Zebrafish (Danio rerio) are increasingly used to investigate spine development, growth, and for studying the etiology of spinal deformity, such as scoliosis. Here, we present a micro-computed tomography-based pipeline for visualizing the zebrafish skeleton. We describe steps for sample preparation, imaging, data management, and processing. We then detail analysis of vertebral and spine morphology using open-source software. This protocol will be useful for scientists using zebrafish to understand spine development and disease. For complete details on the use and execution of this protocol, please refer to Bearce et al. (2022)..
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| http://dx.doi.org/10.1016/j.xpro.2023.102739 | DOI Listing |
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Visualization and quantitation of spine deformity in zebrafish models of scoliosis by micro-computed tomography.
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December 2023
Institute of Molecular Biology, Department of Biology, University of Oregon, Eugene, OR 97403, USA. Electronic address:
Zebrafish (Danio rerio) are increasingly used to investigate spine development, growth, and for studying the etiology of spinal deformity, such as scoliosis. Here, we present a micro-computed tomography-based pipeline for visualizing the zebrafish skeleton. We describe steps for sample preparation, imaging, data management, and processing.
View Article and Find Full Text PDFHypophosphatasia (HPP), caused by loss-of-function mutations in the gene encoding tissue-nonspecific alkaline phosphatase (TNAP), is characterized by skeletal and dental hypomineralization that can vary in severity from life-threatening to milder manifestations only in adulthood. PHOSPHO1 deficiency leads to early-onset scoliosis, osteomalacia, and fractures that mimic pseudo-HPP. Asfotase alfa, a life-saving enzyme replacement therapy approved for pediatric-onset HPP, requires subcutaneous injections 3 to 6 times per week.
View Article and Find Full Text PDFAbnormal morphological features of osteocyte lacunae in adolescent idiopathic scoliosis: A large-scale assessment by ultra-high-resolution micro-computed tomography.
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SH Ho Scoliosis Research Laboratory, Joint Scoliosis Research Centre of the Chinese University of Hong Kong and Nanjing University Department of Orthopaedics and Traumatology, The Chinese University of Hong Kong, Hong Kong, China; Li Ka Shing Institute of Health Sciences, The Chinese University of Hong Kong, Hong Kong, China. Electronic address:
Aim: Abnormal osteocyte lacunar morphology in adolescent idiopathic scoliosis (AIS) has been reported while the results were limited by the number of osteocyte lacunae being quantified. The present study aimed to validate previous findings through (a) comparing morphological features of osteocyte lacunae between AIS patients and controls in spine and ilium using a large-scale assessment, and (b) investigating whether there is an association between the acquired morphological features of osteocyte lacunae and disease severity in AIS.
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The orthopedic characterization of cfap298 mutants validate zebrafish to faithfully model human AIS.
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Orthopedic Surgery and Trauma Center, Pitié-Salpêtrière Teaching Hospital, 47 Boulevard de l'Hôpital, 75013, Paris, France.
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