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Simultaneous presence of Brugada and overgrowth syndromes.

Authors :
Segreti A
Piccirillo F
Crispino SP
Cocchia F
Martucciello A
Calabrese V
Gurrieri F
Grigioni F
Source :
Monaldi archives for chest disease = Archivio Monaldi per le malattie del torace [Monaldi Arch Chest Dis] 2023 Apr 27; Vol. 94 (1). Date of Electronic Publication: 2023 Apr 27.
Publication Year :
2023

Abstract

In the present article, we describe the case of a 21-year-old male presenting to the emergency department following a syncopal episode. Physical examination revealed a distinctive facial appearance in the context of an overgrowth syndrome. Also, an ajmaline test was performed because of the evidence of an incomplete right bundle branch block with ST-T segment elevation in the right precordial derivations, revealing a type-1 Brugada electrocardiographic pattern. Considering the high cardiovascular risk phenotype, the patient underwent subcutaneous cardiac defibrillator implantation. The subsequent comprehensive genomic testing analysis led to the diagnosis of a variant of uncertain significance of the nuclear receptor binding SET domain protein 1 (NSD1) gene and a heterozygous mutation of the calsequestrin 2 (CASQ2) gene. NSD1 gene alterations are usually responsible for the Sotos syndrome, characterized by distinctive facial appearance, learning disability, and overgrowth, in addition to cardiac anomalies ranging from single self-limiting alterations to more severe, complex cardiac abnormalities. On the contrary, a compound heterozygous or homozygous alteration of the CASQ2 gene is usually associated with catecholaminergic polymorphic ventricular tachycardia; however, the significance of a merely heterozygous alteration in the CASQ2 gene, as in the present case report, is not yet clear. In conclusion, to the best of our knowledge, this is the first description of the coexisting presence of Brugada and overgrowth syndromes in a single patient.

Details

Language :
English
ISSN :
1122-0643
Volume :
94
Issue :
1
Database :
MEDLINE
Journal :
Monaldi archives for chest disease = Archivio Monaldi per le malattie del torace
Publication Type :
Academic Journal
Accession number :
37114354
Full Text :
https://doi.org/10.4081/monaldi.2023.2521