Paediatric Behcet's disease presenting with recurrent papillitis and episcleritis: A case report

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Abstract

Introduction: Behcet'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 Behcet'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 Behcet's disease. Infliximab is an effective, new therapeutic approach for Behcet's disease that is refractory to the conventional corticosteroid and immunosuppressive therapy.

Original languageEnglish
Pages (from-to)81
Number of pages1
JournalJournal of Medical Case Reports
DOIs
Publication statusAccepted/In press - Feb 25 2011

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Scleritis
Papilledema
Behcet Syndrome
Pediatrics
Adrenal Cortex Hormones
Retinal Vasculitis
Posterior Uveitis
Iridocyclitis
Retinal Vessels
Optic Neuritis
compound A 12
Cheek
Uveitis
Immunosuppressive Agents
Vasculitis
Cyclosporine
Therapeutics
Joints
Inflammation
Infliximab

ASJC Scopus subject areas

  • Medicine(all)

Cite this

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title = "Paediatric Behcet's disease presenting with recurrent papillitis and episcleritis: A case report",
abstract = "Introduction: Behcet'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 Behcet'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 Behcet's disease. Infliximab is an effective, new therapeutic approach for Behcet's disease that is refractory to the conventional corticosteroid and immunosuppressive therapy.",
author = "Fulvio Parentin and Loredana Lepore and Ingrid Rabach and Stefano Pensiero",
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AU - Pensiero, Stefano

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N2 - Introduction: Behcet'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 Behcet'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 Behcet's disease. Infliximab is an effective, new therapeutic approach for Behcet's disease that is refractory to the conventional corticosteroid and immunosuppressive therapy.

AB - Introduction: Behcet'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 Behcet'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 Behcet's disease. Infliximab is an effective, new therapeutic approach for Behcet's disease that is refractory to the conventional corticosteroid and immunosuppressive therapy.

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