Literature DB >> 19036115

Exogenous TERC alone can enhance proliferative potential, telomerase activity and telomere length in lymphocytes from dyskeratosis congenita patients.

Michael Kirwan1, Richard Beswick, Tom Vulliamy, Amit C Nathwani, Amanda J Walne, Colin Casimir, Inderjeet Dokal.   

Abstract

Dyskeratosis congenita (DC) is an inherited multi-system disorder characterised by muco-cutaneous abnormalities, bone marrow failure and a predisposition to malignancy. Bone marrow failure is the principal cause of mortality and is thought to be the result of premature cell death in the haematopoietic compartment because DC cells age prematurely and tend to have short telomeres. DC is genetically heterogeneous and patients have mutations in genes that encode components of the telomerase complex (DKC1, TERC, TERT, NOP10 and NHP2), and telomere shelterin complex (TINF2), both important in telomere maintenance. Here, we transduced primary T lymphocytes and B lymphocyte lines established from patients with TERC and DKC1 mutations with wild type TERC-bearing lentiviral vectors. We found that transduction with exogenous TERC alone was capable of increasing telomerase activity in mutant T lymphocytes and B lymphocyte lines and improved the survival and thus overall growth of B-lymphocyte lines over a prolonged period, regardless of their disease mutation. Telomeres in TERC-treated lines were longer than in the untreated cultures. This is the first study of its kind in DC lymphocytes and the first to demonstrate that transduction with TERC alone can improve cell survival and telomere length without the need for exogenous TERT.

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Year:  2008        PMID: 19036115     DOI: 10.1111/j.1365-2141.2008.07516.x

Source DB:  PubMed          Journal:  Br J Haematol        ISSN: 0007-1048            Impact factor:   6.998


  18 in total

1.  Small-Molecule PAPD5 Inhibitors Restore Telomerase Activity in Patient Stem Cells.

Authors:  Neha Nagpal; Jianing Wang; Jing Zeng; Emily Lo; Diane H Moon; Kevin Luk; Roman O Braun; Lauri M Burroughs; Sioban B Keel; Christopher Reilly; R Coleman Lindsley; Scot A Wolfe; Albert K Tai; Patrick Cahan; Daniel E Bauer; Yick W Fong; Suneet Agarwal
Journal:  Cell Stem Cell       Date:  2020-04-21       Impact factor: 24.633

2.  Poly(A)-specific ribonuclease (PARN) mediates 3'-end maturation of the telomerase RNA component.

Authors:  Diane H Moon; Matthew Segal; Baris Boyraz; Eva Guinan; Inga Hofmann; Patrick Cahan; Albert K Tai; Suneet Agarwal
Journal:  Nat Genet       Date:  2015-10-19       Impact factor: 38.330

3.  Posttranscriptional manipulation of TERC reverses molecular hallmarks of telomere disease.

Authors:  Baris Boyraz; Diane H Moon; Matthew Segal; Maud Z Muosieyiri; Asli Aykanat; Albert K Tai; Patrick Cahan; Suneet Agarwal
Journal:  J Clin Invest       Date:  2016-08-02       Impact factor: 14.808

Review 4.  Recent progress in dyskeratosis congenita.

Authors:  Nobuhiro Nishio; Seiji Kojima
Journal:  Int J Hematol       Date:  2010-10-01       Impact factor: 2.490

5.  A common variant in the telomerase RNA component is associated with short telomere length.

Authors:  Omer T Njajou; Elizabeth H Blackburn; Ludmila Pawlikowska; Massimo Mangino; Coleen M Damcott; Pui-Yan Kwok; Timothy D Spector; Anne B Newman; Tamara B Harris; Steven R Cummings; Richard M Cawthon; Alan R Shuldiner; Ana M Valdes; Wen-Chi Hsueh
Journal:  PLoS One       Date:  2010-09-27       Impact factor: 3.240

Review 6.  InTERTpreting telomerase structure and function.

Authors:  Haley D M Wyatt; Stephen C West; Tara L Beattie
Journal:  Nucleic Acids Res       Date:  2010-05-11       Impact factor: 16.971

7.  Telomerase reverse transcriptase-dependent telomere equilibration mitigates tissue dysfunction in mTert heterozygotes.

Authors:  Marie Meznikova; Natalie Erdmann; Rich Allsopp; Lea A Harrington
Journal:  Dis Model Mech       Date:  2009-10-19       Impact factor: 5.758

Review 8.  Advances in the understanding of dyskeratosis congenita.

Authors:  Amanda J Walne; Inderjeet Dokal
Journal:  Br J Haematol       Date:  2009-02-04       Impact factor: 6.998

9.  Too short stature, too many stigmata.

Authors:  Lydia Kossiva; George Vartzelis; Marieta Harisi; Ioanna Logotheti; Anastasia Garoufi
Journal:  BMJ Case Rep       Date:  2010-11-18

10.  Proliferative defects in dyskeratosis congenita skin keratinocytes are corrected by expression of the telomerase reverse transcriptase, TERT, or by activation of endogenous telomerase through expression of papillomavirus E6/E7 or the telomerase RNA component, TERC.

Authors:  Francoise A Gourronc; mckaylee M Robertson; Annie K Herrig; Peter M Lansdorp; Frederick D Goldman; Aloysius J Klingelhutz
Journal:  Exp Dermatol       Date:  2009-06-23       Impact factor: 3.960

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