Congenital diaphragmatic hernia-associated pulmonary hypertension

Semin Perinatol. 2020 Feb;44(1):151167. doi: 10.1053/j.semperi.2019.07.006. Epub 2019 Jul 30.

Abstract

Congenital diaphragmatic hernia (CDH) is a neonatal pathology in which intrathoracic herniation of abdominal viscera via diaphragmatic defect results in aberrant pulmonary and cardiovascular development. Despite decades of study and many advances in the diagnosis and treatment of CDH, morbidity and mortality remain high, largely due to pulmonary hypertension (PH), along with pulmonary hypoplasia and cardiac dysfunction. In patients with CDH, hypoplastic pulmonary vasculature and alterations in multiple molecular pathways lead to pathophysiologic pulmonary vasculopathy and, for severe CDH, sustained, elevated pulmonary arterial pressures. This review addresses the multiple anatomic and physiologic changes that underlie CDH-associated PH (CDH-PH), along with the multimodal treatment strategies that exist currently and future therapies currently under investigation.

Keywords: Cardiac dysfunction; Congenital diaphragmatic hernia; Pulmonary hypertension; Pulmonary vasculature.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Endothelins / metabolism
  • Hernias, Diaphragmatic, Congenital / complications
  • Hernias, Diaphragmatic, Congenital / metabolism
  • Hernias, Diaphragmatic, Congenital / physiopathology*
  • Humans
  • Hypertension, Pulmonary / etiology
  • Hypertension, Pulmonary / metabolism
  • Hypertension, Pulmonary / physiopathology*
  • Infant, Newborn
  • Lung / abnormalities
  • Lung / embryology
  • Lung / physiopathology*
  • MicroRNAs / metabolism
  • Nitric Oxide / metabolism
  • Pulmonary Artery / abnormalities
  • Pulmonary Artery / embryology
  • Pulmonary Artery / physiopathology*
  • Pulmonary Veins / abnormalities
  • Pulmonary Veins / embryology
  • Pulmonary Veins / physiopathology*
  • Signal Transduction
  • Tretinoin / metabolism
  • Vascular Endothelial Growth Factor A / metabolism
  • Vascular Remodeling
  • Ventricular Dysfunction / etiology
  • Ventricular Dysfunction / physiopathology*

Substances

  • Endothelins
  • MicroRNAs
  • Vascular Endothelial Growth Factor A
  • Nitric Oxide
  • Tretinoin