Granulomatosis with Polyangiitis Complicated by Severe Exudative Retinal Detachment and Orbital Granuloma Successfully Controlled with Rituximab: A Case Report.

We report a rare case of severe exudative retinal detachment with orbital granuloma associated with granulomatosis with polyangiitis (GPA). A 42-year-old man developed bilateral conjunctival hyperemia and eye pain 15 months before presenting to us. Because vitreous cells and retinal detachment were detected in his left eye, he was referred to us for further evaluation. The left eye showed scleral edema, cells in the anterior chamber and anterior vitreous, exudative retinal detachment, and elevated white subretinal lesions from the nasal to the inferior parts of the eye fundus. Orbital contrast-enhanced magnetic resonance imaging revealed a granulomatous lesion, retinal detachment, and fluid retention in the left eyeball. Comprehensive rheumatological evaluation revealed proteinase 3 anti-neutrophil cytoplasmic antibody positivity and a history of otitis media, leading to a GPA diagnosis. Methylprednisolone 1,000 mg/day was administered intravenously for 3 days, followed by oral prednisolone and intravenous cyclophosphamide. Although the retinal detachment decreased, scleritis and choroidal detachment relapse were observed in the left eye after the fifth cyclophosphamide administration. After switching from cyclophosphamide to rituximab, the scleritis and choroidal detachment resolved. Remission was successfully maintained with biannual rituximab administration. In this case, we conclude that rituximab was important to re-induce and maintain remission after recurrence. Collaboration with a rheumatologist is essential for proper treatment in related cases. This is the first report of ultra-widefield and multimodal imaging for retinal detachment associated with GPA.

[Clinical Survey of Uveitis in Tokyo Medical and Dental University--Comparison between the Periods of 1998-2001 and 2007-2011].

PURPOSE: To investigate a clinical survey of uveitis in Tokyo Medical and Dental University Hospital. SUBJECTS AND METHODS: The clinical records of patients with uveitis who were treated from October 1998 to December 2001 and from January 2007 to December 2011 were reviewed. The clinical results of both periods were compared. RESULTS: A total number of 455 patients (707 eyes) and 1091 patients (1716 eyes) were analyzed in this survey. The mean age of the first period was 45.4, and of the second 50.3 years old. The incidences of sarcoidosis (11.0%, 13.7%) and Vogt-Koyanagi-Harada disease (5.7%, 5.9%) were virtually unchanged in the two series. However, the incidence of Behçet's disease was higher in the first period (10.6%) than in the second (5.8%). The frequency of herpetic iritis (1.8%, 4.7%) and intraocular lymphoma (0.4%, 2.6%)showed apparent increase. Unclassified uveitis decreased from 55.6% to 47.5%. CONCLUSIONS: The etiologies of uveitis have changed with the progress of diagnostic techniques and the establishment of new disease concepts.

Clinical course of patients with Behçet's uveitis following discontinuation of infliximab therapy.

PURPOSE: To investigate the clinical course of Behçet's uveitis patients following discontinuation of infliximab therapy. METHODS: This retrospective chart review study examined Behçet's disease patients who received infliximab treatment between 2000 and 2012. Medical records of patients whose infliximab treatment was discontinued were reviewed, with special focus on the frequency of uveitis attacks in the period before initiation, during treatment and after cessation of the infliximab therapy. Mean visual acuities were evaluated for each treatment period. RESULTS: Out of the 43 patients treated with infliximab at our uveitis clinic, ten were discontinued due to adverse events or inefficiency. Data for seven patients followed for more than 12 months before initiation and after cessation of infliximab were analyzed. Frequency of acute uveitis attacks was 7.43 per 12 months before initiation of infliximab, 2.86 during treatment and 0.57 after cessation. A statistically higher frequency of uveitis attacks was observed before initiation of infliximab compared to during (p < 0.05) and after cessation of treatment (p < 0.05). There was no statistical significance observed between the period during treatment and after cessation (p = 0.29). The mean logMAR was 0.79 at baseline, 0.68 during treatment, and 0.88 at 12 months after cessation. These differences were not statistically significant. CONCLUSIONS: A satisfactory clinical course with well-controlled ocular inflammation was found after discontinuation of infliximab therapy in Behçet's uveitis patients. These results suggest that a safe, pre-planned discontinuation of infliximab therapy can be performed in patients with Behçet's uveitis.

Choroidal thickness in convalescent vogt-koyanagi-harada disease.

