PUBLICATION
Expression of acid-labile subunit (als) in developing and adult zebrafish and its role in dorso-ventral patterning during development
- Authors
- Landi, E., Karabatas, L., Scaglia, P., Pisciottano, F., Gutierrez, M., Ramirez, L., Bergadá, I., Rey, R., Guillermo Jasper, H., Mario Domené, H., Viviana Plazas, P., Domené, S.
- ID
- ZDB-PUB-200825-2
- Date
- 2020
- Source
- General and comparative endocrinology 299: 113591 (Journal)
- Registered Authors
- Keywords
- Acid-labile subunit, dorso-ventral pattern, knockdown phenotype, morpholinos, zebrafish development
- MeSH Terms
-
- Animals
- Carrier Proteins/metabolism*
- Glycoproteins/metabolism*
- Mutation
- Zebrafish/growth & development*
- PubMed
- 32828812 Full text @ Gen. Comp. Endocrinol.
Citation
Landi, E., Karabatas, L., Scaglia, P., Pisciottano, F., Gutierrez, M., Ramirez, L., Bergadá, I., Rey, R., Guillermo Jasper, H., Mario Domené, H., Viviana Plazas, P., Domené, S. (2020) Expression of acid-labile subunit (als) in developing and adult zebrafish and its role in dorso-ventral patterning during development. General and comparative endocrinology. 299:113591.
Abstract
Mammalian acid-labile subunit (ALS) is a serum protein that binds binary complexes between Insulin-like growth factors (IGFs) and insulin-like growth factor-binding proteins (IGFBPs) extending their half-life and keeping them in the vasculature. Human ALS deficiency (ACLSD), due to homozygous or compound heterozygous mutations in IGFALS, leads to moderate short stature with reduced levels of IGF-I and IGFBP-3. There is only one corresponding zebrafish ortholog gene and it has not yet been studied. In this study we elucidate the role of igfals during zebrafish development. In zebrafish embryos igfals mRNA is expressed throughout development, mainly in the brain and subsequently also in the gut and swimbladder. To determine its role during development, we knocked down igfals gene product using morpholinos (MOs). Igfals morphant embryos displayed dorsalization in different degrees of severity, including a shortened trunk and loss of tail. Furthermore, co-injection of human IGFALS mRNA (hIGFALS) was able to rescue the MO-induced phenotype. Finally, overexpression of either hIGFALS or zebrafish igfals mRNA (zigfals) leads to ventralization of embryos including a reduced head and enlarged tail. These findings suggest that als plays an important role in dorso-ventral patterning during zebrafish development.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping