Inborn errors of immunity affecting the MHC class I pathway for antigen presentation

Inborn errors of immunity affecting the major histocompatibility complex (MHC) class I pathway for antigen presentation represent a rare group of human disease syndromes. Here, we review symptoms associated with such conditions, which manifest as chronic respiratory infections, granulomatous skin lesions, and related severe symptoms. We highlight the potential for misdiagnosis with autoimmune conditions such as granulomatosis with polyangiitis and emphasize the necessity of infection-focused treatment, given the risks associated with immunosuppressive therapy. Furthermore, we present novel long-term follow-up data on TAP-deficient patients, revealing new insights into disease progression, including an increased risk of skin cancer and severe herpesvirus infections. Additionally, we discuss cases of a few individuals with significant MHC class I deficiency who remain largely asymptomatic, underscoring the variability in clinical presentation. Our findings emphasize the importance of further genetic research and immunopathological analysis to identify predictive markers and optimize individualized treatment approaches. Long-term patient surveillance remains critical to understanding late-onset complications and refining clinical management strategies.

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Publication Details

Journal
Journal of Human Immunity
Published
2025-06-11
DOI
https://doi.org/10.70962/jhi.20250029
Citations
2
Primary Topic
Immune Cell Function and Interaction
Type
article
Field-Weighted Citation Impact
1.59
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article

Inborn errors of immunity affecting the MHC class I pathway for antigen presentation

Ömür Ardeniz, Jacques Zimmer, Angélica Cuapio, Hans‐Gustaf Ljunggren et al.
2 citations
Journal of Human Immunity
Immune Cell Function and Interaction
1.59
article

Inborn errors of immunity affecting the MHC class I pathway for antigen presentation

Ömür Ardeniz, Jacques Zimmer, Angélica Cuapio, Hans‐Gustaf Ljunggren, Stephan D. Gadola
article en
2 citations

Abstract

Inborn errors of immunity affecting the major histocompatibility complex (MHC) class I pathway for antigen presentation represent a rare group of human disease syndromes. Here, we review symptoms associated with such conditions, which manifest as chronic respiratory infections, granulomatous skin lesions, and related severe symptoms. We highlight the potential for misdiagnosis with autoimmune conditions such as granulomatosis with polyangiitis and emphasize the necessity of infection-focused treatment, given the risks associated with immunosuppressive therapy. Furthermore, we present novel long-term follow-up data on TAP-deficient patients, revealing new insights into disease progression, including an increased risk of skin cancer and severe herpesvirus infections. Additionally, we discuss cases of a few individuals with significant MHC class I deficiency who remain largely asymptomatic, underscoring the variability in clinical presentation. Our findings emphasize the importance of further genetic research and immunopathological analysis to identify predictive markers and optimize individualized treatment approaches. Long-term patient surveillance remains critical to understanding late-onset complications and refining clinical management strategies.

Journal of Human ImmunityVol. 1(2)
Twitter (United States) (US), Bethesda Hospital (IN), Karolinska Institutet (SE), Ege University (TR), Luxembourg Institute of Health (LU), Integrated Cardio Metabolic Centre (SE)
Openalex Percentile: Top 16%
Immune Cell Function and Interaction
1.59
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