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PubMed2001

An association between Vogt-Koyanagi-Harada disease and Guillain-Barré syndrome.

PURPOSE: To describe an association between Vogt-Koyanagi-Harada disease and Guillain-Barré syndrome. METHODS: Case series, describing three patients. RESULTS: In two patients, the disorders had their onsets within 2 weeks of each other; in the third patient, Vogt-Koyanagi-Harada disease occurred after 3 months, as Guillain-Barré syndrome resolved. All three patients had bilateral panuveitis typical of Vogt-Koyanagi-Harada disease. Each also developed well-accepted manifestations of Guillain-Barré syndrome, including paresis of the lower extremities (all patients), paresis of the upper extremities (two patients), paresis of cranial nerves (two patients), areflexia (all patients), and abnormal electromyography findings (two patients). CONCLUSIONS: Vogt-Koyanagi-Harada disease may follow or occur simultaneously with Guillain-Barré syndrome. The fact that these two autoimmune disorders occur together in some patients suggest that they may share common disease mechanisms.

Adult↗
PubMed2003

Laser flare photometry and complications of chronic uveitis in children.

PURPOSE: To investigate possible relationships between laser flare photometry values, complications of uveitis, and outcomes in children with chronic uveitis. DESIGN: Retrospective chart review. METHOD: We evaluated patients with active, noninfectious anterior, intermediate, or panuveitis who were 16 years of age or younger and who had laser flare photometry at one of two academic institutions. Complications enumerated at baseline and during follow-up were compared with laser flare photometry values and to anterior chamber cell counts. RESULTS: At least one laser flare photometry value ("flare"), defined as baseline measurement, was available for 59 patients (41 girls, 18 boys; mean age, 10.3 +/- 3.5 years); 38 of these patients had at least one additional measurement during follow-up (median 11 months). Complications of uveitis were present in 35 patients (59%) at baseline. There was a positive association between increased laser flare photometry values and complications at baseline (any complication [P =.007], posterior synechiae [P =.003]). The development of complications during follow-up was associated with the presence of complications at baseline (P =.018). A subgroup of patients with low flare at baseline had no complications during follow-up regardless of treatment given. CONCLUSIONS: There is a positive relationship between laser flare photometry values and the prevalence of complications of uveitis in children. Laser flare photometry provides a novel way to monitor children with uveitis. Future study will be needed to determine whether values have prognostic importance and whether a treatment strategy that minimizes flare results in fewer uveitic complications.

Aqueous Humor↗
PubMed1996

Ocular manifestation of hairy cell leukemia with dramatic response to 2-chloro-deoxy-adenosine.

PURPOSE: To demonstrate that hairy cell leukemia may involve the eye. METHODS: We examined a patient who had a unique ocular manifestation of hairy cell leukemia with severe panuveitis in one eye and leukemic retinopathy in the fellow eye. RESULTS: Therapy with 2-chloro-deoxy-adenosine, a drug recently introduced for treatment of hairy cell leukemia, led to dramatic improvement of the ocular complications. CONCLUSIONS: Hairy cell leukemia may involve the eye and ocular symptoms may respond to systemic treatment with 2-chloro-deoxy-adenosine.

Aged↗
PubMed1996

Progressive subretinal fibrosis and blindness in patients with multifocal granulomatous chorioretinitis.

PURPOSE: To describe the clinical and histopathologic findings in four eyes of three patients who became blind because of multifocal choroiditis and massive subretinal fibrosis. METHODS: Clinicopathologic correlative study. RESULTS: During a period of several years, three healthy elderly patients developed severe visual loss only partly explained by multifocal chorioretinitis and massive subretinal fibrosis. Histopathologic examination of four eyes from these patients disclosed widespread destruction of the outer retina and retinal pigment epithelium, massive areas of subretinal fibrous tissue proliferation, granulomatous inflammation centered around degenerated and fragmented Bruch's membrane, and chronic uveitis. No infectious organisms were identified by special stains or electron microscopy in one eye. CONCLUSIONS: Clinical and histopathologic findings in these three patients were consistent with an autoimmune disease process directed at the retina, retinal pigment epithelium, inner choroid, or all three. Cellular injury in this location can result in massive subretinal fibrosis. Subretinal fibroplasia, however, is probably a nonspecific reparative response to injury. The pathogenesis of this blinding disorder in elderly patients may be similar to the less severe disease usually occurring in younger patients with multifocal choroiditis, panuveitis, and punctate inner choroiditis.

