Literature DB >> 28932937

FOXN1 Deficiency: from the Discovery to Novel Therapeutic Approaches.

Vera Gallo1, Emilia Cirillo1, Giuliana Giardino1, Claudio Pignata2.   

Abstract

Since the discovery of FOXN1 deficiency, the human counterpart of the nude mouse, a growing body of evidence investigating the role of FOXN1 in thymus and skin, has been published. FOXN1 has emerged as fundamental for thymus development, function, and homeostasis, representing the master regulator of thymic epithelial and T cell development. In the skin, it also plays a pivotal role in keratinocytes and hair follicle cell differentiation, although the underlying molecular mechanisms still remain to be fully elucidated. The nude severe combined immunodeficiency phenotype is indeed characterized by the clinical hallmarks of athymia with severe T cell immunodeficiency, congenital alopecia, and nail dystrophy. In this review, we summarize recent discoveries in the field and give interesting perspective about new and promising therapeutic approaches for disorders of immune system with athymia.

Entities:  

Keywords:  Athymia; FOXN1; Nude SCID phenotype; Nude mouse; Severe combined immunodeficiency; T cell development; Thymus transplantation

Mesh:

Substances:

Year:  2017        PMID: 28932937     DOI: 10.1007/s10875-017-0445-z

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.317


  67 in total

Review 1.  Role of thymic organ structure and stromal composition in steady-state postnatal T-cell production.

Authors:  Howard T Petrie
Journal:  Immunol Rev       Date:  2002-11       Impact factor: 12.988

2.  Brain alteration in a Nude/SCID fetus carrying FOXN1 homozygous mutation.

Authors:  Stefania Amorosi; Ilaria Vigliano; Ennio Del Giudice; Luigi Panico; Giuseppe M Maruotti; Anna Fusco; Mario Quarantelli; Carla Ciccone; Matilde V Ursini; Pasquale Martinelli; Claudio Pignata
Journal:  J Neurol Sci       Date:  2010-11-15       Impact factor: 3.181

Review 3.  Journey through the thymus: stromal guides for T-cell development and selection.

Authors:  Yousuke Takahama
Journal:  Nat Rev Immunol       Date:  2006-02       Impact factor: 53.106

4.  Absence of thymus in a mouse mutant.

Authors:  E M Pantelouris
Journal:  Nature       Date:  1968-01-27       Impact factor: 49.962

5.  Two genetically separable steps in the differentiation of thymic epithelium.

Authors:  M Nehls; B Kyewski; M Messerle; R Waldschütz; K Schüddekopf; A J Smith; T Boehm
Journal:  Science       Date:  1996-05-10       Impact factor: 47.728

6.  Nail dystrophy associated with a heterozygous mutation of the nude/SCID human FOXN1 (WHN) gene.

Authors:  Luigi Auricchio; Marsilio Adriani; Jorge Frank; Rosanna Busiello; Angela Christiano; Claudio Pignata
Journal:  Arch Dermatol       Date:  2005-05

7.  A forkhead-domain gene is mutated in a severe speech and language disorder.

Authors:  C S Lai; S E Fisher; J A Hurst; F Vargha-Khadem; A P Monaco
Journal:  Nature       Date:  2001-10-04       Impact factor: 49.962

8.  New member of the winged-helix protein family disrupted in mouse and rat nude mutations.

Authors:  M Nehls; D Pfeifer; M Schorpp; H Hedrich; T Boehm
Journal:  Nature       Date:  1994-11-03       Impact factor: 49.962

9.  Anomalies in the hormonal status of athymic nude mice.

Authors:  P Köpf-Maier; V F Mboneko
Journal:  J Cancer Res Clin Oncol       Date:  1990       Impact factor: 4.553

10.  Regulation of keratinocyte terminal differentiation by integrin-extracellular matrix interactions.

Authors:  F M Watt; M D Kubler; N A Hotchin; L J Nicholson; J C Adams
Journal:  J Cell Sci       Date:  1993-09       Impact factor: 5.285

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  11 in total

1.  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

2.  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

Review 3.  Thymic stromal cells: Roles in atrophy and age-associated dysfunction of the thymus.

