Literature DB >> 28209770

Are Patients With Loeys-Dietz Syndrome Misdiagnosed With Beals Syndrome?

Rebecca Woolnough1, Andrew Dhawan2, Kimberly Dow1, Jagdeep S Walia3.   

Abstract

Beals syndrome, also known as congenital contractural arachnodactyly (Online Mendelian Inheritance in Man: 121050), is an autosomal dominant disorder caused by a mutation in FBN2 that is typically characterized by congenital contractures and arachnodactyly. It shares a number of phenotypic features with Loeys-Dietz syndrome (Online Mendelian Inheritance in Man: 609192). Loeys-Dietz syndrome, initially described in 2005, is associated with mutations for the transforming growth factor β receptor and is characterized by findings of cerebral, thoracic, and abdominal arterial aneurysms. This report describes a 17-year-old male patient with a typical neonatal diagnosis of Beals syndrome. At age 15 years, an echocardiogram conducted in response to an aortic dissection in his father showed moderate aortic root dilation, prompting comprehensive testing for aortopathies, revealing a mutation in TGFBR1, thereby changing the diagnosis to Loeys-Dietz syndrome. Previously published reports have not implicated any mutation of the transforming growth factor β receptor genes in cases of Beals syndrome. This case underscores that due to significant phenotypic overlap, there is utility in a full panel of testing, including genes for hereditary connective tissue disorders with vascular involvement, as well as FBN2. Likewise, young patients who have tested negative for FBN2 should be tested for hereditary connective tissue disorders with vascular involvement.
Copyright © 2017 by the American Academy of Pediatrics.

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Year:  2017        PMID: 28209770     DOI: 10.1542/peds.2016-1281

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  3 in total

Review 1.  Neonatal presentation of Loeys-Dietz syndrome: two case reports and review of the literature.

Authors:  Francesco Baldo; Laura Morra; Agnese Feresin; Flavio Faletra; Yasmin Al Naber; Luigi Memo; Laura Travan
Journal:  Ital J Pediatr       Date:  2022-06-06       Impact factor: 3.288

Review 2.  The genetics and biomechanics of thoracic aortic diseases.

Authors:  Amer Harky; Ka Siu Fan; Ka Hay Fan
Journal:  Vasc Biol       Date:  2019-10-15

3.  A Genomic Approach to Delineating the Occurrence of Scoliosis in Arthrogryposis Multiplex Congenita.

Authors:  Xenia Latypova; Stefan Giovanni Creadore; Noémi Dahan-Oliel; Anxhela Gjyshi Gustafson; Steven Wei-Hung Hwang; Tanya Bedard; Kamran Shazand; Harold J P van Bosse; Philip F Giampietro; Klaus Dieterich
Journal:  Genes (Basel)       Date:  2021-07-08       Impact factor: 4.096

  3 in total

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