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PubMed2002

[Efficacy of intravenous immunoglobulin in the treatment of Birdshot retinochoroiditis].

AIM: To evaluate the efficacy of intravenous immunoglobulin (IVIg) in the treatment of Birdshot retinochoroiditis. PATIENTS AND METHODS: Eighteen patients were followed-up prospectively in a total number of 37 patients recorded. Inclusion criteria were birdshot retinochoroiditis (BRC) according to the criteria defined by Ryan et al., and a decrease in visual acuity (VA). Efficacy was assessed by measurements of visual acuity and a decrease in inflammation and macular edema on fluorescein angiograms. RESULTS: Sex ratio=1, mean age was 51 (range 29 to 72 years), HLA A29 was positive in 100% of the patients. VA at baseline was 0.6+/-2.4 (range 0.25 to 0.9). Angiography showed retinal vasculitis in 32 patients (86.48%) and cystoid macular edema in 16 patients (43.24%). IVIg was administered at a dosage of 0.4g/kg/d for 4 days then 0.6g/kg/d for 2 days every 4 weeks. Follow-up lasted a mean of 2.7+/-2.0 years (range, 4 months to 5.6 years). Visual acuity of 35 out of 66 eyes (53%) increased by 2.6+/-1.5 (range 1 to 5). In 19 eyes out of 66 (29%), VA remained stable and in 12 eyes out of 66 (18%), VA decreased by -1.8+/-0.8 (ranges-4 to -1). Inflammation on fluorescein angiography improved in 17 patients (81%) and cystoid macular edema decreased in 65% of the cases. Side effects were rare. Treatment was discontinued for side effects in only 3 patients. DISCUSSION: BRC is a chronique disease threatening vision. Treatments including systemic corticosteroids and cyclosporin A may result in severe long-term side effects. IVIg is well tolerated and may be therapeutic alternative for the treatment of BRC.

Adult↗
PubMed2002

[Approach to the patient with acute anterior uveitis].

Uveitis refers to inflammation of the uveal tract, which includes the iris, ciliary body, and choroid. Common usage has expanded this definition, however, to include intraocular inflammation of any type. Hence, retinitis, retinal vasculitis, and papillitis are also grouped under uveitis, even in the absence of significant uveal tract inflammation. Uveitis can also be secondary to corneal, scleral, or orbital inflammation. Experience has shown that identifying the primary focus of intraocular inflammation is important, both for establishing a diagnosis and as a predictor of natural history and response to therapy. A number of ocular and systemic syndromes are recognized to cause anterior uveitis. Clues to the diagnosis of the more commonly encountered anterior uveitis syndromes are outlined below. It is important to pay particular attention to the severity of the anterior chamber inflammation, and to note the presence of anterior granulomatous findings since the intensity and character of the inflammation can sometimes suggest a diagnosis.

Acute Disease↗
PubMed2002

[Onset of Behçet's disease as meningoencephalitis and sudden blindness].

CASE REPORT: A 41-year-old man presented a clinical picture characterized by lymphocytic meningoencephalitis, visual loss in both eyes and transverse sinus thrombosis. This picture was treated with prednisone and anticoagulation. Fundus examination showed complete occlusive arteritis, periphlebitis, peripheral ischemia and perfusion macular defects affecting both eyes. The picture was suggestive of Behçet's disease. Azathioprine was added to the treatment without improvement in visual acuity. Four months later oral aphthous ulcers developed, confirming the suspected diagnosis. DISCUSSION: Behçet's disease may appear with the sudden onset of visual loss secondary to massive occlusive retinal vasculitis. The critical state of neuro-Behçet disease may delay the diagnosis. This combination of visual and neurological symptoms is associated with a poor visual prognosis (Arch Soc Esp Oftalmol 2002; 77: 275-278).

Adult↗
PubMed1992

[Cyclosporin A in therapy of chronic uveitis].

48 patients with chronic severe uveitis of pressumed noninfectious origin resistent to corticosteroids have been treated with Cyclosporin A. All patients had an initial loading dose of 5 mg/kg/day followed by a dose reduction according to ocular inflammatory activity and tolerability and according the guidelines by BenEzra, Nussenblatt and Timonen. Most of the patients received additional corticosteroids in a low dose. 35 out of these 48 patients (13 suffering from intermediate uveitis, 11 from retinal vasculitis, 5 panuveitis, 4 "pressumed histoplasmosis" and 2 sympathetic ophthalmia) were treated for 1 year and observed after withdrawing of Sandimmun for at least 6 months. The majority of these patients have manifested a positive therapeutic response to Cyclosporine, in particular patients suffering from vasculitis, panuveitis and sympathetic ophthalmia. All 35 patients were treated before for a long time with steroids without success, 19 out of these 35 in addition with cytotoxic agents. The immunosuppressive effect of Cyclosporine was not permanent, frequently the inflammation relapsed on reduction of dosage or withdrawing of the drug. Guidelines for combined regimen (Cyclosporine and corticosteroids and vitrectomy) were given. Although a large variety of side effects were reported the compliance was good.

