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Neonatal Medicine ; : 126-130, 2018.
Artículo en Inglés | WPRIM | ID: wpr-716553

RESUMEN

Parkes Weber syndrome is a rare congenital vascular anomaly, related to the RAS p21 protein activator 1 (RASA1) gene. It is characterized by capillary cutaneous malformations, bony and soft tissue hyperplasia, and multiple arteriovenous fistulas throughout the affected upper or lower extremity. These arteriovenous fistulas can be associated with life-threatening complications such as bleeding, thrombosis, and high output heart failure. In this report, we present a neonate who had a disproportionately hypertrophied left upper limb with port-wine stain, dystrophy of the left humerus, and hypertrophy of the left clavicle on X-ray, and arteriovenous malformation and massive dilatation of the left subclavian artery on magnetic resonance angiography. Exome sequencing analysis revealed a novel heterozygous splicing mutation (c.1776+2T>A) in the RASA1 gene. To the best of our knowledge, this report is the first case of RASA1-related Parkes Weber syndrome in Korea.


Asunto(s)
Humanos , Recién Nacido , Fístula Arteriovenosa , Malformaciones Arteriovenosas , Capilares , Clavícula , Dilatación , Exoma , Insuficiencia Cardíaca , Hemorragia , Húmero , Hiperplasia , Hipertrofia , Corea (Geográfico) , Extremidad Inferior , Angiografía por Resonancia Magnética , Mancha Vino de Oporto , Síndrome de Sturge-Weber , Arteria Subclavia , Trombosis , Extremidad Superior
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