Aged↗
PubMed1987

Changing patterns of uveitis.

We conducted a retrospective analysis of 600 patients with uveitis seen at the Estelle Doheny Eye Center to determine the frequency of occurrence of the various forms of uveitis and to see if the causes of uveitis have changed as compared with previous studies. In 402 cases (67.0%) we established a specific diagnosis based on history, physical findings, and laboratory studies: 167 cases (27.8%) involved primarily the anterior segment, 230 (38.4%) the posterior segment, and 111 (18.4%) occurred as panuveitis. Intermediate uveitis (pars planitis) was the single most frequently diagnosed uveitic entity and accounted for 92 cases (15.4%). We compared our findings with those of previously published studies and found that, as new diseases occur and improved diagnostic techniques become available, the differential diagnosis of uveitis continues to change.

Choroiditis↗
PubMed1991

Analysis of local antibody production in the vitreous humor of patients with severe uveitis.

We analyzed the local antibody production in vitreous humor samples collected during vitrectomy in patients with severe vision-threatening uveitis. In 24 patients, paired serum and undiluted vitreous humor samples were collected and tested for antibodies against Toxoplasma gondii, herpes simplex virus, varicella-zoster virus, cytomegalovirus, Epstein-Barr virus, and Toxocara canis. Total IgG and the Goldmann-Witmer coefficient were determined. The initial diagnosis of ocular toxoplasmosis could be confirmed in six of the seven patients. The seventh patient showed a local antibody production against herpes simplex virus. One of the three patients with chronic panuveitis at initial diagnosis showed a local antibody production against T. gondii. These last two findings resulted in a change in medical treatment. Analysis of local antibody production in vitreous humor samples is a valuable diagnostic tool.

Animals↗
PubMedDate not supplied

Analysis of 87 cases with Vogt-Koyanagi-Harada disease.

PURPOSE: Vogt-Koyanagi-Harada (VKH) disease is known to have varied manifestations in different ethnic groups. In order to analyze the clinical profile of VKH cases in the Indian population, we studied 87 consecutive cases of VKH disease treated in an uveitis clinic in South India between 1985 and 1996. METHODS: Retrospective analysis and review of charts of consecutive new VKH cases diagnosed in a referral clinic. RESULTS: VKH disease comprised 2.2% of all uveitis referrals. Extraocular symptoms or signs were seen in 64% of cases at the time of presentation. Most common was meningism (95.9%). However, subsequently all patients developed extraocular manifestations. Panuveitis (92%) was the commonest presentation. Systemic corticosteroid was the usual form of therapy (50.3%) followed by immunosuppressive therapy (39%); surgical treatment was needed in 8% of the cases. Complicated cataract (33%) and glaucoma (24%) were major complications. Final vision was between 6/60 and 6/18 in 88% of the cases and 6/18 and better in 15.4%; there was no improvement in 11% of the cases. CONCLUSIONS: VKH disease occurs less frequently in India than in Japan and about as commonly as in the United States. Extraocular signs are far less common than in the Japanese population. Visual prognosis is good in patients presenting within 1 month of onset of symptoms. Immunosuppressive agents and vitreoretinal surgery are needed in advanced cases and in cases reported later. Jpn J Ophthalmol 2000;44:296-301

Adolescent↗
PubMed2001

Patients with Multiple Sclerosis and concomitant uveitis/periphlebitis retinae are not distinct from those without intraocular inflammation.