PURPOSE: To evaluate the subfoveal choroidal thickness (SCT) at the convalescent stage of Vogt-Koyanagi-Harada disease and to investigate the correlations among SCT, the presence of the sunset glow fundus, and size of the peripapillary atrophy (PPA). METHODS: The medical records of consecutive patients with Vogt-Koyanagi-Harada disease without active intraocular inflammation were reviewed, and one eye was randomly chosen for analyses. The disease duration was more than 3 years. Enhanced depth imaging optical coherence tomography was performed to measure SCT. The area of PPA was measured using the PDT/MPS software. Sunset glow fundi were classified into two groups according to the degree of depigmentation. RESULTS: Nineteen eyes with Vogt-Koyanagi-Harada disease were studied. The mean SCT of 12 eyes with severe sunset glow fundus was 144 ± 72 µm which was thinner than that of the 7 eyes with no or mild depigmentation (P = 0.0057). The SCT was inversely correlated with the disease duration (P = 0.048) and the PPA area (P = 0.0002). The PPA area was positively correlated with the disease duration (P = 0.007). CONCLUSION: The thinner choroid and larger PPA areas were correlated with the degree of depigmentation or disease duration and might be caused by latent choroidal inflammation in the convalescent stage of Vogt-Koyanagi-Harada disease.

Role of IL-22- and TNF-α-producing Th22 cells in uveitis patients with Behcet's disease.

Behçet's disease is a systemic inflammatory disorder with recurrent episodes of oral ulceration, skin lesions, genital ulceration, and intraocular inflammation (uveitis). The intraocular inflammation is strictly associated with Th effector cells. IL-22 is a member of the IL-10 cytokine family that is involved in inflammatory processes. Recently, Th22 cells were identified as a Th cell population that produces IL-22 and TNF-α and are distinct from Th1, Th2, and Th17 cells. In this study, we established Th22-type T cell clones from ocular samples taken from Behçet's disease patients with active uveitis. These clones produced large amounts of IL-22 and TNF-α but not the Th1 cytokine IFN-γ and the Th17 cytokine IL-17. CD4(+) T cells from the peripheral blood of Behçet's disease patients differentiated into Th22 cells in the presence of IL-6 and TNF-α in vitro. The polarized Th22 cell lines produced large amounts of IL-22, and the polarized Th1 and Th17 cells also produced IL-22. In the presence of anti-TNF-α- and anti-IL-6-blocking Abs, Behçet's disease Th22-type T cells failed to produce IL-22. In addition, infliximab-pretreated Th22 cells and Th22-type ocular T cells produced less IL-22 and TNF-α. Moreover, IL-22-producing T cells were isolated from mice with experimental autoimmune uveitis, an animal model of Behçet's disease, and the intraocular T cells from uveitis models produced large amounts of IL-22 in the presence of retinal Ags. Our results suggest that inflammatory cytokines IL-22 and TNF-α may play a key role in the ocular immune response in Behçet's disease.

Human immunodeficiency virus-related retinal microangiopathy and systemic cytomegalovirus disease association.

PURPOSE: To determine whether there is a significant association between human immunodeficiency virus (HIV)-related retinal microangiopathy and systemic cytomegalovirus (CMV) disease in HIV-infected patients. METHODS: Participants in this single-center, cross-sectional, retrospective study were 383 HIV-infected patients assessed for ocular manifestations before the beginning of antiretroviral therapy. The presence of HIV-related retinal microangiopathy, the presence of systemic CMV disease, laboratory data, and demographic information were determined by referring to medical records. The significance of any association between HIV-related retinal microangiopathy and systemic CMV disease was determined by use of the Chi-squared test and by multivariate analysis. RESULTS: HIV-related retinal microangiopathy was present in 85 patients, and was significantly associated with systemic CMV disease both by use of the Chi-squared test (P = 0.006) and by multivariate analysis (P = 0.045, odds ratio 2.03, 95 % confidence interval 1.02-4.06 adjusted for CD4+ cell count and plasma HIV-RNA level). CONCLUSIONS: These findings indicate that microangiopathy may be involved in the development of CMV disease in HIV-infected patients. Thus, detection of the presence of HIV-related retinal microangiopathy is important in the management of HIV-infected patients.

Regression of optic disc neovascularization in patients with Behçet's uveoretinitis after infliximab therapy.

