Literature DB >> 8911612

Congenital Alopecia and nail dystrophy associated with severe functional T-cell immunodeficiency in two sibs.

C Pignata1, M Fiore, V Guzzetta, A Castaldo, G Sebastio, F Porta, A Guarino.   

Abstract

We report on two sisters affected by congenital alopecia, nail dystrophy, and a severe T-cell immunodeficiency, presumably inherited as an autosomal-recessive disorder. The T-cell defect was characterized by severe functional impairment, as shown by the lack of proliferative response and upregulation of activation markers following mitogen stimulation. The functional abnormality occurred in spite of the presence of phenotypically mature of the defect. This is the first observation reported on an ectodermal disorder, characterized by alopecia and nail dystrophy, observed at birth, in association with a primary immunodeficiency. The hypothesis that these two events may be casually related is discussed.

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Year:  1996        PMID: 8911612     DOI: 10.1002/(SICI)1096-8628(19961016)65:2<167::AID-AJMG17>3.0.CO;2-O

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  42 in total

1.  Laboratory technology for population-based screening for severe combined immunodeficiency in neonates: the winner is T-cell receptor excision circles.

Authors:  Jennifer M Puck
Journal:  J Allergy Clin Immunol       Date:  2012-01-29       Impact factor: 10.793

2.  First use of thymus transplantation therapy for FOXN1 deficiency (nude/SCID): a report of 2 cases.

Authors:  M Louise Markert; José G Marques; Bénédicte Neven; Blythe H Devlin; Elizabeth A McCarthy; Ivan K Chinn; Adriana S Albuquerque; Susana L Silva; Claudio Pignata; Geneviève de Saint Basile; Rui M Victorino; Capucine Picard; Marianne Debre; Nizar Mahlaoui; Alain Fischer; Ana E Sousa
Journal:  Blood       Date:  2010-10-26       Impact factor: 22.113

Review 3.  [Genetically induced hair diseases].

Authors:  T Wiederholt; P Poblete-Gutiérrez; J Frank
Journal:  Hautarzt       Date:  2003-07-04       Impact factor: 0.751

4.  A Novel FOXN1 Variant Is Identified in Two Siblings with Nude Severe Combined Immunodeficiency.

Authors:  Sinem Firtina; Funda Cipe; Yuk Yin Ng; Ayca Kiykim; Ozden Hatirnaz Ng; Tugce Sudutan; Cigdem Aydogmus; Safa Baris; Gulyuz Ozturk; Elif Aydiner; Ahmet Ozen; Muge Sayitoglu
Journal:  J Clin Immunol       Date:  2019-03-22       Impact factor: 8.317

Review 5.  FOXN1 Transcription Factor in Epithelial Growth and Wound Healing.

Authors:  Anna I Grabowska; Tomasz Wilanowski
Journal:  Mol Cell Biol       Date:  2017-08-11       Impact factor: 4.272

6.  FOXN1 compound heterozygous mutations cause selective thymic hypoplasia in humans.

Authors:  Qiumei Du; Larry K Huynh; Fatma Coskun; Erika Molina; Matthew A King; Prithvi Raj; Shaheen Khan; Igor Dozmorov; Christine M Seroogy; Christian A Wysocki; Grace T Padron; Tyler R Yates; M Louise Markert; M Teresa de la Morena; Nicolai Sc van Oers
Journal:  J Clin Invest       Date:  2019-11-01       Impact factor: 14.808

7.  Heterozygous FOXN1 Variants Cause Low TRECs and Severe T Cell Lymphopenia, Revealing a Crucial Role of FOXN1 in Supporting Early Thymopoiesis.

Authors:  Marita Bosticardo; Yasuhiro Yamazaki; Jennifer Cowan; Giuliana Giardino; Cristina Corsino; Giulia Scalia; Rosaria Prencipe; Melanie Ruffner; David A Hill; Inga Sakovich; Irma Yemialyanava; Jonathan S Tam; Nurcicek Padem; Melissa E Elder; John W Sleasman; Elena Perez; Hana Niebur; Christine M Seroogy; Svetlana Sharapova; Jennifer Gebbia; Gary Ira Kleiner; Jane Peake; Jordan K Abbott; Erwin W Gelfand; Elena Crestani; Catherine Biggs; Manish J Butte; Nicholas Hartog; Anthony Hayward; Karin Chen; Jennifer Heimall; Filiz Seeborg; Lisa M Bartnikas; Megan A Cooper; Claudio Pignata; Avinash Bhandoola; Luigi D Notarangelo
Journal:  Am J Hum Genet       Date:  2019-08-22       Impact factor: 11.025

8.  Hypomorphic phenotype of Foxn1 gene-modified rats by CRISPR/Cas9 system.

Authors:  Teppei Goto; Hiromasa Hara; Hiromitsu Nakauchi; Shinichi Hochi; Masumi Hirabayashi
Journal:  Transgenic Res       Date:  2016-03-02       Impact factor: 2.788

9.  Combined immunodeficiency associated with DOCK8 mutations.

Authors:  Qian Zhang; Jeremiah C Davis; Ian T Lamborn; Alexandra F Freeman; Huie Jing; Amanda J Favreau; Helen F Matthews; Joie Davis; Maria L Turner; Gulbu Uzel; Steven M Holland; Helen C Su
Journal:  N Engl J Med       Date:  2009-09-23       Impact factor: 91.245

Review 10.  Genetics of SCID.

Authors:  Fausto Cossu
Journal:  Ital J Pediatr       Date:  2010-11-15       Impact factor: 2.638

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