Labeling of heterochronic ribosomes reveals C1ORF109 and SPATA5 control a late step in human ribosome assembly

Chunyang Ni, Daniel A. Schmitz, Jeon Lee, Krzysztof Pawłowski, Jun Wu, Michael Buszczak

Research output: Contribution to journalArticlepeer-review

2 Scopus citations


Although features of ribosome assembly are shared between species, our understanding of the diversity, complexity, dynamics, and regulation of ribosome production in multicellular organisms remains incomplete. To gain insights into ribosome biogenesis in human cells, we perform a genome-wide loss-of-function screen combined with differential labeling of pre-existing and newly assembled ribosomes. These efforts identify two functionally uncharacterized genes, C1orf109 and SPATA5. We provide evidence that these factors, together with CINP and SPATA5L1, control a late step of human pre-60S maturation in the cytoplasm. Loss of either C1orf109 or SPATA5 impairs global protein synthesis. These results link ribosome assembly with neurodevelopmental disorders associated with recessive SPATA5 mutations. Based on these findings, we propose that the expanded repertoire of ribosome biogenesis factors likely enables multicellular organisms to coordinate multiple steps of ribosome production in response to different developmental and environmental stimuli.

Original languageEnglish (US)
Article number110597
JournalCell Reports
Issue number13
StatePublished - Mar 29 2022


  • C1ORF109
  • CINP
  • CP: Molecular biology
  • microcephaly
  • mRNA translation
  • Ribosome biogenesis
  • RSL24D1
  • SPATA5
  • SPATA5L1

ASJC Scopus subject areas

  • Biochemistry, Genetics and Molecular Biology(all)


Dive into the research topics of 'Labeling of heterochronic ribosomes reveals C1ORF109 and SPATA5 control a late step in human ribosome assembly'. Together they form a unique fingerprint.

Cite this