PURPOSE: Optic disc neovascularization (NVD) in patients with Behçet's uveoretinitis is a relatively uncommon but severe complication that lacks standardized treatments. We report 2 cases of Behçet's uveoretinitis that achieved partial regression of NVD after infliximab therapy. METHODS: Intravenous infliximab infusions were administrated to 2 immunosuppressive therapy-resistant Behçet's uveoretinitis patients with severe NVD accompanied by vitreous hemorrhage (both eyes in case 1 and the left eye in case 2 ). Best-corrected visual acuity, funduscopic findings, and fluorescein angiography were evaluated before and after the treatments. RESULTS: After being switched to infliximab therapy, NVD in both cases stopped developing and regressed partially, as confirmed by fluorescein angiography and resolution of vitreous hemorrhage, which led to an improvement of the best-corrected visual acuity. CONCLUSIONS: Results suggest that infliximab administration might be an effective treatment in NVD patients with Behçet's uveoretinitis.

Indocyanine green angiography findings in initial acute pretreatment Vogt-Koyanagi-Harada disease in Japanese patients.

PURPOSE: Indocyanine green angiography (IA) is a highly sensitive method to evaluate choroidal inflammatory lesions. We present standardized IA findings of initial acute Vogt-Koyanagi-Harada (VKH) disease in Japanese patients before therapeutical intervention. METHODS: Medical records of patients with VKH disease at Tokyo Medical and Dental University Hospital and Miyata Eye Hospital were retrospectively analyzed. We analyzed six IA signs: choroidal perfusion inhomogeneity, early hyperfluorescent stromal vessels, hypofluorescent dark dots (HDDs), fuzzy or lost pattern of large stromal vessels, disc hyperfluorescence, and diffuse late choroidal hyperfluorescence. RESULTS: Ten patients from the two hospitals were studied. The most constant findings present in all eyes were early hyperfluorescent stromal vessels, HDDs, and either fuzzy or lost pattern of large stromal vessels. Disc hyperfluorescence was present in 18 eyes. Choroidal perfusion inhomogeneity was seen in six patients, and diffuse late choroidal hyperfluorescence was seen to a certain degree in all eyes. CONCLUSIONS: Four of the analyzed signs, including early hyperfluorescent stromal vessels, HDDs, fuzzy or lost pattern of large stromal vessels, and disc hyperfluorescence were consistent findings in Japanese VKH patients. Because the primary lesion is situated in the choroid, IA is the method of choice to monitor disease activity in VKH disease.

[Effects and safety of infliximab administration in refractory uveoretinitis with Behçet's disease].

PURPOSE: To evaluate the efficacy and safety of infliximab administration in refractory uveoretinitis in Behçet's disease (BD). METHODS: The subjects were 22 consecutive BD patients with refractory uveoretinitis treated with infliximab. Three patients dropped out from the therapy. The other 19 patients received the therapy for 6 months or longer. The efficacy was evaluated by the ocular attacks and best corrected visual acuity at remission in the 19 patients and the safety was evaluated in all 22 patients. RESULTS: The incidence of ocular attacks in the 6 months before and after the infliximab therapy were 3.0+/-2.6 and 0.4+/-1.1, respectively (p<0.0005). Infliximab was very effective in 7 patients where no ocular attacks occurred throughout infliximab therapy, effective in 10 patients where ocular attacks was reduced, and non-effective in 2 patients. The vision was either improved or unchanged in all eyes at the 6th months therapy. Various adverse effects were seen in 14 patients, but non were serious. CONCLUSIONS: Infiximab therapy is effective in managing the refractory uveoretinitis in BD, but elucidation of the safety needs more patients and a longer follow-up period.

Comparison of infliximab versus ciclosporin during the initial 6-month treatment period in Behçet disease.

AIM: To compare the efficacy and safety of infliximab versus ciclosporin A (CsA) in refractory uveoretinitis in Behçet disease. METHODS: In this retrospective clinical chart review of patients with Behçet disease who were treated with CsA or infliximab, we collected information on the number of uveitis attacks, visual acuity and adverse side effects that occurred during the 6 months prior to and after the initiation of CsA (n=20) or infliximab (n=17). RESULTS: The number of acute episodes of uveitis during the 6 months before and after initiation of CsA were 3.3+/-2.4 and 1.2+/-1.2, and those of infliximab were 3.1+/-2.7 and 0.4+/-1.0, respectively (p<0.005). The number of episodes after infliximab administration was significantly lower than that seen for CsA (p<0.05). During the 6-month treatment period, there were no significant differences noted in the improvement of the visual acuity between the two therapies. After CsA administration, neurological symptoms and renal toxicity were seen in one patient each, while after the infliximab administration, an infusion reaction and leucopenia were seen in one patient each. CONCLUSION: During the initial 6 months of treatment, infliximab proved to be more effective in reducing acute episodes of uveitis in Behçet disease.

