Abnormalities of developmental cell death in Dad1-deficient mice

Kiyomasa Nishii, Teruhisa Tsuzuki, Madoka Kumai, Naoki Takeda, Hideya Koga, Shinichi Aizawa, Takeharu Nishimoto, Yosaburo Shibata

Research output: Contribution to journalArticlepeer-review

39 Citations (Scopus)


Background: Dad1, the defender against apoptotic cell death, comprises the oligosaccharyltransferase complex and is well conserved among eukaryotes. In hamster BHK21-derived tsBN7 cells, loss of Dad1 causes apoptosis which cannot be prevented by Bcl-2. Results: To determine the role of Dad1 function in vivo, we prepared by gene targeting, mice harbouring a disrupted Dad1 gene. Homozygous mutants died shortly after they were implanted with the characteristic features of apoptosis. In an in vitro blastocyst culture system, Dad1-null cells displayed abnormalities which were comparable to those obtained in vivo. However, oligosaccharyltransferase activity was apparently retained even after the Dad1-null cells were destined to die. Some live-born heterozygous mutants displayed soft-tissue syndactyly. Mild thymic hypoplasia was also indicated in heterozygotes. Conclusion: These results suggest the involvement of the Dad1 gene in the acquisition of a common syndactyly phenotype, as well as in the control of programmed cell death during development.

Original languageEnglish
Pages (from-to)243-252
Number of pages10
JournalGenes to Cells
Issue number4
Publication statusPublished - 1999

All Science Journal Classification (ASJC) codes

  • Genetics
  • Cell Biology


Dive into the research topics of 'Abnormalities of developmental cell death in Dad1-deficient mice'. Together they form a unique fingerprint.

Cite this