Literature DB >> 33399177

Proteus syndrome caused by novel somatic AKT1 duplication.

Talal AlAnzi1, Eman Al-Mashharawi, Amal Alhashem.   

Abstract

Proteus syndrome (PS) is a rare overgrowth disorder that presents with asymmetrical growth of the bone and fat tissues following a mosaic pattern mutation. The estimated worldwide incidence is approximately one in one million live births. Proteus syndrome causes disfigurement and psychological impact through its effects on somatic tissue. Due to its rarity and diversity of tissues involved, it represents a significant challenge to caregivers and multidisciplinary medical teams. Here, we report a Saudi girl, with a large left cervical mass discovered antenatally. This mass was identified as a growing cystic hygroma, and she had features of overgrowth and hemangiomas. Whole exome sequencing was negative from the blood lymphocytes and affected tissue sample.  However, deletion duplication analysis from tissue shows a novel mosaic somatic mutation of the AKT1 gene. Somatic mutation remains an obstacle, and the geneticist has an essential role in its management, providing an established genetic diagnosis, prognosis, and family counselling.

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Year:  2021        PMID: 33399177      PMCID: PMC7989324          DOI: 10.15537/smj.2021.1.25618

Source DB:  PubMed          Journal:  Saudi Med J        ISSN: 0379-5284            Impact factor:   1.484


  6 in total

Review 1.  Reassessment of the Proteus syndrome literature: application of diagnostic criteria to published cases.

Authors:  Joyce T Turner; M Michael Cohen; Leslie G Biesecker
Journal:  Am J Med Genet A       Date:  2004-10-01       Impact factor: 2.802

Review 2.  Proteus syndrome review: molecular, clinical, and pathologic features.

Authors:  M Michael Cohen
Journal:  Clin Genet       Date:  2013-10-23       Impact factor: 4.438

3.  A mosaic activating mutation in AKT1 associated with the Proteus syndrome.

Authors:  Marjorie J Lindhurst; Julie C Sapp; Jamie K Teer; Jennifer J Johnston; Erin M Finn; Kathryn Peters; Joyce Turner; Jennifer L Cannons; David Bick; Laurel Blakemore; Catherine Blumhorst; Knut Brockmann; Peter Calder; Natasha Cherman; Matthew A Deardorff; David B Everman; Gretchen Golas; Robert M Greenstein; B Maya Kato; Kim M Keppler-Noreuil; Sergei A Kuznetsov; Richard T Miyamoto; Kurt Newman; David Ng; Kevin O'Brien; Steven Rothenberg; Douglas J Schwartzentruber; Virender Singhal; Roberto Tirabosco; Joseph Upton; Shlomo Wientroub; Elaine H Zackai; Kimberly Hoag; Tracey Whitewood-Neal; Pamela G Robey; Pamela L Schwartzberg; Thomas N Darling; Laura L Tosi; James C Mullikin; Leslie G Biesecker
Journal:  N Engl J Med       Date:  2011-07-27       Impact factor: 91.245

4.  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

5.  Proteus Syndrome: a difficult diagnosis and management plan.

Authors:  M D Popescu; G Burnei; L Draghici; I Draghici
Journal:  J Med Life       Date:  2014 Oct-Dec

6.  Orthopaedic manifestations of Proteus syndrome in a child with literature update.

Authors:  Tamer Ahmed El-Sobky; Solaf M Elsayed; Dalia M E El Mikkawy
Journal:  Bone Rep       Date:  2015-09-26
  6 in total
  1 in total

Review 1.  Genetic and Molecular Determinants of Lymphatic Malformations: Potential Targets for Therapy.

Authors:  Su Yeon Lee; Emma Grace Loll; Abd-Elrahman Said Hassan; Mingyu Cheng; Aijun Wang; Diana Lee Farmer
Journal:  J Dev Biol       Date:  2022-02-08
  1 in total

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