Suboptimal therapy controls clinically apparent disease but not subclinical progression of Vogt-Koyanagi-Harada disease.

PURPOSE: To evaluate clinical and angiographic differences in patients with Vogt-Koyanagi-Harada (VKH) disease during the early 4-month treatment phase with high- or medium-dose systemic corticosteroid therapy. METHODS: VKH patients treated at the Centre for Ophthalmic Specialized Care, Lausanne, Switzerland (n = 4), or the Department of Ophthalmology, Tokyo Medical and Dental University, Tokyo, Japan (n = 5), underwent a pre-treatment indocyanine green angiography (ICGA) and a follow-up ICGA four months after treatment began. Lausanne patients received high-dose, systemic corticosteroid therapy, with or without immunosuppressive therapy. Tokyo patients received medium-dose systemic corticosteroid therapy that included 3 days of intravenous pulse methylprednisolone. ICGA signs including choroidal stromal vessel hyperfluorescence and leakage, hypofluorescent dark dots (HDD), fuzzy vascular pattern of large stromal vessels and disc hyperfluorescence were retrospectively compared. RESULTS: The pre-treatment ICGA demonstrated that each of the nine patients had choroidal inflammatory foci, as indicated by HDD. At 4-month follow-up, clinical and fluorescein findings had improved almost equally in both groups. HDD had resolved in the Lausanne group but persisted in the Tokyo group. Sunset glow fundus occurred in three of the Tokyo patients and none of the Lausanne patients. CONCLUSIONS: Submaximal doses of inflammation suppressive therapy are sufficient to suppress clinically apparent disease but not the underlying lesion process. This explains the propensity for sunset glow fundus in seemingly controlled disease.

Seroprevalence of Bartonella henselae in patients with uveitis and healthy individuals in Tokyo.

PURPOSE: To compare the seroprevalence of Bartonella henselae, a pathogen of cat scratch disease, in patients with uveitis and in healthy individuals. METHODS: Serum samples were collected from 197 consecutive patients with various entities of uveitis at Tokyo Medical and Dental University Hospital, and from 83 healthy age- and sex-matched volunteers. Anti-Bartonella IgG and IgM antibodies were examined by indirect immunofluorescent antibody. RESULTS: Serum antibodies to B. henselae were positive in 39 of 197 (19.8%) patients with uveitis, and in 21 of 83 (25.3%) of the healthy volunteers. There was no statistical difference in the seroprevalence between the two groups. The overall seroprevalence of B. henselae was 60 of 280 (21.4%). CONCLUSIONS: The seroprevalence of B. henselae was similar in both patients with uveitis and in healthy volunteers in Tokyo, Japan. These data indicate that a significant number of healthy individuals are asymptomatic carriers of B. henselae, which should be kept in mind when a diagnosis of cat scratch disease is made.

A clinical survey of uveitis in HTLV-1 endemic region.

PURPOSE: To investigate a clinical survey of uveitis in southern Kyushu of Japan, where human T-lymphotropic virus type 1 (HTLV-1) and toxoplasmosis is highly endemic. METHODS: The clinical records of patients with uveitis between 1975 and 2007 at Miyata Eye Hospital were reviewed. RESULTS: A total number of 1338 patients (2012 eyes), consisting of 526 men and 812 women with mean age of 50.5 years old, were analyzed. The most common clinical entity was HTLV-1 uveitis (17.1%), followed by Vogt-Koyanagi-Harada disease (9.9%), sarcoidosis (7.2%), toxoplasmosis (7.1%), Behçet's disease (4.3%) and others. Unclassified uveitis comprised 41.1% in the series. Anterior uveitis was seen in 30.8%, intermediate uveitis in 17.3%, posterior uveitis in 9.3%, and pan-uveitis in 42.6%. CONCLUSIONS: HTLV-1 uveitis and toxoplasmosis were the major clinical entities in southern Kyushu of Japan. This relates to the high seroprevalence of the infectious agents in this region of Japan.

Interleukin-17 causes neutrophil mediated inflammation in ovalbumin-induced uveitis in DO11.10 mice.

