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PubMed1991

[The antiphospholipid syndrome. The neurological complications and the therapeutic possibilities].

For several months a 23-year-old woman had been suffering from increasingly frequent attacks of bilateral amaurosis fugax. They affected only individual segments of a monocular visual field and finally occurred several times daily. Physical and cardiological examinations as well as echocardiography were within normal limits. Laboratory tests revealed a slight increase in erythrocyte sedimentation rate and high titres of serum antibodies against phospholipids. Fundoscopy did not show any vascular changes. Because vasculitis--possibly as an expression of systemic lupus erythematodes--was suspected, high-dosage treatment with steroids was commenced, but failed to influence the visual disorder. While steroid dosage was gradually decreased, administration of acetylsalicylic acid (for three months 100 mg daily, then three times daily 100 mg) brought about complete disappearance of the visual signs. Their cause was probably a reversible platelet aggregation induced by antiphospholipid antibodies which, because of the physiological characteristics of the terminal retinal vascular bed brought about the isolated sign of recurrent amaurosis fugax.

Adult↗
PubMed2001

[Central serous chorioretinopathy and systemic steroid therapy].

BACKGROUND: The manifestations of the ocular toxicity of systemic corticosteroids include posterior subcapsular cataracts and glaucoma. We describe 14 cases of serous detachment of the macula due to central serous chorioretinopathy in patients given long-term steroid therapy, which may be another potential ocular side effect of corticosteroid. CASES REPORT: The 14 (9 men and 5 women) patients were aged from 39 to 55 year old. Their systemic diseases were allergic thrombopenic purpura, optic neuritis, kidney or heart transplant, Churg and Strauss vasculitis, facial palsy, rheumatoid arthritis, systemic lupus and a kidney tumor. None of the patients had hypertension. RESULTS: Serous detachment occurred between 6 days and 10 years after the start of steroid treatment. The higher the doses, the earlier the onset of ocular disease. All patients were symptomatic, with rapid onset of blurred vision. Serous detachment was bilateral in two cases. The fluorescein angiographic finding was in most cases a single small focal hyperfluorescent leak from the retinal pigment epithelium which appeared early in the angiogram and increased in size and intensity. No diffuse degradation of the retinal pigment epithelium was seen on the fluorescein angiogram. Five patients underwent laser photocoagulation of the leaking area followed by resorption of subretinal fluid. In other patients, the symptoms disappeared as the doses of steroid were reduced. CONCLUSION: The pathogenesis of central serous chorioretinopathy remains unclear and is controversial. Corticosteroids are known to worsen the prognosis of idiopathic central serous chorioretinopathy, and serous detachment has been reported after renal transplantation. In most of these cases, chorioretinopathy was combined with diffuse leakage from the choriocapillaris. We discuss the relationship between steroid therapy and focal leakage as seen in idiopathic central serous chorioretinopathy. In conclusion, we describe 14 cases of central serous retinopathy whose clinical and fluorescein angiography were fairly typical, without obvious diffuse degradation of the retinal pigment epithelium. All these patients had been given long-term steroid therapy for various diseases.

Adrenal Cortex Hormones↗
PubMed2006

[A new approach towards giant cell arteritis].

Giant cell arteritis is a rare systemic vasculitis affecting large- and medium-sized arteries. Focal arteries lesions, include mononuclear cells infiltration of the vessel wall with giant cell formation. It is a disease of elderly persons and can result in a wide variety of systemic, neurological and ophthalmic complications, due to ischemia. The incidence of visual loss and ocular involvement varies between 14-88%, but one of the most common and severe complications is anterior ischemic optic neuropathy. The other ocular ischemic lesions include: central retinal artery occlusion, choroidal ischemia, diplopia, ocular motor paresis, anterior uveitis, cataract, ocular hypotony, corneal oedema and ulcerations, episcleritis and anterior scleritis, orbital cellulitis and pseudotumor. Because giant cell arteritis is potentially blinding disease, early diagnosis and immediate treatment with high dose corticosteroids may prevent further damage to the affected eye and prevent visual loss in the opposite eye. The purpose of this review is to revise established knowledge and to highlight the recent developments in diagnosis and management of giant cell arteritis.

Aged↗
PubMed2001

Unilateral frosted branch angiitis with vitreous haemorrhage.

