Predictors of Rapid Aortic Root Dilation and Referral for Aortic Surgery in Marfan Syndrome View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2018-06-11

AUTHORS

Arvind Hoskoppal, Shaji Menon, Felicia Trachtenberg, Kristin M. Burns, Julie De Backer, Bruce D. Gelb, Marie Gleason, Jeanne James, Wyman W. Lai, Aimee Liou, Lynn Mahony, Aaron K. Olson, Reed E. Pyeritz, Angela M. Sharkey, Mario Stylianou, Stephanie Burns Wechsler, Luciana Young, Jami C. Levine, Elif Seda Selamet Tierney, Ronald V. Lacro, Timothy J. Bradley, Pediatric Heart Network Investigators

ABSTRACT

Few data exist regarding predictors of rapid aortic root dilation and referral for aortic surgery in Marfan syndrome (MFS). To identify independent predictors of the rate of aortic root (AoR) dilation and referral for aortic surgery, we investigated the data from the Pediatric Heart Network randomized trial of atenolol versus losartan in young patients with MFS. Data were analyzed from the echocardiograms at 0, 12, 24, and 36 months read in the core laboratory of 608 trial subjects, aged 6 months to 25 years, who met original Ghent criteria and had an AoR z-score (AoRz) > 3. Repeated measures linear and logistic regressions were used to determine multivariable predictors of AoR dilation. Receiver operator characteristic curves were used to determine cut-points in AoR dilation predicting referral for aortic surgery. Multivariable analysis showed rapid AoR dilation as defined by change in AoRz/year > 90th percentile was associated with older age, higher sinotubular junction z-score, and atenolol use (R2 = 0.01) or by change in AoR diameter (AoRd)/year > 90th percentile with higher sinotubular junction z-score and non-white race (R2 = 0.02). Referral for aortic root surgery was associated with higher AoRd, higher ascending aorta z-score, and higher sinotubular junction diameter:ascending aorta diameter ratio (R2 = 0.17). Change in AoRz of 0.72 SD units/year had 42% sensitivity and 92% specificity and change in AoRd of 0.34 cm/year had 38% sensitivity and 95% specificity for predicting referral for aortic surgery. In this cohort of young patients with MFS, no new robust predictors of rapid AoR dilation or referral for aortic root surgery were identified. Further investigation may determine whether generalized proximal aortic dilation and effacement of the sinotubular junction will allow for better risk stratification. Rate of AoR dilation cut-points had high specificity, but low sensitivity for predicting referral for aortic surgery, limiting their clinical use. Clinical Trial Number ClinicalTrials.gov number, NCT00429364. More... »

PAGES

1453-1461

References to SciGraph publications

Journal

TITLE

Pediatric Cardiology

ISSUE

7

VOLUME

39

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s00246-018-1916-6

DOI

http://dx.doi.org/10.1007/s00246-018-1916-6

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1104528265

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/29948025


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    "description": "Few data exist regarding predictors of rapid aortic root dilation and referral for aortic surgery in Marfan syndrome (MFS). To identify independent predictors of the rate of aortic root (AoR) dilation and referral for aortic surgery, we investigated the data from the Pediatric Heart Network randomized trial of atenolol versus losartan in young patients with MFS. Data were analyzed from the echocardiograms at 0, 12, 24, and 36\u00a0months read in the core laboratory of 608 trial subjects, aged 6\u00a0months to 25 years, who met original Ghent criteria and had an AoR z-score (AoRz)\u2009>\u20093. Repeated measures linear and logistic regressions were used to determine multivariable predictors of AoR dilation. Receiver operator characteristic curves were used to determine cut-points in AoR dilation predicting referral for aortic surgery. Multivariable analysis showed rapid AoR dilation as defined by change in AoRz/year\u2009>\u200990th percentile was associated with older age, higher sinotubular junction z-score, and atenolol use (R2\u2009=\u20090.01) or by change in AoR diameter (AoRd)/year\u2009>\u200990th percentile with higher sinotubular junction z-score and non-white race (R2\u2009=\u20090.02). Referral for aortic root surgery was associated with higher AoRd, higher ascending aorta z-score, and higher sinotubular junction diameter:ascending aorta diameter ratio (R2\u2009=\u20090.17). Change in AoRz of 0.72 SD units/year had 42% sensitivity and 92% specificity and change in AoRd of 0.34\u00a0cm/year had 38% sensitivity and 95% specificity for predicting referral for aortic surgery. In this cohort of young patients with MFS, no new robust predictors of rapid AoR dilation or referral for aortic root surgery were identified. Further investigation may determine whether generalized proximal aortic dilation and effacement of the sinotubular junction will allow for better risk stratification. Rate of AoR dilation cut-points had high specificity, but low sensitivity for predicting referral for aortic surgery, limiting their clinical use. Clinical Trial Number ClinicalTrials.gov number, NCT00429364.", 
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