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Centrioles and Centrosomes01:13

Centrioles and Centrosomes

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Most animal cells comprise a pair of centrioles together called a centrosome. The cell duplicates its centrosome and contains two centrosomes side-by-side, which begin to move apart during the prophase. As the centrosomes migrate to two different sides of the cell, microtubules start extending from each centrosome toward the other end. The mitotic spindle is composed of the centrosomes and their emerging microtubules.
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The primary microtubule organizing center (MTOC) in animal cells is the centrosome. A centrosome has two cylindrical centrioles at its core. Each centriole consists of nine sets of three microtubules held together by proteins. The centrioles are positioned at right angles to each other and surrounded by a shapeless protein cloud called the pericentriolar matrix, or pericentriolar material (PCM).
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The primary cilium, made up of microtubules, acts as antennae on the cell surfaces for relaying external stimuli into the cells. These fine hair-like structures are present, generally one per cell. These are non-motile cilia in a 9+0 microtubules arrangement, where the central pair of microtubules are absent. The primary cilia arise from the basal body embedded in the cell membrane. Intraflagellar transport (IFT) carries requisite proteins from the cytoplasm to the cilium because the primary...
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The cytoskeleton is an essential cell component that plays several structural and functional roles. However, the filaments that make up the cytoskeleton cannot function independently and depend on the accessory or ancillary proteins to effectively carry out their function. Accessory proteins associate with cytoskeletal filaments and their monomers, aiding filament formation and function. They also help in the cross-communication among cytoskeletal filaments. Cytoskeletal accessory proteins are...
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Histone variants are the histone proteins with structural and sequence variations. These variants may be regarded as “mutant” forms that replace their canonical histone counterparts in the nucleosomes. Specific post-translational modifications on the histone variants enable further chromatin complexity and regulate tissue-specific gene expression. The most common histone variants are from histone H2A, H2B, and linker histone H1 families. However, several variants of histone H3...
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Catenins are characterized by multiple binding domains and dynamic structures that allow them to function as linker proteins in cell junction complexes. All catenins, except α-catenin, contain a characteristic protein sequence called the armadillo repeat and are therefore also called armadillo proteins.
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Related Experiment Video

Updated: Oct 30, 2025

Quantitative Immunofluorescence Assay to Measure the Variation in Protein Levels at Centrosomes
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Centriolar Protein C2cd3 Is Required for Craniofacial Development.

Ching-Fang Chang1,2, Kari M Brown1,2, Yanfen Yang1,2

  • 1Division of Developmental Biology, Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, United States.

Frontiers in Cell and Developmental Biology
|July 2, 2021
PubMed
Summary

Investigating C2cd3, a protein crucial for primary cilia, revealed its role in craniofacial development. Mouse models showed C2cd3 mutations cause ciliopathies, with specific domains impacting embryonic development and craniofacial structures.

Keywords:
C2cd3ciliopathiescraniofacial developmentneural crestprimary cilia

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Area of Science:

  • Cell Biology
  • Developmental Biology
  • Genetics

Background:

  • Primary cilia are vital microtubule-based organelles; their dysfunction causes ciliopathies.
  • C2 domain containing 3 (C2cd3) is essential for ciliogenesis, and its mutations link to Oral-Facial-Digital syndrome type 14 (OFD14).

Purpose of the Study:

  • To investigate the role of C2cd3 in ciliogenesis and craniofacial development using murine models.
  • To understand the molecular mechanisms underlying C2cd3-related ciliopathies.

Main Methods:

  • Generation of multiple murine models targeting C2cd3, including a conditional allelic series.
  • Analysis of tissue-specific C2cd3 isoforms and domain-specific functions.
  • Evaluation of C2cd3 expression patterns during embryonic development.

Main Results:

  • Loss of C2cd3 in mice resulted in developmental abnormalities including exencephaly and pericardial edema.
  • Genetic background influenced phenotypic severity, highlighting its role in ciliopathy variation.
  • Specific C2cd3 domains (N-terminal C2CD3N-C2) were critical for both overall embryonic development and craniofacial development.

Conclusions:

  • C2cd3 is essential for embryonic development and ciliogenesis, with distinct domains playing critical roles.
  • Understanding C2cd3 function and mutation effects provides insights into craniofacial ciliopathies like OFD14.
  • Novel murine models offer a platform for future research into ciliopathy pathogenesis.