Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report

Abstract Objective To describe a case of Behçet disease (BD) uveitis manifesting with sequential bilateral neuroretinitis associated with prepapillary inflammatory vitreous exudate (PIVE). Material and methods A single case report documented with multimodal imaging. Results A 37-year-old man develop...

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Main Authors: Imen Ksiaa, Safa Ben Aoun, Sourour Zina, Dhouha Nefzi, Sana Khochtali, Moncef Khairallah
Format: Article
Language:English
Published: SpringerOpen 2020-12-01
Series:Journal of Ophthalmic Inflammation and Infection
Subjects:
Online Access:https://doi.org/10.1186/s12348-020-00226-y
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author Imen Ksiaa
Safa Ben Aoun
Sourour Zina
Dhouha Nefzi
Sana Khochtali
Moncef Khairallah
author_facet Imen Ksiaa
Safa Ben Aoun
Sourour Zina
Dhouha Nefzi
Sana Khochtali
Moncef Khairallah
author_sort Imen Ksiaa
collection DOAJ
description Abstract Objective To describe a case of Behçet disease (BD) uveitis manifesting with sequential bilateral neuroretinitis associated with prepapillary inflammatory vitreous exudate (PIVE). Material and methods A single case report documented with multimodal imaging. Results A 37-year-old man developed neuroretinitis with associated PIVE in the left eye. He was diagnosed with ocular toxoplasmosis and treated accordingly based on positive serologic testing and negative work-up for other entities, including BD. The disease course was favorable, but 1 year later a similar neuroretinitis developed in the right eye. Extraocular features of BD became evident only at the time of the second eye involvement, and the patient received corticosteroid and immunosuppressive therapy. Swept source (SS) OCT showed at the acute phase in both eyes a typical “mushroom-shaped” prepapillary hyperreflectivity of the PIVE. SS OCT angiography (OCTA) demonstrated a corresponding prepapillary hypointense area due to shadowing effect, decreasing in size while scanning deeper layers. It also detected peripapillary retinal hypervascularity in both eyes and a sectoral area of flow signal loss in the first involved left eye. Visual acuity improved following the resolution of the PIVE and associated acute inflammatory changes in both eyes. The left eye showed residual optic disc pallor and retinal nerve fiber layer defects. Conclusion Sequential bilateral neuroretinitis associated with PIVE may occur before other clinical features of BD become evident. SS OCT and OCTA can provide useful information for the diagnosis and management of this rare, but typical, ocular manifestation of BD uveitis.
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spelling doaj.art-974c0aaa378a4e83bd391d8250492fda2022-12-21T18:56:07ZengSpringerOpenJournal of Ophthalmic Inflammation and Infection1869-57602020-12-011011610.1186/s12348-020-00226-ySequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case reportImen Ksiaa0Safa Ben Aoun1Sourour Zina2Dhouha Nefzi3Sana Khochtali4Moncef Khairallah5Department of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of MonastirDepartment of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of MonastirDepartment of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of MonastirDepartment of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of MonastirDepartment of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of MonastirDepartment of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of MonastirAbstract Objective To describe a case of Behçet disease (BD) uveitis manifesting with sequential bilateral neuroretinitis associated with prepapillary inflammatory vitreous exudate (PIVE). Material and methods A single case report documented with multimodal imaging. Results A 37-year-old man developed neuroretinitis with associated PIVE in the left eye. He was diagnosed with ocular toxoplasmosis and treated accordingly based on positive serologic testing and negative work-up for other entities, including BD. The disease course was favorable, but 1 year later a similar neuroretinitis developed in the right eye. Extraocular features of BD became evident only at the time of the second eye involvement, and the patient received corticosteroid and immunosuppressive therapy. Swept source (SS) OCT showed at the acute phase in both eyes a typical “mushroom-shaped” prepapillary hyperreflectivity of the PIVE. SS OCT angiography (OCTA) demonstrated a corresponding prepapillary hypointense area due to shadowing effect, decreasing in size while scanning deeper layers. It also detected peripapillary retinal hypervascularity in both eyes and a sectoral area of flow signal loss in the first involved left eye. Visual acuity improved following the resolution of the PIVE and associated acute inflammatory changes in both eyes. The left eye showed residual optic disc pallor and retinal nerve fiber layer defects. Conclusion Sequential bilateral neuroretinitis associated with PIVE may occur before other clinical features of BD become evident. SS OCT and OCTA can provide useful information for the diagnosis and management of this rare, but typical, ocular manifestation of BD uveitis.https://doi.org/10.1186/s12348-020-00226-yBehçet uveitisPrepapillary exudateSwept source OCTOCT angiographyCase report
spellingShingle Imen Ksiaa
Safa Ben Aoun
Sourour Zina
Dhouha Nefzi
Sana Khochtali
Moncef Khairallah
Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report
Journal of Ophthalmic Inflammation and Infection
Behçet uveitis
Prepapillary exudate
Swept source OCT
OCT angiography
Case report
title Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report
title_full Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report
title_fullStr Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report
title_full_unstemmed Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report
title_short Sequential bilateral Behçet’s neuroretinitis associated with prepapillary vitreous exudate: case report
title_sort sequential bilateral behcet s neuroretinitis associated with prepapillary vitreous exudate case report
topic Behçet uveitis
Prepapillary exudate
Swept source OCT
OCT angiography
Case report
url https://doi.org/10.1186/s12348-020-00226-y
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