Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab

The classic entity of autosomal dominant Blau syndrome (BS) consists of arthritis, dermatitis, and uveitis, occurring as a result of mutations in the NOD2 gene pattern recognition receptor. Sporadic cases are those in which no known gene mutation is identifiable. Uveitis in BS can be refractory to c...

Descripción completa

Detalles Bibliográficos
Autores principales: Naik, Anmol U, Annamalai, Radha, Biswas, Jyotirmay
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Medknow Publications & Media Pvt Ltd 2018
Materias:
Case Reports
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6173012/
https://www.ncbi.nlm.nih.gov/pubmed/30249847
http://dx.doi.org/10.4103/ijo.IJO_629_18
_version_ 1783361055458590720
author Naik, Anmol U
Annamalai, Radha
Biswas, Jyotirmay
author_facet Naik, Anmol U
Annamalai, Radha
Biswas, Jyotirmay
author_sort Naik, Anmol U
collection PubMed
description The classic entity of autosomal dominant Blau syndrome (BS) consists of arthritis, dermatitis, and uveitis, occurring as a result of mutations in the NOD2 gene pattern recognition receptor. Sporadic cases are those in which no known gene mutation is identifiable. Uveitis in BS can be refractory to conventional therapy. We report a case of sporadic Blau uveitis managed with adalimumab monotherapy after failing to respond to topical steroids, systemic steroids, methotrexate, and infliximab therapy sequentially. Uveitis resolved completely with adalimumab and the patient has had a disease-free period over a 2-year follow-up with bi-monthly injections for arthritis control.
format Online
Article
Text
id pubmed-6173012
institution National Center for Biotechnology Information
language English
publishDate 2018
publisher Medknow Publications & Media Pvt Ltd
record_format MEDLINE/PubMed
spelling pubmed-61730122018-10-10 Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab Naik, Anmol U Annamalai, Radha Biswas, Jyotirmay Indian J Ophthalmol Case Reports The classic entity of autosomal dominant Blau syndrome (BS) consists of arthritis, dermatitis, and uveitis, occurring as a result of mutations in the NOD2 gene pattern recognition receptor. Sporadic cases are those in which no known gene mutation is identifiable. Uveitis in BS can be refractory to conventional therapy. We report a case of sporadic Blau uveitis managed with adalimumab monotherapy after failing to respond to topical steroids, systemic steroids, methotrexate, and infliximab therapy sequentially. Uveitis resolved completely with adalimumab and the patient has had a disease-free period over a 2-year follow-up with bi-monthly injections for arthritis control. Medknow Publications & Media Pvt Ltd 2018-10 /pmc/articles/PMC6173012/ /pubmed/30249847 http://dx.doi.org/10.4103/ijo.IJO_629_18 Text en Copyright: © 2018 Indian Journal of Ophthalmology http://creativecommons.org/licenses/by-nc-sa/4.0 This is an open access journal, and articles are distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as appropriate credit is given and the new creations are licensed under the identical terms.
spellingShingle Case Reports
Naik, Anmol U
Annamalai, Radha
Biswas, Jyotirmay
Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab
title Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab
title_full Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab
title_fullStr Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab
title_full_unstemmed Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab
title_short Uveitis in sporadic Blau syndrome: Long-term follow-up of a refractory case treated successfully with adalimumab
title_sort uveitis in sporadic blau syndrome: long-term follow-up of a refractory case treated successfully with adalimumab
topic Case Reports
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6173012/
https://www.ncbi.nlm.nih.gov/pubmed/30249847
http://dx.doi.org/10.4103/ijo.IJO_629_18
work_keys_str_mv AT naikanmolu uveitisinsporadicblausyndromelongtermfollowupofarefractorycasetreatedsuccessfullywithadalimumab
AT annamalairadha uveitisinsporadicblausyndromelongtermfollowupofarefractorycasetreatedsuccessfullywithadalimumab
AT biswasjyotirmay uveitisinsporadicblausyndromelongtermfollowupofarefractorycasetreatedsuccessfullywithadalimumab

Ejemplares similares