Literature DB >> 1887849

Chromosome instability and X-ray hypersensitivity in a microcephalic and growth-retarded child.

G Barbi1, J M Scheres, D Schindler, R D Taalman, K Rodens, K Mehnert, M Müller, H Seyschab.   

Abstract

We report on a microcephalic, growth-retarded newborn girl without major anomalies who has chromosome instability in lymphocytes and fibroblasts. Frequent involvement of bands 7p13, 7q34, 14q11, and 14q32 suggested the diagnosis of ataxia telangiectasia (AT) or a related disorder. Supportive evidence was radioresistant DNA synthesis in fibroblasts and radiation hypersensitivity of short-term lymphocyte cultures. Follow-up for nearly 4 years showed largely normal development, and no signs of telangiectasia, ataxia, or immunodeficiency. Serum AFP levels turned from elevated at age 5 months to normal at age 2 years. We propose that our patient belongs to the expanding category of "AT-related" genetic disorders, probably to the Nijmegen breakage syndrome.

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Year:  1991        PMID: 1887849     DOI: 10.1002/ajmg.1320400109

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  18 in total

1.  A variant of the Nijmegen breakage syndrome with unusual cytogenetic features and intermediate cellular radiosensitivity.

Authors:  R Tupler; G L Marseglia; M Stefanini; E Prosperi; L Chessa; T Nardo; A Marchi; P Maraschio
Journal:  J Med Genet       Date:  1997-03       Impact factor: 6.318

2.  Severe intrauterine growth retardation with increased mitomycin C sensitivity, or Nijmegen breakage syndrome?

Authors:  V M Der Kaloustian; A M Elliott; P Eydoux
Journal:  J Med Genet       Date:  1995-12       Impact factor: 6.318

3.  ATM protein-dependent phosphorylation of Rad50 protein regulates DNA repair and cell cycle control.

Authors:  Magtouf Gatei; Burkhard Jakob; Philip Chen; Amanda W Kijas; Olivier J Becherel; Nuri Gueven; Geoff Birrell; Ji-Hoon Lee; Tanya T Paull; Yaniv Lerenthal; Shazrul Fazry; Gisela Taucher-Scholz; Reinhard Kalb; Detlev Schindler; Regina Waltes; Thilo Dörk; Martin F Lavin
Journal:  J Biol Chem       Date:  2011-07-14       Impact factor: 5.157

Review 4.  Mechanisms of DNA-protein crosslink repair.

Authors:  Julian Stingele; Roberto Bellelli; Simon J Boulton
Journal:  Nat Rev Mol Cell Biol       Date:  2017-06-28       Impact factor: 94.444

Review 5.  Nijmegen breakage syndrome.

Authors:  I van der Burgt; K H Chrzanowska; D Smeets; C Weemaes
Journal:  J Med Genet       Date:  1996-02       Impact factor: 6.318

Review 6.  Nijmegen breakage syndrome. The International Nijmegen Breakage Syndrome Study Group.

Authors: 
Journal:  Arch Dis Child       Date:  2000-05       Impact factor: 3.791

7.  G2-phase radiation response in lymphoblastoid cell lines from Nijmegen breakage syndrome.

Authors:  A Antoccia; A di Masi; P Maraschio; M Stumm; R Ricordy; C Tanzarella
Journal:  Cell Prolif       Date:  2002-04       Impact factor: 6.831

8.  Human RAD50 deficiency in a Nijmegen breakage syndrome-like disorder.

Authors:  Regina Waltes; Reinhard Kalb; Magtouf Gatei; Amanda W Kijas; Markus Stumm; Alexandra Sobeck; Britta Wieland; Raymonda Varon; Yaniv Lerenthal; Martin F Lavin; Detlev Schindler; Thilo Dörk
Journal:  Am J Hum Genet       Date:  2009-04-30       Impact factor: 11.025

9.  Simultaneous measurement, using flow cytometry, of radiosensitivity and defective mitogen response in ataxia telangiectasia and related syndromes.

Authors:  H Seyschab; D Schindler; R Friedl; G Barbi; E Boltshauser; J P Fryns; F Hanefeld; R Korinthenberg; I Krägeloh-Mann; J M Scheres
Journal:  Eur J Pediatr       Date:  1992-10       Impact factor: 3.183

10.  Severe microcephaly with normal intellectual development: the Nijmegen breakage syndrome.

Authors:  A J Green; J R Yates; A M Taylor; P Biggs; G M McGuire; C M McConville; C J Billing; N D Barnes
Journal:  Arch Dis Child       Date:  1995-11       Impact factor: 3.791

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