Literature DB >> 29299618

Heart Transplantation in Children with Turner Syndrome: Analysis of a Linked Dataset.

Joshua D Chew1, Jonathan H Soslow2, Cary Thurm3, Matt Hall3, Debra A Dodd2, Brian Feingold4, Jill Simmons5, Justin Godown2.   

Abstract

Turner syndrome (TS) patients with hypoplastic left heart syndrome (HLHS) have poor single ventricle palliation outcomes; therefore, consideration of other potential management strategies is important. Little is known about heart transplantation (HTx) in this group, as standard HTx databases do not allow for identification of TS. This study describes experiences and outcomes of HTx in TS using a unique linkage between the Scientific Registry of Transplant Recipients and the Pediatric Health Information System databases. All pediatric HTx recipients (2002-2016) with TS were identified in the database using ICD-9 code 758.6 (gonadal dysgenesis) in conjunction with female sex. Patient characteristics and outcomes were described. Fourteen patients with TS were identified who underwent 16 HTx procedures at eight centers. For initial HTx, HLHS was the most common indication (10/14) with a median age of 10 months (IQR 3-73 months). Median transplant-free survival following initial HTx was 4.1 years (IQR 16 days-10.5 years), with all deaths occurring in the first year post-HTx. For patients that survived past 1 year (8/14), follow-up ranged from 4.1 to 10.9 years (median 8.0 years) with no deaths observed. Our cohort demonstrates that while there is a clear risk for early mortality, there is the potential for favorable outcomes following HTx in patients with TS. Therefore, TS should not be viewed as an absolute contraindication to HTx, but careful assessment of candidate risk is needed. Primary palliation with HTx for HLHS and TS may be a reasonable consideration given the poor outcomes of single ventricle palliation in this group. Further research is needed to fully delineate the outcomes and characteristics of this unique population.

Entities:  

Keywords:  Heart transplant; Hypoplastic left heart syndrome; Outcomes; Pediatric; Turner syndrome

Mesh:

Year:  2018        PMID: 29299618      PMCID: PMC5831528          DOI: 10.1007/s00246-017-1801-8

Source DB:  PubMed          Journal:  Pediatr Cardiol        ISSN: 0172-0643            Impact factor:   1.655


  29 in total

1.  Standardized multidisciplinary evaluation yields significant previously undiagnosed morbidity in adult women with Turner syndrome.

Authors:  Kim Freriks; Janneke Timmermans; Catharina C M Beerendonk; Chris M Verhaak; Romana T Netea-Maier; Barto J Otten; Didi D M Braat; Dominique F C M Smeets; Dirk H P M Kunst; Ad R M M Hermus; Henri J L M Timmers
Journal:  J Clin Endocrinol Metab       Date:  2011-07-13       Impact factor: 5.958

Review 2.  The Registry of the International Society for Heart and Lung Transplantation: Twentieth Pediatric Heart Transplantation Report-2017; Focus Theme: Allograft ischemic time.

Authors:  Joseph W Rossano; Wida S Cherikh; Daniel C Chambers; Samuel Goldfarb; Kiran Khush; Anna Y Kucheryavaya; Bronwyn J Levvey; Lars H Lund; Bruno Meiser; Roger D Yusen; Josef Stehlik
Journal:  J Heart Lung Transplant       Date:  2017-07-20       Impact factor: 10.247

Review 3.  Scientific Registry of Transplant Recipients: collecting, analyzing, and reporting data on transplantation in the United States.

Authors:  Susan Leppke; Tabitha Leighton; David Zaun; Shu-Cheng Chen; Melissa Skeans; Ajay K Israni; Jon J Snyder; Bertram L Kasiske
Journal:  Transplant Rev (Orlando)       Date:  2013-03-06       Impact factor: 3.943

4.  Frequency and outcomes of cardiac operations and catheter interventions in Turner syndrome.

Authors:  Erin Madriago; Thuan Nguyen; Megan McFerson; E Virgil Larson; Nathan Airhart; James H Moller; Michael Silberbach
Journal:  Am J Cardiol       Date:  2012-05-22       Impact factor: 2.778

5.  Outcome of infants with hypoplastic left heart and Turner syndromes.

Authors:  P M Reis; M R Punch; E L Bove; C J van de Ven
Journal:  Obstet Gynecol       Date:  1999-04       Impact factor: 7.661

6.  Insulin resistance is an intrinsic defect independent of fat mass in women with Turner's syndrome.

Authors:  Burak Salgin; Rakesh Amin; Kevin Yuen; Rachel M Williams; Peter Murgatroyd; David B Dunger
Journal:  Horm Res       Date:  2006-01-10

7.  Incidence of chromosome aberrations among 11148 newborn children.

Authors:  J Nielsen; I Sillesen
Journal:  Humangenetik       Date:  1975-10-20

8.  Vascular involvement of the liver in Turner's syndrome.

Authors:  Dominique Roulot; Claude Degott; Olivier Chazouillères; Frédéric Oberti; Paul Calès; Nicolas Carbonell; Said Benferhat; Solange Bresson-Hadni; Dominique Valla
Journal:  Hepatology       Date:  2004-01       Impact factor: 17.425

9.  The spectrum of congenital heart disease and outcomes after surgical repair among children with Turner syndrome: a single-center review.

Authors:  Jonathan W Cramer; Peter J Bartz; Pippa M Simpson; Steven D Zangwill
Journal:  Pediatr Cardiol       Date:  2013-08-10       Impact factor: 1.655

10.  Clinical predictors of autoimmune and severe atopic disease in pediatric heart transplant recipients.

Authors:  Jessica H Mouledoux; Erin L Albers; Zengqi Lu; Benjamin R Saville; Daniel J Moore; Debra A Dodd
Journal:  Pediatr Transplant       Date:  2013-12-28
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  1 in total

1.  Expanding analytic possibilities in pediatric solid organ transplantation through linkage of administrative and clinical registry databases.

Authors:  Justin Godown; Matt Hall; Bryn Thompson; Cary Thurm; Kathy Jabs; Lynette A Gillis; Einar T Hafberg; Sophoclis Alexopoulos; Seth J Karp; Jonathan H Soslow
Journal:  Pediatr Transplant       Date:  2019-02-21
  1 in total

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