Recent models of experimental autoimmune encephalomyelitis (EAE) have indicated that antigens co-expressed in the retina and uvea might be of pathogenetic relevance in Multiple Sclerosis (MS). We investigated the clinical spectrum and magnetic resonance imaging of 11 MS patients with concomitant uveitis, and determined the frequency of clinically silent intraocular inflammation in a prospective series of 50 patients. Two of the 11 patients had panuveitis, seven had anterior, and the remaining two had intermediate uveitis. The onset of uveitis preceded that of neurological symptoms by a mean of 8.5 years (range 1-20). None of the 50 MS patients studied prospectively by using slit lamp examinations and dilated funduscopy showed any evidence of uveitis but six patients had signs of retinal inflammation ("periphlebitis retinae"). Cranial MRI did not reveal "atypical" lesional distribution in MS patients with uveitis or periphlebitis retinae. No correlation between the type of MS and uveitis, or between the degree of neurological disability and the type of uveitis was found.

Adult↗
PubMedDate not supplied

Vogt-Koyanagi-Harada syndrome.

The Vogt-Koyanagi-Harada syndrome (VKH) is a bilateral, diffuse granulomatous uveitis associated with poliosis, vitiligo, alopecia, and central nervous system and auditory signs. These manifestations are variable and race dependent. This inflammatory syndrome is probably the result of an autoimmune mechanism, influenced by genetic factors, and appears to be directed against melanocytes. On histopathologic examination typical cases show nonnecrotizing diffuse granulomatous panuveitis with initial sparing and late involvement of the choriocapillaris and formation of Dalen-Fuchs' nodules. Fluorescein angiography, lumbar puncture, and echography are useful adjuncts in the diagnosis and management of VKH syndrome. Patients with this syndrome are treated generally with high dose systemic corticosteroids or, when necessary, with cyclosporine or cytotoxic agents. The prognosis of patients with VKH syndrome is fair, with nearly 60% of patients retaining vision of 20/30 or better. The complications of VKH syndrome that lead to visual loss include cataracts in about 25% of patients, glaucoma in 33%, and subretinal neovascular membranes (SRNVMs) in about 10%; the latter, however, are an important cause of late visual loss. These complications usually require medical and/or surgical intervention, including photocoagulation. The major risk factor for the development of cataracts, SRNVMs, and, to some extent, glaucoma, is chronic recurrent intraocular inflammation that may be resistant to corticosteroid therapy. It appears that initial treatment with high dose corticosteroids, combined with prolonged corticosteroid therapy at appropriate dosage, may minimize these complications and may improve visual prognosis.

Fluorescein Angiography↗
PubMed2000

Evaluation of foldable intraocular lenses in patients with uveitis.

OBJECTIVE: To evaluate various foldable posterior chamber intraocular lenses (IOLs) after phacoemulsification in patients with uveitis. DESIGN: A prospective, noncomparative, interventional case series. PARTICIPANTS: Forty-nine consecutive patients (60 eyes) with various types of uveitis (anterior, n = 20; posterior, n = 1; panuveitis, n = 37, intermediate, n = 2). INTERVENTION: All patients underwent phacoemulsification with foldable posterior chamber IOL implantation. All eyes were free of active inflammation at the time of surgery. A variety of IOL biomaterials were implanted: acrylic (n = 30), silicone (n = 17), and hydrogel (n = 13). MAIN OUTCOME MEASURES: Detailed examination was performed by one masked observer. Several parameters were compared for each implant biomaterial, including level of best corrected Snellen visual acuity at final follow-up, presence of posterior synechiae, anterior capsular phimosis, posterior capsule opacification, and the degree of cellular deposits on the IOL optic. RESULTS: There were 26 males and 23 females, aged 9 to 83 years (mean, 48 years). Follow-up ranged from 1 to 33 months (mean, 17.03 months). At final follow-up, 56 eyes (93.3%) had an improvement in visual acuity compared with preoperative levels as follows: 34 eyes (56.6%) achieved an improvement of four or more Snellen lines, and 44 eyes (73.3%) achieved 20/30 or better. Giant cells, observed on the IOL optic in 19 eyes (31.7%), were most often seen on the acrylic biomaterial at the 1-month follow-up, although this was not found to be statistically significant. Scratch marks produced by the lens-introducing forceps were seen in 24 eyes (40.0%), mainly on the acrylic and hydrogel optics. Posterior capsule opacification (PCO) occurred in 49 eyes (81.7%), with only 5 eyes requiring laser capsulotomy. There was no association between PCO and the various lens biomaterials. Other causes for reduced visual acuity included glaucomatous optic neuropathy (n = 5) and cystoid macular edema (n = 8). CONCLUSIONS: The use of foldable IOLs in eyes with uveitis is safe, but the optimal biomaterial has yet to be found.

