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Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report

INTRODUCTION: Behçet's disease is a chronic multisystem vasculitis characterized by mucocutaneous, articular, neurological, gastrointestinal and ophthalmological lesions. Ocular involvement is mainly represented by recurrent uveitis, especially posterior uveitis; however, iridocyclitis, retinal...

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Autores principales: Parentin, Fulvio, Lepore, Loredana, Rabach, Ingrid, Pensiero, Stefano
Formato: Texto
Lenguaje:English
Publicado: BioMed Central 2011
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3058087/
https://www.ncbi.nlm.nih.gov/pubmed/21352523
http://dx.doi.org/10.1186/1752-1947-5-81
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author Parentin, Fulvio
Lepore, Loredana
Rabach, Ingrid
Pensiero, Stefano
author_facet Parentin, Fulvio
Lepore, Loredana
Rabach, Ingrid
Pensiero, Stefano
author_sort Parentin, Fulvio
collection PubMed
description INTRODUCTION: Behçet's disease is a chronic multisystem vasculitis characterized by mucocutaneous, articular, neurological, gastrointestinal and ophthalmological lesions. Ocular involvement is mainly represented by recurrent uveitis, especially posterior uveitis; however, iridocyclitis, retinal and choroidal vasculitis, optic neuritis and retinal vascular occlusion can also occur. CASE PRESENTATION: A 12-year-old Caucasian boy with a history of recurrent buccal aphthosis and nonspecific gastrointestinal symptoms was admitted to our hospital with blurred vision associated with acute episcleritis and papillitis. The patient's pathergy test was positive, suggesting a diagnosis of Behçet's disease. Corticosteroid and cyclosporine therapy was started, but further episodes were noted in both eyes. The patient was then switched to intravenous infliximab, with complete resolution of the inflammation after the second infusion. CONCLUSION: Episcleritis and papillitis should be added to the list of uncommon manifestations of pediatric Behçet's disease. Infliximab is an effective, new therapeutic approach for Behçet's disease that is refractory to the conventional corticosteroid and immunosuppressive therapy.
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spelling pubmed-30580872011-03-16 Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report Parentin, Fulvio Lepore, Loredana Rabach, Ingrid Pensiero, Stefano J Med Case Reports Case Report INTRODUCTION: Behçet's disease is a chronic multisystem vasculitis characterized by mucocutaneous, articular, neurological, gastrointestinal and ophthalmological lesions. Ocular involvement is mainly represented by recurrent uveitis, especially posterior uveitis; however, iridocyclitis, retinal and choroidal vasculitis, optic neuritis and retinal vascular occlusion can also occur. CASE PRESENTATION: A 12-year-old Caucasian boy with a history of recurrent buccal aphthosis and nonspecific gastrointestinal symptoms was admitted to our hospital with blurred vision associated with acute episcleritis and papillitis. The patient's pathergy test was positive, suggesting a diagnosis of Behçet's disease. Corticosteroid and cyclosporine therapy was started, but further episodes were noted in both eyes. The patient was then switched to intravenous infliximab, with complete resolution of the inflammation after the second infusion. CONCLUSION: Episcleritis and papillitis should be added to the list of uncommon manifestations of pediatric Behçet's disease. Infliximab is an effective, new therapeutic approach for Behçet's disease that is refractory to the conventional corticosteroid and immunosuppressive therapy. BioMed Central 2011-02-25 /pmc/articles/PMC3058087/ /pubmed/21352523 http://dx.doi.org/10.1186/1752-1947-5-81 Text en Copyright ©2011 Parentin et al; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Parentin, Fulvio
Lepore, Loredana
Rabach, Ingrid
Pensiero, Stefano
Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report
title Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report
title_full Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report
title_fullStr Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report
title_full_unstemmed Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report
title_short Paediatric Behçet's disease presenting with recurrent papillitis and episcleritis: a case report
title_sort paediatric behçet's disease presenting with recurrent papillitis and episcleritis: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3058087/
https://www.ncbi.nlm.nih.gov/pubmed/21352523
http://dx.doi.org/10.1186/1752-1947-5-81
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