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      Biomarkers for Pediatric Pulmonary Arterial Hypertension – A Call to Collaborate

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          Abstract

          Therapeutic approaches in pediatric pulmonary arterial hypertension (PAH) are based primarily on clinician experience, in contrast to the evidence-based approach in adults with pulmonary hypertension. There is a clear and present need for non-invasive and objective biomarkers to guide the accurate diagnosis, treatment, and prognosis of this disease in children. The multifaceted spectrum of disease, clinical presentation, and association with other diseases makes this a formidable challenge. However, as more progress is being made in the understanding and management of adult PAH, the potential to apply this knowledge to children has never been greater. This review explores the state of the art with regard to non-invasive biomarkers in PAH, with an eye toward those adult PAH biomarkers potentially suitable for application in pediatric PAH.

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          Most cited references139

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          Pulmonary arterial hypertension: baseline characteristics from the REVEAL Registry.

          The Registry to EValuate Early And Long-term pulmonary arterial hypertension disease management (REVEAL Registry) was established to provide updated characteristics of patients with pulmonary arterial hypertension (PAH) and to improve diagnosis, treatment, and management. Fifty-four US centers enrolled consecutively screened patients with World Health Organization group I PAH who met expanded hemodynamic criteria of mean pulmonary arterial pressure (PAP) > 25 mm Hg at rest (30 mm Hg with exercise), pulmonary capillary wedge pressure (PCWP) or= 240 dynes x s x cm(-5). Patients meeting the traditional hemodynamic definition (PCWP
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            Guidelines for the diagnosis and treatment of pulmonary hypertension.

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              An epidemiological study of pulmonary arterial hypertension.

              All hospitalisations for pulmonary arterial hypertension (PAH) in the Scottish population were examined to determine the epidemiological features of PAH. These data were compared with expert data from the Scottish Pulmonary Vascular Unit (SPVU). Using the linked Scottish Morbidity Record scheme, data from all adults aged 16-65 yrs admitted with PAH (idiopathic PAH, pulmonary hypertension associated with congenital heart abnormalities and pulmonary hypertension associated with connective tissue disorders) during the period 1986-2001 were identified. These data were compared with the most recent data in the SPVU database (2005). Overall, 374 Scottish males and females aged 16-65 yrs were hospitalised with incident PAH during 1986-2001. The annual incidence of PAH was 7.1 cases per million population. On December 31, 2002, there were 165 surviving cases, giving a prevalence of PAH of 52 cases per million population. Data from the SPVU were available for 1997-2006. In 2005, the last year with a complete data set, the incidence of PAH was 7.6 cases per million population and the corresponding prevalence was 26 cases per million population. Hospitalisation data from the Scottish Morbidity Record scheme gave higher prevalences of pulmonary arterial hypertension than data from the expert centres (Scotland and France). The hospitalisation data may overestimate the true frequency of pulmonary arterial hypertension in the population, but it is also possible that the expert centres underestimate the true frequency.
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                Author and article information

                Contributors
                Journal
                Front Pediatr
                Front Pediatr
                Front. Pediatr.
                Frontiers in Pediatrics
                Frontiers Media S.A.
                2296-2360
                22 December 2013
                03 February 2014
                2014
                : 2
                : 7
                Affiliations
                [1] 1Department of Bioengineering, University of Colorado Denver , Aurora, CO, USA
                [2] 2Department of Pediatrics-Critical Care, University of Colorado Denver , Aurora, CO, USA
                [3] 3Cardiovascular Pulmonary Research, University of Colorado Denver , Aurora, CO, USA
                [4] 4Linda Crnic Institute for Down Syndrome, University of Colorado Denver , Aurora, CO, USA
                [5] 5Children’s Hospital Denver , Aurora, CO, USA
                Author notes

                Edited by: Michael David Shields, Queen’s University Belfast, Northern Ireland

                Reviewed by: Nael McCarty, Emory University School of Medicine, USA; Ignacio Tapia, The Children’s Hospital of Philadelphia, USA; Michael David Shields, Queen’s University Belfast, Northern Ireland

                *Correspondence: Michael E. Yeager, Departments of Bioengineering and Pediatrics-Critical Care, Cardiovascular Pulmonary Research, Linda Crnic Institute for Down Syndrome, University of Colorado Denver, 12700 East 19th Avenue, Box B131, Aurora, CO 80045, USA e-mail: michael.yeager@ 123456ucdenver.edu

                Melanie J. Dufva and Ryan P. Delaney have contributed equally to this work.

                This article was submitted to Pediatric Pulmonology, a section of the journal Frontiers in Pediatrics.

                Article
                10.3389/fped.2014.00007
                3910125
                ab1b1af2-b5b6-46ea-8aab-4b1c613989be
                Copyright © 2014 Colvin, Dufva, Delaney, Ivy, Stenmark and Yeager.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                : 11 November 2013
                : 21 January 2014
                Page count
                Figures: 1, Tables: 0, Equations: 0, References: 147, Pages: 12, Words: 11363
                Categories
                Pediatrics
                Review Article

                biomarkers,right ventricle,pulmonary arterial hypertension,magnetic resonance,pediatric,echocardiography,imaging

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