Literature DB >> 27566612

A novel deletion mutation in IL2RG gene results in X-linked severe combined immunodeficiency with an atypical phenotype.

Wenjun Mou1,2, Jianxin He1,3, Xi Chen1,2, Hui Zhang1,2, Xiaoya Ren1,2, Xunyao Wu1,2, Xin Ni1,4, Baoping Xu1,3, Jingang Gui5,6.   

Abstract

Severe combined immunodeficiency (SCID) is the most serious disorder among primary immunodeficiency diseases threatening children's life. Atypical SCID variant, presenting with mild reduced T cells subsets, is often associated with infection susceptibility but poor clinical diagnosis. The atypical X-SCID patient in the present study showed a mild clinical presentation with a TlowNK+B+ immunophenotype. The patient has reduced T- cell subpopulations with a subdued thymic output measured by sjTRECs. Further analysis showed that T cells maintained a normal proliferation and a broad Vβ repertoire. NK cells, however, exhibited a skewed development toward immature CD3-CD16+CD56- cells. Genetic analysis revealed a novel deletion at nucleotide 52 in exon 1 of IL2RG gene. Sequence alignment predicted a truncated IL2RG protein missing signal peptide derived from a possible alternative reading frame. The novel mutation in IL2RG gene identified in our study may help the early diagnosis of atypical X-SCID.

Entities:  

Keywords:  Atypical SCID; Frameshift; IL2RG gene; Primary immunodeficiency diseases; Signal peptide; X-linked SCID

Mesh:

Substances:

Year:  2016        PMID: 27566612     DOI: 10.1007/s00251-016-0949-3

Source DB:  PubMed          Journal:  Immunogenetics        ISSN: 0093-7711            Impact factor:   2.846


  30 in total

1.  Detection of maternal DNA in placental/umbilical cord blood by locus-specific amplification of the noninherited maternal HLA gene.

Authors:  A Scaradavou; C Carrier; N Mollen; C Stevens; P Rubinstein
Journal:  Blood       Date:  1996-08-15       Impact factor: 22.113

2.  Using CLUSTAL for multiple sequence alignments.

Authors:  D G Higgins; J D Thompson; T J Gibson
Journal:  Methods Enzymol       Date:  1996       Impact factor: 1.600

3.  Cutting edge: the common gamma-chain is an indispensable subunit of the IL-21 receptor complex.

Authors:  H Asao; C Okuyama; S Kumaki; N Ishii; S Tsuchiya; D Foster; K Sugamura
Journal:  J Immunol       Date:  2001-07-01       Impact factor: 5.422

Review 4.  Interleukin 15: A key cytokine for immunotherapy.

Authors:  Manoj Patidar; Naveen Yadav; Sarat K Dalai
Journal:  Cytokine Growth Factor Rev       Date:  2016-06-07       Impact factor: 7.638

Review 5.  Clinical and immunological manifestations of patients with atypical severe combined immunodeficiency.

Authors:  Kerstin Felgentreff; Ruy Perez-Becker; Carsten Speckmann; Klaus Schwarz; Krzysztof Kalwak; Gasper Markelj; Tadej Avcin; Waseem Qasim; E G Davies; Tim Niehues; Stephan Ehl
Journal:  Clin Immunol       Date:  2011-05-30       Impact factor: 3.969

6.  Human lymphoid development in the absence of common γ-chain receptor signaling.

Authors:  Lisa A Kohn; Christopher S Seet; Jessica Scholes; Felicia Codrea; Rebecca Chan; Sania Zaidi-Merchant; Yuhua Zhu; Satiro De Oliveira; Neena Kapoor; Ami Shah; Hisham Abdel-Azim; Donald B Kohn; Gay M Crooks
Journal:  J Immunol       Date:  2014-04-25       Impact factor: 5.422

7.  Arrested rearrangement of TCR V beta genes in thymocytes from children with X-linked severe combined immunodeficiency disease.

Authors:  J W Sleasman; T O Harville; G B White; J F George; D J Barrett; M M Goodenow
Journal:  J Immunol       Date:  1994-07-01       Impact factor: 5.422

8.  Engrafted maternal T cells in a severe combined immunodeficiency patient express T-cell receptor variable beta segments characterized by a restricted V-D-J junctional diversity.