Administration, Oral↗
PubMed2003

[Eales' disease with bilateral brain strokes and jaw-closing dystonia].

Eales' disease is an idiopathic occlusive vasculopathy of the retina, which is characterized by extensive peripheral non-perfusion, perivascular sheathing, and neovascularization. It is associated with recurrent vitreous hemorrhages. Both eyes are affected consecutively in 80% to 90% of the patients. In spite of the multiple theories that have been proposed, it continues to have unknown origin and its diagnosis relies on exclusion of other causes of retinal vasculopathy. In some occasions, these patients develop complications of the central nervous system, above all brain infarcts. We present the case of a 38 year old woman with Eales' disease who developed bilateral brain infarcts associated with occlusion or stenosis of the middle cerebral arteries. The cerebral angiography showed beading of the Silvian arteries, suggestive of an underlying inflammatory disorder. Early corticosteroid therapy could avoid contralateral retinal involvement and neurological complications. The patient also presented delayed jaw closing dystonia secondary to basal ganglia infarct which followed a benign course with spontaneous resolution in a few days.

Adult↗
PubMed2004

[Endogenous uveitis during pregnancy--a report of 4 cases].

PURPOSE: To estimate the influence of pregnancy on the clinical course of endogenous uveitis. MATERIAL AND METHODS: Four pregnant women (6 eyes) aged 16-21 yrs with uveitis of unknown etiology were observed. In two cases uveitis was bilateral while other two patients developed unilateral intraocular inflammation. At the time when the pregnancy was diagnosed systemic steroids were discontinued. Ophthalmic examination was performed every 1-2 months and then 3-8 months after delivery. RESULTS: In all cases, before pregnancy, the active stage of uveitis was observed; vitreous flare and vitreous cells were present in all cases (6 eyes). Iritis was present in one patient (1 eye), inflammatory chorioretinal lesions in 2 cases (3 eyes), while retinal vasculitis was observed in one case (1 eye). During pregnancy as well as within 3-8 months of follow-up after delivery, neither progression nor recurrences of uveitis were observed. In all patients gradual, total regression of intraocular inflammation with the improvement of visual acuity in 3 cases were noted. CONCLUSIONS: Our observations can suggest positive influence of pregnancy on uveitis activity.

Adolescent↗
PubMed2005

[Ocular manifestations of rickettsial diseases].

Rickettsial infection may cause severe systemic disease. The ocular manifestations of rickettsial infection may include sub-conjunctival hemorrhages, corneal abscesses, uveitis, optic nerve head edema, retinal vasculitis hemorrhages and infiltrates and endophthalmitis. Ocular complications may result in significant visual loss. Ocular manifestations may precede other systemic signs and symptoms and assist in clinical diagnosis. This report describes two cases with ocular involvement of rickettsial disease.

Eye Diseases↗
PubMed1987

Ischemic optic neuropathy secondary to intracarotid infusion of BCNU.

A 39-year-old man with a left parietal malignant glioma was treated with intracarotid infusion of 1,3-bis(2-chloroethyl)-1-nitrosourea (BCNU). Five weeks after a second infusion, he developed inferior altitudinal visual field loss and pallid optic disc edema ipsilateral to the injection. Retinal vasculitis is a well-recognized complication of intra-arterial BCNU infusion. It is believed that this patient suffered a similar vasculopathy in the posterior ciliary artery circulation.

Adult↗
PubMed1988

Human retinal pigment epithelial cells differentially express MHC class II (HLA, DP, DR and DQ) antigens in response to in vitro stimulation with lymphokine or purified IFN-gamma.

A possible role for retinal pigment epithelial cells (RPE) as local antigen presenting cells in immune inflammatory eye disease was investigated by studying the in vitro response of human RPE cells to stimulation with purified IFN-gamma or Con A induced lymphokine. RPE cells cultured with a single dose of 50-1000 u/ml IFN-gamma for up to 8 days to allow maximal Class II gene transcription, expressed HLA DP, DR and DQ antigens in a dose-dependent manner with 80% or more of cells positive for each antigen at the higher concentration. After removal of a suboptimal IFN-gamma stimulus, HLA-DR antigen expression persisted for at least 15 days. HLA-DP and DQ antigens persisted only after maximal IFN-gamma stimulation. Lymphokine from Con A stimulated lymphocytes induced higher levels of DR and DQ expression (80%) over DP (15%) implying complex interactions with other mediators present in the lymphocyte culture supernatant. Since RPE cells phagocytose and recycle autoantigen-rich retinal rod outer segments and co-express HLA DR and DQ Class II antigens in response to IFN-gamma stimulation, an immunoregulatory role in conditions in which retinal autoimmunity is implicated, such as chronic idiopathic posterior uveitis and retinal vasculitis is postulated for these cells.

