Characterisation of cartilage intermediate layer protein (CILP)-induced arthropathy in mice

Yao, Zihao; Nakamura, Hiroshi; Masuko, Kayo; Suzuki-Kurokawa, Manae; Nishioka, Kusuki; Kato, Tomohiro

doi:10.1136/ard.2003.008045

Cited by 16 publications

(7 citation statements)

References 27 publications

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“…We also found very specific expression of Cilp in the outermost one- to two-cell layers of the SZ. Although Cilp is known as cartilage intermediate layer protein and was initially thought to be specifically expressed in MZ (25,26,27), others have demonstrated Cilp primarily in SZ (28). Only two markers were identified for the MZ, both of which showed only moderate specificity, consistent with a transitional nature of the MZ.…”

Section: Discussionmentioning

confidence: 99%

Spatial regulation of gene expression during growth of articular cartilage in juvenile mice

et al. 2014

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Background: In juvenile mammals, the epiphyses of long bones grow by chondrogenesis within the articular cartilage. A better understanding of the molecular mechanisms that regulate the growth of articular cartilage may give insight into the antecedents of joint disease, such as osteoarthritis. Methods: We used laser-capture microdissection to isolate chondrocytes from the superficial, middle, and deep zones of growing tibial articular cartilage in the 1-wk old mouse and then investigated expression patterns by microarray. To identify molecular markers for each zone of the growing articular cartilage, we found genes showing zone-specific expression and confirmed by real-time PCR and in situ hybridization. Results: Bioinformatic analyses implicated ephrin receptor signaling, Wnt signaling, and BMP signaling in the spatial regulation of chondrocyte differentiation during growth. Molecular markers were identified for superficial (e.g. Cilp, Prg4), middle (Cxcl14, Tnn), and deep zone (Sfrp5, Frzb). Comparison between juvenile articular and growth plate cartilage revealed that the superficial-to-deep zone transition showed similarity with the hypertrophic-to-resting zone transition. Conclusion: Laser capture microdissection combined with microarray analysis identified novel signaling pathways that are spatially regulated in growing mouse articular cartilage and revealed similarities between the molecular architecture of the growing articular cartilage and that of growth plate cartilage.

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Section: Discussionmentioning

confidence: 99%

Spatial regulation of gene expression during growth of articular cartilage in juvenile mice

et al. 2014

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“…Not surprisingly, there was down-regulation of other markers of osteoblast function, including Il6st (IL-6 signal transducer), a gene that encodes the gp130 subunit of the IL-6 receptor (32) and is responsible for signal transduction in osteoblasts by IL-6 (33, 34) and other cytokines (35). The transcript exhibiting the sharpest drop coded for Cilp, cartilage intermediate layer protein, a structural protein thought to play a role in matrix disposition during endochondral bone formation (36,37).…”

Section: Fig 6 Analysis Of Selected Proteins By Western Blotmentioning

confidence: 99%

Mutation of Prkar1a Causes Osteoblast Neoplasia Driven by Dysregulation of Protein Kinase A

Pavel¹,

Nadella²,

Towns³

et al. 2008

Molecular Endocrinology

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Carney complex (CNC) is an autosomal dominant neoplasia syndrome caused by inactivating mutations in PRKAR1A, the gene encoding the type 1A regulatory subunit of protein kinase A (PKA). This genetic defect induces skin pigmentation, endocrine tumors, myxomas, and schwannomas. Some patients with the complex also develop myxoid bone tumors termed osteochondromyxomas. To study the link between the PRKAR1A mutations and tumor formation, we generated a mouse model of this condition. Prkar1a(+/-) mice develop bone tumors with high frequency, although these lesions have not yet been characterized, either from human patients or from mice. Bone tumors from Prkar1a(+/-) mice were heterogeneous, including elements of myxomatous, cartilaginous, and bony differentiation that effaced the normal bone architecture. Immunohistochemical analysis identified an osteoblastic origin for the abnormal cells associated with islands of bone. To better understand these cells at the biochemical level, we isolated primary cultures of tumoral bone and compared them with cultures of bone from wild-type animals. The tumor cells exhibited the expected decrease in Prkar1a protein and exhibited increased PKA activity. At the phenotypic level, we observed that tumor cells behaved as incompletely differentiated osteoblasts and were able to form tumors in immunocompromised mice. Examination of gene expression revealed down-regulation of markers of bone differentiation and increased expression of locally acting growth factors, including members of the Wnt signaling pathway. Tumor cells exhibited enhanced growth in response to PKA-stimulating agents, suggesting that tumorigenesis in osteoblast precursor cells is driven by effects directly mediated by the dysregulation of PKA.

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“…Downregulation of other markers includes Il6st (interleukin-6 signal transducer, MGI:96560), a gene that codes for gp130 subunit of the IL-6 receptor [47]. However, the transcript with the most significant drop is CILP (cartilage intermediate layer protein, MGI:2444507), a structural protein that plays a role in matrix composition and bone formation [48], [49].…”

Section: Geneticsmentioning

confidence: 99%

Osteochondromyxoma: Review of a rare carney complex criterion

Golden

Siordia

2016

Journal of Bone Oncology

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Osteochondromyxoma is an extremely rare bone tumor associated with 1% of Carney complex patients and constitutes one of its 11 diagnostic criteria. This narrative review of osteochondromyxoma is based on a search of all references to the topic in PubMed, Web Of Science, SCOPUS, ScienceDirect, and JSTOR databases. Special attention was focused on case reports, leading to a review encompassing the case reports to date, as well as related animal model studies. This review covers the current understanding of osteochondromyxoma, highlighting its variability while providing consensus on the most common clinical presentation, pathological findings, and genetic features of this rare bone tumor.

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Characterisation of cartilage intermediate layer protein (CILP)-induced arthropathy in mice

Cited by 16 publications

References 27 publications

Spatial regulation of gene expression during growth of articular cartilage in juvenile mice

Spatial regulation of gene expression during growth of articular cartilage in juvenile mice

Mutation of Prkar1a Causes Osteoblast Neoplasia Driven by Dysregulation of Protein Kinase A

Osteochondromyxoma: Review of a rare carney complex criterion

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