P02-031 - Phenotype of V198M and Q703K NLRP3 variants

نویسندگان

  • V Messia
  • M Pardeo
  • R Nicolai
  • C Bracaglia
  • F De Benedetti
  • A Insalaco
چکیده

Introduction The term CAPS (cryopyrin-associated periodic syndromes) identifies a spectrum of autoinflammatory diseases caused by heterozygous mutations of the CIAS1/ NLRP3. Affected individuals may present three different phenotypes: familial cold autoinflammatory syndrome (FCAS), Muckle-Wells syndrome (MWS) and CINCA syndrome, the most severe form of the clinical spectrum. Clinical manifestations include urticaria-like rash, recurrent fever, arthralgia, conjunctivitis; chronic aseptic meningitis, cerebral atrophy and bone malformations in the severe cases.

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PW02-040 - Low-penetrance NLRP3 variants

Methods This multi-center observational study included 44 patients (25 children and 19 adults). All patients were symptomatic with some symptoms suggesting possible CAPS at the time of baseline examination. Genetic analysis detected one of the following NLRP3 variants: Q703K (n=18), R488K (n=6), and V198M (n=20). Clinical phenotypes were described and laboratory markers were analyzed. In order ...

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P02-024 - Clinical impact of V198M mutation in NLRP3 gene

Results The V198M mutation was found in 13 screened patients: 10 were heterozygous for the mutation only. In one patient with a typical MWS phenotype the V198M variant was associated with the Q703K and the D303N mutation of the same gene. In a patient a low-penetrance mutation of TNFRSF1A gene (P46L) was also found, while another one carried the A91V mutation of Pfr1 gene. Out of the 10 patient...

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Expanding spectrum of neurologic manifestations in patients with NLRP3 low-penetrance mutations

OBJECTIVE To evaluate the frequency of the cryoporin/NLRP3 low-penetrance mutations V198M and Q703K in patients who reported at least 2 symptoms compatible with cryopyrin-associated periodic syndromes (CAPS) and to characterize the phenotype in mutation-positive patients. METHODS The frequency of the V198M and Q703K mutations was investigated in a selected cohort of 108 patients from our neur...

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A functional inflammasome activation assaydifferentiates patients with pathogenic NLRP3mutations and symptomatic patients with lowpenetrance variants

Cryopyrin-associated periodic syndromes (CAPS) are characterized by recurrent episodes of systemic inflammation caused by mutations in the NLRP3 gene. Besides confirmed pathogenic NLRP3 mutations, patients with CAPS-like symptoms frequently show low penetrance variants in NLRP3. The disease relevance of these variants is inconsistent. In this study, we investigated if an inflammasome activation...

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PReS-FINAL-2200: Phenotype of V198M and Q703K NLRP3 variants

Introduction The term CAPS (Cryopyrin-Associated Periodic Syndromes) identifies a spectrum of autoinflammatory diseases caused by heterozygous mutations of the CIAS1/NLRP3. Affected individuals may present three different phenotypes: Familial Cold Autoinflammatory Syndrome (FCAS), Muckle-Wells Syndrome (MWS) and CINCA syndrome, the most severe form of the clinical spectrum. Clinical manifestati...

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عنوان ژورنال:

دوره 11  شماره 

صفحات  -

تاریخ انتشار 2013