T cell-mediated uveitis is strongly associated with many systemic inflammatory disorders. Th17 cells are a novel T cell subset characterized by production of interleukin (IL)-17. In this study, we used DO11.10 mice to investigate the role of IL-17 in the pathogenesis of uveitis. CD4(+) T cells in DO11.10 mice are genetically engineered to react with ovalbumin (OVA). IL-17 expression was determined by real-time PCR and ELISPOT. Uveitis was induced by intravitreal injection of OVA, and ocular inflammation was evaluated by intravital microscopy. OVA challenge significantly induced IL-17 production by DO11.10 splenocytes in vitro. Next, we examined whether OVA challenge could elicit local inflammation and induce IL-17 in vivo. OVA elicited marked neutrophil-predominant inflammatory cell infiltration in the eyes. This leukocyte influx was mediated by CD4(+) lymphocytes as evidenced by significant inhibition of the ocular inflammation by CD4+ depleting antibody. Compared to control mice, OVA treatment induced IL-17 expression. Moreover, anti-IL-17 antibody markedly reduced OVA-mediated ocular inflammation. Finally, the neutralization of IL-17 attenuated ocular expression of CXCL2 and CXCL5, two cytokines which are chemotactic for neutrophils. Our study suggests that IL-17 is implicated in the pathogenesis of this T cell-mediated model of uveitis in part through neutrophil chemotaxis as a downstream effect of IL-17.

Nucleotide oligomerization domain-2 (NOD2)-induced uveitis: dependence on IFN-gamma.

PURPOSE: Nucleotide oligomerization domain-2 (NOD2) plays an important role in innate immunity to sense muramyl dipeptide (MDP), a component of bacterial cell walls. Notably, NOD2 is linked to eye inflammation because mutations in NOD2 cause a granulomatous type of uveitis called Blau syndrome. A mouse model of NOD2-dependent ocular inflammation was employed to test the role of a cytokine strongly implicated in granuloma formation, IFN-gamma, in order to gain insight into downstream functional consequences of NOD2 activation within the eye triggering uveitis. METHODS: Mice deficient in IFN-gamma, NOD2, or CD11b and their wild-type controls were treated with intravitreal injection of MDP in the presence or absence of IFN-gamma. IFN-gamma production in the eye was measured by ELISA. The intravascular inflammatory response within the iris was quantified by intravital microscopy. RESULTS: NOD2 activation resulted in the production of IFN-gamma within the eye. Deficiency in IFN-gamma diminished the development of MDP-induced uveitis, indicating its crucial role in downstream inflammatory events triggered by NOD2. Moreover, exogenous IFN-gamma markedly exacerbated MDP-induced ocular inflammation in a NOD2-dependent mechanism. The potential of IFN-gamma to enhance inflammation required the adhesion molecule CD11b because CD11b-deficient mice failed to show the synergistic effects of IFN-gamma and MDP cotreatment on adhering and infiltrating cells. CONCLUSIONS: IFN-gamma was identified as a downstream mediator of NOD2-driven inflammation and the capacity of IFN-gamma in vivo to enhance the inflammatory potential of NOD2 was demonstrated. Extrapolation of these findings in mice suggests that the dysregulation of IFN-gamma may occur in patients with Blau syndrome, thereby contributing to the granulomatous nature of the disease.

Therapy for acute retinal necrosis.

Acute retinal necrosis is a progressive necrotizing retinopathy caused by herpes simplex virus (HSV) or varicella zoster virus (VZV). The mainstay of its treatment is antiviral therapy against these pathogenic organisms, such as intravenous acyclovir or oral valacyclovir. Systemic and topical corticosteroids together with antiviral therapy are used as an anti-inflammatory treatment to minimize damages to the optic nerve and retinal blood vessels. Because the majority of severe cases of the disease show occlusive retinal vasculitis, a low dosage of aspirin is used as anti-thrombotic treatment. Vitreo-retinal surgery is useful to repair rhegmatogenous retinal detachment, one of the main late-stage complications. Moreover, recent articles have reported some encouraging results of prophylactic vitrectomy before rhegmatogenous retinal detachment occurs. The efficacy of laser photocoagulation to prevent the development or extension of rhegmatogenous retinal detachment is controversial. Despite these treatments, the visual prognosis of acute retinal necrosis is still poor, in particular VZV-induced acute retinal necrosis.

In vivo imaging of the immune response in the eye.

The immune system is governed by dynamic events involving in part direct intercellular interactions between an immune cell and other cells or the cell's environment. Owing to its unique optical characteristics, the eye offers remarkable opportunities for the analysis of the immune system by intravital microscopy. In this review, we present a brief overview of the current state of knowledge of leukocyte trafficking in each of three anatomically distinct and medically important regions of the eye (cornea, iris, retina) as determined by the application of intravital microscopy to animal models of disease. Additionally, we discuss the use of ocular imaging in patients and volunteers. Finally, we examine the future prospects for this field in terms of its potential for impacting our understanding of fundamental immunological phenomena.