Acrylic Resins↗
PubMed2000

Fluocinolone acetonide sustained drug delivery device to treat severe uveitis.

PURPOSE: Uveitis is often a chronic disease requiring long-term medical therapy. In this report, we describe a pilot safety and efficacy trial of a novel sustained drug delivery system containing fluocinolone acetonide to treat patients with severe uveitis. DESIGN: Prospective, noncomparative, interventional case series PARTICIPANTS: Patients with severe uveitis. METHODS: Sustained drug delivery devices designed to release fluocinolone acetonide for at least 2.5 years were implanted through the pars plana into the vitreous cavity of seven eyes of five patients. All patients had severe uveitis not well controlled with, or intolerant to, repeated periocular corticosteroid injections, systemic corticosteroids, nonsteroidal immunosuppressive agents, or a combination thereof at the time of device implantation. Before device implantation, patients underwent complete evaluation including history, ophthalmologic examination, fluorescein angiography, visual field testing, and electroretinography. After surgery, patients were reexamined at 1 week, 2 weeks, 4 weeks, and at 1- to 3-month intervals. Visual fields, electroretinograms, and fluorescein angiography were repeated at 3- to 6-month intervals. MAIN OUTCOME MEASURES: Preoperative and postoperative visual acuity, ocular inflammation, anti-inflammatory medication use, and intraocular pressure. RESULTS: Patients had a diagnosis of Behçet's syndrome (two eyes), or idiopathic panuveitis (five eyes, including two with necrotizing retinitis, two with progressive chorioretinitis, and one with iridocyclitis and intermediate uveitis). Patients were observed an average of 10 months (range, 5-19 months). All eyes had stabilized or improved visual acuity after device implantation, and four of seven eyes had an improvement of three lines or more. The mean initial visual acuity, measured by Snellen chart, was 20/207, and the mean final visual acuity was 20/57 (P = 0.02). After surgery, at the final visit, no eye had clinically detectable inflammation, and all seven eyes had a marked reduction in systemic, topical, and periocular anti-inflammatory medication use. Four eyes had increased intraocular pressure 6 weeks to 6 months after device implantation. Intraocular pressure has been controlled on topical medications. No patient experienced intraoperative complications. CONCLUSIONS: A fluocinolone acetonide sustained drug delivery device is a promising new therapy for the treatment of severe uveitis. Intraocular pressure must be carefully monitored long after device implantation. Based on these data, a randomized study of a larger group of patients is warranted.

Adolescent↗
PubMed2001

Uveitis masquerade syndromes.

BACKGROUND: Incorrect diagnosis of the uveitic masquerade syndromes (UMS) may have severe consequences. In this study, the frequency, clinical manifestations, and informative diagnostic tests for UMS are described. DESIGN: Retrospective observational case series. PARTICIPANTS: Forty patients with UMS were identified in a cohort of 828 consecutive patients with uveitis. The mean follow-up was 4.5 years. METHODS: A review of clinical, laboratory, photographic, and angiographic records was performed. MAIN OUTCOME MEASURES: Clinical features, associated systemic diseases, diagnostic procedures and their role in the diagnostic process, and systemic and visual outcomes. RESULTS: Nineteen patients had intraocular malignancy (48% of all with UMS; 2.3% of all with uveitis), mainly intraocular lymphoma (n = 13) and leukemia (n = 3). The ophthalmologist was the first to recognize malignant disease in 11 of 19 patients (58%). Of 21 patients with nonmalignant UMS, 10 had an ocular vascular disease and 5 a hereditary ocular disorder. The patients with malignant UMS were older than those with nonmalignant UMS (average age, 50 vs 44 years, P: < 0.05). During follow-up, 9 of 19 patients with malignant UMS died. The most informative diagnostic procedure for malignant UMS was intraocular fluid analysis; for nonmalignant UMS, fluorescein angiography. The cytologic analysis of intraocular fluids yielded the best results for diagnosing intraocular malignancies (7 of 11 patients, 64%); the association of negative cytologic results with the recent administration of systemic corticosteroids was noted. Immunophenotyping of the aqueous confirmed the diagnosis of hematologic malignancy for 3 of 5 patients examined. Panuveitis was the most frequent manifestation of malignant UMS. Intraocular lymphomas presented with isolated vitreitis (n = 6), chorioretinal lesions (n = 5) and iris infiltration (n = 2). Clinical presentation of nonmalignant UMS was diverse but consisted mainly of abnormalities of the retinal vasculature. CONCLUSIONS: UMS was diagnosed in 5% of the patients with uveitis at a tertiary center. Despite the variety of underlying disorders and different clinical presentations, a high frequency of malignant and vascular diseases was found. Awareness of the clinical manifestations of UMS and application of the correct diagnostic procedures should promote timely diagnosis and treatment, which are essential not only for visual acuity but also for the life of the patient.

