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      Acute Macular Neuroretinopathy in a 15-Year-Old Boy: Optical Coherence Tomography and Visual Acuity Findings


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          Purpose: To report a case of acute macular neuroretinopathy (AMN) in a 15-year-old boy. Methods: Images were obtained with fundus photography and optical coherence tomography (OCT). Results: The patient complained of blurred vision and a small central scotoma in the left eye. Left visual acuity was 0.3. Fundus photographs revealed a small dark area in the fovea of the left eye. OCT showed attenuation of the photoreceptor inner segment (IS)/outer segment (OS) line and the OS/retinal pigment epithelium (RPE) line in the left eye. One week after the initial visit, left visual acuity had improved to 0.6, and there was partial recovery of the IS/OS line with focal thinning of the OS/RPE line. One month later, left visual acuity had improved to 0.8, and OCT showed complete restoration of the IS/OS line and the OS/RPE line. Conclusions: To our knowledge, this is the youngest patient reported with a diagnosis of AMN. Changes in microstructural findings and visual acuity were analogous during recovery of AMN in our patient.

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          Most cited references15

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          Paracentral acute middle maculopathy: a new variant of acute macular neuroretinopathy associated with retinal capillary ischemia.

          With the advent of more sophisticated imaging systems, such as spectral domain optical coherence tomography (SD-OCT), disruption of the inner segment/outer segment (IS/OS) band, and thinning of the outer nuclear layer (ONL) have been identified in association with acute macular neuroretinopathy (AMN). To characterize a new SD-OCT presentation of AMN as a paracentral acute middle maculopathy and to describe multimodal imaging findings that implicate an underlying pathogenesis related to retinal capillary ischemia. Retrospective observational case series (January 1, 2012, to January 1, 2013) reviewing clinical and imaging data from 9 patients (11 eyes) with AMN at 6 tertiary referral centers. Lesions were classified as type 1 or 2 in relation to the SD-OCT location of the lesion above (type 1) or below (type 2) the outer plexiform layer (OPL) at 6 tertiary referral centers. Of the 9 patients, 5 were female and 4 were male (mean age, 47.6 years; range, 21-65 years). All patients presented with an acute paracentral scotoma and demonstrated a classic dark gray paracentral lesion with near-infrared imaging. Visual acuity ranged from 20/15 to 20/30. Six eyes (5 patients) had type 1 SD-OCT lesions, also referred to as paracentral acute middle maculopathy, and 5 eyes (4 patients) had type 2 SD-OCT lesions. Although type 1 lesions lead to inner nuclear layer (INL) thinning, type 2 lesions resulted in ONL thinning. Type 2 lesions were always associated with significant outer macular defects, including disruption of the inner segment/outer segment and outer segment/retinal pigment epithelium bands, whereas type 1 lesions spared the outer macula. Paracentral acute middle maculopathy may represent a novel variant of AMN that affects the middle layers of the macula above the OPL as diagnosed with SD-OCT imaging. Two types of AMN lesions may be seen with SD-OCT occurring above and below the OPL. Type 1 refers to hyperreflective bands in the OPL/INL region with subsequent INL thinning. Type 2 is hyperreflective bands in the OPL/ONL region with subsequent ONL thinning. Type 2 lesions may be associated with concomitant defects of the inner segment/outer segment layer. We propose that each of these lesions may be explained by occlusion of either the superficial capillary plexus (type 1) or deep capillary plexus (type 2) located in the innermost and outermost portion of the INL, respectively, immediately adjacent to each corresponding lesion type.
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            Acute macular neuroretinopathy.

            An unrecognized acute macular affection occurred in four women, 24 to 35 years old, using oral contraceptives who complained of a sudden decrease of visual acuity or paracentral scotomas. Three patients had bilateral lesions and one patient had unilateral lesions. These lesions consisted of darkish brown-red, wedge-shaped dots in the macula pointing to the fovea. These dots were located mostly on the nasal side to the macula. Biomicroscoby showed these lesions were located in the superficial layers of the retina. The retinal vessels, pigment epithelium, and optic disk showed no distinct pathologic features. Fluorescein angiography, performed repeatedly, showed some questionably dilated perimacular capillaries without leakage in two cases. Static perimetry delineated dense paracentral scotomas. Recovery was slow or absent, confirmed py perimetric observation. Ophthalmoscopic, fluorescein angiography, and perimetric details excluded an affection of the pigment epithelium, the nerve fiber layer, and the optic disk. Since the affection appears to be localized superficially in the retina, we called this specific entity acute macular neuroretinopathy.
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              Acute macular neuroretinopathy: long-term insights revealed by multimodal imaging.

              To report the structural and functional changes in acute macular neuroretinopathy (AMN) and their long-term evolution. Multimodal retinal imaging was acquired, including Fourier domain optical coherence tomography (OCT), infrared (IR) reflectance, and near IR autofluorescence (NIA). In this retrospective observational case series, detailed clinical history and multimodal imaging are reported in eight patients with AMN. Manual segmentation of the Fourier domain OCT volume scans was done in one patient with the largest AMN lesion to yield retinal sublayer topographic maps. Two patients were seen within the first 1 to 2 days of symptoms, and both showed outer nuclear and outer plexiform layer hyperreflectivity. Both patients developed enlargement of the lesion over the first week on IR reflectance imaging with a corresponding lateral extension of the outer retinal disruption on Fourier domain OCT. Thinning of the outer nuclear layer persisted in all patients with lesions >100 μm width, and in one patient this thinning worsened over the course of follow-up, as noted on the sublayer maps. This structural abnormality correlated with long-term functional deficits, persisting up to 14 months after the initial episode. Infrared reflectance highlights the lesion best, and abnormalities on near IR autofluorescence may be present. Acute macular neuroretinopathy acutely affects the outer nuclear and plexiform layers manifesting as OCT hyperreflectivity. The hallmark long-term changes are outer nuclear thinning on Fourier domain OCT and a fading dark lesion on IR reflectance imaging. These changes correspond to focal disruption of the outer segment/retinal pigment epithelium junction on OCT, and not the inner segment/outer segment junction, as previously reported. Optical coherence tomography and near IR autofluorescence abnormalities suggest previously unrecognized melanin and retinal pigment epithelium derangements in this condition.

                Author and article information

                Case Reports in Ophthalmology
                S. Karger AG
                January – April 2014
                08 January 2014
                : 5
                : 1
                : 11-15
                Department of Ophthalmology, Jichi Medical University, Shimotsuke, Japan
                Author notes
                *Shinji Makino, Department of Ophthalmology, Jichi Medical University, 3311-1 Yakushiji, Shimotsuke, Tochigi 329-0498 (Japan), E-Mail makichan@jichi.ac.jp
                358166 PMC3934695 Case Rep Ophthalmol 2014;5:11-15
                © 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: 5
                Published: January 2014

                Vision sciences,Ophthalmology & Optometry,Pathology
                Photoreceptor inner segment/outer segment junction,Acute macular neuroretinopathy,Retinal pigment epithelium,Optical coherence tomography


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