Idiopathic retinal frosted branch angiitis is a rare bilateral condition and presents with an acute reduction of vision. Unilateral affection is rare. We report an unusual case of unilateral frosted branch angiitis where the patient developed total vitreous haemorrhage.

Fluorescein Angiography↗
PubMed2026

Whole exome sequencing of paediatric patients with Cogan's syndrome to identify monogenic mimics.

OBJECTIVES: Cogan's syndrome (CS) is a rare variable vessel vasculitis, describing sensorineural hearing loss (SNHL), inflammatory ocular disease and vestibular dysfunction. We hypothesized that within paediatric-onset (p)CS, a proportion would have monogenic disease, either autoinflammatory and/or associated with SNHL. METHODS: Whole exome sequencing (WES) was performed and analysed using an in-house pipeline incorporating virtual gene panels for inflammation and SNHL; copy number variant analysis (ExomeDepth); and phenotype-driven variant prioritization (Exomiser). Genetic variants were interpreted by a multi-disciplinary team according to American College of Medical Genetics and Genomics guidelines. RESULTS: Ten patients with a clinical diagnosis of pCS were enrolled. Three/10 (30%) had a monogenic contribution to the phenotype based on Class 4/5 variants: de novo NLRP3 p.T915R (n = 1) associated with Cryopyrin-associated periodic syndrome; MYO7A p.K542Qfs*5 (n = 1) causing SNHL; and HBB homozygous p.E7V causing sickle cell disease (associated with hearing loss and uveitis). A further two cases had possible monogenic contribution with the following rare variants of uncertain significance (class 3): ADGRV1 compound heterozygous variants (n = 1) associated with Usher syndrome; and a novel ALPK1 p.H735P (n = 1), associated with Retinal dystrophy Optic nerve oedema Splenomegaly Anhidrosis Headache (ROSAH) syndrome. CONCLUSIONS: In children presenting with features suggesting CS, genetic screening should be considered before conferring this rare diagnostic label since at least 30% had an alternative monogenic contribution to the phenotype rather than true pCS, with implications for treatment and prognosis. We thus advocate for genetic testing using next-generation sequencing for patients presenting with pCS.

Humans↗
PubMed2004

[Eales disease and tuberculous allergy].

The authors report the case of a 25-year-old woman who presented unilateral Eales disease associated with biologically confirmed pulmonary tuberculosis. The patient, from a family with a history of tuberculosis, showed a painless and abrupt decrease in visual acuity of the right eye with venous and arterial vasculitis. Fluorescein angiography confirmed the existence of peripheral ischemia and vascular abnormalities. The biological exam showed a positive PPD and the BK was isolated. Antibiotic and corticosteroid drugs were used in the treatment. The clinical, immunopathological and therapeutic aspects of this disease are discussed.

Adult↗
PubMed1983

Neuro-ophthalmologic signs in the angiitic form of neurosarcoidosis.

We studied eight patients with CNS sarcoidosis, including two patients with postmortem evidence of sarcoid granulomatous angiitis. Neuro-ophthalmologic findings included perivenular sheathing of retinal veins resembling the dripping of candle wax, vitreous cells, anterior uveitis, optic disk edema or granulomas, and conjunctival granulomas. Perivenous sheathing in the retina and the perivascular meningeal lesions in sarcoid angiitis were identical.

Adult↗
PubMed1998

Arterial macroaneurysms in peripheral multifocal chorioretinitis associated with sarcoidosis.

OBJECTIVE: This study aimed to describe the clinical presentation, angiographic findings, and natural history of acquired arterial macroaneurysms diagnosed in 8 of 48 patients with peripheral multifocal chorioretinitis (PMC) (panuveitis with multiple peripheral punched-out chorioretinal lesions). DESIGN: A retrospective case-control study. PARTICIPANTS: Forty-eight consecutive patients with PMC, consisting of 13 patients with histologically or radiologically proven sarcoidosis, 11 with laboratory evidence of (presumed) sarcoidosis, and 24 with no evidence of systemic disease, participated in the study. INTERVENTION: A review of the clinical, photographic, and angiographic records was performed. MAIN OUTCOME MEASURES: Arterial macroaneurysms, clinical symptoms, and associations with systemic disease were measured. RESULTS: Of 48 patients with PMC, 8 (17%) had arterial macroaneurysms: 3 (38%) of 8 with histologically proven sarcoidosis, 1 (20%) of 5 with radiologically proven sarcoidosis, 2 (18%) of 11 with laboratory evidence of sarcoidosis, and 2 (8%) of 24 patients with no evidence of systemic sarcoidosis. All eight patients were white females older than 60 years of age. Macroaneurysms appeared to be unrelated to the systemic hypertension (5 of 17 compared to 3 of 31; P = 0.11); however, the frequency of cerebrovascular accidents or myocardial infarctions was higher among those with macroaneurysms (5 of 8 vs. 2 of 40; P = 0.0000). Three of the eight patients reported visual complaints related to macroaneurysms; only one patient required laser treatment. Fluorescein angiography showed no evidence of previous vasculitis in the involved area. CONCLUSIONS: Arterial macroaneurysms, which occur in elderly female patients with PMC, are associated with severe cardiovascular disease. Whether the macroaneurysms are a complication of an inflammatory vascular process remains speculative.

