PURPOSE: To report a case of fulminant toxoplasmic chorioretinitis following intravitreal dexamethasone implantation monotherapy in a stabilized toxoplasmic chorioretinitis patient with initial treatment. CASE SUMMARY: A 60-year-old healthy female presented with decreased visual acuity in the left eye. On fundus examination, focal chorioretinitis and yellow-white infiltration were observed. Laboratory work-up, including blood chemistry, complete blood count, and serum serology, was negative; however, toxoplasmic chorioretinitis could not be ruled out. The primary lesion improved with antibiotics and prednisolone treatment. However, the patient did not come in for her follow-up visit, as she had already received an intravitreal dexamethasone implant for recurrent vitreous inflammation elsewhere. On her return, she presented with necrotic retinitis with extensive infiltration. She underwent diagnostic vitrectomy and implant removal. A diagnosis of toxoplasma antigen was confirmed by polymerase chain reaction analysis; the lesions stabilized after anti-toxoplasmic therapy. CONCLUSIONS: Intravitreal dexamethasone implant monotherapy with stabilized toxoplasmic chorioretinitis without systemic antibiotics can lead to fulminant toxoplasmic chorioretinitis and should be used with caution.
Anti-Bacterial Agents ; Blood Cell Count ; Chemistry ; Chorioretinitis ; Dexamethasone ; Diagnosis ; Female ; Follow-Up Studies ; Humans ; Inflammation ; Intravitreal Injections ; Middle Aged ; Polymerase Chain Reaction ; Prednisolone ; Retinitis ; Toxoplasma ; Toxoplasmosis ; Visual Acuity ; Vitrectomy

PURPOSE: This study sought to describe the different clinical features and presentations of primary ocular toxoplasmosis in a setting not demonstrating an outbreak of disease.METHODS: This was a retrospective review of patients presenting to uveitis management services in Auckland and Hamilton, New Zealand between 2003 to 2018 with uveitis and positive toxoplasmosis immunoglobulin M serology.RESULTS: We identified 16 patients with primary acquired toxoplasmosis infection and ocular involvement. The mean age was 53 years. Systemic symptoms were reported in 56% (9 / 16). Visual acuity was reduced to 20 / 30 or less in 50% of patients (8 / 16). A single focus of retinitis without a pigmented scar was the salient clinical feature in 69% (11 / 16). Optic nerve inflammation was the sole clinical finding in 19% (3 / 16). Bilateral arterial vasculitis was the sole clinical finding in 13% (2 / 16). A delay in the diagnosis of toxoplasmosis of more than two weeks occurred in 38% (6 / 16) due to an initial alternative diagnosis. Antibiotic therapy was prescribed in all cases. Vision was maintained or improved in 69% (11 / 16) at the most recent follow-up visit (15 months to 10 years). Relapse occurred in 69% (11 / 16), typically within four years from the initial presentation.CONCLUSIONS: Primary ocular toxoplasmosis presenting in adulthood is a relatively uncommon cause of posterior uveitis in New Zealand. This condition should be considered in any patient presenting with retinitis or optic nerve inflammation without a retinochoroidal scar. This disease tends to relapse; thus, close follow-up is required.
Cicatrix ; Diagnosis ; Follow-Up Studies ; Humans ; Immunoglobulin M ; Inflammation ; New Zealand ; Optic Nerve ; Recurrence ; Retinitis ; Retrospective Studies ; Toxoplasmosis ; Toxoplasmosis, Ocular ; Uveitis ; Uveitis, Posterior ; Vasculitis ; Visual Acuity

OBJECTIVETo investigate optical coherence tomography (OCT) characteristics of tuberculous serpiginous-like choroiditis (Tb-SLC) and serpiginous choroiditis (SC) and to perform OCT to differentiate between these conditions.

METHODSThis retrospective, case-control study examined consecutively enrolled patients with active Tb-SLC or SC. Patients underwent comprehensive ocular examinations and imaging (OCT, color fundus photography, autofluorescence imaging, fluorescein angiography, and indocyanine green angiography). Findings were examined and compared between eyes with SC and Tb-SLC.

