Uveitis in paediatric Behçet disease: a large multicentric Italian cohort

Background: Behçet disease (BD) is a rare disease in childhood and its uveitis may lead to blindness if not properly treated. Objectives: We aim to describe a cohort of paediatric BD patients with uveitis. Design: This is a multicentric retrospective study. Methods: Six paediatric rheumatology units...

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Main Authors: Ilaria Maccora, Sara Immacolata Orsini, Romina Gallizzi, Davide Montin, Marco Cattalini, Francesco La Torre, Alessandra Spagnolo, Federico Diomeda, Gabriele Simonini
Format: Article
Language:English
Published: SAGE Publishing 2024-12-01
Series:Therapeutic Advances in Musculoskeletal Disease
Online Access:https://doi.org/10.1177/1759720X241275822
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author Ilaria Maccora
Sara Immacolata Orsini
Romina Gallizzi
Davide Montin
Marco Cattalini
Francesco La Torre
Alessandra Spagnolo
Federico Diomeda
Gabriele Simonini
author_facet Ilaria Maccora
Sara Immacolata Orsini
Romina Gallizzi
Davide Montin
Marco Cattalini
Francesco La Torre
Alessandra Spagnolo
Federico Diomeda
Gabriele Simonini
author_sort Ilaria Maccora
collection DOAJ
description Background: Behçet disease (BD) is a rare disease in childhood and its uveitis may lead to blindness if not properly treated. Objectives: We aim to describe a cohort of paediatric BD patients with uveitis. Design: This is a multicentric retrospective study. Methods: Six paediatric rheumatology units in Italy were involved including children with a diagnosis of paediatric BD according to the International Criteria for BD Criteria and/or to the International Study Group Criteria for BD, or Paediatric BD classification criteria if they had uveitis. Demographic, laboratory and clinical data were collected and followed up to March 2023. Ocular characteristics and treatment response were assessed according to Standardization Uveitis Nomenclature. Results: Among the 97 children with BD followed, 33 (34%) had uveitis (22 males, 66.7%). The median age at onset of BD and uveitis were, respectively, 11.5 years (2.5–17.1) and 11 years (3–17.3). Uveitis preceded BD diagnosis in 18 children (54.5%). Seventeen children had HLA B51 positivity (51.5%). Uveitis was bilateral in 25 (75.8%), and panuveitis in 16 (48.5%). All the patients received at least 1 systemic treatment for uveitis: 25 adalimumab, 2 tocilizumab, 1 abatacept, 3 infliximab, 4 azathioprine, 1 methotrexate and 1 corticosteroid. The remission was achieved with 30/35 treatments (85.7%) after a median time of 8 months (6–24). Six children had a relapse in therapy after the achievement of remission (20%). Fourteen patients stopped the therapy for persistent remission, but 5 relapsed (35.7%) after a median time of 9 months (range 1–48). Conclusion: Uveitis in BD is a sight-threatening condition, and it is more frequently a panuveitis. Biologic treatments seem to be often required to control ocular inflammation.
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spelling doaj-art-58b96686b6374b7da11b82aa720ca6f22025-08-20T02:31:30ZengSAGE PublishingTherapeutic Advances in Musculoskeletal Disease1759-72182024-12-011610.1177/1759720X241275822Uveitis in paediatric Behçet disease: a large multicentric Italian cohortIlaria MaccoraSara Immacolata OrsiniRomina GallizziDavide MontinMarco CattaliniFrancesco La TorreAlessandra SpagnoloFederico DiomedaGabriele SimoniniBackground: Behçet disease (BD) is a rare disease in childhood and its uveitis may lead to blindness if not properly treated. Objectives: We aim to describe a cohort of paediatric BD patients with uveitis. Design: This is a multicentric retrospective study. Methods: Six paediatric rheumatology units in Italy were involved including children with a diagnosis of paediatric BD according to the International Criteria for BD Criteria and/or to the International Study Group Criteria for BD, or Paediatric BD classification criteria if they had uveitis. Demographic, laboratory and clinical data were collected and followed up to March 2023. Ocular characteristics and treatment response were assessed according to Standardization Uveitis Nomenclature. Results: Among the 97 children with BD followed, 33 (34%) had uveitis (22 males, 66.7%). The median age at onset of BD and uveitis were, respectively, 11.5 years (2.5–17.1) and 11 years (3–17.3). Uveitis preceded BD diagnosis in 18 children (54.5%). Seventeen children had HLA B51 positivity (51.5%). Uveitis was bilateral in 25 (75.8%), and panuveitis in 16 (48.5%). All the patients received at least 1 systemic treatment for uveitis: 25 adalimumab, 2 tocilizumab, 1 abatacept, 3 infliximab, 4 azathioprine, 1 methotrexate and 1 corticosteroid. The remission was achieved with 30/35 treatments (85.7%) after a median time of 8 months (6–24). Six children had a relapse in therapy after the achievement of remission (20%). Fourteen patients stopped the therapy for persistent remission, but 5 relapsed (35.7%) after a median time of 9 months (range 1–48). Conclusion: Uveitis in BD is a sight-threatening condition, and it is more frequently a panuveitis. Biologic treatments seem to be often required to control ocular inflammation.https://doi.org/10.1177/1759720X241275822
spellingShingle Ilaria Maccora
Sara Immacolata Orsini
Romina Gallizzi
Davide Montin
Marco Cattalini
Francesco La Torre
Alessandra Spagnolo
Federico Diomeda
Gabriele Simonini
Uveitis in paediatric Behçet disease: a large multicentric Italian cohort
Therapeutic Advances in Musculoskeletal Disease
title Uveitis in paediatric Behçet disease: a large multicentric Italian cohort
title_full Uveitis in paediatric Behçet disease: a large multicentric Italian cohort
title_fullStr Uveitis in paediatric Behçet disease: a large multicentric Italian cohort
title_full_unstemmed Uveitis in paediatric Behçet disease: a large multicentric Italian cohort
title_short Uveitis in paediatric Behçet disease: a large multicentric Italian cohort
title_sort uveitis in paediatric behcet disease a large multicentric italian cohort
url https://doi.org/10.1177/1759720X241275822
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