[Detection of herpesvirus genome by multiplex polymerase chain reaction (PCR) and real-time PCR in ocular fluids of patients with acute retinal necrosis].

PURPOSE: The human herpesvirus (HHV) family consists of types 1 to 8 (HHV1-8). The purpose of this study was to investigate the detection of HHV DNA, especially HSV1 (herpes simplex virus 1, HHV1), HSV2 (herpes simplex virus 2, HHV2), and VZV (varicella-zoster virus, HHV3) in ocular fluids of patients with acute retinal necrosis(ARN). METHODS: The intraocular genome for HHV1-8 was determined in 19 ocular fluid samples (12 vitreous fluid and 7 aqueous humor samples) taken from ARN patients (n=14). The samples were tested for the presence of virus DNA by two systems of polymerase chain reaction (PCR): the multiplex PCR screening test and real-time quantitative PCR. RESULTS: Multiplex PCR demonstrated VZV (n=16, 84%), HSV1 (n = 1.5%) or HSV2 (n = 2.11%)genomic DNA in all the samples. In real-time PCR, a high copy number of virus DNA was detected. The virus DNA-positive samples contained Epstein-Barr virus (EBV, HHV4) DNA in 9 of 19 samples (47%). No HHV6-8 DNA was detected in the ocular samples, and no virus DNA was detected in the serum samples. CONCLUSIONS: The genome for HHV1-3 was detected in the patients with ARN. All cases contained a high copy number for the virus DNA that indicates viral replication. PCR systems are useful for determing whether virus infections are associated with uveitis.

Work-up, diagnosis and management of acute Vogt-Koyanagi-Harada disease: a case of acute myopization with granulomatous uveitis.

PURPOSE: In its typical form and when seen at onset, Vogt-Koyanagi-Harada (VKH) is characterized by easily recognizable signs that allow diagnosis without difficulty. In cases that do not have acute onset, that are seen at a later stage or that do not show the complete set of signs, appraisal is more difficult and diagnosis may cause difficulties. We present here a case of bilateral granulomatous uveitis compatible with VKH disease in order to allow several experts to give their opinion on the most appropriate manner to confirm or reject the diagnosis and their approach to the management of the case. CASE PRESENTATION: A 17-year-old female patient consulted her ophthalmologist for blurred vision OU following an episode of a flu-like disease with malaise, fever and headaches. A bilateral anterior granulomatous uveitis with a right papillitis was diagnosed and the patient presented with a bilateral acute myopization. Fluorescein angiography showed right disc hyperfluorescence with late leakage and slight left disc hyperfluorescence. The patient was given a course of one week of peroral corticosteroid therapy followed by an intramuscular injection of Bentelan twice weekly. In the absence of significant improvement the patient was sent six weeks later to a specialized center where a complete work-up was performed. EXPERT OPINION: The diagnostic work-up, investigational tests, and differential diagnosis to confirm or reject the diagnosis of VKH as well as the management of the case will be described by the experts.

Kinetics of aqueous flare, intraocular pressure and virus-DNA copies in a patient with cytomegalovirus iridocyclitis without retinitis.

BACKGROUND: A case report of recurrent unilateral granulomatous iridocyclitis with ocular hypertension without retinitis caused by cytomegalovirus (CMV) in an immunocompetent patient. METHODS: Aqueous humor was analysed by multiplex PCR to detect viral DNA, and real-time PCR was used to evaluate virus copies before and after anti-virus treatments. Inflammation of the anterior chamber was evaluated by a laser flare photometry. RESULTS: Genomic DNA of CMV - but not of other herpes viruses - was detected in the aqueous humor. Quantitative real-time PCR revealed 2.3 x 10(5) copies/ml of CMV DNA from the specimen. Oral valganciclovir was added to the ongoing treatment, which consisted of topical corticosteroid, timolol and latanoprost as well as systemic acetazolamide, resulting in the reduction of aqueous flare correlated with the reduction of virus copies in aqueous humor. CONCLUSIONS: In this case of CMV-related iridocyclitis in an immunocompetent patient, specific additional anti-viral therapy was effective in controlling inflammation of anterior chamber but, as is so often the case, it was unable to control intraocular pressure. We show that inflammatory activity correlated well with the number of virus copies in the aqueous humor.