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Loss of lysyl oxidase-like 3 causes cleft palate and spinal deformity in mice.

Zhang J, Yang R, Liu Z, Hou C, Zong W, Zhang A, Sun X, Gao J - Hum. Mol. Genet. (2015)

Bottom Line: Failure of any one of these processes can result in embryonic malformation.We found that the obvious decrease of collagen cross-links in palate and spine that was induced by the lack of LOXL3 resulted in cleft palate and spinal deformity.The Loxl3 gene may be a candidate disease gene resulting in cleft palate and spinal deformity.

View Article: PubMed Central - PubMed

Affiliation: Institute of Developmental Biology, School of Life Science, Shandong University, 27 Shanda Nanlu, Jinan 250100, China.

No MeSH data available.


Related in: MedlinePlus

Collagen defect in the cartilage primordia of the thoracic vertebrae of Loxl3−/− fetuses (Sirius red staining). (A and B) In LOXL3 knockout mice at E14.5, chondrocytes in the cartilage primordia were loosely organized because of a reduction in the collagen fibres around the chondrocytes. (C and D) Collagen fibres around the columnar chondrocytes in LOXL3 knockout mice were still sparse at E16.5. (E) Whether at E14.5 or at E16.5, the collagen fibre density around the chondrocytes in Loxl3−/− mice was significantly less than that in Loxl3+/+ mice. **P < 0.01. Blue arrows: collagen fibres. Left bar: 100 μm. Right bar: 10 μm.
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DDV333F9: Collagen defect in the cartilage primordia of the thoracic vertebrae of Loxl3−/− fetuses (Sirius red staining). (A and B) In LOXL3 knockout mice at E14.5, chondrocytes in the cartilage primordia were loosely organized because of a reduction in the collagen fibres around the chondrocytes. (C and D) Collagen fibres around the columnar chondrocytes in LOXL3 knockout mice were still sparse at E16.5. (E) Whether at E14.5 or at E16.5, the collagen fibre density around the chondrocytes in Loxl3−/− mice was significantly less than that in Loxl3+/+ mice. **P < 0.01. Blue arrows: collagen fibres. Left bar: 100 μm. Right bar: 10 μm.

Mentions: Mature collagen fibres consist of multiple collagen fibrils. Collagen fibrils are formed from multiple tropocollagen molecules through covalent cross-linking catalysed by LOXs. Thus we examined the collagen fibres in palate shelves and cartilage primordia of the thoracic vertebrae. Masson's trichrome staining showed that collagen fibres in Loxl3−/− palate shelves compared with Loxl3+/+ palate shelves were significantly decreased at E14.5 (P < 0.01, Fig. 8). The thoracic vertebrae of Loxl3−/−mice started bending at E14.5, and this phenotype became very obvious around E16.5. Sagittal sections and Sirius red staining were used to check the collagen fibres in the thoracic vertebrae at E14.5 and E16.5. The chondrocytes in the cartilage primordia of the thoracic vertebrae were more dispersed in Loxl3−/−mice than in wild-type mice at E14.5 (Fig. 9A and B). Sparse collagen fibres around the columnar chondrocytes were also observed in Loxl3−/− mice at E16.5 (Fig. 9C and D). The amount of collagen fibres around the chondrocytes was significantly decreased in Loxl3−/−mice at E14.5 or E16.5 (P < 0.01, Fig. 9E).Figure 8.


Loss of lysyl oxidase-like 3 causes cleft palate and spinal deformity in mice.

Zhang J, Yang R, Liu Z, Hou C, Zong W, Zhang A, Sun X, Gao J - Hum. Mol. Genet. (2015)

Collagen defect in the cartilage primordia of the thoracic vertebrae of Loxl3−/− fetuses (Sirius red staining). (A and B) In LOXL3 knockout mice at E14.5, chondrocytes in the cartilage primordia were loosely organized because of a reduction in the collagen fibres around the chondrocytes. (C and D) Collagen fibres around the columnar chondrocytes in LOXL3 knockout mice were still sparse at E16.5. (E) Whether at E14.5 or at E16.5, the collagen fibre density around the chondrocytes in Loxl3−/− mice was significantly less than that in Loxl3+/+ mice. **P < 0.01. Blue arrows: collagen fibres. Left bar: 100 μm. Right bar: 10 μm.
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Related In: Results  -  Collection

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getmorefigures.php?uid=PMC4599675&req=5

DDV333F9: Collagen defect in the cartilage primordia of the thoracic vertebrae of Loxl3−/− fetuses (Sirius red staining). (A and B) In LOXL3 knockout mice at E14.5, chondrocytes in the cartilage primordia were loosely organized because of a reduction in the collagen fibres around the chondrocytes. (C and D) Collagen fibres around the columnar chondrocytes in LOXL3 knockout mice were still sparse at E16.5. (E) Whether at E14.5 or at E16.5, the collagen fibre density around the chondrocytes in Loxl3−/− mice was significantly less than that in Loxl3+/+ mice. **P < 0.01. Blue arrows: collagen fibres. Left bar: 100 μm. Right bar: 10 μm.
Mentions: Mature collagen fibres consist of multiple collagen fibrils. Collagen fibrils are formed from multiple tropocollagen molecules through covalent cross-linking catalysed by LOXs. Thus we examined the collagen fibres in palate shelves and cartilage primordia of the thoracic vertebrae. Masson's trichrome staining showed that collagen fibres in Loxl3−/− palate shelves compared with Loxl3+/+ palate shelves were significantly decreased at E14.5 (P < 0.01, Fig. 8). The thoracic vertebrae of Loxl3−/−mice started bending at E14.5, and this phenotype became very obvious around E16.5. Sagittal sections and Sirius red staining were used to check the collagen fibres in the thoracic vertebrae at E14.5 and E16.5. The chondrocytes in the cartilage primordia of the thoracic vertebrae were more dispersed in Loxl3−/−mice than in wild-type mice at E14.5 (Fig. 9A and B). Sparse collagen fibres around the columnar chondrocytes were also observed in Loxl3−/− mice at E16.5 (Fig. 9C and D). The amount of collagen fibres around the chondrocytes was significantly decreased in Loxl3−/−mice at E14.5 or E16.5 (P < 0.01, Fig. 9E).Figure 8.

Bottom Line: Failure of any one of these processes can result in embryonic malformation.We found that the obvious decrease of collagen cross-links in palate and spine that was induced by the lack of LOXL3 resulted in cleft palate and spinal deformity.The Loxl3 gene may be a candidate disease gene resulting in cleft palate and spinal deformity.

View Article: PubMed Central - PubMed

Affiliation: Institute of Developmental Biology, School of Life Science, Shandong University, 27 Shanda Nanlu, Jinan 250100, China.

No MeSH data available.


Related in: MedlinePlus