Age of Onset↗
PubMed1999

[Churg-Strauss syndrome].

BACKGROUND: The Churg-Strauss syndrome, also known as allergic granulomatosis with angiitis, is a rare necrotizing vasculitis with unknown pathogenesis. The necrotizing granulomatous vasculitis involves small and medium-sized arteries, capillaries and veins, leading to the characteristic changes of intra- and extravascular, eosiniphilic granulomas, accompanied by clinically symptoms: bronchial asthma, hypereosinophilia and fever. Ocular manifestations are rarely reported. CASE REPORT: A 53-year-old woman suffered from bronchial asthma, relapsing lung infiltrates and sinusites since 1994. In August 1997 she suddenly disclosed vasculitic skin manifestations on both legs and a mononeuritis of the left peroneus nerve. At the same time visual acuity of the right eye decreased, before she had shown some attacks of amaurosis fugax. The funduscopy showed a central retinal artery occlusion. LABORATORY FINDINGS: blood eosinophilia of 20%, elevated IgE value to 396 kU/l (normal value < 120 kU/l), and negative parameters for antineutrophil cytoplasmatic antibodies (p- and c-ANCA). CONCLUSION: The clinical and laboratory findings are characteristic signs for the Churg-Strauss syndrome. Without such typical manifestations the histologic examination leads to the diagnosis and helps to differentiate this disease from other necrotizing vasculitides, e.g. panarteriitis nodosa or Wegener's granulomatosis.

Azathioprine↗
PubMed1983

Acute multifocal ischaemic choroidopathy and the choriocapillaris.

Acute multifocal ischaemic choroidopathy (AMIC) seems to be a better name than acute posterior multifocal placoid pigment epitheliopathy (A.P.M.P.P.E.). Fluorescein angiography indicates obstruction of the precapillary choroidal arterioles leading to non perfused choriocapillaris lobules and focal swelling of the retinal pigment epithelium at the site of the lesions. Furthermore in many fluorescein angiograms of human patients the specific mosaic pattern of the choriocapillaris can be observed, which was described in monkeys by Hayreh & Baines (7). The choroid has, indeed, an endarterial system. The special conditions in which a well-defined appearance of the choriocapillaris occurs are listed.

Choroid↗
PubMed1996

[Giant cell arteritis and normal sedimentation rate: more than an exception!].

BACKGROUND: Giant cell arteritis (GCA) is a systemic segmental vasculitis of unknown etiology, typically affecting elderly patients. Elevated erythrocyte-sedimentation rate (ESR) is usually found in such patients. PATIENTS AND METHODS: One hundred and twenty three patients underwent temporal artery biopsy in our institution between 1977 and 1995. Among them, 66 (53.7%) biopsies were positive (i.e. histologic findings were very suggestive of GCA). The clinical charts from all patients with positive biopsies were retrieved and 47 were eligible for our study (inadequate data in 19 cases). RESULTS: Seven of the 47 patients with positive biopsies (15%) had a normal ESR and 70% (33/47 cases) had neuro-ophthalmic complications including anterior ischemic optic neuropathy, central retinal artery occlusion, choroidal ischemia and extraocular muscle and/or cranial nerve palsy (III, IV, VI). No differences were found between the groups with normal or elevated ESR as 87.5% (6/7 cases) of the group with normal ESR exhibited neuro-ophthalmic complications. CONCLUSIONS: ESR was normal in 15% of our GCA patients and these patients had the same frequency of neuro-ophthalmic complications as the GCA patients with elevated ESR. Thus, our study does not support the previous concept that patients with higher ESR are more at risk for neuro-ophthalmic complications. GCA with normal ESR is not rare and such patients should be investigated with other blood studies (C-reactive protein) and with fluorescein angiography.

