ArticleThe Journal of clinical investigation2020
Rathke's cleft-like cysts arise from Isl1 deletion in murine pituitary progenitors.
Article in The Journal of clinical investigation, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers, 1 of them a synthesis that pooled it.
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Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
12 citing papers in PubMed, 1 synthesis or guideline pooled it, 20 citations in OpenAlex.
- A Potential Concomitant Sellar Embryonic Remnant-Associated Collision Tumor: Systematic Review.Frontiers in oncology · 2021Pooled it
- Loss of primary cilia in late pituitary organogenesis does not cause endocrine dysfunction.Journal of anatomy · 2026Article
- Hypopituitarism and Rathke's cleft cyst in 48,XXYY Syndrome: new insights into sex chromosome aneuploidies.Einstein (Sao Paulo, Brazil) · 2025Article
- Recent Advances in the Understanding of Gonadotrope Lineage Differentiation in the Developing Pituitary.Neuroendocrinology · 2025Review
- Long Noncoding RNAs Expressed in Mouse Pituitary Development and Mature Hormone-Producing Cells.Endocrinology · 2024Article
- Missense mutation of ISL1 (E283D) is associated with the development of type 2 diabetes.Diabetologia · 2024Article
- Heterozygous variants in SIX3 and POU1F1 cause pituitary hormone deficiency in mouse and man.Human molecular genetics · 2023Article
- Pituitary Stem Cell Regulation by Zeb2 and BMP Signaling.Endocrinology · 2023Article
- Cellular interactions in the pituitary stem cell niche.Cellular and molecular life sciences : CMLS · 2022Review
- Analysis of the Clinical Characteristics and Pituitary Function of Patients in Central China With Rathke's Cleft Cysts.Frontiers in endocrinology · 2022Article
- Multi-omic profiling of pituitary thyrotropic cells and progenitors.BMC biology · 2021Article
- Establishing a papillary craniopharyngioma cell line by SV40LT-mediated immortalization.Pituitary · 2021Article
Corrections and comments
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Authors and funding
7 authors at 3 institutions in 3 countries.
Funding
Abstract
The transcription factor ISL1 is expressed in pituitary gland stem cells and the thyrotrope and gonadotrope lineages. Pituitary-specific Isl1 deletion causes hypopituitarism with increased stem cell apoptosis, reduced differentiation of thyrotropes and gonadotropes, and reduced body size. Conditional Isl1 deletion causes development of multiple Rathke's cleft-like cysts, with 100% penetrance. Foxa1 and Foxj1 are abnormally expressed in the pituitary gland and associated with a ciliogenic gene-expression program in the cysts. We confirmed expression of FOXA1, FOXJ1, and stem cell markers in human Rathke's cleft cyst tissue, but not craniopharyngiomas, which suggests these transcription factors are useful, pathological markers for diagnosis of Rathke's cleft cysts. These studies support a model whereby expression of ISL1 in pituitary progenitors drives differentiation into thyrotropes and gonadotropes and without it, activation of FOXA1 and FOXJ1 permits development of an oral epithelial cell fate with mucinous cysts. This pituitary-specific Isl1 mouse knockout sheds light on the etiology of Rathke's cleft cysts and the role of ISL1 in normal pituitary development.
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Registered trials
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