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Updated: Feb 16, 2026

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Constitutive Cyclin O deficiency results in penetrant hydrocephalus, impaired growth and infertility

Marc Núnez-Ollé1, Carole Jung2, Berta Terré3

  • 1Apoptosis Signalling Group, IMIM (Institut Hospital del Mar d'Investigacions Mèdiques), Barcelona, Spain.

Oncotarget
|December 17, 2017
PubMed

Insights

Constitutive loss of Cyclin O (CCNO) in mice causes growth impairment, infertility, and hydrocephalus. This CCNO gene is crucial for normal development and may be linked to human infertility and hydrocephalus.

Area of Science:

  • Developmental Biology
  • Genetics
  • Cell Biology

Background:

  • Cyclin O (CCNO) is a cyclin family member regulating ciliogenesis and apoptosis.
  • CCNO mutations cause Reduced Generation of Multiple Motile Cilia (RGMC) in humans.
  • Previous mouse models showed CCNO's role in motile cilia development and hydrocephalus.

Purpose of the Study:

  • To investigate the in vivo functions of CCNO using a constitutive knockout mouse model.
  • To characterize the developmental and aging phenotypes associated with complete CCNO loss.

Main Methods:

  • Generation of a constitutive CCNO knockout mouse model (Ccno-/-).
  • Phenotypic analysis of Ccno-/- mice during aging, including growth, CNS, and reproductive assessments.
  • Histological examination of cilia in female reproductive tracts.

Main Results:

  • Ccno-/- mice exhibited growth impairment and high-penetrance hydrocephalus.
  • Additional CNS defects, including cortical thinning and hippocampal abnormalities, were observed.
  • Both male and female Ccno-/- mice were infertile, with females showing reduced oviductal cilia.

Conclusions:

  • CCNO is essential for normal development, impacting CNS formation, cilia function, and fertility.
  • Constitutive CCNO loss leads to severe developmental defects and infertility in mice.
  • Heterozygous CCNO mutations may contribute to hydrocephalus and diminished fertility in humans.
Keywords:
Cyclin OGerotargetciliogenesisdevelopmenthydrocephalusneurogenesis

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