Authors:  Sergio Cepeda; Ann V Griffith
Journal:  Exp Gerontol       Date:  2017-12-24       Impact factor: 4.032

4.  Editorial, Journal of Clinical Immunology.

Authors:  Jean-Laurent Casanova; Vincent Bonagura
Journal:  J Clin Immunol       Date:  2019-11       Impact factor: 8.317

Review 5.  [Ectopic thymic tissue and ectopic thymic tumors].

Authors:  A Marx; T Rüdiger; E Rößner; A Tzankov; V T de Montpréville; R R Rieker; P Ströbel; C-A Weis
Journal:  Pathologe       Date:  2018-09       Impact factor: 1.011

Review 6.  When Screening for Severe Combined Immunodeficiency (SCID) with T Cell Receptor Excision Circles Is Not SCID: a Case-Based Review.

Authors:  David Buchbinder; Jolan E Walter; Manish J Butte; Wan-Yin Chan; Maria Chitty Lopez; Victoria R Dimitriades; Morna J Dorsey; Diane J Nugent; Jennifer M Puck; Jasjit Singh; Cathleen A Collins
Journal:  J Clin Immunol       Date:  2021-01-07       Impact factor: 8.542

7.  Clinical, Immunological, and Molecular Features of Typical and Atypical Severe Combined Immunodeficiency: Report of the Italian Primary Immunodeficiency Network.

Authors:  Emilia Cirillo; Caterina Cancrini; Chiara Azzari; Silvana Martino; Baldassarre Martire; Andrea Pession; Alberto Tommasini; Samuele Naviglio; Andrea Finocchi; Rita Consolini; Paolo Pierani; Irene D'Alba; Maria Caterina Putti; Antonio Marzollo; Giuliana Giardino; Rosaria Prencipe; Federica Esposito; Fiorentino Grasso; Alessia Scarselli; Gigliola Di Matteo; Enrico Attardi; Silvia Ricci; Davide Montin; Fernando Specchia; Federica Barzaghi; Maria Pia Cicalese; Giuseppe Quaremba; Vassilios Lougaris; Silvia Giliani; Franco Locatelli; Paolo Rossi; Alessandro Aiuti; Raffaele Badolato; Alessandro Plebani; Claudio Pignata
Journal:  Front Immunol       Date:  2019-08-13       Impact factor: 7.561

8.  Development of the Nude Rabbit Model.

Authors:  Jun Song; Mark Hoenerhoff; Dongshan Yang; Ying Yang; Cheng Deng; Luan Wen; Linyuan Ma; Brooke Pallas; Changzhi Zhao; Yui Koike; Tomonari Koike; Patrick Lester; Bo Yang; Jifeng Zhang; Y Eugene Chen; Jie Xu
Journal:  Stem Cell Reports       Date:  2021-02-18       Impact factor: 7.765

9.  P63 targeted deletion under the FOXN1 promoter disrupts pre-and post-natal thymus development, function and maintenance as well as induces severe hair loss.

Authors:  Heather E Stefanski; Yan Xing; Jemma Nicholls; Leslie Jonart; Emily Goren; Patricia A Taylor; Alea A Mills; Megan Riddle; John McGrath; Jakub Tolar; Georg A Hollander; Bruce R Blazar
Journal:  PLoS One       Date:  2022-01-25       Impact factor: 3.752

10.  Fatal and Unresponsive Cytomegalovirus Infection in a New Homozygous FOXN1 Gene Variation Causing Nude SCID.

Authors:  Herberto Jose Chong-Neto; Gesmar Rodrigues Silva Segundo; Nelson Augusto Rosário
Journal:  J Clin Immunol       Date:  2022-01-22       Impact factor: 8.542

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