Cellular and molecular mechanisms breaking immune tolerance in inborn errors of immunity

G Sogkas, F Atschekzei, IR Adriawan… - Cellular & Molecular …, 2021 - nature.com
In addition to susceptibility to infections, conventional primary immunodeficiency disorders
(PIDs) and inborn errors of immunity (IEI) can cause immune dysregulation, manifesting as …

Interplay between epigenetic and genetic alterations in inborn errors of immunity

J Rodríguez-Ubreva, CL Calvillo, LRF Satter… - Trends in …, 2023 - cell.com
Inborn errors of immunity (IEIs) comprise a variety of immune conditions leading to
infections, autoimmunity, allergy, and cancer. Some IEIs have no identified mutation (s) …

Biochemically deleterious human NFKB1 variants underlie an autosomal dominant form of common variable immunodeficiency

J Li, WT Lei, P Zhang, F Rapaport… - Journal of Experimental …, 2021 - rupress.org
Autosomal dominant (AD) NFKB1 deficiency is thought to be the most common genetic
etiology of common variable immunodeficiency (CVID). However, the causal link between …

Primary immune regulatory disorders with an autoimmune lymphoproliferative syndrome-like phenotype: immunologic evaluation, early diagnosis and management

M López-Nevado, LI González-Granado… - Frontiers in …, 2021 - frontiersin.org
Primary immune regulatory disorders (PIRD) are associated with autoimmunity,
autoinflammation and/or dysregulation of lymphocyte homeostasis. Autoimmune …

Establishing the molecular diagnoses in a cohort of 291 patients with predominantly antibody deficiency by targeted next-generation sequencing: experience from a …

J Rojas-Restrepo, A Caballero-Oteyza… - Frontiers in …, 2021 - frontiersin.org
Predominantly antibody deficiencies (PAD) are a heterogeneous group of disorders
characterized by dysfunctional antibody production, low immunoglobulin levels in serum …

Common variable immunodeficiency-associated cancers: the role of clinical phenotypes, immunological and genetic factors

L Bruns, V Panagiota, S von Hardenberg… - Frontiers in …, 2022 - frontiersin.org
Objective The aim of this study was to investigate the prevalence of cancer and associating
clinical, immunological, and genetic factors in a German cohort of patients with common …

[HTML][HTML] Long-term follow up of families with pathogenic NFKB1 variants reveals incomplete penetrance and frequent inflammatory sequelae

EA Tuovinen, O Kuismin, L Soikkonen, T Martelius… - Clinical …, 2023 - Elsevier
Nuclear factor κ light-chain enhancer of activated B cells (NF-κB) family of evolutionarily
conserved transcription factors are involved in key cellular signaling pathways. Previously …

A Pathogenic Missense Variant in NFKB1 Causes Common Variable Immunodeficiency Due to Detrimental Protein Damage

M Fliegauf, R Krüger, S Steiner, LG Hanitsch… - Frontiers in …, 2021 - frontiersin.org
In common variable immunodeficiency (CVID), heterozygous damaging NFKB1 variants
represent the most frequent monogenic cause. NFKB1 encodes the precursor p105, which …

How do nuclear factor kappa B (NF-κB) 1 and NF-κB2 defects lead to the incidence of clinical and immunological manifestations of inborn errors of immunity?

N Fathi, H Mojtahedi, M Nasiri… - Expert Review of …, 2023 - Taylor & Francis
Introduction Genetic defects affect the manner of the immune system's development,
activation, and function. Nuclear factor-kappa B subunit 1 (NF-κB1) and NF-κB2 are involved …

Detrimental NFKB1 missense variants affecting the Rel-homology domain of p105/p50

M Fliegauf, M Kinnunen, S Posadas-Cantera… - Frontiers in …, 2022 - frontiersin.org
Most of the currently known heterozygous pathogenic NFKB1 (Nuclear factor kappa B
subunit 1) variants comprise deleterious defects such as severe truncations, internal …