TY - JOUR
T1 - Identification and functional characterisation of a novel DNASE1L3 variant (c.572A>G, p.Asn191Ser) in three Emirati families with systemic lupus erythematosus and hypocomplementaemic urticarial vasculitis
AU - Aljaberi, Najla
AU - Bharathan, Anjali
AU - Gopal, Remya Prajesh
AU - Mohammed, Ekhlass
AU - Shibli, Fatema Al
AU - Tabouni, Mohammed
AU - Alhmoudi, Sara
AU - Kizhakkedath, Praseetha
AU - Baydoun, Ibrahim
AU - Allam, Mushal
AU - Mustafa, Noor
AU - Aljasmi, Fatma
AU - Dhaheri, Afra Al
AU - Alblooshi, Hiba
N1 - Publisher Copyright:
© Author(s) (or their employer(s)) 2025.
PY - 2025/2/13
Y1 - 2025/2/13
N2 - Objectives To evaluate the functional impact of a novel DNASE1L3 variant (c.572A>G, p.Asn191Ser) in three families with SLE and hypocomplementaemic urticarial vasculitis (HUV) from the United Arab Emirates. Methods Whole-exome sequencing was performed on affected patients and findings were confirmed using Sanger sequencing in family members. DNASE1L3 protein expression, secretion and enzymatic activity were assessed in HEK293 cell lines. Plasma smear assay for neutrophil extracellular traps (NETs) was evaluated in patients, family members and healthy control. Results A total of seven patients diagnosed with both SLE and HUV were identified from three unrelated families. All affected individuals were found to carry a homozygous c.572A>G, p.Asn191Ser (191S) variant in DNASE1L3. The variant 191S was shown to impact the secretion and activity of DNASE1L3. Patients homozygous for 191S variant had significantly higher burden (p=0.0409) of NET structure in comparison to heterozygous and healthy control. Conclusions We functionally evaluated the effect of a novel DNASE1L3 (c.572A>G, p.Asn191Ser) in familial SLE with a consistent pattern of HUV across seven patients. This variant resulted in impaired secretion and enzymatic activity of DNASE1L3 along with increased NETosis in patients with homozygous genotype.
AB - Objectives To evaluate the functional impact of a novel DNASE1L3 variant (c.572A>G, p.Asn191Ser) in three families with SLE and hypocomplementaemic urticarial vasculitis (HUV) from the United Arab Emirates. Methods Whole-exome sequencing was performed on affected patients and findings were confirmed using Sanger sequencing in family members. DNASE1L3 protein expression, secretion and enzymatic activity were assessed in HEK293 cell lines. Plasma smear assay for neutrophil extracellular traps (NETs) was evaluated in patients, family members and healthy control. Results A total of seven patients diagnosed with both SLE and HUV were identified from three unrelated families. All affected individuals were found to carry a homozygous c.572A>G, p.Asn191Ser (191S) variant in DNASE1L3. The variant 191S was shown to impact the secretion and activity of DNASE1L3. Patients homozygous for 191S variant had significantly higher burden (p=0.0409) of NET structure in comparison to heterozygous and healthy control. Conclusions We functionally evaluated the effect of a novel DNASE1L3 (c.572A>G, p.Asn191Ser) in familial SLE with a consistent pattern of HUV across seven patients. This variant resulted in impaired secretion and enzymatic activity of DNASE1L3 along with increased NETosis in patients with homozygous genotype.
KW - Genetic
KW - Polymorphism
KW - Systemic Lupus Erythematosus
KW - Vasculitis
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U2 - 10.1136/lupus-2024-001477
DO - 10.1136/lupus-2024-001477
M3 - Article
AN - SCOPUS:85218155494
SN - 2053-8790
VL - 12
JO - Lupus Science and Medicine
JF - Lupus Science and Medicine
IS - 1
M1 - e001477
ER -