Adult↗
PubMed2004

Tumor necrosis factor alpha blockade with infliximab for refractory uveitis and scleritis.

OBJECTIVE: To assess the efficacy and safety of the anti-tumor necrosis factor alpha agent infliximab in treatment-resistant uveitis and scleritis. DESIGN: Retrospective, noncomparative interventional case series. PARTICIPANTS: Seven patients with noninfectious ocular inflammatory disease that was refractory to alternative immunosuppression. These included one patient with idiopathic retinal vasculitis and panuveitis, one patient with intermediate uveitis, one patient with chronic juvenile anterior uveitis, three patients with scleritis, and one patient with scleritis and peripheral ulcerative keratitis. Four patients had an underlying systemic disease that was in remission in three cases. INTERVENTION: Infusions of infliximab, 200 mg, were given at 4-week to 8-week intervals, depending on the clinical response. MAIN OUTCOME MEASURES: Clinical response, including symptoms, visual acuity, degree of scleral vascular engorgement, corneal thinning, anterior chamber activity, and posterior segment inflammation, reduction in concomitant immunosuppression, and adverse effects. RESULTS: The mean patient age was 47 years (range, 24-78), and four patients were female. The mean number of infliximab infusions was seven (range, 2-19), and the mean follow-up period was 12 months (range, 4-22 months). Six patients experienced a clinical improvement, with five achieving remission and significant reduction in immunosuppression. One patient showed an initial response but developed a delayed hypersensitivity response that precluded further treatment. No other adverse effects occurred. CONCLUSIONS: Infliximab seems to be an effective and safe treatment for noninfectious uveitis and scleritis and may be indicated as rescue therapy for relapses of ocular inflammation or as maintenance therapy when conventional immunosuppression has failed. Further investigation of infliximab for treatment-resistant scleritis and uveitis is warranted.

Adult↗
PubMed1986

Sympathetic ophthalmia. Immunopathological findings.

Ocular tissue from six patients with a clinical diagnosis of sympathetic ophthalmia (SO) was examined using immunohistochemical techniques. All patients presented with a history of bilateral panuveitis after penetrating ocular injury or multiple intraocular surgeries and clinical features of SO. In four cases, classic histopathological features of SO were observed, including granulomatous uveal tract infiltration and subretinal pigment epithelium (RPE) collections of inflammatory cells (Dalen-Fuchs nodules). Bone marrow derived monocytes were the major cellular components in these granulomas. In two cases, histopathology failed to demonstrate typical Dalen-Fuchs nodules or granulomas in the choroid. However, the choroidal infiltrates were composed primarily of T-helper and B lymphocytes, without macrophages or epithelioid cells. The eyes examined in this report indicate that a varied spectrum of immunopathological and histopathological findings may occur in clinically diagnosed SO.

Adult↗
PubMed1993

Immunocytologic findings in a case of Vogt-Koyanagi-Harada syndrome.

