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Fibrillin-1 Gene Mutations in Left Ventricular Non-compaction Cardiomyopathy.

Authors :
Parent JJ
Towbin JA
Jefferies JL
Source :
Pediatric cardiology [Pediatr Cardiol] 2016 Aug; Vol. 37 (6), pp. 1123-6. Date of Electronic Publication: 2016 May 09.
Publication Year :
2016

Abstract

Left ventricular non-compaction cardiomyopathy (LVNC) is a unique cardiomyopathy with a current yield of about 30-40 % in identifying a causative gene mutation. A retrospective review of all patients with LVNC at our institution was performed and genetic testing was reviewed. Echocardiographic and cardiac magnetic resonance imaging was reviewed to corroborate the reported phenotype. We present a series of patients with LVNC dilated phenotype associated with fibrillin-1 gene mutations. Fifty-one patients were identified as having LVNC with reduced left ventricular function and/or left ventricular dilation. We retrospectively reviewed gene testing in this cohort when available and identified 5 patients (10 %) with an FBN1 gene mutation. Syndrome breakdown as follows: 3 with Marfan, 1 with Shprintzen-Goldberg, and 1 with no identifiable syndrome. Derangements in fibrillin-1 may impact the compaction process resulting in LVNC. Although causation has not been proven by our report, it certainly raises interest in a possible mechanistic relationship between fibrillin-1 and LVNC given the increased prevalence of Marfan syndrome and fibrillin-1 gene mutations in this cohort.

Details

Language :
English
ISSN :
1432-1971
Volume :
37
Issue :
6
Database :
MEDLINE
Journal :
Pediatric cardiology
Publication Type :
Academic Journal
Accession number :
27160103
Full Text :
https://doi.org/10.1007/s00246-016-1404-9