Literature DB >> 25782637

Proteus syndrome: Report of a case with AKT1 mutation in a dental cyst.

Marie-Cécile Valéra1, Fréderic Vaysse2, Eric Bieth3, Michel Longy4, Claude Cances5, Isabelle Bailleul-Forestier6.   

Abstract

Proteus syndrome (PS) is a sporadic and rare congenital disorder characterized by a patchy or mosaic postnatal overgrowth, sometimes involving the face. The onset of overgrowth typically occurs in infancy and can commonly involve skin, connective tissue, central nervous system, eyes and viscera. The progressive overgrowth causes severe complications, such as skeletal deformities, cystic lung disease, invasive lipomas, connective tissue hyperplasia, benign and malignant tumours and deep venous thrombosis with pulmonary embolism, which can cause premature death. This disorder is caused by somatic mosaicism for a specific activating AKT1 mutation that would be lethal in a non-mosaic state. In this report, current knowledge of the aetiology, the diagnosis and the craniofacial manifestations of the disorder are reviewed. The short-term management of a 7-year-old patient with unusual oral manifestations is described. For the first time mutation of AKT1 (c.49G > A) gene was detected both in cranial exostosis and in central odontogenic fibroma of the lower jaw.
Copyright © 2015 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  AKT1; Central odontogenic fibroma; Craniofacial symptoms; Oral symptoms; Proteus syndrome

Mesh:

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Year:  2015        PMID: 25782637     DOI: 10.1016/j.ejmg.2015.02.008

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  3 in total

1.  Phenotype and Surgical Treatment in a Case of Proteus Syndrome With Craniofacial and Oral Findings.

Authors:  Reinhard E Friedrich
Journal:  In Vivo       Date:  2021 May-Jun       Impact factor: 2.155

2.  Postmortem Diagnosis of the Proteus Syndrome by Next Generation Sequencing of Affected Brain Tissue.

Authors:  Tiffany G Baker; William B Glen; Robert C Wilson; Nicholas I Batalis; Daynna J Wolff; Cynthia T Welsh
Journal:  Acad Forensic Pathol       Date:  2022-05-05

3.  Gingival Biopsy to Detect Mosaicism in Overgrowth Syndromes: Report of Two Cases of Megalencephaly-Capillary Malformation Syndrome with Periodontal Anomalies.

Authors:  Mathieu Marty; Carole Bonnaud; Natalie Jones; Michel Longy; Frédéric Vaysse; Eric Bieth; Isabelle Bailleul-Forestier
Journal:  Case Rep Dent       Date:  2020-09-12
  3 in total

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