Autoimmune Diseases↗
PubMed1984

Raccoon ascarid larvae (Baylisascaris procyonis) as a cause of ocular larva migrans.

Larvae of the common raccoon roundworm, Baylisascaris procyonis, are known causes of visceral larva migrans and CNS disease in animals and human beings. In the present experiments we examined the ability of B. procyonis to cause ocular larva migrans (OLM) in subhuman primates, as an indication of its possible ocular zoonotic importance. Squirrel monkeys given 5,000 or 10,000 infective B. procyonis eggs per os and cynomolgus monkeys given 20,000 eggs had clinical and histologic evidence of OLM, beginning 7 days after inoculation. Clinically, multifocal retinal hemorrhages, white spots, chorioretinitis, inflammatory tracks, vascular sheathing, diffuse retinal degeneration, and motile intraretinal larvae were seen. Histologically, primarily subretinal larvae caused varying degrees of retinal disruption, degeneration and necrosis, retinitis, vasculitis, and perivascular sheathing, primarily with eosinophils. Larvae were also present in choroidal granulomas. It was concluded that B. procyonis larvae have marked ability to produce OLM in subhuman primates following oral infection and should be considered as a possible etiology in human ocular disease.

Animals↗
PubMed1982

Systemic inflammatory complications following jejuno-ileal bypass.

The clinical and laboratory features of three patients with systemic inflammatory complications following jejuno-ileal bypass surgery for morbid obesity are described. The constellation of an asymmetrical polyarthritis, tenosynovitis, sterile skin pustules, mucous membrane ulceration and retinal vasculitis resembled Behçet's syndrome. Serum complement levels were normal and circulating immune complexes not regularly detected, although hypocomplementaemia, elevations in Clq and rheumatoid factor binding materials were noted prior to clinical relapse in one patient. Antibiotic therapy proved disappointing, whereas dapsone produced a sustained remission. Bacterial overgrowth was not invariably found which together with recurrence of symptoms following bypass reversal, suggest that bacteria may only be required to initiate, not perpetuate inflammatory complications.

Arthritis↗
PubMed1994

Clinical features of HTLV-I associated uveitis determined in multicenter collaborative study. Study Group for HTLV-I Associated Ocular Diseases.

The elucidation of the clinical features of human T-lymphotropic virus type I (HTLV-I) associated uveitis (HAU) was advanced by the results of a multicenter study performed by the collaboration of university hospitals throughout Kyushu and Okinawa and two institutions in the Tokyo-Yokohama metropolitan areas of Japan. A total of 426 cases of endogenous uveitis were registered during the five-month study period between September 1992 and January 1993. The etiology and clinical entity remained unknown for about half of these cases. A series of 50 cases of HTLV-I seropositive, entity-undefined uveitis were suspected to be HAU, and analyses of these cases provided the following clinical features of this recently defined uveitis entity. 1) middle-aged adult of either sex develops acute, inflammatory uveal disease and presents with visual haze or floaters, or both; 2) active disease consists of granulomatous or nongranulomatous reactions accompanied by vitreous opacities and retinal vasculitis with rare retinochoroidal exudative changes; 3) the disease resolves with response to corticosteroids, although leaving sequelae in occasional cases; 4) the disease affects one or both eyes, and up to half of cases show recurrent disease within years; 5) the visual outcome is usually favorable; 6) the disease occurs as an isolated disorder, but may sometimes be complicated by HTLV-I associated myelopathy. Hyperthyroidism is also associated in occasional cases; 7) the titer of serum HTLV-I antibodies is significantly high compared with that of HTLV-I carriers, suggesting an immune mechanism as the pathogenesis of HAU.

Adolescent↗
PubMed1994

[A multicenter clinico-epidemiological study of HTLV-I associated uveitis].

