and 20/200 in the left eye at the end of the follow-up. Atrophic changes in the retinal pigment epithelium developed around the SRNVM treated with photodynamic therapy (PDT). CONCLUSIONS: Pseudoxanthoma elasticum may be associated with T2AIJT complicated by bilateral SRNVM. Further studies are needed to assess both efficacy and appropriate PDT regime in T2AIJT-related SRNVM, especially if associated with other conditions.

Coloring Agents↗
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.

Aged↗
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↗
PubMed2004

Transpupillary thermotherapy for subfoveal neovascularization secondary to group 2A idiopathic juxtafoveolar telangiectasis.

PURPOSE: To evaluate the efficacy and safety of transpupillary thermotherapy (TTT) for subfoveal neovascularization (SRNVM) in patients with group 2A Idiopathic Juxtafoveolar Telangiectasis (IJFT). DESIGN: Nonrandomized interventional case series. METHODS: We performed TTT for subfoveal SRNVM in 14 eyes of 13 patients with group 2A IJFT, who were referred to our tertiary care center. We evaluated visual outcome and SRNVM closure rate in these patients. RESULTS: After a mean follow-up period of 8.65 months, 92.3% of treated eyes had stabilization or improvement in visual acuity as well as regression of SRNVM by fluorescein angiography (FA). One SRNVM showed persistent leakage. One patient worsened by more than 2 Snellen lines; one required retreatment. CONCLUSION: Transpupillary thermotherapy may be a safe and useful alternative treatment option for patients with group 2A IJFT with subfoveal SRNVM.

Adult↗
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.

Aged↗
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↗