ArticleMolecular and cellular endocrinology2021
The X-linked acrogigantism-associated gene gpr101 is a regulator of early embryonic development and growth in zebrafish.
Article in Molecular and cellular endocrinology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 12 citations in OpenAlex.
- Constitutive activity among orphan G protein-coupled receptors: Molecular mechanisms and pharmacological perspectives.Molecular pharmacology · 2026Review
- Comparing models and experimental structures of the GPR101 receptor: Artificial intelligence yields highly accurate models.Journal of molecular graphics & modelling · 2025Article
- Chromatin conformation capture in the clinic: 4C-seq/HiC distinguishes pathogenic from neutral duplications at the GPR101 locus.Genome medicine · 2024Article
- Structure of GPR101-Gs enables identification of ligands with rejuvenating potential.Nature chemical biology · 2024Article
- Brain Imaging and Registration in Larval Zebrafish.Methods in molecular biology (Clifton, N.J.) · 2024Article
- A Critical Review of Zebrafish Neurological Disease Models-2. Application: Functional and Neuroanatomical Phenotyping Strategies and Chemical Screens.Oxford open neuroscience · 2023Article
- GPR101 loss promotes insulin resistance and diet-induced obesity risk.Neuroscience applied · 2023Article
- Review
- Duplications disrupt chromatin architecture and rewire GPR101-enhancer communication in X-linked acrogigantism.American journal of human genetics · 2022Article
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Authors and funding
9 authors at 3 institutions in 3 countries.
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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.
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