Articular cartilage and growth plate defects are associated with chondrocyte cytoskeletal abnormalities in Tg737orpk mice lacking the primary cilia protein polaris

McGlashan, Susan R.; Haycraft, CJ; Jensen, Cynthia G.; Yoder, BK; Poole, CA

doi:10.1016/j.matbio.2006.12.003

Cited by 101 publications

(126 citation statements)

References 62 publications

(90 reference statements)

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“…Disturbance of the intraflagellar transport system leads to a depletion of cells with primary cilia and defects in the growth plate such as disruption of cell shape and lack of columnar orientation, which suggests defects in cell movement/rotation. 3,11,12 Figure 3 Electron micrographs of the primary cilium and the centrioles. The cilium is observed with a similar ultrastructure in the cells of the growth plate (a-c) and osteochondroma (d-f).…”

Section: Discussionmentioning

confidence: 99%

“…Primary cilia have been implicated as recipients of some signaling pathways essential for the regulation of the growth plate, such as the non-canonical Wnt and Indian Hedgehog pathways. 3,[11][12][13][14] KIF3A deficiency has been shown to cause abnormal topography of Hedgehog signaling and growth plate dysfunction. 15 In several tissues, the organization of primary cilia has been shown to reflect cell polarity.…”

mentioning

confidence: 99%

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Primary cilia organization reflects polarity in the growth plate and implies loss of polarity and mosaicism in osteochondroma

Andrea

Wiweger

Prins

et al. 2010

Laboratory Investigation

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Primary cilia are specialized cell surface projections found on most cell types. Involved in several signaling pathways, primary cilia have been reported to modulate cell and tissue organization. Although they have been implicated in regulating cartilage and bone growth, little is known about the organization of primary cilia in the growth plate cartilage and osteochondroma. Osteochondromas are bone tumors formed along the growth plate, and they are caused by mutations in EXT1 or EXT2 genes. In this study, we show the organization of primary cilia within and between the zones of the growth plate and osteochondroma. Using confocal and electron microscopy, we found that in both tissues, primary cilia have a similar formation but a distinct organization. The shortest ciliary length is associated with the proliferative state of the cells, as confirmed by Ki-67 immunostaining. Primary cilia organization in the growth plate showed that nonpolarized chondrocytes (resting zone) are becoming polarized (proliferating and hypertrophic zones), orienting the primary cilia parallel to the longitudinal axis of the bone. The alignment of primary cilia forms one virtual axis that crosses the center of the columns of chondrocytes reflecting the polarity axis of the growth plate. We also show that primary cilia in osteochondromas are found randomly located on the cell surface. Strikingly, the growth plate-like polarity was retained in sub-populations of osteochondroma cells that were organized into small columns. Based on this, we propose the existence of a mixture ('mosaic') of normal lining (EXT þ /À or EXT wt/wt ) and EXT À/À cells in the cartilaginous cap of osteochondromas.

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Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

Primary cilia organization reflects polarity in the growth plate and implies loss of polarity and mosaicism in osteochondroma

Andrea

Wiweger

Prins

et al. 2010

Laboratory Investigation

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“…Tg737 orpk (orpk) mice carry a hypomorphic mutation in IFT88. In orpk mice, primary cilia are significantly shorter than wild-type cilia and are often difficult to detect (McGlashan et al 2007). The growth plate of orpk mice is significantly smaller.…”

Section: Primary Ciliamentioning

confidence: 99%

Developmental Regulation of the Growth Plate and Cranial Synchondrosis

Wei

Mishina

et al. 2016

J Dent Res

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Long bones and the cranial base are both formed through endochondral ossification. Elongation of long bones is primarily through the growth plate, which is a cartilaginous structure at the end of long bones made up of chondrocytes. Growth plate chondrocytes are organized in columns along the longitudinal axis of bone growth. The cranial base is the growth center of the neurocranium. Synchondroses, consisting of mirror-image growth plates, are critical for cranial base elongation and development. Over the last decade, considerable progress has been made in determining the roles of the parathyroid hormone-related protein, Indian hedgehog, fibroblast growth factor, bone morphogenetic protein, and Wnt signaling pathways in various aspects of skeletal development. Furthermore, recent evidence indicates the important role of the primary cilia signaling pathway in bone elongation. Here, we review the development of the growth plate and cranial synchondrosis and the regulation by the above-mentioned signaling pathways, highlighting the similarities and differences between these 2 structures.

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“…11,46 Interestingly, the organization of proliferative chondrocytes is also evident at the subcellular level in the planar alignment of the primary cilium of each chondrocyte at the center of the column. 47 Thus, as in the limb bud, cartilage growth is associated with planar alignment of cell axes, oriented cell division and oriented cell movements.…”

confidence: 99%

Cell polarity

Romereim

Dudley

2011

Organogenesis

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Articular cartilage and growth plate defects are associated with chondrocyte cytoskeletal abnormalities in Tg737orpk mice lacking the primary cilia protein polaris

Cited by 101 publications

References 62 publications

Primary cilia organization reflects polarity in the growth plate and implies loss of polarity and mosaicism in osteochondroma

Primary cilia organization reflects polarity in the growth plate and implies loss of polarity and mosaicism in osteochondroma

Developmental Regulation of the Growth Plate and Cranial Synchondrosis

Cell polarity

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