Literature DB >> 17304050

Genotype/phenotype correlation in 325 individuals referred for a diagnosis of tuberous sclerosis complex in the United States.

Kit Sing Au1, Aimee T Williams, E Steve Roach, Lori Batchelor, Steven P Sparagana, Mauricio R Delgado, James W Wheless, James E Baumgartner, Benjamin B Roa, Carolyn M Wilson, Teresa K Smith-Knuppel, Min-Yuen C Cheung, Vicky H Whittemore, Terri M King, Hope Northrup.   

Abstract

Tuberous sclerosis complex is an autosomal dominant neurocutaneous disorder marked by hamartoma growth in multiple organ systems. We performed mutational analyses on 325 individuals with definite tuberous sclerosis complex diagnostic status. We identified mutations in 72% (199/257) of de novo and 77% (53/68) of familial cases, with 17% of mutations in the TSC1 gene and 50% in the TSC2 gene. There were 4% unclassified variants and 29% with no mutation identified. Genotype/phenotype analyses of all observed tuberous sclerosis complex findings in probands were performed, including several clinical features not analyzed in two previous large studies. We showed that patients with TSC2 mutations have significantly more hypomelanotic macules and learning disability in contrast to those with TSC1 mutations, findings not noted in previous studies. We also observed results consistent with two similar studies suggesting that individuals with mutations in TSC2 have more severe symptoms. On performing meta-analyses of our data and the other two largest studies in the literature, we found significant correlations for several features that individual studies did not have sufficient power to conclude. Male patients showed more frequent neurologic and eye symptoms, renal cysts, and ungual fibromas. Correlating genotypes with phenotypes should facilitate the disease management of tuberous sclerosis complex.

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Year:  2007        PMID: 17304050     DOI: 10.1097/gim.0b013e31803068c7

Source DB:  PubMed          Journal:  Genet Med        ISSN: 1098-3600            Impact factor:   8.822


  113 in total

1.  Biallelic TSC gene inactivation in tuberous sclerosis complex.

Authors:  Peter B Crino; Eleonora Aronica; Gordon Baltuch; Katherine L Nathanson
Journal:  Neurology       Date:  2010-05-25       Impact factor: 9.910

2.  Characterisation of TSC1 promoter deletions in tuberous sclerosis complex patients.

Authors:  Ans M W van den Ouweland; Peter Elfferich; Bernard A Zonnenberg; Willem F Arts; Tjitske Kleefstra; Mark D Nellist; Jose M Millan; Caroline Withagen-Hermans; Anneke J A Maat-Kievit; Dicky J J Halley
Journal:  Eur J Hum Genet       Date:  2010-09-29       Impact factor: 4.246

3.  Resting and task-modulated high-frequency brain rhythms measured by scalp encephalography in infants with tuberous sclerosis complex.

Authors:  Catherine Stamoulis; Vanessa Vogel-Farley; Geneva Degregorio; Shafali S Jeste; Charles A Nelson
Journal:  J Autism Dev Disord       Date:  2015-02

4.  Clinical utility gene card for: tuberous sclerosis complex (TSC1, TSC2).

Authors:  Karin Mayer; Christa Fonatsch; Katharina Wimmer; Ans M W van den Ouweland; Anneke J A Maat-Kievit
Journal:  Eur J Hum Genet       Date:  2013-06-12       Impact factor: 4.246

5.  Tuberous sclerosis complex diagnostic criteria update: recommendations of the 2012 Iinternational Tuberous Sclerosis Complex Consensus Conference.

Authors:  Hope Northrup; Darcy A Krueger
Journal:  Pediatr Neurol       Date:  2013-10       Impact factor: 3.372

6.  MRI findings reveal three different types of tubers in patients with tuberous sclerosis complex.

Authors:  Anne Gallagher; Ellen P Grant; Neel Madan; Delma Y Jarrett; David A Lyczkowski; Elizabeth A Thiele
Journal:  J Neurol       Date:  2010-03-30       Impact factor: 4.849

Review 7.  Thoracoabdominal imaging of tuberous sclerosis.

Authors:  Cara E Morin; Nicholas P Morin; David N Franz; Darcy A Krueger; Andrew T Trout; Alexander J Towbin
Journal:  Pediatr Radiol       Date:  2018-08-04

8.  A reliable cell-based assay for testing unclassified TSC2 gene variants.

Authors:  Ricardo Coevoets; Sermin Arican; Marianne Hoogeveen-Westerveld; Erik Simons; Ans van den Ouweland; Dicky Halley; Mark Nellist
Journal:  Eur J Hum Genet       Date:  2008-10-15       Impact factor: 4.246

9.  Rhinencephalon changes in tuberous sclerosis complex.

Authors:  Renzo Manara; Davide Brotto; Samuela Bugin; Maria Federica Pelizza; Stefano Sartori; Margherita Nosadini; Sara Azzolini; Giorgio Iaconetta; Cecilia Parazzini; Alessandra Murgia; Angela Peron; Paola Canevini; Francesca Labriola; Aglaia Vignoli; Irene Toldo
Journal:  Neuroradiology       Date:  2018-06-17       Impact factor: 2.804

10.  MetaSeq: privacy preserving meta-analysis of sequencing-based association studies.

Authors:  Angad Pal Singh; Samreen Zafer; Itsik Pe'er
Journal:  Pac Symp Biocomput       Date:  2013
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