Unique Pulmonary Hypertensive Vascular Diseases Associated with Heart and Lung Developmental Defects

  • Hidekazu Ishida
    Department of Pediatrics, Osaka University Graduate School of Medicine, 2-2 Yamadaoka, Suita 565-0871, Osaka, Japan
  • Hiroyuki Yamagishi
    Department of Pediatrics, Keio University of Medicine, 35 Shinanomachi, Shinjuku-ku 160-8582, Tokyo, Japan
  • Jun Maeda
    Department of Cardiology, Tokyo Metropolitan Children’s Medical Center, 2-8-29 Musashidai, Fuchu 183-8561, Tokyo, Japan
  • Keiko Uchida
    Department of Pediatrics, Keio University of Medicine, 35 Shinanomachi, Shinjuku-ku 160-8582, Tokyo, Japan

書誌事項

公開日
2023-08-03
資源種別
journal article
権利情報
  • https://creativecommons.org/licenses/by/4.0/
DOI
  • 10.3390/jcdd10080333
公開者
MDPI AG

説明

<jats:p>Although pediatric pulmonary hypertension (PH) shares features and mechanisms with adult PH, there are also some significant differences between the two conditions. Segmental PH is a unique pediatric subtype of PH with unclear and/or multifactorial pathophysiological mechanisms, and is often associated with complex congenital heart disease (CHD), pulmonary atresia with ventricular septal defect, and aortopulmonary collateral arteries. Some cases of complex CHD, associated with a single ventricle after Fontan operation, show pathological changes in the small peripheral pulmonary arteries and pulmonary vascular resistance similar to those observed in pulmonary arterial hypertension (PAH). This condition is termed as the pediatric pulmonary hypertensive vascular disease (PPHVD). Recent advances in genetics have identified the genes responsible for PAH associated with developmental defects of the heart and lungs, such as TBX4 and SOX17. Targeted therapies for PAH have been developed; however, their effects on PH associated with developmental heart and lung defects remain to be established. Real-world data analyses on the anatomy, pathophysiology, genetics, and molecular biology of unique PPHVD cases associated with developmental defects of the heart and lungs, using nationwide and/or international registries, should be conducted in order to improve the treatments and prognosis of patients with these types of pediatric PH.</jats:p>

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