Table 2.

IEI with predominant rheumatologic manifestations

ClassificationClinical syndromeRheumatologic/autoimmune manifestationsExamples of genes identified
Disorders of immune dysregulation APECED Chronic mucocutaneous candidiasis, hypoparathyroidism, primary adrenal insufficiency, diabetes mellitus, and JIA AIRE 
ALPS Lymphoproliferation, lymphadenopathy, splenomegaly, and autoimmune cytopenias FAS and FADD 
Tregopathies IPEX (T1D, autoimmune enteropathy, eczema, dermatitis, alopecia, and psoriasis-like lesions) FOXP3, CD25, STAT5B, LRBA, CTLA4, and DEF6 
Multi-organ lymphocytic infiltration 
T1D, hypoparathyroidism, and JIA 
Disorders of cytokine signaling T1D, cytopenias, SLE, lymphadenopathy, and multi-organ autoimmunity STAT1, STAT3, STAT4, STAT6, JAK1, SOCS1, PTPN2, ISG15, and USP18 
HLH Fever, cytopenias, elevated ferritin, hypertriglyceridemia, and encephalitis PRF1, STX11, STXBP2, and UNC13D 
Innate immune defects Monogenic lupus SLE Complement genes, DNASE1, DNASE1L3, TLR7, and UNC93B1 
Disorders of complement regulators Vasculopathies and aHUS CFH (factor H), CFI (factor I), CD45 (membrane cofactor protein), and autoantibodies targeting factor H 
Autoinflammatory disorders Interferonopathies Severe neurologic findings AGS (TREX1, RNAASEH2A, RNASEH2B, RNASEH2C, TREX1, ADAR, and IFIH1), COPA, STING1, and ADA2 
Skin manifestations (chilblain-like lesions) 
Interstitial lung disease 
Inflammasomopathies Periodic fevers, peritonitis, synovitis, and pleuritis MEFV (FMF), NLRP3, PLCG2, and NLRC4 
Non-inflammasomopathy inflammatory disorders Oral and genital ulcers, arthritis, erythema nodosum, recurrent fevers, and very early onset IBD TNFRSF1A (TRAPS) TNFAIP3 (A20), RELA, and IKBKG 
VEXAS syndrome Alveolitis, chondritis, thromboembolisms, dermatoses, and cutaneous vasculitis UBA1 (somatic) 
ClassificationClinical syndromeRheumatologic/autoimmune manifestationsExamples of genes identified
Disorders of immune dysregulation APECED Chronic mucocutaneous candidiasis, hypoparathyroidism, primary adrenal insufficiency, diabetes mellitus, and JIA AIRE 
ALPS Lymphoproliferation, lymphadenopathy, splenomegaly, and autoimmune cytopenias FAS and FADD 
Tregopathies IPEX (T1D, autoimmune enteropathy, eczema, dermatitis, alopecia, and psoriasis-like lesions) FOXP3, CD25, STAT5B, LRBA, CTLA4, and DEF6 
Multi-organ lymphocytic infiltration 
T1D, hypoparathyroidism, and JIA 
Disorders of cytokine signaling T1D, cytopenias, SLE, lymphadenopathy, and multi-organ autoimmunity STAT1, STAT3, STAT4, STAT6, JAK1, SOCS1, PTPN2, ISG15, and USP18 
HLH Fever, cytopenias, elevated ferritin, hypertriglyceridemia, and encephalitis PRF1, STX11, STXBP2, and UNC13D 
Innate immune defects Monogenic lupus SLE Complement genes, DNASE1, DNASE1L3, TLR7, and UNC93B1 
Disorders of complement regulators Vasculopathies and aHUS CFH (factor H), CFI (factor I), CD45 (membrane cofactor protein), and autoantibodies targeting factor H 
Autoinflammatory disorders Interferonopathies Severe neurologic findings AGS (TREX1, RNAASEH2A, RNASEH2B, RNASEH2C, TREX1, ADAR, and IFIH1), COPA, STING1, and ADA2 
Skin manifestations (chilblain-like lesions) 
Interstitial lung disease 
Inflammasomopathies Periodic fevers, peritonitis, synovitis, and pleuritis MEFV (FMF), NLRP3, PLCG2, and NLRC4 
Non-inflammasomopathy inflammatory disorders Oral and genital ulcers, arthritis, erythema nodosum, recurrent fevers, and very early onset IBD TNFRSF1A (TRAPS) TNFAIP3 (A20), RELA, and IKBKG 
VEXAS syndrome Alveolitis, chondritis, thromboembolisms, dermatoses, and cutaneous vasculitis UBA1 (somatic) 

A summary of the major IEI that present with rheumatologic, autoimmune, and autoinflammatory conditions as their primary manifestations. FMF, familial Mediterranean fever; TRAPS, tumor necrosis factor receptor-associated periodic syndrome.

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