BACKGROUND: Vogt-Koyanagi-Harada (VKH) syndrome is a form of chronic panuveitis that often has a poor long-term visual outcome. To the authors' knowledge, there have been only two previous reports describing immunocytologic findings in the eyes of patients with VKH syndrome, all studied in relatively late stages of the disease. METHODS: The authors performed immunocytologic studies on the enucleated eyes of a patient with active VKH syndrome, using monoclonal antibodies to mononuclear subsets, major histocompatibility antigens, and viral and treponemal agents, to better understand the pathophysiologic mechanisms underlying the disease. Emulsified chorioretinal tissue also was processed for viral isolation in tissue culture and for Treponema pallidum by rabbit inoculation studies. RESULTS: The dense uveal infiltrates in active VKH syndrome are composed predominantly of T lymphocytes and HLA-DR+ macrophages. Scattered T cells and plasma cells were present in the retina. In addition, nondendritic-appearing CD1 (Leu-6) positive cells were localized in the choroid, in close proximity to choroidal melanocytes. No microbial agents were identified. CONCLUSION: Whereas the etiology of the VKH syndrome remains an enigma, the authors' immunocytologic findings are consistent with a T-cell-mediated disorder. In demonstrating CD1 (Leu-6) positive cells in the infiltrates, the authors propose a potential role for these cells in the etiology of VKH syndrome.

Cell Count↗
PubMed1993

Incidence and management of glaucoma in Vogt-Koyanagi-Harada syndrome.

BACKGROUND: The Vogt-Koyanagi-Harada syndrome is a bilateral panuveitis associated with neurologic and dermatologic manifestations. METHODS: The authors reviewed the charts of all patients with Vogt-Koyanagi-Harada syndrome seen at their institution over the past decade to determine the incidence of glaucoma, as well as the results of medical and surgical therapy for glaucoma, in this group of patients. FINDINGS: Of 42 patients diagnosed with Vogt-Koyanagi-Harada syndrome, evidence of glaucoma requiring either medical or surgical intervention occurred in 16 patients (38.1%). Of these, nine (56.3%) had open-angle glaucoma and seven (43.7%) had angle-closure secondary to pupillary block. In 5 (31.3%) of the 16 patients, medical therapy alone was sufficient to control intraocular pressure. Eleven patients (68.7%) required surgical intervention, consisting of laser iridotomy, surgical iridectomy, trabeculectomy with or without 5-fluorouracil, and/or Molteno implantation. CONCLUSION: The relative success of each of these procedures in this group of patients is discussed. Glaucoma is a common complication in the Vogt-Koyanagi-Harada syndrome, and one that is often difficult to control.

Adolescent↗
PubMed1995

Association between Vogt-Koyanagi-Harada syndrome and HLA-DR1 and -DR4 in Hispanic patients living in southern California.

BACKGROUND: Vogt-Koyanagi-Harada (VKH) syndrome is associated with human leukocyte antigen (HLA)-B54, -DR4, -DR beta 1*0405, -DQ4, and -DR53 in Japanese patients. Disease-associated HLA specificities may differ among races. This study examined HLA associations with VKH syndrome in Hispanic patients living in southern California, a racial subgroup at increased risk for the disease. METHODS: Human leukocyte antigen specificities were determined on 25 Hispanic patients with VKH syndrome and compared with HLA specificities of 217 healthy Hispanic control subjects. Inclusion criteria for study patients were nontraumatic panuveitis with exudative retinal detachments, with or without extraocular manifestations. Tests were performed using standard cytotoxic assays. RESULTS: HLA-DR4 was present in 14 (56%) patients with VKH syndrome and in 62(29%) control subjects (relative risk = 1.96). HLA-DR1 was present in 9 (36%) patients with VKH syndrome and in 19 (9%) control subjects (relative risk = 4.11). HLA-DR1 and DR4 share a common epitope within the DR beta 1 gene. HLA-DR1 and/or DR4 were present in 21 (84%) patients with VKH syndrome and in 76 (35%) control subjects (relative risk = 2.40). CONCLUSIONS: HLA-DR1 and -DR4 were found in a significantly disproportionate number of Hispanic patients with VKH syndrome living in southern California. HLA-DR4, although not HLA-DR1, has been previously associated with VKH syndrome in other groups. These associations suggest a common immunogenic predisposition to VKH among different racial groups, and suggest that a common epitope shared by DR1 and DR4 may be involved in the pathogenesis of the disease.

California↗