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PubMed2006

Wegener's granulomatosis: ophthalmic manifestations and management.

OBJECTIVES: To provide an up-to-date and comprehensive review of Wegener's granulomatosis (WG) as a disease entity, focusing on the ophthalmic manifestations and management options. METHODS: A search of Medline was undertaken between 1966 and 2005 regarding WG, systemic vasculitis, and the ocular manifestations of WG. Major ophthalmic and medical textbooks also were reviewed for content, as well as original references. RESULTS: Involvement of ocular and orbital structures in patients with WG is common and may be a presenting feature. The ocular manifestations range from mild conjunctivitis and episcleritis to more severe inflammation with keratitis, scleritis, uveitis, and retinal vasculitis. Involvement of the nasolacrimal system and orbital tissues also can occur. Except for some cases of anterior segment inflammation, the ocular involvement will not respond to topical agents, but rather to systemic antiinflammatory and immunosuppressive regimens. Surgical intervention may be of value for obtaining tissue diagnosis, in achieving orbital decompression in cases of significant orbital disease with optic nerve compromise, or in cases of nasolacrimal duct obstruction. CONCLUSION: WG is an important clinical entity that needs to be recognized early and treated appropriately. Ophthalmic manifestations are frequently encountered and can result in significant morbidity and even blindness. The management is challenging and often requires a multidisciplinary approach.

Diagnosis, Differential↗
PubMed1998

[Measurement of thrombomodulin values in the serum and eyes of experimental autoimmune uveoretinitis].

Thrombomodulin (TM) is a protein, present on intact endothelial cell surfaces, that plays a major role in the protein C anticoagulant system. Plasma TM is produced by injured endothelium, and is recognized in circulating blood or urine as a sign of endothelium cell damage. The localization of TM within the eye and its kinetics in the eye and the serum were investigated in rats with experimental autoimmune uveoretinitis (EAU). An immuno-histochemical study showed the presence of TM at sites of fibrin in the anterior chamber and retinal vasculitis. The amount of TM in the eye increased with progression of intraocular inflammation, but the serum level of TM did not significantly differ from the values in the control group. These data suggest that TM may play a role in preventing thrombosis in cases of vasculitis and fibrin formation in the anterior chamber in EAU, thus maintaining blood flow and the fluidity of aqueous humor.

Animals↗
PubMed1994

[Rapidly progressive outer retinal necrosis in a patient with acquired immunodeficiency syndrome].

We report a case of rapidly progressive varicella zoster virus retinitis, which is distinct from acute retinal necrosis syndrome. The patient was a 52-year-old male and suffered acquired immunodeficiency syndrome. Two months after the varicella zoster dermatitis in the distribution of the first division of the left trigeminal nerve, pseudodendritic keratitis and iridocyclitis were observed in the left eye. After 5 weeks, multifocal and patchy white exudates were observed in the peripheral deeper layer of the retina in the left eye, but retinal vasculitis in the exudative lesions was slight. Despite systemic administration of acyclovir, white exudates progressed confluently from the periphery to the post pole of the retina and reached the macula in 10 weeks. Eight weeks after the observation of lesions in the left eye, we found the same lesions in the right eye. After the white exudative lesions disappeared, the retina became atrophic and the retinal vessels were narrowed, but no retinal detachment was observed. Recently, Foster and associates described the rapidly progressive outer retinal necrosis as a new entity of varicella zoster virus retinitis in AIDS patients. We think our case was very similar to the rapidly progressive outer retinal necrosis. This case shows that we must carefully follow up the rapidly progressive outer retinal necrosis in the AIDS patients with a varicella zoster dermatitis.

AIDS-Related Opportunistic Infections↗
PubMedDate not supplied

Immunology of multisystem ocular disease.

In this review based on Prof. James' 1983 George Nelms Wise Lecture, the authors summarize proven and speculated relationships involved in the immunology of multisystem ocular disease. Extensive tables elucidate the mechanisms underlying such conditions as, systemic lupus erythematosus, polyarteritis nodosa, Sjögren's syndrome, Wegener's granulomatosis, scleritis, sarcoidosis, Behcet's disease, idiopathic retinal vasculitis, sympathetic ophthalmia, Vogt-Koyanagi-Harada syndrome, and dysthyroid eye disease.

Antigen-Antibody Complex↗
PubMed2006

Atypical manifestations of acute posterior multifocal placoid pigment epitheliopathy.