RESULTSNine patients with active Tb-SLC (14 eyes) and 8 with active SC (12 eyes) were included. The following OCT findings were observed significantly more often in the Tb-SLC group than in the SC group: vitreal hyper-reflective spots [5 Tb-SLC eyes (36%), no SC eyes; P = 0.02], intraretinal edema [11 Tb-SLC eyes (79%), 3 SC eyes (25%); P = 0.01], sub-retinal pigment epithelium (RPE) drusenoid deposits [11 Tb-SLC eyes (79%), 2 SC eyes (17%); P < 0.01], and choroidal granulomas [8 Tb-SLC eyes (57%), 2 SC eyes (17%); P = 0.03]. A hyporeflective, wedge-shaped band was observed more often in the SC group [5 Tb-SLC eyes (36%), 9 SC eyes (75%); P = 0.045] than in the Tb-SLC group. The incidence of other OCT signs did not differ between the groups and included outer nuclear layer hyper-reflection, outer retinal tabulation, and choriocapillaris point-like hyper-reflection.

CONCLUSIONVitreal hyper-reflective spots, intraretinal fluid, sub-RPE drusenoid deposits, and choroidal granulomas on OCT images may indicate Tb-SLC. Additionally, a hyporeflective, wedge-shaped band may indicate SC. Therefore, OCT is likely helpful in differentiating between Tb-SLC and SC.

Adult ; Case-Control Studies ; Choroiditis ; diagnostic imaging ; Female ; Humans ; Male ; Middle Aged ; Retrospective Studies ; Tomography, Optical Coherence ; Tuberculosis, Ocular ; diagnostic imaging ; pathology

PURPOSE: The aim of this study was to evaluate the clinical characteristics of children diagnosed as cryopyrin-associated periodic syndrome (CAPS) in Korea. METHODS: Diagnosis was made based on clinical features and confirmed by a mutation in the cold-induced autoinflammatory syndrome 1 (CIAS1) gene. Especially, osteocartilaginous overgrowth in the patella or distal femur was so characteristic that its presence warranted a diagnosis of chronic infantile neurologic cutaneous and articular/NOMID. RESULTS: We observed the clinical features of 9 Korean CAPS patients. All the patients suffered from an urticarial rash with recurrent fever. Among the 9 patients, 6 presented with rash and 4 with fever on the 1st or 2nd days of birth. Eight patients showed myalgia, and 7 patients showed arthralgia in the joints, and 6 patients showed radiologic findings of arthropathy including cupping of the metaphysis, excessive growth of the epiphysis, osteopenia or overgrowth of the cartilage. Four patients showed brain atrophy, enlarged ventricles or leptomeningeal enhancement on magnetic resonance imaging. Intellectual disability was observed in 1 patient. Five patients had eye involvement as conjunctivitis, uveitis, chorioretinitis, avascular area or papillary edema, and 3 patients showed progressive hearing loss. All 9 patients showed increased C-reactive protein (CRP) and erythrocyte sedimentation rate (ESR). CONCLUSIONS: All the patients carried a mutation on exon 3 of the CIAS1 gene. After the anakinra (interleukin-1 receptor antagonist) therapy, the fever and rash immediately disappeared, and CRP and ESR were improved.
Arthralgia ; Atrophy ; Blood Sedimentation ; Bone Diseases, Metabolic ; Brain ; C-Reactive Protein ; Cartilage ; Child ; Chorioretinitis ; Conjunctivitis ; Cryopyrin-Associated Periodic Syndromes ; Diagnosis ; Edema ; Epiphyses ; Exanthema ; Exons ; Femur ; Fever ; Hearing Loss ; Humans ; Intellectual Disability ; Interleukin 1 Receptor Antagonist Protein ; Joints ; Korea ; Magnetic Resonance Imaging ; Myalgia ; Parturition ; Patella ; Uveitis

