Juvenile-Onset Immunodeficiency Secondary to Anti-Interferon-Gamma Autoantibodies

Liew, Woei-Kang; Thoon, Koh-Cheng; Chong, Chia-Yin; Tan, Natalie Wh; Cheng, Duo-Tong; Chan, Bianca S W; Ng, Michelle S Y; Das, Lena; Arkachaisri, Thaschawee; Huang, Chiung‐Hui; Kuan, Jyn-Ling; Chai, Louis Yi Ann; Koh, Mark Jean Aan

doi:10.1007/s10875-019-00652-1

Cited by 18 publications

(9 citation statements)

References 19 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Additionally, no intrinsic defect in IFNg production or IFNg response was detected. Anti-cytokine autoantibodies are known to be a cause of adult-onset infection susceptibility (also called "phenocopies" of IEI) (1,(11)(12)(13), but the presence of anti-IFNg or anti-GM-CSF has been excluded.…”

Section: Discussionmentioning

confidence: 99%

“…The second situation concerns cases associated with acquired immunodeficiency to IFNg pathways that are described as a phenocopy of IEI. They lead to opportunistic infections (fungi, parasites, and bacteria), tuberculosis, and infection due to nontuberculosis mycobacteria (1)(2)(3)(4). Finally, reactivations are described in immunocompromised patients with human immunodeficiency virus (HIV) (5,6), and cases of secondary disseminated BCG disease (BCGosis) are also reported under BCG therapy for bladder malignancy (7,8).…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy

et al. 2021

View full text Add to dashboard Cite

ContextDisseminated infections due to Mycobacterium bovis Bacillus Calmette-Guérin (BCG) are unusual and occur mostly in patients with inborn error of immunity (IEI) or acquired immunodeficiency. However, cases of secondary BCGosis due to intravesical BCG instillation have been described. Herein, we present a case of severe BCGosis occurring in an unusual situation.Case DescriptionWe report one case of severe disseminated BCG disease occurring after hematological malignancy in a 48-year-old man without BCG instillation and previously vaccinated in infancy with no complication. Laboratory investigations demonstrated that he was not affected by any known or candidate gene of IEI or intrinsic cellular defect involving IFNγ pathway. Whole genome sequencing of the BCG strain showed that it was most closely related to the M. bovis BCG Tice strain, suggesting an unexpected relationship between the secondary immunodeficiency of the patient and the acquired BCG infection.ConclusionThis case highlights the fact that, in addition to the IEI, physicians, as well as microbiologists and pharmacists should be aware of possible acquired disseminated BCG disease in secondary immunocompromised patients treated in centers that administrate BCG for bladder cancers.

show abstract

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy

et al. 2021

View full text Add to dashboard Cite

show abstract

“…Nevertheless, adolescents and young adults with disseminated opportunistic pathogenic infections with anti-IFN-γ AAbs have been reported recently. 30,31 The majority of patients reported to have anti-IFN-γ AAbs have been narrowed to Asia-born Asians, particularly those from Southeast Asian populations with human leukocyte antigen (HLA)−DRB1 and DQB1 alleles, especially HLA−DRB1*15:01/16:02 and DQB1*05:01/05:02. 32–34 However, opportunistic pathogenic infections have been reported in Caucasian patients with anti-IFN-γ AAbs living in the UK, USA, Germany as well as in patients of African ancestry.…”

Section: Anti-ifn-γ Aabs and Pathologymentioning

confidence: 99%

Minireview: Insights into anti-interferon-γ autoantibodies

Chawansuntati

Rattanathammethee

Wipasa

2021

Exp Biol Med (Maywood)

View full text Add to dashboard Cite

The association between the presence of anti-interferon-γ autoantibodies and the onset of immunodeficiency with intracellular infections has been clearly established. No standard regimen to control the production of these pathogenic autoantibodies, apart from antimicrobial therapy to eliminate infections, contributes to the medical burden of this syndrome, which sometimes has a fatal outcome. In this review, we summarize the findings on anti-interferon-γ autoantibodies to facilitate further research and to provide guidance for treatment strategies.

show abstract

“…Almost all patients are adults in the range from 40 to 70 years with no sex predominance. However, more recently, two young patients presenting at 10 years and 16 years, respectively, have been reported (Liew et al 2019). In addition to NTM infections, some nAIGAs patients also were developing Tuberculosis, which may precede, be concomitant with, or follow the diagnosis of NTM infections (Browne et al 2012;Chi et al 2016;Kampitak et al 2011).…”

Section: Anti-interferon-γ Autoantibodies As An Etiology In Mycobacterial Infections In Adultsmentioning

confidence: 99%

Autoantibodies against cytokines: phenocopies of primary immunodeficiencies?

Chi

Bernuth

et al. 2020

Hum Genet

View full text Add to dashboard Cite

Anti-cytokine autoantibodies may cause immunodeficiency and have been recently recognized as ‘autoimmune phenocopies of primary immunodeficiencies’ and are found in particular, but not exclusively in adult patients. By blocking the cytokine’s biological function, patients with anti-cytokine autoantibodies may present with a similar clinical phenotype as the related inborn genetic disorders. So far, autoantibodies to interferon (IFN)-γ, GM-CSF, to a group of TH-17 cytokines and to IL-6 have been found to be causative or closely associated with susceptibility to infection. This review compares infectious diseases associated with anti-cytokine autoantibodies with primary immunodeficiencies affecting similar cytokines or related pathways.

show abstract

Juvenile-Onset Immunodeficiency Secondary to Anti-Interferon-Gamma Autoantibodies

Cited by 18 publications

References 19 publications

Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy

Case Report: Acquired Disseminated BCG in the Context of a Delayed Immune Reconstitution After Hematological Malignancy

Minireview: Insights into anti-interferon-γ autoantibodies

Autoantibodies against cytokines: phenocopies of primary immunodeficiencies?

Contact Info

Product

Resources

About