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Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report

Thyroid eye disease (TED) characterizes by inflammation and remodeling of orbital tissues. Although the majority of pediatric TED is mild, some children present progressive and severe disease. The approach to severe disease in this group of patients, especially when noncortico-responsive, is challen...

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Autores principales: Moleiro, Ana Filipa, Vilares-Morgado, Rodrigo, Alves, Gonçalo Coutinho, Rodrigues, Mariana, Falcão-Reis, Fernando, Magalhães, Augusto, Leal, Vitor, Meira, Jorge
Formato: Online Artículo Texto
Lenguaje:English
Publicado: S. Karger AG 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9710467/
https://www.ncbi.nlm.nih.gov/pubmed/36466065
http://dx.doi.org/10.1159/000526546
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author Moleiro, Ana Filipa
Vilares-Morgado, Rodrigo
Alves, Gonçalo Coutinho
Rodrigues, Mariana
Falcão-Reis, Fernando
Magalhães, Augusto
Leal, Vitor
Meira, Jorge
author_facet Moleiro, Ana Filipa
Vilares-Morgado, Rodrigo
Alves, Gonçalo Coutinho
Rodrigues, Mariana
Falcão-Reis, Fernando
Magalhães, Augusto
Leal, Vitor
Meira, Jorge
author_sort Moleiro, Ana Filipa
collection PubMed
description Thyroid eye disease (TED) characterizes by inflammation and remodeling of orbital tissues. Although the majority of pediatric TED is mild, some children present progressive and severe disease. The approach to severe disease in this group of patients, especially when noncortico-responsive, is challenging. The purpose of this work was to describe the effective use of tocilizumab as second-line therapy in steroid-resistant pediatric TED. A 13-year-old female with a history of Graves' disease presented with right eye proptosis for at least 8 months associated with mild pain on eye movements and ocular surface complaints. The ophthalmologic evaluation revealed Hertel exophthalmometry readings of 22 mm on the right eye (OD) and 19 mm on the left (OS). The remaining ophthalmic examination was unremarkable. Intravenous methylprednisolone pulses of 500 mg were initiated without any improvement after 4 treatments. Following multidisciplinary team discussion, therapy was switched to monthly tocilizumab injections at 4 mg/kg. Significant reduction of proptosis and resolution of pain and ocular surface complaints were noted immediately after the treatment switch. Exophthalmometry readings after the end of treatment that included 4 tocilizumab injections were 20 mm on OD and 19 mm on OS. No side effects were reported during the entire follow-up. Six months after treatment cessation, the patient remains stable, without any signs of orbitopathy relapse and no ophthalmologic complaints. This is the second case report showing the effectiveness of tocilizumab in pediatric TED and the first one showing its efficacy when steroids fail. Our results support the potential safety and efficacy of this immunosuppressor in children with TED.
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spelling pubmed-97104672022-12-01 Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report Moleiro, Ana Filipa Vilares-Morgado, Rodrigo Alves, Gonçalo Coutinho Rodrigues, Mariana Falcão-Reis, Fernando Magalhães, Augusto Leal, Vitor Meira, Jorge Case Rep Ophthalmol Case Report Thyroid eye disease (TED) characterizes by inflammation and remodeling of orbital tissues. Although the majority of pediatric TED is mild, some children present progressive and severe disease. The approach to severe disease in this group of patients, especially when noncortico-responsive, is challenging. The purpose of this work was to describe the effective use of tocilizumab as second-line therapy in steroid-resistant pediatric TED. A 13-year-old female with a history of Graves' disease presented with right eye proptosis for at least 8 months associated with mild pain on eye movements and ocular surface complaints. The ophthalmologic evaluation revealed Hertel exophthalmometry readings of 22 mm on the right eye (OD) and 19 mm on the left (OS). The remaining ophthalmic examination was unremarkable. Intravenous methylprednisolone pulses of 500 mg were initiated without any improvement after 4 treatments. Following multidisciplinary team discussion, therapy was switched to monthly tocilizumab injections at 4 mg/kg. Significant reduction of proptosis and resolution of pain and ocular surface complaints were noted immediately after the treatment switch. Exophthalmometry readings after the end of treatment that included 4 tocilizumab injections were 20 mm on OD and 19 mm on OS. No side effects were reported during the entire follow-up. Six months after treatment cessation, the patient remains stable, without any signs of orbitopathy relapse and no ophthalmologic complaints. This is the second case report showing the effectiveness of tocilizumab in pediatric TED and the first one showing its efficacy when steroids fail. Our results support the potential safety and efficacy of this immunosuppressor in children with TED. S. Karger AG 2022-11-17 /pmc/articles/PMC9710467/ /pubmed/36466065 http://dx.doi.org/10.1159/000526546 Text en Copyright © 2022 by The Author(s). Published by S. Karger AG, Basel https://creativecommons.org/licenses/by-nc/4.0/This article is licensed under the Creative Commons Attribution-NonCommercial-4.0 International License (CC BY-NC) (http://www.karger.com/Services/OpenAccessLicense). Usage and distribution for commercial purposes requires written permission.
spellingShingle Case Report
Moleiro, Ana Filipa
Vilares-Morgado, Rodrigo
Alves, Gonçalo Coutinho
Rodrigues, Mariana
Falcão-Reis, Fernando
Magalhães, Augusto
Leal, Vitor
Meira, Jorge
Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report
title Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report
title_full Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report
title_fullStr Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report
title_full_unstemmed Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report
title_short Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report
title_sort tocilizumab as a useful tool for thyroid eye disease in pediatric population: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9710467/
https://www.ncbi.nlm.nih.gov/pubmed/36466065
http://dx.doi.org/10.1159/000526546
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