We report four patients with acute posterior multifocal placoid pigment epitheliopathy (APMPPE) with atypical features which were unilaterality, serous detachment of the neurosensory retina, retinal vasculitis and papillitis. All patients responded well to oral corticosteroids. We suggest that systemic corticosteroids could be useful in selected cases of APMPPE with atypical clinical features.

Administration, Oral↗
PubMed1987

Cytomegalovirus infection in a healthy adult associated with recurrent branch retinal artery occlusion.

The authors report a 16-year-old healthy male who developed branch retinal artery occlusions sequentially in both eyes. A few vitreous cells and retinal vasculitis were associated findings. A complete work-up of the patient for an etiology for the occlusions failed to show any possible embolic source. Spinal fluid studies showed increased protein content and lymphocytic pleocytosis. Serologic studies showed a tenfold rise in the cytomegalovirus (CMV) titer. Other viral titers did not rise. CMV could not be cultured from the patient's body. The case represents a possible cause of the so-called idiopathic recurrent branch retinal artery occlusion syndrome.

Acute Disease↗
PubMed2006

[Melanoma-associated retinopathy with unknown primary site in a Japanese woman].

BACKGROUND: We report the clinical features of the first case of a Japanese person with melanoma-associated retinopathy. CASE: A 44-year-old woman complained of photopsia and blurred vision in her right eye, and was treated with steroids for uveitis by an ophthalmologist. She was referred to our hospital for further examination. After one month of treatment, she still complained of photopsia in her right eye. The best corrected visual acuity in the right eye was 0.8 and these was sensitivity loss in the central visual field test. Ophthalmoscopy and fluorescein angiography showed some retinal vasculitis in the right eye. A full-field electroretinogram demonstrated a negative-type electroretinogram (ERG) waveform with attenuation of the b-wave amplitude in the right eye. A dark adaptation test revealed sensitivity loss of the rods. The lymph nodes on the right side of her neck were examined and the diagnosis was made of metastic cutaneous melanoma with unknown primary site; her visual dysfunction was diagnosed as melanoma-associated retinopathy. The retinal inflammation improved after steroid treatment, but her visual dysfunction remained. Chemotherapy and an immunotherapy regimen was begun, but 36 months later she died of metastatic melanoma in the lungs. CONCLUSIONS: A woman treated for uveitis without any prior systemic and ocular diseases was diagnosed with melanoma-associated retinopathy and metastatic melanoma in the cervical lymph nodes of unknown primary origin. The first ocular symptoms were photopsia and blurred vision, not night blindness. ERG was useful for diagnosing this rare ocular condition in an early stage.

Adult↗
PubMedDate not supplied

A change in ocular involvement in a patient suffering from ankylosing spondylitis and Behçet's disease.

We report a case of a young man suffering from both ankylosing spondylitis and Behçet's disease in whom the character of the ocular involvement changed according to the predominant disease at a given time. When the clinical picture was one of ankylosing spondylitis, only anterior uveitis was observed, while the clinical picture of Behçet's disease occurred with panuveitis and retinal vasculitis. To the best of our knowledge, this is the first report in the English literature of two different patterns of ocular involvement in the same patient with two seronegative arthritides.

Adult↗
PubMed1999

Cushing's disease masking coincidental steroid-responsive diseases.

Two cases of Cushing's disease are presented. In both cases successful treatment was followed by the development of a steroid-responsive disease condition, a seronegative arthritis in the first case and retinal vasculitis in the second. It is likely that both these conditions were unmasked by the fall in the endogenous steroid levels following the successful treatment of the Cushing's disease by trans-sphenoidal hypophysectomy.

Adrenocorticotropic Hormone↗
PubMedDate not supplied

Indocyanine green angiographic findings in acute retinal necrosis.

PURPOSE: To clarify indocyanine green (IA) angiographic features in patients with acute retinal necrosis (ARN). METHODS: Two patients with ARN were examined by fluorescein angiography (FA) and IA, and findings from both were compared. RESULTS: Fundus examination revealed widespread retinal hemorrhages and yellowish-white patches in the periphery, characteristic of ARN. In both cases, FA showed diffuse dye leakage from all retinal veins and the optic disc, and vascular obstruction in the peripheral fundus. In IA, dye leakage was localized, and extravasation of dye was evident only from the lower temporal retinal vein and the lower half of the optic disc. This pattern of indocyanine green dye leakage appeared to be continuous from the optic disc toward the lower temporal retinal vein. Also, IA clearly demonstrated choroidal vascular filling delay in one case in the early phase of the angiogram. CONCLUSIONS: While FA showed diffuse dye leakage from all retinal veins, IA identified only the retinal vessels with the most prominent vascular damage. IA also identified choroidal vascular lesions in these patients with ARN. The information obtained by IA might be useful to detect retinal vasculitis with prominent inflammation and to determine the extent of choroidal inflammation in patients with ARN.

