Cardiothoracic imaging findings of Proteus syndrome.


Journal

Scientific reports
ISSN: 2045-2322
Titre abrégé: Sci Rep
Pays: England
ID NLM: 101563288

Informations de publication

Date de publication:
22 03 2021
Historique:
received: 25 11 2020
accepted: 08 03 2021
entrez: 23 3 2021
pubmed: 24 3 2021
medline: 26 10 2021
Statut: epublish

Résumé

In this work, we sought to delineate the prevalence of cardiothoracic imaging findings of Proteus syndrome in a large cohort at our institution. Of 53 individuals with a confirmed diagnosis of Proteus syndrome at our institution from 10/2001 to 10/2019, 38 individuals (men, n = 23; average age = 24 years) underwent cardiothoracic imaging (routine chest CT, CT pulmonary angiography and/or cardiac MRI). All studies were retrospectively and independently reviewed by two fellowship-trained cardiothoracic readers. Disagreements were resolved by consensus. Differences between variables were analyzed via parametric and nonparametric tests based on the normality of the distribution. The cardiothoracic findings of Proteus syndrome were diverse, but several were much more common and included: scoliosis from bony overgrowth (94%), pulmonary venous dilation (62%), band-like areas of lung scarring (56%), and hyperlucent lung parenchyma (50%). In addition, of 20 individuals who underwent cardiac MRI, 9/20 (45%) had intramyocardial fat, mostly involving the endocardial surface of the left ventricular septal wall. There was no statistically significant difference among the functional cardiac parameters between individuals with and without intramyocardial fat. Only one individual with intramyocardial fat had mildly decreased function (LVEF = 53%), while all others had normal ejection fraction.

Identifiants

pubmed: 33753828
doi: 10.1038/s41598-021-86029-0
pii: 10.1038/s41598-021-86029-0
pmc: PMC7985501
doi:

Types de publication

Journal Article Research Support, N.I.H., Intramural

Langues

eng

Sous-ensembles de citation

IM

Pagination

6577

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Auteurs

S Mojdeh Mirmomen (SM)

Cardiovascular and Pulmonary Branch, National Heart Lung and Blood Institute, National Institutes of Health, Building 10, Room B1D416, 10 Center Drive, Bethesda, MD, 20814, USA.

Andrew E Arai (AE)

Cardiovascular and Pulmonary Branch, National Heart Lung and Blood Institute, National Institutes of Health, Building 10, Room B1D416, 10 Center Drive, Bethesda, MD, 20814, USA.

Evrim B Turkbey (EB)

Radiology and Imaging Sciences, National Institutes of Health, Building 10, Room 1C336, Bethesda, MD, 20814, USA.

Andrew J Bradley (AJ)

Cardiovascular and Pulmonary Branch, National Heart Lung and Blood Institute, National Institutes of Health, Building 10, Room B1D416, 10 Center Drive, Bethesda, MD, 20814, USA.

Julie C Sapp (JC)

Genetic Disease Research Branch, National Human Genome Research Institute, National Institutes of Health, Building 10, Room 8D47E, Bethesda, MD, 20814, USA.

Leslie G Biesecker (LG)

Genetic Disease Research Branch, National Human Genome Research Institute, National Institutes of Health, Building 10, Room 8D47E, Bethesda, MD, 20814, USA.

Arlene Sirajuddin (A)

Cardiovascular and Pulmonary Branch, National Heart Lung and Blood Institute, National Institutes of Health, Building 10, Room B1D416, 10 Center Drive, Bethesda, MD, 20814, USA. arlene.sirajuddin@nih.gov.

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