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Significance of Premature Vertebral Mineralization in Zebrafish Models in Mechanistic and Pharmaceutical Research on
Judith Van Wynsberghe1,2,3, Olivier M Vanakker1,2,3
1Center for Medical Genetics, Ghent University Hospital, 9000 Ghent, Belgium.
Abstract:
Zebrafish are increasingly becoming an important model organism for studying the pathophysiological mechanisms of human diseases and investigating how these mechanisms can be effectively targeted using compounds that may open avenues to novel treatments for patients. The zebrafish skeleton has been particularly instrumental in modeling bone diseases as-contrary to other model organisms-the lower load on the skeleton of an aquatic animal enables mutants to survive to early adulthood. In this respect, the axial skeletons of zebrafish have been a good read-out for congenital spinal deformities such as scoliosis and degenerative disorders such as osteoporosis and osteoarthritis, in which aberrant mineralization in humans is reflected in the respective zebrafish models. Interestingly, there have been several reports of hereditary multisystemic diseases that do not affect the vertebral column in human patients, while the corresponding zebrafish models systematically show anomalies in mineralization and morphology of the spine as their leading or, in some cases, only phenotype. In this review, we describe such examples, highlighting the underlying mechanisms, the already-used or potential power of these models to help us understand and amend the mineralization process, and the outstanding questions on how and why this specific axial type of aberrant mineralization occurs in these disease models.
Insights
Zebrafish models reveal unexpected spinal mineralization defects in hereditary diseases that don't affect human spines. These models offer insights into disease mechanisms and potential therapeutic targets for bone disorders.
Area of Science:
- Comparative biology
- Genetics
- Biomedical research
Background:
- Zebrafish are valuable models for human disease research due to their genetic tractability and physiological similarities.
- Their unique skeletal structure, under lower load, allows mutants to survive, making them ideal for studying bone diseases.
- The zebrafish axial skeleton effectively models human spinal conditions like scoliosis, osteoporosis, and osteoarthritis.
Purpose of the Study:
- To review examples of hereditary multisystemic diseases where zebrafish models exhibit distinct spinal phenotypes.
- To explore the mechanisms underlying aberrant mineralization in these zebrafish models.
- To highlight the potential of these models for understanding and treating human bone diseases.
Main Methods:
- Literature review of zebrafish models for hereditary multisystemic diseases.
- Analysis of reported phenotypes, focusing on spinal mineralization and morphology.
- Discussion of underlying genetic and molecular mechanisms.
Main Results:
- Zebrafish models of certain hereditary diseases show prominent spinal anomalies, even when human patients lack vertebral defects.
- Aberrant mineralization and morphological changes in the zebrafish spine are often the primary or sole phenotype.
- These models provide a unique window into disease processes not readily observable in other model organisms.
Conclusions:
- Zebrafish models can reveal critical insights into disease mechanisms, particularly concerning spinal mineralization, even in cases where human phenotypes are less severe or absent.
- Further investigation into these specific zebrafish models is crucial for understanding the fundamental processes of mineralization.
- These models hold significant potential for developing novel therapeutic strategies targeting bone and spinal health.
