Literature DB >> 8211379

Sensitivity of Roberts syndrome cells to gamma radiation, mitomycin C, and protein synthesis inhibitors.

D J Van den Berg1, U Francke.   

Abstract

Roberts syndrome (RS) is a rare autosomal recessive disorder characterized by pre- and postnatal growth retardation, limb reduction abnormalities, and craniofacial anomalies. Mitotic chromosomes from RS individuals display repulsion of heterochromatin regions or centromere splitting, leading to a railroad-track appearance of mitotic chromosomes. Abnormalities in metaphase duration, anaphase progression, nuclear morphology, and increased frequency of micronucleation have been reported in RS cells. Cells from RS heterozygotes are normal in these respects, and in vitro complementation of the defects in somatic cell hybrids has been reported. Therefore, in preparation for the isolation of cDNAs that complement the RS defect, we investigated various drug treatments to identify an agent that specifically involves the growth of RS cells. Based on the cytogenetic and cell biologic findings, we chose agents that increase micronucleation or inhibit protein synthesis. We found that RS cells are hypersensitive to gamma radiation, mitomycin C, G418 and hygromycin B, but not to colcemid or streptonigrin when compared to normal cells. DNA content and cell viability analysis confirmed that the sensitivity to gamma irradiation was primarily due to increased cell death.

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Year:  1993        PMID: 8211379     DOI: 10.1007/bf01232749

Source DB:  PubMed          Journal:  Somat Cell Mol Genet        ISSN: 0740-7750


  7 in total

1.  2012 William Allan Award: Adventures in cytogenetics.

Authors:  Uta Francke
Journal:  Am J Hum Genet       Date:  2013-03-07       Impact factor: 11.025

2.  Mapping of a single locus capable of complementing the defective heterochromatin phenotype of Roberts syndrome cells.

Authors:  Lisa D McDaniel; Darrell J Tomkins; Eric J Stanbridge; Martin J Somerville; Errol C Friedberg; Roger A Schultz
Journal:  Am J Hum Genet       Date:  2005-05-10       Impact factor: 11.025

3.  Inactivating mutations in ESCO2 cause SC phocomelia and Roberts syndrome: no phenotype-genotype correlation.

Authors:  Birgitt Schüle; Angelica Oviedo; Kathreen Johnston; Shashidhar Pai; Uta Francke
Journal:  Am J Hum Genet       Date:  2005-10-31       Impact factor: 11.025

Review 4.  Can corruption of chromosome cohesion create a conduit to cancer?

Authors:  Huiling Xu; Jonathan M Tomaszewski; Michael J McKay
Journal:  Nat Rev Cancer       Date:  2011-02-17       Impact factor: 60.716

5.  Diagnostic Overlap between Fanconi Anemia and the Cohesinopathies: Roberts Syndrome and Warsaw Breakage Syndrome.

Authors:  Petra van der Lelij; Anneke B Oostra; Martin A Rooimans; Hans Joenje; Johan P de Winter
Journal:  Anemia       Date:  2010-07-18

6.  Cohesin mediates Esco2-dependent transcriptional regulation in a zebrafish regenerating fin model of Roberts Syndrome.

Authors:  Rajeswari Banerji; Robert V Skibbens; M Kathryn Iovine
Journal:  Biol Open       Date:  2017-12-15       Impact factor: 2.422

7.  The cellular phenotype of Roberts syndrome fibroblasts as revealed by ectopic expression of ESCO2.

Authors:  Petra van der Lelij; Barbara C Godthelp; Wouter van Zon; Djoke van Gosliga; Anneke B Oostra; Jûrgen Steltenpool; Jan de Groot; Rik J Scheper; Rob M Wolthuis; Quinten Waisfisz; Firouz Darroudi; Hans Joenje; Johan P de Winter
Journal:  PLoS One       Date:  2009-09-07       Impact factor: 3.240

  7 in total

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