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      • Record: found
      • Abstract: found
      • Article: found
      Is Open Access

      Acute Retinal Necrosis in Childhood

      a-c , * , a-c
      Case Reports in Ophthalmology
      S. Karger AG
      Retinal necrosis, Children, Methylprednisolone

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          Background: Acute retinal necrosis (ARN) is a viral syndrome consisting of uveitis/vitritis, occlusive vasculitis and peripheral necrosis. Few incidents are reported in children. The etiology is reactivated herpes simplex virus (HSV) or varicella-zoster virus (VZV). Treatment with acyclovir is often used. The administration of oral glucocorticosteroids is of unproven benefit. Prognosis is variable but poor. Methods: Three weeks after contracting mild chickenpox, a healthy 4-year-old girl developed blurred vision in her right eye. Severely reduced visual acuity was noted, together with anterior uveitis, ‘mutton-fat' precipitates and vitral flare. Retinal vasculitis with necrosis was present. Serology for toxoplasma, cytomegalovirus and HIV was negative, while HSV and VZV IgG antibodies were positive. She was treated with 30 mg/kg of intravenous methylprednisolone (3 days), 30 mg of oral prednisone (3 days), and tapering for 8 weeks. Intravenous acyclovir was given for 10 days, followed by oral acyclovir for 4 months. Aspirin (100 mg/day) was given for 4 months. Results: At 12 months, the girl felt good. Her right eye acuity was 6/9, with an intraocular pressure of 17 mm Hg. The peripheral retina showed scarring but no detachment. Conclusions: This is the first report of a once-daily high-dose methylprednisolone pulse therapy in one of the youngest known ARN cases. Pulsed steroid therapy was based on its known effectiveness in vasculitis, which is the main pathophysiology in ARN. There was no evidence of steroid-related viral over-replication. Our case achieved an excellent clinical and ophthalmic recovery in spite of the poor prognosis. The positive result of this case report provides a basis for further evaluation of high-dose steroid pulse therapy in ARN.

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          Most cited references16

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          Endogenous bacterial endophthalmitis. Report of a ten-year retrospective study.

          The purpose of this study is to report the predisposing factors, timing of symptoms, timing of diagnosis, causative organisms, source of infection, and visual outcome in cases of endogenous bacterial endophthalmitis. The records of 28 patients with endogenous bacterial endophthalmitis who presented to our combined ophthalmology and medicine services over a 10-year period were reviewed. Ninety percent of the patients had prior medical conditions, including diabetes mellitus, gastrointestinal disorders, hypertension, cardiac disorders, and malignancy. Acute ocular symptoms were the most common reasons why the patient went to the physician (usually an ophthalmologist) rather than systemic symptoms. A correct initial diagnosis was made in 50% of patients, with a delay in diagnosis of 4 days or more in 29% of patients. Organisms were identified in 27 of the 28 patients (96% identification rate), two thirds of which were gram-positive organisms. Streptococcal species were the most common group (32% of patients), although the single most common organism was Staphylococcus aureus (25% of patients). Sources of infection were identified in 93% of patients, with endocarditis and the gastrointestinal tract being the most common. In the majority of patients, visual outcome was poor. However, six eyes that received intravenous and intravitreal antibiotics, as well as therapeutic vitrectomy, achieved visual acuities of 20/50 or better. These results provide a further understanding of the manner of presentation, organisms involved, and sources of infection in endogenous bacterial endophthalmitis. They also suggest that improved outcome may be associated with early initiation of combined medical and surgical treatment.
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            Standard diagnostic criteria for the acute retinal necrosis syndrome. Executive Committee of the American Uveitis Society.

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              Behçet's syndrome: disease manifestations, management, and advances in treatment.

              The acne lesions characteristic of Behçet's syndrome are not sterile and are commonly observed in combination with arthritis. The two main nodular skin lesions--superficial thrombophlebitis and erythema nodosum--are equally frequent, and rather difficult to distinguish. Superficial thrombophlebitis is usually observed in combination with thrombosis in large veins, and thrombosis of the large veins usually clusters with dural sinus thrombi, which make up approximately 20% of all central nervous system (CNS) lesions of Behçet's syndrome. The remaining CNS lesions are parenchymal, mainly located in the brainstem, and associated with a graver prognosis than dural sinus thrombi. The presence of clinical clusters indicates that there are at least two pathogenetic pathways in Behçet's syndrome: a reactive arthritis pathway and a thrombophilia pathway. Research into the pathogenesis of Behçet's syndrome has shown that the most consistent genetic marker of Behçet's syndrome is HLA-B51; however, the genetic association of this true-to-form 'complex' disorder with HLA-B51 is only 20%, and a whole-genome study showed associations with 16 different loci. The severity of Behçet's syndrome and the mortality associated with it tend to decrease with time, and there is no associated increase in incidence of atherosclerosis. Although treatment of skin-mucosa manifestations, eye disease and pulmonary artery aneurysms has improved significantly in the past decades, the treatment of CNS lesions and thrombophilia are still problematic.

                Author and article information

                Case Reports in Ophthalmology
                S. Karger AG
                May – August 2014
                14 May 2014
                : 5
                : 2
                : 138-143
                aTzameret Medical Track, Hebrew University, Jerusalem, bDepartment of Ophthalmology, Ziv Medical Center, and cFaculty of Medicine, Bar Ilan University, Safed, Israel
                Author notes
                *Yoav Y. Pikkel, Hebrew University, Haarazim 12/34, IL-9618221 Jerusalem (Israel), E-Mail yoav.pikkel@mail.huji.ac.il
                363130 PMC4049010 Case Rep Ophthalmol 2014;5:138-143
                © 2014 S. Karger AG, Basel

                Open Access License: This is an Open Access article licensed under the terms of the Creative Commons Attribution-NonCommercial 3.0 Unported license (CC BY-NC) ( http://www.karger.com/OA-license), applicable to the online version of the article only. Distribution permitted for non-commercial purposes only. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                Page count
                Figures: 2, Pages: 6
                Published: May 2014

                Vision sciences,Ophthalmology & Optometry,Pathology
                Retinal necrosis,Children,Methylprednisolone
                Vision sciences, Ophthalmology & Optometry, Pathology
                Retinal necrosis, Children, Methylprednisolone


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