Literature DB >> 33753828

Cardiothoracic imaging findings of Proteus syndrome.

S Mojdeh Mirmomen1, Andrew E Arai1, Evrim B Turkbey2, Andrew J Bradley1, Julie C Sapp3, Leslie G Biesecker3, Arlene Sirajuddin4.   

Abstract

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.

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Year:  2021        PMID: 33753828      PMCID: PMC7985501          DOI: 10.1038/s41598-021-86029-0

Source DB:  PubMed          Journal:  Sci Rep        ISSN: 2045-2322            Impact factor:   4.996


  29 in total

Review 1.  The challenges of Proteus syndrome: diagnosis and management.

Authors:  Leslie Biesecker
Journal:  Eur J Hum Genet       Date:  2006-08-02       Impact factor: 4.246

Review 2.  Proteus syndrome: diagnostic criteria, differential diagnosis, and patient evaluation.

Authors:  L G Biesecker; R Happle; J B Mulliken; R Weksberg; J M Graham; D L Viljoen; M M Cohen
Journal:  Am J Med Genet       Date:  1999-06-11

3.  Myocardial fat overgrowth in Proteus syndrome.

Authors:  H Hannoush; V Sachdev; A Brofferio; A E Arai; G LaRocca; J Sapp; S Sidenko; C Brenneman; L G Biesecker; K M Keppler-Noreuil
Journal:  Am J Med Genet A       Date:  2014-11-06       Impact factor: 2.802

4.  Pulmonary manifestations and management of proteus syndrome.

Authors:  Chia-Ying Li; Yih-Leong Chang; Wei-Chou Chen; Yung-Chie Lee
Journal:  J Formos Med Assoc       Date:  2010-05       Impact factor: 3.282

Review 5.  CT and MRI in diseases of the aorta.

Authors:  Diana Litmanovich; Alexander A Bankier; Luce Cantin; Vassilios Raptopoulos; Phillip M Boiselle
Journal:  AJR Am J Roentgenol       Date:  2009-10       Impact factor: 3.959

6.  Myocardial Fat Imaging.

Authors:  Peter Kellman; Diego Hernando; Andrew E Arai
Journal:  Curr Cardiovasc Imaging Rep       Date:  2010-03-11

7.  Multiecho dixon fat and water separation method for detecting fibrofatty infiltration in the myocardium.

Authors:  Peter Kellman; Diego Hernando; Saurabh Shah; Sven Zuehlsdorff; Renate Jerecic; Christine Mancini; Zhi-Pei Liang; Andrew E Arai
Journal:  Magn Reson Med       Date:  2009-01       Impact factor: 4.668

8.  Macrodactyly, hemihypertrophy, and connective tissue nevi: report of a new syndrome and review of the literature.

Authors:  S A Temtamy; J G Rogers
Journal:  J Pediatr       Date:  1976-12       Impact factor: 4.406

9.  Early Recognition of Proteus Syndrome.

Authors:  Dorothy L Rodenbeck; Laura A Greyling; John H Anderson; Loretta S Davis
Journal:  Pediatr Dermatol       Date:  2016-07-04       Impact factor: 1.588

10.  A dyadic genotype-phenotype approach to diagnostic criteria for Proteus syndrome.

Authors:  Julie C Sapp; Anna Buser; Jasmine Burton-Akright; Kim M Keppler-Noreuil; Leslie G Biesecker
Journal:  Am J Med Genet C Semin Med Genet       Date:  2019-11-06       Impact factor: 3.359

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  2 in total

1.  Precapillary pulmonary arterial hypertension in a patient with Proteus syndrome.

Authors:  Akash Mathavan; Akshay Mathavan; Cyrus Vahdatpour; Christina Eagan; Saminder Singh Kalra; Ali Ataya
Journal:  Pulm Circ       Date:  2022-04-01       Impact factor: 2.886

2.  Late-onset Proteus syndrome with cerebriform connective tissue nevus and subsequent development of intraductal papilloma.

Authors:  Emily W Modlin; Anne M Slavotinek; Thomas N Darling; Stanley Lipkowitz; Frederic G Barr; Pamela N Munster; Leslie G Biesecker; Christopher A Ours
Journal:  Am J Med Genet A       Date:  2022-04-20       Impact factor: 2.578

  2 in total

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