Aged↗
PubMed2006

[Temporal arteritis (giant cell arteritis). Clinical picture, histology, and treatment].

Giant cell arteritis (arteritis temporalis) is the most common form of systemic vasculitis in the elderly. A series of symptoms such as new-onset headache, jaw claudication, proximal myalgia, weight loss, and fever may lead to the diagnosis. However, there is also a silent or occult presentation with minor or no systemic symptoms, especially no headache. A number of laboratory values (erythrocyte sedimentation rate, CRP, fibrinogen, thrombocytes, and cardiolipin antibodies) indicate giant cell arteritis, but none of this proves the diagnosis. Temporal artery biopsy is the gold standard for diagnosis of giant cell arteritis. Due to skip lesions, a negative result does not exclude the diagnosis. The most important complication of giant cell arteritis is visual loss in one or both eyes due to AION or retinal artery occlusion. Usually, visual loss is irreversible even with therapy. Corticosteroids are the drug of choice to treat giant cell arteritis. Therapy is required for a long time, monitored by parameters of inflammation (ESR, CRP).

Adrenal Cortex Hormones↗
PubMed1983

Churg-Strauss syndrome (allergic granulomatous angiitis). Neuro-ophthalmologic manifestations.

Two patients with severe bronchial asthma, hypereosinophilia, and peripheral neuropathy were initially observed with neuro-ophthalmologic signs and symptoms that included amaurosis fugax, superior oblique palsy, ischemic optic neuropathy, and scattered areas of retinal infarction. Clinical investigation led to the diagnosis of Churg-Strauss syndrome (allergic granulomatous angiitis). As in other collagen vascular diseases, ocular signs or symptoms may occasionally be the most prominent manifestation of the disease. Recognition of this systemic disorder by the ophthalmologist may minimize systemic and ocular complications.

Asthma↗
PubMed2004

[Syphilitic chorioretinitis in the immunocompromised patient: a case report].

Syphilitic chorioretinitis is a rare manifestation of secondary syphilis. The Authors report a case of a patient tested positive for human immune deficiency virus. A 26-Year-old man presented with a history of suddenly blurred vision in the right eye. Because of a central scotoma, visual acuity was reduced to counting fingers. Slit-lamp examination found one-plus anterior chamber cells and one-plus vitreous cells. Results of ophthalmoscopic examination showed a large yellow lesion in the macular area with a papillar edema and peripheral vasculitis. Serum and cerebrospinal fluid were positive for syphilitic serology. Visual acuity improved to 20/20 after three courses of intravenous ceftriaxone (2g/day for 15 days). The fundus lesion resolved almost completely, resulting in slight pigmentary changes in the macular area. This case report highlights the need for syphilitic serology in cases of uveitis in immunocompromised patients.

Adult↗
PubMed2003

Indocyanine green angiography in chorioretinal diseases: indications and interpretation: an evidence-based update.

UNLABELLED: TOPIC/PURPOSE: To assess the clinical usefulness and relevance of indocyanine green angiography (ICG) in the investigation of chorioretinal disorders and assess specifically in what conditions it may add useful information to that obtained using standard fluorescein angiography. CLINICAL RELEVANCE: Many publications on ICG have appeared in recent years touting its use in ophthalmology. These publications have led to increasing use of this technique and to its application in numerous retinal diseases in which the fluorescein angiographic findings have been thoroughly described. METHODS/LITERATURE REVIEWED: During this systematic literature review, we identified and reviewed a total of 376 articles, from among which we selected 92 that we considered most relevant to our purpose of evaluating published evidence as to the efficacy of ICG. We excluded many articles with weak study designs and those that simply duplicated previously published information. Our literature search used PubMed and was confined to articles in English or that included an English abstract. RESULTS: Our systematic review suggests that ICG has relatively few specific indications for use as justified by previously published peer-reviewed studies. In keeping with the requirements for this Journal's evidence-based articles, we have divided our clinical recommendations for the use of ICG into three categories: (A) strongly recommended and supported by strong evidence; (B) recommended with moderately strong supporting evidence; (C) not recommended at present because supported only by anecdotal or group consensus evidence. We highly recommended ICG for (1) identification of polypoidal choroidal vasculopathy, (2) occult choroidal neovascularization, (3) neovascularization associated with pigment epithelial detachments, and (4) recurrent choroidal neovascular membranes. These are all conditions in which ICG contributes to the identification of lesions that may be treatable. We recommend ICG with some enthusiasm for identifying feeder vessels in age-related macular degeneration, choroidal neovascular membranes, chronic central serous retinopathy, multiple evanescent white dot syndrome, vasculitis, acute multifocal placoid pigment epitheliopathy, Vogt-Koyanagi-Harada syndrome, macular lesions associated with angioid streaks, and birdshot retinopathy. In all these conditions, ICG may help establish a diagnosis and provide some useful guidance for therapy. At present, we do not recommend ICG for scleritis and posterior scleritis, drusen differentiation, Behçet's disease, or sarcoidosis, because it has not been demonstrated to add useful clinical information. CONCLUSIONS: ICG, although now a well established technique, has clear advantage over fluorescein angiography in relatively few chorioretinal disorders. It has, however, contributed to the understanding of pathologic processes in many ocular diseases. As yet, no published randomized controlled clinical trials show any benefit to the use of ICG in the management of any specific ocular disease.

