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Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies

BACKGROUND: Nontuberculous mycobacteria (NTM) are found ubiquitously in the environment. Since exposure to NTM is universal, infection likely represents underlying host susceptibility factors. Anti-IFN-ɣ autoantibodies have been described previously in patients with NTM. Up to 88 % of patients with...

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Autores principales: Lee, Tian Loon, Agrawal, Rupesh, Tan, Jackie Yu-Ling, Ong, Kiat Hoe, Wong, Chen Seong, Ho, Su Ling
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Berlin Heidelberg 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5075326/
https://www.ncbi.nlm.nih.gov/pubmed/27771870
http://dx.doi.org/10.1186/s12348-016-0106-z
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author Lee, Tian Loon
Agrawal, Rupesh
Tan, Jackie Yu-Ling
Ong, Kiat Hoe
Wong, Chen Seong
Ho, Su Ling
author_facet Lee, Tian Loon
Agrawal, Rupesh
Tan, Jackie Yu-Ling
Ong, Kiat Hoe
Wong, Chen Seong
Ho, Su Ling
author_sort Lee, Tian Loon
collection PubMed
description BACKGROUND: Nontuberculous mycobacteria (NTM) are found ubiquitously in the environment. Since exposure to NTM is universal, infection likely represents underlying host susceptibility factors. Anti-IFN-ɣ autoantibodies have been described previously in patients with NTM. Up to 88 % of patients with disseminated NTM or other opportunistic infections have high-titer anti-IFN-ɣ autoantibodies, compared with 2 % of patients with TB and healthy controls. FINDINGS: We report a unique presentation of a patient with anti-IFN-ɣ autoantibodies with disseminated NTM infection who presents with panuveitis with multifocal retinitis and vasculitis. Treatment with systemic anti tubercular therapy resulted in complete clinical resolution with good visual recovery. CONCLUSIONS: Patients with anti-IFN-ɣ autoantibodies present with a novel syndrome that links autoimmunity and immunodeficiency. This case emphasizes the importance of testing for anti-IFN-ɣ autoantibodies in patients with disseminated mycobacterial infection.
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spelling pubmed-50753262016-11-04 Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies Lee, Tian Loon Agrawal, Rupesh Tan, Jackie Yu-Ling Ong, Kiat Hoe Wong, Chen Seong Ho, Su Ling J Ophthalmic Inflamm Infect Brief Report BACKGROUND: Nontuberculous mycobacteria (NTM) are found ubiquitously in the environment. Since exposure to NTM is universal, infection likely represents underlying host susceptibility factors. Anti-IFN-ɣ autoantibodies have been described previously in patients with NTM. Up to 88 % of patients with disseminated NTM or other opportunistic infections have high-titer anti-IFN-ɣ autoantibodies, compared with 2 % of patients with TB and healthy controls. FINDINGS: We report a unique presentation of a patient with anti-IFN-ɣ autoantibodies with disseminated NTM infection who presents with panuveitis with multifocal retinitis and vasculitis. Treatment with systemic anti tubercular therapy resulted in complete clinical resolution with good visual recovery. CONCLUSIONS: Patients with anti-IFN-ɣ autoantibodies present with a novel syndrome that links autoimmunity and immunodeficiency. This case emphasizes the importance of testing for anti-IFN-ɣ autoantibodies in patients with disseminated mycobacterial infection. Springer Berlin Heidelberg 2016-10-22 /pmc/articles/PMC5075326/ /pubmed/27771870 http://dx.doi.org/10.1186/s12348-016-0106-z Text en © The Author(s). 2016 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.
spellingShingle Brief Report
Lee, Tian Loon
Agrawal, Rupesh
Tan, Jackie Yu-Ling
Ong, Kiat Hoe
Wong, Chen Seong
Ho, Su Ling
Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies
title Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies
title_full Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies
title_fullStr Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies
title_full_unstemmed Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies
title_short Disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-IFN-ɣ autoantibodies
title_sort disseminated nontuberculous mycobacterial infection with multifocal retinitis and vasculitis in an immunocompromised patient with anti-ifn-ɣ autoantibodies
topic Brief Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5075326/
https://www.ncbi.nlm.nih.gov/pubmed/27771870
http://dx.doi.org/10.1186/s12348-016-0106-z
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