Authors:  A Sottini; E Quiròs-Roldan; L D Notarangelo; A Malagoli; D Primi; L Imberti
Journal:  Blood       Date:  1995-04-15       Impact factor: 22.113

Review 9.  Cytokines and immunodeficiency diseases: critical roles of the gamma(c)-dependent cytokines interleukins 2, 4, 7, 9, 15, and 21, and their signaling pathways.

Authors:  Panu E Kovanen; Warren J Leonard
Journal:  Immunol Rev       Date:  2004-12       Impact factor: 12.988

Review 10.  TREC Based Newborn Screening for Severe Combined Immunodeficiency Disease: A Systematic Review.

Authors:  Jet van der Spek; Rolf H H Groenwold; Mirjam van der Burg; Joris M van Montfrans
Journal:  J Clin Immunol       Date:  2015-04-17       Impact factor: 8.317

View more
  5 in total

1.  The IL-2RG R328X nonsense mutation allows partial STAT-5 phosphorylation and defines a critical region involved in the leaky-SCID phenotype.

Authors:  A Arcas-García; M Garcia-Prat; M Magallón-Lorenz; A Martín-Nalda; O Drechsel; S Ossowski; L Alonso; J G Rivière; P Soler-Palacín; R Colobran; J Sayós; M Martínez-Gallo; C Franco-Jarava
Journal:  Clin Exp Immunol       Date:  2020-01-19       Impact factor: 4.330

2.  Family History of Early Infant Death Correlates with Earlier Age at Diagnosis But Not Shorter Time to Diagnosis for Severe Combined Immunodeficiency.

Authors:  Anderson Dik Wai Luk; Pamela P Lee; Huawei Mao; Koon-Wing Chan; Xiang Yuan Chen; Tong-Xin Chen; Jian Xin He; Nadia Kechout; Deepti Suri; Yin Bo Tao; Yong Bin Xu; Li Ping Jiang; Woei Kang Liew; Orathai Jirapongsananuruk; Tassalapa Daengsuwan; Anju Gupta; Surjit Singh; Amit Rawat; Amir Hamzah Abdul Latiff; Anselm Chi Wai Lee; Lynette P Shek; Thi Van Anh Nguyen; Tek Jee Chin; Yin Hsiu Chien; Zarina Abdul Latiff; Thi Minh Huong Le; Nguyen Ngoc Quynh Le; Bee Wah Lee; Qiang Li; Dinesh Raj; Mohamed-Ridha Barbouche; Meow-Keong Thong; Maria Carmen D Ang; Xiao Chuan Wang; Chen Guang Xu; Hai Guo Yu; Hsin-Hui Yu; Tsz Leung Lee; Felix Yat Sun Yau; Wilfred Hing-Sang Wong; Wenwei Tu; Wangling Yang; Patrick Chun Yin Chong; Marco Hok Kung Ho; Yu Lung Lau
Journal:  Front Immunol       Date:  2017-07-12       Impact factor: 7.561

3.  Gene analysis of seven cases of primary immunodeficiency.

Authors:  Ying Zhu; Li Li; Guoshun Mao; Lei Zhang; Jing Wang; Nannan Li
Journal:  Transl Pediatr       Date:  2020-04

4.  Novel Hemizygous IL2RG p.(Pro58Ser) Mutation Impairs IL-2 Receptor Complex Expression on Lymphocytes Causing X-Linked Combined Immunodeficiency.

Authors:  Elina A Tuovinen; Juha Grönholm; Tiina Öhman; Sakari Pöysti; Raine Toivonen; Anna Kreutzman; Kaarina Heiskanen; Luca Trotta; Sanna Toiviainen-Salo; John M Routes; James Verbsky; Satu Mustjoki; Janna Saarela; Juha Kere; Markku Varjosalo; Arno Hänninen; Mikko R J Seppänen
Journal:  J Clin Immunol       Date:  2020-02-19       Impact factor: 8.317

5.  Somatic Reversion of a Novel IL2RG Mutation Resulting in Atypical X-Linked Combined Immunodeficiency.

Authors:  Yujuan Hou; Hans Peter Gratz; Guillermo Ureña-Bailén; Paul G Gratz; Karin Schilbach-Stückle; Tina Renno; Derya Güngör; Daniel A Mader; Elke Malenke; Justin S Antony; Rupert Handgretinger; Markus Mezger
Journal:  Genes (Basel)       Date:  2021-12-23       Impact factor: 4.096

  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.