Literature DB >> 30264496

Hyper IgE syndromes: clinical and molecular characteristics.

Taha Al-Shaikhly1, Hans D Ochs2,3.   

Abstract

Hyper IgE syndromes comprise a group of rare primary immunodeficiency disorders characterized by a triad of atopic dermatitis, recurrent skin and lung infections along with elevated IgE levels. Job syndrome or autosomal dominant hyper IgE syndrome because of heterozygous loss-of-function mutations with dominant negative effect in signal transducer and activator of transcription-3 is the prototype of these disorders. However, several other genetically characterized immunodeficiency disorders have been identified over the past decade and joined the umbrella of hyper IgE syndromes including autosomal recessive mutations in the DOCK8, ZNF431 and PGM3 genes and heterozygous mutations with dominant negative effect in the CARD11 gene. Moreover, a number of phenotypically distinct immunodeficiency disorders can mimic hyper IgE syndromes, adding to the diagnostic challenge. Herein, we will concisely review these disorders, their molecular bases, highlighting key distinguishing clinical and laboratory findings and therapeutic options.
© 2018 Australasian Society for Immunology Inc.

Entities:  

Keywords:  zzm321990HIESzzm321990; zzm321990IPEXzzm321990; 22q11 deletion; CARD11; Comel-Netherton syndrome; DOCK8; DiGeorge syndrome; Omenn syndrome; PGM3; SPINK5; STAT3; Tyk2; Wiskott-Aldrich Syndrome; ZNF431; atopic dermatitis; eczema; hyper IgE syndromes

Year:  2018        PMID: 30264496     DOI: 10.1111/imcb.12209

Source DB:  PubMed          Journal:  Immunol Cell Biol        ISSN: 0818-9641            Impact factor:   5.126


  18 in total

1.  Congenital iRHOM2 deficiency causes ADAM17 dysfunction and environmentally directed immunodysregulatory disease.

Authors:  Satoshi Kubo; Jill M Fritz; Hayley M Raquer-McKay; Rhea Kataria; Ivan Vujkovic-Cvijin; Ahmad Al-Shaibi; Yikun Yao; Lixin Zheng; Juan Zou; Alex D Waldman; Xinyi Jing; Taylor K Farley; Ann Y Park; Andrew J Oler; Adrian K Charles; Melanie Makhlouf; Eman H AbouMoussa; Reem Hasnah; Luis R Saraiva; Sundar Ganesan; Abdulrahman Ahmed Al-Subaiey; Helen Matthews; Emilio Flano; Hyun Hee Lee; Alexandra F Freeman; Asena Pınar Sefer; Ersin Sayar; Erkan Çakır; Elif Karakoc-Aydiner; Safa Baris; Yasmine Belkaid; Ahmet Ozen; Bernice Lo; Michael J Lenardo
Journal:  Nat Immunol       Date:  2021-12-22       Impact factor: 25.606

2.  Ex vivo effect of JAK inhibition on JAK-STAT1 pathway hyperactivation in patients with dominant-negative STAT3 mutations.

Authors:  Pilar Blanco Lobo; Paloma Guisado-Hernández; Isabel Villaoslada; Beatriz de Felipe; Carmen Carreras; Hector Rodriguez; Begoña Carazo-Gallego; Ana Méndez-Echevarria; José Manuel Lucena; Pilar Ortiz Aljaro; María José Castro; José Francisco Noguera-Uclés; Joshua D Milner; Katelyn McCann; Ofer Zimmerman; Alexandra F Freeman; Michail S Lionakis; Steven M Holland; Olaf Neth; Peter Olbrich
Journal:  J Clin Immunol       Date:  2022-05-04       Impact factor: 8.542

3.  Pulmonary Infections and Surgical Complications in a Young Girl with Signal Transducer and Activator of Transcription 3 Loss-of-Function Mutation Hyperimmunoglobulin E Syndrome: A Case Report.

Authors:  Crhistian Toribio-Dionicio; Dania Cubas-Guzmán; Pedro Guerra-Canchari; Vanuza García-Sánchez; Wilmer Córdova-Calderón
Journal:  Pediatr Allergy Immunol Pulmonol       Date:  2021-03       Impact factor: 1.349

Review 4.  STAT3 Hyper-IgE Syndrome-an Update and Unanswered Questions.

Authors:  Christo Tsilifis; Alexandra F Freeman; Andrew R Gennery
Journal:  J Clin Immunol       Date:  2021-05-01       Impact factor: 8.317

5.  A delayed diagnosis of atypical immune dysregulation, polyendocrinopathy, enteropathy, X-linked (IPEX) syndrome: A case report.

Authors:  Ying Zhang; Hanmin Liu; Tao Ai; Wanmin Xia; Tingting Chen; Lei Zhang; Xiulan Luo; Yaping Duan
Journal:  Medicine (Baltimore)       Date:  2021-03-26       Impact factor: 1.817

6.  First Case of Patient With Two Homozygous Mutations in MYD88 and CARD9 Genes Presenting With Pyogenic Bacterial Infections, Elevated IgE, and Persistent EBV Viremia.

Authors:  Maria Chiriaco; Gigliola Di Matteo; Francesca Conti; Davide Petricone; Maia De Luca; Silvia Di Cesare; Cristina Cifaldi; Rita De Vito; Matteo Zoccolillo; Jessica Serafinelli; Noemi Poerio; Maurizio Fraziano; Immacolata Brigida; Fabio Cardinale; Paolo Rossi; Alessandro Aiuti; Caterina Cancrini; Andrea Finocchi
Journal:  Front Immunol       Date:  2019-02-14       Impact factor: 7.561

Review 7.  Definition of Opportunistic Infections in Immunocompromised Children on the Basis of Etiologies and Clinical Features: A Summary for Practical Purposes.

Authors:  Niccolò Riccardi; Gioacchino Andrea Rotulo; Elio Castagnola
Journal:  Curr Pediatr Rev       Date:  2019

8.  The clinical, immunological and genetic features of 12 Chinese patients with STAT3 mutations.

Authors:  Li Lin; Ying Wang; Bijun Sun; Luyao Liu; Wenjing Ying; Wenjie Wang; Qinhua Zhou; Jia Hou; Haili Yao; Liyuan Hu; Jinqiao Sun; Xiaochuan Wang
Journal:  Allergy Asthma Clin Immunol       Date:  2020-07-22       Impact factor: 3.406

9.  A Novel STAT3 Mutation in a Patient with Hyper-IgE Syndrome Diagnosed with a Severe Necrotizing Pulmonary Infection.

Authors:  Ran Zhao; Chao Wang; Chao Sun; Kun Jiang; Shengnan Wu; Fen Pan; Zeyu Zeng; Yijing Hu; Xiaoyan Dong
Journal:  J Asthma Allergy       Date:  2021-03-12

10.  STAT3 deficiency in B cells exacerbates uveitis by promoting expansion of pathogenic lymphocytes and suppressing regulatory B cells (Bregs) and Tregs.

Authors:  Favour O Oladipupo; Cheng-Rong Yu; Ezekiel Olumuyide; Yingyos Jittaysothorn; Jin Kyeong Choi; Charles E Egwuagu
Journal:  Sci Rep       Date:  2020-10-01       Impact factor: 4.996

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