Choroid↗
PubMed1997

Nocardia scleritis.

PURPOSE: To describe a case of Nocardia scleritis, an unusual ocular infection. METHODS: Case report and review of pertinent literature. RESULTS: An 83-year-old man with leukocytoclastic vasculitis was initially examined for infectious necrotizing scleritis after explantation of an extruded scleral buckle. The patient was successfully treated with sulfonamides. CONCLUSIONS: Nocardia asteroides may cause infectious scleritis in the absence of cataract surgery or trauma. Treatment with sulfonamides can result in a satisfactory outcome.

Aged↗
PubMed1989

Transient ocular motor paresis associated with acute internal carotid artery occlusion.

While sudden monocular blindness and occlusion of the central retinal artery associated with acute thrombosis of the internal carotid artery are well reported, concurrent unilateral ophthalmoparesis is not. We studied 3 adult men who did not have other major signs of vascular disease but who exhibited the complete syndrome. The initial paresis of the oculomotor, trochlear, and abducens nerves varied in each individual and slowly cleared over days to weeks in all, but vision did not return in any. Each had mild-to-moderate signs of hemispheric dysfunction. In each, the thrombus extended from the origin of the internal carotid to its intracranial bifurcation into the anterior and middle cerebral arteries. It also occluded the proximal one-half to two-thirds of the ophthalmic artery. None had evidence of vasculitis or compromise of the posterior circulation.

Adult↗
PubMed1990

Cerebrovascular and neurologic disease associated with antiphospholipid antibodies: 48 cases.

Lupus anticoagulants and anticardiolipin antibodies are antiphospholipid antibodies (APLAb) with related antigenic specificities and are newly recognized markers for an increased risk of thrombosis. We studied 48 patients who presented with cerebral or visual dysfunction associated with APLAb to help clarify the diagnostic, clinical, laboratory, radiologic, and pathologic features in these patients. Most patients presented with transient cerebral ischemia or cerebral infarction. Recurrent and stereotypic events were frequent. Visual disturbances resulted from amaurosis fugax, retinal arterial or venous occlusion, occipital ischemia, diplopia, and migraine-like disturbances. Three patients presented with severe atypical classic migraine. Recurrent infarcts of brain and eye were significantly associated with the presence of cigarette smoking, hyperlipidemia, and a positive antinuclear antibody. During 44.4 patient-years of prospective follow-up, the combined stroke and systemic thrombotic event rate was 0.27 events per patient-year and was 0.54 events per patient-year if TIA and death were included. Forty (83%) of the patients did not have systemic lupus erythematosus (SLE). Thrombocytopenia was present in 15 (31%) and a false-positive VDRL in 11 (23%) of the patients. Cerebral angiography was normal or revealed large-vessel occlusion or stenosis without changes suggestive of vasculitis. Patients with only transient dysfunction generally had normal radiologic studies, including angiography. Organs and arterial vessels studied pathologically revealed thrombotic occlusive disease without vasculitis. APLAb are strongly associated with an immune-mediated thrombotic tendency, generally in the absence of SLE. Other stroke risk factors may add to the risk of recurrent ischemic events in patients with APLAb.

Adult↗