ArticleEndocrinology2017
TRH Action Is Impaired in Pituitaries of Male IGSF1-Deficient Mice.
Article in Endocrinology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 40 citations in OpenAlex.
- ZEB1 Inhibits LHβ Subunit Transcription When Overexpressed, but Is Dispensable for LH Synthesis in Mice.Endocrinology · 2024Article
- The hypothalamic-pituitary-thyroid axis is intact in male insulin receptor substrate 4 knockout mice.European thyroid journal · 2024Article
- Dynamics of thyroid diseases and thyroid-axis gland masses.Molecular systems biology · 2022Article
- IGSF1 Deficiency Leads to Reduced TSH Production Independent of Alterations in Thyroid Hormone Action in Male Mice.Endocrinology · 2022Article
- Deletion of Gαq/11 or Gαs Proteins in Gonadotropes Differentially Affects Gonadotropin Production and Secretion in Mice.Endocrinology · 2022Article
- The extant immunoglobulin superfamily, member 1 gene results from an ancestral gene duplication in eutherian mammals.PloS one · 2022Article
- Article
- Genetic mapping of renal glutathione suggests a novel regulatory locus on the murine X chromosome and overlap with hepatic glutathione regulation.Free radical biology & medicine · 2021Article
- IGSF1 Does Not Regulate Spermatogenesis or Modify FSH Synthesis in Response to Inhibins or Activins.Journal of the Endocrine Society · 2021Article
- Diagnosis and Management of Central Congenital Hypothyroidism.Frontiers in endocrinology · 2021Review
- Hypoprolactinemia as a Clue to Diagnosis of Mild Central Hypothyroidism due toJournal of clinical research in pediatric endocrinology · 2020Article
- IGSF1 Deficiency Results in Human and Murine Somatotrope Neurosecretory Hyperfunction.The Journal of clinical endocrinology and metabolism · 2020Article
- Recent advances in research on isolated congenital central hypothyroidism.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2019Article
- A novel IGSF1 mutation in a large Irish kindred highlights the need for familial screening in the IGSF1 deficiency syndrome.Clinical endocrinology · 2018Article
- Mutations in IRS4 are associated with central hypothyroidism.Journal of medical genetics · 2018Article
- From Consternation to Revelation: Discovery of a Role for IGSF1 in Pituitary Control of Thyroid Function.Journal of the Endocrine Society · 2018Review
- The short mRNA isoform of the immunoglobulin superfamily, member 1 gene encodes an intracellular glycoprotein.PloS one · 2017Article
- Congenital hypothyroidism: insights into pathogenesis and treatment.International journal of pediatric endocrinology · 2017Review
Corrections and comments
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Authors and funding
13 authors at 5 institutions in 4 countries.
Funding
Abstract
Loss-of-function mutations in the X-linked immunoglobulin superfamily, member 1 (IGSF1) gene cause central hypothyroidism. IGSF1 is a transmembrane glycoprotein of unknown function expressed in thyrotropin (TSH)-producing thyrotrope cells of the anterior pituitary gland. The protein is cotranslationally cleaved, with only its C-terminal domain (CTD) being trafficked to the plasma membrane. Most intragenic IGSF1 mutations in humans map to the CTD. In this study, we used CRISPR-Cas9 to introduce a loss-of-function mutation into the IGSF1-CTD in mice. The modified allele encodes a truncated protein that fails to traffic to the plasma membrane. Under standard laboratory conditions, Igsf1-deficient males exhibit normal serum TSH levels as well as normal numbers of TSH-expressing thyrotropes. However, pituitary expression of the TSH subunit genes and TSH protein content are reduced, as is expression of the receptor for thyrotropin-releasing hormone (TRH). When challenged with exogenous TRH, Igsf1-deficient males release TSH, but to a significantly lesser extent than do their wild-type littermates. The mice show similarly attenuated TSH secretion when rendered profoundly hypothyroid with a low iodine diet supplemented with propylthiouracil. Collectively, these results indicate that impairments in pituitary TRH receptor expression and/or downstream signaling underlie central hypothyroidism in IGSF1 deficiency syndrome.
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