Literature DB >> 20979174

A child with severe form of dyskeratosis congenita and TINF2 mutation of shelterin complex.

Nazan Sarper1, Emine Zengin, Suar Çakı Kılıç.   

Abstract

A 26-month-old male presented with bone marrow failure and dystrophic nail lesions mimicking onychomycosis. There was no skin finding. Treatment with androgen and methylprednisolone was started due to unavailability of a matched-related hematopoietic stem cell donor. After 30 months, transfusion support was required. TINF2 mutation was identified at the age of five and dyskeratosis congenita (DC) was confirmed. TIN2 mutation analysis must be carried out in patients younger than 10 years presenting with bone marrow failure even if characteristic physical anomalies of DC is missing. Genetic confirmation of DC prevents ineffective immunotherapy with misdiagnosis of acquired aplastic anemia.

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Year:  2010        PMID: 20979174     DOI: 10.1002/pbc.22624

Source DB:  PubMed          Journal:  Pediatr Blood Cancer        ISSN: 1545-5009            Impact factor:   3.167


  12 in total

1.  Three novel truncating TINF2 mutations causing severe dyskeratosis congenita in early childhood.

Authors:  G S Sasa; A Ribes-Zamora; N D Nelson; A A Bertuch
Journal:  Clin Genet       Date:  2011-04-07       Impact factor: 4.438

Review 2.  Genetics of human telomere biology disorders.

Authors:  Patrick Revy; Caroline Kannengiesser; Alison A Bertuch
Journal:  Nat Rev Genet       Date:  2022-09-23       Impact factor: 59.581

3.  Isolation of chromatin from dysfunctional telomeres reveals an important role for Ring1b in NHEJ-mediated chromosome fusions.

Authors:  Cristina Bartocci; Jolene K Diedrich; Iliana Ouzounov; Julia Li; Andrea Piunti; Diego Pasini; John R Yates; Eros Lazzerini Denchi
Journal:  Cell Rep       Date:  2014-05-09       Impact factor: 9.423

Review 4.  The shelterin complex and hematopoiesis.

Authors:  Morgan Jones; Kamlesh Bisht; Sharon A Savage; Jayakrishnan Nandakumar; Catherine E Keegan; Ivan Maillard
Journal:  J Clin Invest       Date:  2016-05-02       Impact factor: 14.808

Review 5.  Dysfunctional telomeres and hematological disorders.

Authors:  Elena Fiorini; Andrea Santoni; Simona Colla
Journal:  Differentiation       Date:  2018-01-04       Impact factor: 3.880

Review 6.  An update on the biology and management of dyskeratosis congenita and related telomere biology disorders.

Authors:  Marena R Niewisch; Sharon A Savage
Journal:  Expert Rev Hematol       Date:  2019-09-10       Impact factor: 2.819

7.  Hematopoietic stem cells are acutely sensitive to Acd shelterin gene inactivation.

Authors:  Morgan Jones; Gail Osawa; Joshua A Regal; Daniel N Weinberg; James Taggart; Hande Kocak; Ann Friedman; David O Ferguson; Catherine E Keegan; Ivan Maillard
Journal:  J Clin Invest       Date:  2013-12-09       Impact factor: 19.456

8.  A TIN2 dyskeratosis congenita mutation causes telomerase-independent telomere shortening in mice.

Authors:  David Frescas; Titia de Lange
Journal:  Genes Dev       Date:  2014-01-15       Impact factor: 11.361

9.  TRF1 negotiates TTAGGG repeat-associated replication problems by recruiting the BLM helicase and the TPP1/POT1 repressor of ATR signaling.

Authors:  Michal Zimmermann; Tatsuya Kibe; Shaheen Kabir; Titia de Lange
Journal:  Genes Dev       Date:  2014-10-24       Impact factor: 12.890

10.  Binding of TPP1 protein to TIN2 protein is required for POT1a,b protein-mediated telomere protection.

Authors:  David Frescas; Titia de Lange
Journal:  J Biol Chem       Date:  2014-07-23       Impact factor: 5.486

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