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Pediatr Dermatol ; 41(4): 697-700, 2024.
Artículo en Inglés | MEDLINE | ID: mdl-38369300

RESUMEN

STING-associated vasculopathy with onset in infancy (SAVI) is a rare, monogenic interferonopathy caused by gain-of-function variants in STING1 (TMEM173) characterized by systemic inflammation, cutaneous vasculopathy, and interstitial lung disease. We report a case of SAVI attributed to a novel STING1 p.R284T variant who demonstrated characteristic cutaneous features including telangiectasias, livedo and acrocyanotic changes on face and extremities, as well as saddle nose deformity, failure to thrive, inflammatory arthritis and notable lack of pulmonary disease or autoantibody positivity. Due to the risk for progressive and irreversible lung and tissue damage and evolving therapeutic landscape involving the use of Janus kinase inhibitors, it is critical to recognize variable clinical phenotypes to diagnose and consider treatment options for SAVI patients early in their disease course.


Asunto(s)
Proteínas de la Membrana , Humanos , Proteínas de la Membrana/genética , Enfermedades Cutáneas Vasculares/genética , Enfermedades Cutáneas Vasculares/diagnóstico , Enfermedades Cutáneas Vasculares/patología , Lactante , Masculino , Femenino , Mutación con Ganancia de Función
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