Literature DB >> 23613309

Novel mutation in TP63 associated with ectrodactyly ectodermal dysplasia and clefting syndrome and T cell lymphopenia.

Philip F Giampietro1, Mei W Baker, Monica J Basehore, Julie R Jones, Christine M Seroogy.   

Abstract

A male child with clinical features consistent with EEC/EECUT plus syndrome (ectrodactyly, ectodermal dysplasia, clefting, urinary tract abnormalities, and thymic abnormalities) including mild ectodermal abnormalities, ectrodactyly of hands and feet, cleft palate, bilateral hydronephrosis, and T cell lymphopenia is reported. He was noted to have T cell receptor excision circle (TREC) analysis below the cutoff for normal on newborn screening and T cell lymphopenia on further immunologic evaluation. A novel, presumably pathogenic de novo 3 bp deletion in exon 7 of TP63 (c.970_972delATT; NCBI Reference Sequence NM_003722.4) was identified. This observation provides supporting evidence for the association between TP63 mutations and EECUT plus syndrome. Clinicians caring for infants presenting with EEC spectrum disorders in the newborn period should also consider the possibility of T cell lymphopenia.
Copyright © 2013 Wiley Periodicals, Inc.

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Year:  2013        PMID: 23613309      PMCID: PMC5484633          DOI: 10.1002/ajmg.a.35885

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  11 in total

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Authors:  C Clare Blackburn; Nancy R Manley
Journal:  Nat Rev Immunol       Date:  2004-04       Impact factor: 53.106

2.  Newborn screening for severe combined immunodeficiency; the Wisconsin experience (2008-2011).

Authors:  James W Verbsky; Mei W Baker; William J Grossman; Mary Hintermeyer; Trivikram Dasu; Benedetta Bonacci; Sreelatha Reddy; David Margolis; James Casper; Miranda Gries; Ken Desantes; Gary L Hoffman; Charles D Brokopp; Christine M Seroogy; John M Routes
Journal:  J Clin Immunol       Date:  2011-11-10       Impact factor: 8.317

Review 3.  p63-associated disorders.

Authors:  Tuula Rinne; Hans G Brunner; Hans van Bokhoven
Journal:  Cell Cycle       Date:  2007-02-03       Impact factor: 4.534

4.  Updating a diagnosis. The EEC/EECUT syndrome.

Authors:  F Hecht
Journal:  Am J Dis Child       Date:  1985-12

5.  Hypoplastic thymus and T-cell reduction in EECUT syndrome.

Authors:  H Frick; D M Münger; J C Fauchère; T Stallmach
Journal:  Am J Med Genet       Date:  1997-03-03

Review 6.  Splitting p63.

Authors:  Hans van Bokhoven; Han G Brunner
Journal:  Am J Hum Genet       Date:  2002-05-30       Impact factor: 11.025

7.  Molecular characterization of iodotyrosine dehalogenase deficiency in patients with hypothyroidism.

Authors:  Gijs Afink; Willem Kulik; Henk Overmars; Janine de Randamie; Truus Veenboer; Arno van Cruchten; Margarita Craen; Carrie Ris-Stalpers
Journal:  J Clin Endocrinol Metab       Date:  2008-09-02       Impact factor: 5.958

8.  Development of a routine newborn screening protocol for severe combined immunodeficiency.

Authors:  Mei W Baker; William J Grossman; Ronald H Laessig; Gary L Hoffman; Charles D Brokopp; Daniel F Kurtycz; Michael F Cogley; Thomas J Litsheim; Murray L Katcher; John M Routes
Journal:  J Allergy Clin Immunol       Date:  2009-05-31       Impact factor: 10.793

9.  Lymphocyte subsets in healthy children from birth through 18 years of age: the Pediatric AIDS Clinical Trials Group P1009 study.

Authors:  William T Shearer; Howard M Rosenblatt; Rebecca S Gelman; Rebecca Oyomopito; Susan Plaeger; E Richard Stiehm; Diane W Wara; Steven D Douglas; Katherine Luzuriaga; Elizabeth J McFarland; Ram Yogev; Mobeen H Rathore; Wende Levy; Bobbie L Graham; Stephen A Spector
Journal:  J Allergy Clin Immunol       Date:  2003-11       Impact factor: 10.793

10.  DeltaNp63 regulates thymic development through enhanced expression of FgfR2 and Jag2.

Authors:  Eleonora Candi; Alessandro Rufini; Alessandro Terrinoni; Alessandro Giamboi-Miraglia; Anna Maria Lena; Roberto Mantovani; Richard Knight; Gerry Melino
Journal:  Proc Natl Acad Sci U S A       Date:  2007-07-11       Impact factor: 11.205

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  1 in total

1.  Newborn screening for severe combined immunodeficiency in 11 screening programs in the United States.

Authors:  Antonia Kwan; Roshini S Abraham; Robert Currier; Amy Brower; Karen Andruszewski; Jordan K Abbott; Mei Baker; Mark Ballow; Louis E Bartoshesky; Francisco A Bonilla; Charles Brokopp; Edward Brooks; Michele Caggana; Jocelyn Celestin; Joseph A Church; Anne Marie Comeau; James A Connelly; Morton J Cowan; Charlotte Cunningham-Rundles; Trivikram Dasu; Nina Dave; Maria T De La Morena; Ulrich Duffner; Chin-To Fong; Lisa Forbes; Debra Freedenberg; Erwin W Gelfand; Jaime E Hale; I Celine Hanson; Beverly N Hay; Diana Hu; Anthony Infante; Daisy Johnson; Neena Kapoor; Denise M Kay; Donald B Kohn; Rachel Lee; Heather Lehman; Zhili Lin; Fred Lorey; Aly Abdel-Mageed; Adrienne Manning; Sean McGhee; Theodore B Moore; Stanley J Naides; Luigi D Notarangelo; Jordan S Orange; Sung-Yun Pai; Matthew Porteus; Ray Rodriguez; Neil Romberg; John Routes; Mary Ruehle; Arye Rubenstein; Carlos A Saavedra-Matiz; Ginger Scott; Patricia M Scott; Elizabeth Secord; Christine Seroogy; William T Shearer; Subhadra Siegel; Stacy K Silvers; E Richard Stiehm; Robert W Sugerman; John L Sullivan; Susan Tanksley; Millard L Tierce; James Verbsky; Beth Vogel; Rosalyn Walker; Kelly Walkovich; Jolan E Walter; Richard L Wasserman; Michael S Watson; Geoffrey A Weinberg; Leonard B Weiner; Heather Wood; Anne B Yates; Jennifer M Puck; Vincent R Bonagura
Journal:  JAMA       Date:  2014-08-20       Impact factor: 56.272

  1 in total

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