Table 1.

Demographic, molecular and clinical data of patients identified to carry rare non-synonymous missense substitutions in UNC93B1

FamilyDisease segregationEthnicityNucleotide substitutionAmino acid substitutiongnomADCombined VEP damageaCADDPhenotype
AGS1861 Four (two M, two F) across three generations North African c.1574_1575delinsCT (Het) Arg525Pro (R525P) 0.81 CBL 
AGS2568 M (history of disease in untested mother, MA, and MG) White European c.989T>G (Het) Leu330Arg (L330R) 8/1,552,226 0.93 29.8 CBL 
AGS2650 F (parental DNA unavailable) Indian c.951C>G (Hom) Ile317Met (I317M) 0.70 24 SLE 
AGS2672 F (mother negative, paternal DNA unavailable) White European c.1398A>C (Het) Arg466Ser (R466S) 0.86 22.3 CBL 
AGS2898 F (variant inherited from asymptomatic mother) White European c.973G>T (Het) Gly325Cys (G325C) 0.93 25.7 SLE 
FamilyDisease segregationEthnicityNucleotide substitutionAmino acid substitutiongnomADCombined VEP damageaCADDPhenotype
AGS1861 Four (two M, two F) across three generations North African c.1574_1575delinsCT (Het) Arg525Pro (R525P) 0.81 CBL 
AGS2568 M (history of disease in untested mother, MA, and MG) White European c.989T>G (Het) Leu330Arg (L330R) 8/1,552,226 0.93 29.8 CBL 
AGS2650 F (parental DNA unavailable) Indian c.951C>G (Hom) Ile317Met (I317M) 0.70 24 SLE 
AGS2672 F (mother negative, paternal DNA unavailable) White European c.1398A>C (Het) Arg466Ser (R466S) 0.86 22.3 CBL 
AGS2898 F (variant inherited from asymptomatic mother) White European c.973G>T (Het) Gly325Cys (G325C) 0.93 25.7 SLE 

CADD, Combined Annotation Dependent Depletion score; F: female; Het: heterozygous; Hom: homozygous; M: male; MA: maternal aunt; MG: maternal grandfather; Pat: paternal. UNC93B1 (ENST00000227471.7/NM_030930.4; NP_112192.2).

a

Based on top-performing variant effect predictors from Livesey and Marsh (2023).

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