To elucidate the clinical and epidemiologic features of HTLV-I associated uveitis (HAU), a multicenter case-control study was performed by collaboration of university hospitals throughout Kyushu and Okinawa and two university hospitals in the central metropolitan area. A total of 426 cases of endogenous uveitis were collected and studied between September 1992 and January 1993; about half of the cases were definable for etiology or clinical entity, and the remaining cases were unknown. Assessment of the serum antibodies to HTLV-I revealed that the group of entity-undefined uveitis had a significantly high prevalence of HTLV-I as compared with the age- and sex-matched control subjects, giving supportive evidence for HAU. The titer of serum HTLV-I antibodies was significantly higher in entity-undefined uveitis than in HTLV-carriers. Assuming that a collection of 50 cases of HTLV-I seropositive, etiology-undefined uveitis represents HAU, its clinical features consisted were: (1) middle-aged, otherwise healthy adults developed acute inflammatory uveal disease and presented with visual haze and/or floaters; (2) the disease showed granulomatous or nongranulomatous anterior uveal reactions accompanied by vitreous opacities and retinal vasculitis; (3) the lesions resolved in response to topical or systemic corticosteroids; (4) the visual outcome was usually favorable; (5) nearly half of the cases had recurrent disease; (6) the cases remained systemically unremarkable, except for two cases of HTLV-I associated myelopathy and eight cases of hyper thyroid disease.

Adolescent↗
PubMed1994

[Tyndallometry with the laser flare cell meter in intraocular inflammation].

The laser flare-cell meter (LFCM) allows noninvasive, quantitative measurement of aqueous flare in human eyes. In this study we analysed changes in aqueous flare in eyes with various forms of intraocular inflammations. PATIENTS. We examined 100 eyes of 100 patients aged (mean +/- SD) 40.2 +/- 19.8 years with acute and chronic intraocular inflammations. The inflammatory diseases were classified according to morphological criteria. In addition, 163 normal eyes of 163 subjects (mean age 44.8 +/- 17.9 years) were evaluated. The eyes were examined by three examiners using the LFCM (Kowa, FC-1000) after pupillary dilatation with mydriatics. The instrument was calibrated with standardized concentrations of human albumin concentrations, and flare values were transformed into human albumin concentration equivalents. For statistical analysis, the non-parametric Wilcoxon-Mann-Whitney test was used. RESULTS. Flare values (mean +/- SD) in the eyes with intraocular inflammations were 4.38 +/- 11.82 mg/ml human albumin equivalents and were significantly higher than in the group of normal eyes (0.11 +/- 0.06 mg/ml, P < 0.0001). In addition, flare values differed in the various inflammation groups, with human albumin concentration equivalents decreasing in the following order: acute iridocyclitis (n = 27, 10.81 +/- 21.12), chronic iridocyclitis (n = 19, 5.19 +/- 6.12), necrotizing retinitis (n = 6, 1.92 +/- 1.11), acute retinochoroiditis (n = 9, 1.46 +/- 2.35), retinal vasculitis (n = 24, 1.03 +/- 0.90), chorioretinitis (n = 5, 0.98 +/- 0.71), Fuchs' uveitis (n = 10, 0.52 +/- 0.25). CONCLUSION. Measurement of aqueous flare with the LFCM is a noninvasive examination that yields objective, quantitative and reproducible results and therefore has advantages over slit-lamp assessment. It gives valuable information about the integrity or impairment of the blood-aqueous barrier. Its possible clinical applications include follow-up examinations and "monitoring" of therapeutic regimens in uveitis.

Adolescent↗
PubMed1994

Human T-lymphotropic virus type I uveitis.

In addition to adult T-cell leukemia (ATL) and chronic myelopathy (HAM/TSP), our current study indicates that human T lymphotropic virus type I (HTLV-I) is a causative agent for a specific type of uveitis with unknown etiology (idiopathic uveitis). The present paper describes the seroepidemiological, clinical, and molecular biological evidences that indicate uveitis seen in HTLV-I asymptomatic carriers (HTLV-I uveitis) is a distinct clinical entity. In an HTLV-I endemic area in Japan, the seroprevalence of HTLV-I in idiopathic uveitis was 38%, while those in uveitis with defined etiology and in non-uveitic ocular diseases were 10% and 19%, respectively. Strikingly, the HTLV-I seroprevalence in younger aged patients (20-49 years) with idiopathic uveitis was 49%, while only 8% in the control group (P < 0.001). A very similar observation was recorded even in HTLV-I less endemic area, suggesting that HTLV-I infection plays a role as a risk factor for idiopathic uveitis. Clinical analysis revealed that an intermediate uveitis characterized by moderate opacities in the vitreous body and retinal vasculitis was seen in the majority the patients. The proviral DNA of HTLV-I was detected from inflammatory cells in the eye of all tested patients using PCR technique. These data thus indicate that HTLV-I is closely related to a certain type of uveitis and the uveitis (HTLV-I uveitis) is a distinct clinical entity.

Adolescent↗