PURPOSE: The purpose of this study is to make a distinction between tuberculous serpiginous-like choroiditis and serpiginous choroiditis, and compare their clinical manifestations. METHODS: We retrospectively reviewed thirty eight eyes of twenty-six patients who visited our institution and were diagnosed with serpiginous choroiditis from January 2005 to December 2014. The patients were divided into two groups, tuberculosis serpiginous- like choroiditis (Tb-SLC) and classic serpiginous choroiditis (classic SC), and were analyzed based on the treatment response, previous history of Bacillus Calmette–Guérin (BCG) vaccination, positive results of tuberculin skin test (TST), chest X-ray, anterior and fundus examination, and fluorescein angiography (FAG). RESULTS: Twenty seven eyes of eighteen patients were serpiginous choroiditis and eleven eyes of eight patients were tuberculosis serpiginous-like choroiditis. There were no significant differences in age, sex, or previous history of BCG vaccination between the two groups. The positive result of the tuberculin skin test and abnormality in the chest X-ray were shown to be significantly higher in the Tb-SLC group. Multi-focal lesions involving periphery observed in fundus examination and FAG were significantly higher in Tb-SLC. CONCLUSIONS: In tuberculosis endemic areas such as Korea, tuberculosis serpiginous-like choroiditis should be considered as a differential diagnosis when the patient is suspicious of serpiginous choroiditis. The understanding of various clinical manifestations of tuberculosis serpiginous-like choroiditis may derive accurate diagnosis and treatment, enhancing patient's prognosis.
Bacillus ; Choroid* ; Choroiditis* ; Diagnosis ; Diagnosis, Differential* ; Fluorescein Angiography ; Humans ; Korea ; Mycobacterium bovis ; Prognosis ; Retrospective Studies ; Skin Tests ; Thorax ; Tuberculin ; Tuberculosis* ; Vaccination

Toxoplasma gondii is an intracellular Apicomplexan parasite and a causative agent of toxoplasmosis in human. It causes encephalitis, uveitis, chorioretinitis, and congenital infection. T. gondii invades the host cell by forming a moving junction (MJ) complex. This complex formation is initiated by intermolecular interactions between the two secretory parasitic proteins—namely, apical membrane antigen 1 (AMA1) and rhoptry neck protein 2 (RON2) and is critically essential for the host invasion process. By this study, we propose two potential leads, NSC95522 and NSC179676 that can efficiently target the AMA1 hydrophobic cleft, which is a hotspot for targeting MJ complex formation. The proposed leads are the result of an exhaustive conformational search-based virtual screen with multilevel precision scoring of the docking affinities. These two compounds surpassed all the precision levels of docking and also the stringent post docking and cumulative molecular dynamics evaluations. Moreover, the backbone flexibility of hotspot residues in the hydrophobic cleft, which has been previously reported to be essential for accommodative binding of RON2 to AMA1, was also highly perturbed by these compounds. Furthermore, binding free energy calculations of these two compounds also revealed a significant affinity to AMA1. Machine learning approaches also predicted these two compounds to possess more relevant activities. Hence, these two leads, NSC95522 and NSC179676, may prove to be potential inhibitors targeting AMA1-RON2 complex formation towards combating toxoplasmosis.
Chorioretinitis ; Drug Design ; Encephalitis ; Humans ; Hydrophobic and Hydrophilic Interactions ; Machine Learning ; Membranes* ; Molecular Docking Simulation ; Molecular Dynamics Simulation ; Neck ; Parasites ; Pliability ; Toxoplasma* ; Toxoplasmosis ; Uveitis

A case of a female patient with local and systemic complications of metallosis, following catastrophic wear of a revised hip arthroplasty, is presented. The patient had a history of a fractured ceramic-on-ceramic implant, exchanged with a metal-on-polyethylene prosthesis. Systemic complications included sarcoidosis-like reactions, presenting as granulomatous lung disease, along with chorioretinitis, erythema nodosum, and cardiomyopathy. High local and circulating cobalt and chromium levels established the diagnosis. The patient underwent extensive debridement and implant revision. One year postoperatively, she had no respiratory symptoms or functional impairment. Local and systemic complications of metallosis after hip arthroplasty should be promptly recognized and treated operatively.
Arthroplasty* ; Arthroplasty, Replacement, Hip ; Cardiomyopathies ; Ceramics ; Chorioretinitis ; Chromium ; Cobalt ; Debridement ; Diagnosis ; Erythema Nodosum ; Female ; Hip* ; Humans ; Lung Diseases* ; Lung* ; Prostheses and Implants ; Reoperation ; Sarcoidosis, Pulmonary

