PUBLICATION

The X-linked acrogigantism-associated gene gpr101 is a regulator of early embryonic development and growth in zebrafish

Authors
Trivellin, G., Tirosh, A., Hernández-Ramírez, L.C., Gupta, T., Tsai-Morris, C.H., Faucz, F.R., Burgess, H.A., Feldman, B., Stratakis, C.A.
ID
ZDB-PUB-201130-10
Date
2020
Source
Molecular and Cellular Endocrinology   520: 111091 (Journal)
Registered Authors
Burgess, Harold, Feldman, Benjamin
Keywords
GPR101, X-linked acrogigantism, embryonic development, growth, zebrafish
MeSH Terms
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
  • Zebrafish/genetics*
  • Zebrafish/growth & development*
  • Ovum/metabolism
  • Hypothalamus/pathology
  • Gigantism/complications
  • Gigantism/genetics*
  • Temperature
  • Fertilization/genetics
  • Acromegaly/complications
  • Acromegaly/genetics*
  • Genetic Diseases, X-Linked/genetics*
  • Mutation/genetics
  • Up-Regulation/genetics
  • Female
  • Transcriptome/genetics
  • Signal Transduction/genetics
  • Zygote/metabolism
  • Gene Expression Regulation, Developmental
  • Embryonic Development/genetics*
  • Gastrulation/genetics
  • Animals
(all 23)
PubMed
33248229 Full text @ Mol. Cell. Endocrinol.
Abstract
We recently described X-linked acrogigantism (X-LAG), a condition of early childhood-onset pituitary gigantism associated with microduplications of the GPR101 receptor. The expression of GPR101 in hyperplastic pituitary regions and tumors in X-LAG patients, and GPR101's normally transient pituitary expression during fetal development, suggest a role in the regulation of growth. Nevertheless, little is still known about GPR101's physiological functions, especially during development. By using zebrafish models, we investigated the role of gpr101 during embryonic development and somatic growth. Transient ectopic gpr101 expression perturbed the embryonic body plan but did not affect growth. Loss of gpr101 led to a significant reduction in body size that was even more pronounced in the absence of maternal transcripts, as well as subfertility. These changes were accompanied by gastrulation and hypothalamic defects. In conclusion, both gpr101 loss- and gain-of-function affect, in different ways, fertility, embryonic patterning, growth and brain development.
Genes / Markers
Figures
Figure Gallery (8 images)
Show all Figures
Expression
Phenotype
Fish Conditions Stage Phenotype Figure
gpr101y629/+ (EKW)standard conditionsPrim-5
gpr101y629/+ (EKW)standard conditionsPrim-5
gpr101y629/+ (EKW)standard conditionsPrim-5
gpr101y629/+ (EKW)standard conditionsDays 21-29
gpr101y629/+ (EKW)standard conditionsDays 30-44
gpr101y629/+ (EKW)standard conditionsDays 30-44
gpr101y629/+ (EKW)standard conditionsDays 45-89
gpr101y629/+ (EKW)standard conditionsDays 45-89
gpr101y629/+; gpr101y630/+ (EKW)standard conditionsDays 30-44
gpr101y629/+; gpr101y630/+ (EKW)standard conditionsDays 30-44
1 - 10 of 48
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
nk73aGtTransgenic Insertion
    nkuasgfp1aTgTransgenic Insertion
      nns6TgTransgenic Insertion
        y444EtTransgenic Insertion
          y517TgTransgenic Insertion
            y629
              		Small Deletion
              y630
                		Small Deletion
                y672TgTransgenic Insertion
                  zf3326TgTransgenic Insertion
                    1 - 9 of 9
                    Show
                    Human Disease / Model
                    No data available
                    Sequence Targeting Reagents
                    Target Reagent Reagent Type
                    gpr101CRISPR1-gpr101CRISPR
                    1 - 1 of 1
                    Show
                    Fish
                    Fish
                    EKW
                    gpr101y629/+ (EKW)
                    gpr101y629/+ (EKW)
                    gpr101y629/+ (EKW)
                    gpr101y629/+; gpr101y630/+ (EKW)
                    gpr101y629/y629 (EKW)
                    gpr101y629/y629 (EKW)
                    gpr101y630/+ (EKW)
                    gpr101y630/+ (EKW)
                    gpr101y630/+ (EKW)
                    1 - 10 of 14
                    Show
                    Antibodies
                    No data available
                    Orthology
                    No data available
                    Engineered Foreign Genes
                    Marker Marker Type Name
                    CFPEFGCFP
                    DsRedEFGDsRed
                    EGFPEFGEGFP
                    GAL4EFGGAL4
                    GAL4FFEFGGAL4FF
                    MCardinalEFGMCardinal
                    1 - 6 of 6
                    Show
                    Mapping
                    No data available
                    Your Input Welcome
                    We welcome your input and comments. Please use this form to recommend updates to the information in ZFIN. We appreciate as much detail as possible and references as appropriate. We will review your comments promptly.
                    Citation
                    Trivellin, G., Tirosh, A., Hernández-Ramírez, L.C., Gupta, T., Tsai-Morris, C.H., Faucz, F.R., Burgess, H.A., Feldman, B., Stratakis, C.A. (2020) The X-linked acrogigantism-associated gene gpr101 is a regulator of early embryonic development and growth in zebrafish. Molecular and Cellular Endocrinology. 520:111091.