Acyclovir↗
PubMedDate not supplied

Histopathological findings in proliferative membrane from a patient with sarcoid uveitis.

BACKGROUND: Sarcoid uveitis is occasionally accompanied by proliferative changes, such as retinal neovascularization and vitreous hemorrhage. Steroid administration, retinal photocoagulation, and vitrectomy may be indicated in such proliferative cases. CASE: A 19-year-old woman presented with proliferative sarcoid uveitis accompanied by recurrent vitreous hemorrhage. OBSERVATIONS: At the initial examination, bilateral vitreous opacity, retinal exudates, mild vitreous hemorrhage, retinal vasculitis, and neovascularization of the retina and optic disc were observed. Although prednisolone was administered and panretinal photocoagulation was performed several times, recurrent vitreous hemorrhage continued. Since the vitreous hemorrhage was not absorbed, pars plana vitrectomy and lensectomy were performed. After surgery, neovascularization and intraocular inflammation decreased, and the corrected visual acuity in the right eye improved to 20/50. Histopathologic analysis of the proliferative membrane removed during surgery revealed substantial neovascularization and numerous neutrophils in the vessels. CONCLUSIONS: Based on these findings, an inflammatory reaction as well as retinal ischemia were thought to be involved in the proliferative changes in this patient.

Adult↗
PubMed2001

[A full thickness macular hole as an uncommon complication of Behçet disease].

The authors report the occurrence of an unusual complication of Behçet's disease: a full thickness macular hole caused by retinal vasculitis resulting in a definite functional impairment. A review of the different ocular complications is provided, stressing the unusual location on the posterior segment and the potential seriousness of these complications.

Adrenal Cortex Hormones↗
PubMed1990

Intraocular tuberculosis.

Twelve patients with a diagnosis of intraocular tuberculosis are described. Nine patients presented with florid ischaemic retinal vasculitis and a marked tendency to neovascularisation. Two patients developed choroidal tubercles; iris nodules were observed in association with anterior uveitis in the remaining patient. The methods of diagnosis, management and possible mechanisms of pathogenesis of these different clinical presentations are discussed.

Adult↗
PubMed1992

Type II essential mixed cryoglobulinaemia: presentation, treatment and outcome in 13 patients.

The long-term clinical course of patients with primary Type II essential mixed cryoglobulinaemia is unclear as many reports fail to separate this group from patients with Type III disease. We have reviewed 13 patients with Type II essential mixed cryoglobulinaemia who presented to the Hammersmith Hospital between 1976 and 1990. All patients had a cryoglobulin level greater than 0.1 mg/ml (range 0.27-6.50 mg/ml), and characterization of the cryoglobulin in all cases revealed the presence of a monoclonal IgM kappa component with rheumatoid factor activity together with polyclonal IgG. All patients had evidence of activation of the classical pathway of complement with greatly reduced levels of C4, while C3 levels were moderately reduced in three patients. All patients had skin disease and joint symptoms were reported by nine patients, with erosive arthritis in one. Eight patients had peripheral sensorimotor neuropathy. Renal disease was observed in 10 patients, manifesting as raised creatinine level, proteinuria or haematuria. Renal tissue was examined in eight patients: in six the appearances were those of a mesangiocapillary glomerulonephritis Type I while in the other two patients there was a mesangioproliferative glomerulonephritis, in one diffuse and in the other focal and segmental. Glomerular capillary 'hyaline thrombi' were found in six biopsies, extracapillary proliferation was found in three and evidence of vasculitis was found in all eight. Liver biopsy showed macronodular cirrhosis in one patient, while a second with recurrent episodes of jaundice showed only chronic inflammatory changes. No patient was positive for hepatitis B surface antigen; however one patient had low titre anti-hepatitis B surface antibody. Normochromic normocytic anaemia was present in nine patients. Bone marrow examination was carried out in 13 patients at presentation to our unit: 10 showed no evidence of a lymphoproliferative disorder, while three suggested the presence of a non-Hodgkin's lymphoma (some years after original presentation in all three). Unusual clinical features included one patient with retinal vasculitis and one patient with severe pulmonary haemorrhage.

Adult↗
PubMedDate not supplied

Behçet's disease.

Behçet's disease is a chronic, relapsing vasculitis that can affect most organ systems. The prevalence varies geographically, and the disease is more common in countries along the ancient Silk Road, including Italy, Turkey, Israel, Saudi Arabia, Iran, China, Korea and Japan. Behçet's is more common in men than in women, and typically affects young adults. The classic finding in Behçet's patients is the presence of recurrent mucocutaneous ulcers, and oral aphthous ulcerations are usually the initial symptom. Other manifestations include genital ulcers, skin lesions, vascular, neurological, articular, and ocular disease. The disease can affect the anterior and/or posterior segments of the eye, and the main manifestations include iridocyclitis, hypopyon, mild to moderate vitreitis, retinal vasculitis and occlusion, optic disc hyperemia, and macular edema. There is no pathognomonic laboratory test in Behçet's disease, and the diagnosis is based in systemic and ocular clinical findings. Treatment of ocular Behçet is based in corticosteroids and immunosuppressive agents, to suppress acute inflammation and reduce its recurrence frequency. Ocular lesions may improve with immunosuppressive therapy, but usually are not fully reversible, and generally progress over time. The prognosis of anterior uveitis is usually good, but patients with posterior lesions tend to have some degree of visual loss, even with adequate treatment.

Behcet Syndrome↗
PubMed1989

Late development of chorioretinal lesions in birdshot retinochoroidopathy.

The clinical manifestations of diffuse retinochoroidopathy after recurrent episodes of vitriitis led to the diagnosis of birdshot retinochoroidopathy in two patients in whom the initial lesions were cystoid macular edema and papillophlebitis. Although the onset and clinical characteristics of the early lesions varied, both patients had manifestations of chronic retinal vasculitis. We observed the late appearance and evolution of the characteristic cream-colored depigmented spots which were not present in the early stages of the disease. In addition the second case also developed choroidal neovascularization. Serial electroretinographic examinations showed that this disorder has a progressive course from focal lesions to widespread diffuse changes.

Adult↗
PubMed1993

Antibodies to endothelial cells in patients with Behçet's disease.

Autoantibodies that bind to endothelial cells have been identified in patients with several forms of vasculitis. Behçet's disease--a multisystem inflammatory disorder of unknown etiology--is associated with thrombosis in addition to systemic manifestations resulting from small and large vessel vasculitis. We studied 72 Turkish patients (33 female, 39 male) with Behçet's disease in order to investigate the prevalence of antiendothelial cell antibodies (AECA) and to examine their possible relationship with clinical and laboratory features of the illness. Sera from 30 healthy Turkish people were used as controls. Human umbilical vein endothelial cells were cultured and used unfixed in a cellular ELISA to detect AECA. IgG and/or IgM AECA were found in 13 (18.1%) patients but not in healthy controls. Antiendothelial cell antibodies did not induce complement-mediated cytotoxicity as assessed by 51Cr release assay and the binding was not due to immune complexes. The prevalences of acute thrombotic events and retinal vasculitis at the time of the AECA assay among patients with AECA were significantly higher than those in patients without AECA. Laboratory parameters of active disease were higher in patients with AECA. There was no correlation between other clinical and laboratory features of Behçet's disease and AECA. Anticardiolipin and antineutrophil cytoplasmic antibodies were negative in our series, excluding a possibility of cross-reaction with AECA. Our results suggest a possible role of AECA in association with thrombosis and vasculitis in patients with Behçet's disease.

Adolescent↗
PubMed1980

Vogt-Koyanagi-Harada syndrome.

We studied 20 patients with Vogt-Koyanagi-Harada syndrome. They represented 3.7% of referred patients with uveitis. American Indian ancestry appears to be the link to Oriental racial type. Five patients had a forme fruste retinal detachment. After extensive treatment with corticosteroids, detachments flattened within an average of two to three months. Anterior inflammation continued to persist chronically. Seven of our patients had retinal vasculitis. Pregnancy had a beneficial effect on disease activity. Those patients with diabetes mellitus and sickle cell hemoglobin did not have an adverse effect. Visual outcome was good; of 20 patients, 16 had visual ascuity of 6/21 (20/70) in at least one eye.

Adolescent↗