PURPOSE: To evaluate the therapeutic effect and safety of mycophenolate mofetil (MMF) on chronic uveitis in Korean patients. METHODS: This study included 25 patients with chronic uveitis who used MMF and were followed up more than 6 months in 2 referral centers from 2010 to 2014. The medical records were analyzed retrospectively. The therapeutic effect was assessed based on control of inflammation, corticosteroid sparing effects, and discontinuation of MMF, and the safety was assessed based on side effects. Control of inflammation was defined as no active inflammation observed on at least 2 consecutive visits 28 days apart or more. RESULTS: The 25 patients consisted of 18 males and 7 females. The mean age of the patients was 47.52 years. The etiology of uveitis was as follows: Behcet's disease in 15 patients (60%), Vogt-Koyanagi-Harada disease in 4 (16%), sympathetic ophthalmia in 2 (8%), systemic lupus erythematosus in 1 (4%), and idiopathic uveitis in 3 (12%). Anatomic classification was anterior uveitis in 20% and posterior uveitis or panuveitis in 80% of patients. Complete control of inflammation was achieved in 44% and 50% of patients within 6 months and 1 year, respectively. Systemic corticosteroid dosage was reduced to 10 mg of prednisone or less while maintaining sustained control of inflammation in 36% and 45% of patients for 6 months and 1 year, respectively. MMF was discontinued in 3 patients (12%) due to side effects and in 2 patients (8%) due to lack of effectiveness. CONCLUSIONS: MMF was effective and side effects were uncommon when managing chronic uveitis in Korean patients.
Classification ; Female ; Humans ; Inflammation ; Lupus Erythematosus, Systemic ; Male ; Medical Records ; Ophthalmia, Sympathetic ; Panuveitis ; Prednisone ; Referral and Consultation ; Retrospective Studies ; Uveitis* ; Uveitis, Anterior ; Uveitis, Posterior ; Uveomeningoencephalitic Syndrome

Sarcoidosis is a granulomatous disease which frequently involves eyes and adnexal tissues. Its prevalence of ocular involvement is reported as about 20%, although it varies widely according to authors, from 12% to 73%. Ocular sarcoidosis is present as various forms - uveitis, conjunctival involvement, optic nerve involvement, and orbital involvement. All the forms of uveitis - anterior uveitis, intermediate uveitis, posterior uveitis and panuveitis - can be shown in sarcoidosis. Because clinical manifestation, prognosis and treatment are determined by the location of uveitis, it is important to classify uveitis. Conjunctival involvement is not a sight-threatening condition in general, its diagnostic value may be of use. Optic nerve is the most commonly involved cranial nerve, which can impair vision. Subjects with visual impairment without any evidence of uveitis, optic nerve involvement should be suspected. Orbital involvement, especially lacrimal gland involvement is common, and it may cause mass-effect. Besides systemic administration of drugs, there are several localized treatments for ocular sarcoidosis - topical eye drops, intra/peri-ocular injection of agents.
Cranial Nerves ; Granuloma ; Lacrimal Apparatus ; Ophthalmic Solutions ; Optic Nerve ; Orbit ; Panuveitis ; Prevalence ; Prognosis ; Sarcoidosis* ; Uveitis ; Uveitis, Anterior ; Uveitis, Intermediate ; Uveitis, Posterior ; Vision Disorders

PURPOSE: Systemic lupus erythematosus (SLE) is a chronic autoimmune disorder with widespread manifestations that rarely include the eye. We present a case of SLE-associated choroidoretinopathy and secondary angle closure attack in both eyes. CASE SUMMARY: A 58-year-old male was admitted into the urologic department complaining of right scrotal swelling, and then consulted with the ophthalmology department regarding both ocular pain and eye injection. The patient was diagnosed with acute angle closure attack using a slit lamp test and tonometry secondary to choroidoretinitis with choroidal detachment at fundus examination in both eyes. The rheumatologist performed systemic evaluation, including serologic tests, and then diagnosed the patient with SLE. After systemic steroid therapy, intraocular pressure was decreased and choroidal detachment disappeared with improvements of choroidoretinitis in both eyes. CONCLUSIONS: Patients with systemic lupus erythematosus choroidopathy can develop secondary angle closure attack, which can be effectively treated using systemic steroid therapy and antiglaucoma drugs.
Choroid ; Choroiditis ; Humans ; Intraocular Pressure ; Lupus Erythematosus, Systemic* ; Male ; Manometry ; Middle Aged ; Ophthalmology ; Serologic Tests ; Slit Lamp