Loss of Snord116 impacts lateral hypothalamus, sleep, and food-related behaviors
| Autor: | Giovanna Zoccoli, Matteo Falappa, Matteo Cerri, Chiara Berteotti, Andrea Freschi, Valter Tucci, Fatemeh Kaveh, Edoardo Balzani, Alfonso Urbanucci, Eivind Hovig, Roberto Amici, Viviana Lo Martire, Marta Pace |
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| Přispěvatelé: | Pace, Marta, Falappa, Matteo, Freschi, Andrea, Balzani, Edoardo, Berteotti, Chiara, Lo Martire, Viviana, Kaveh, Fatemeh, Hovig, Eivind, Zoccoli, Giovanna, Amici, Roberto, Cerri, Matteo, Urbanucci, Alfonso, Tucci, Valter |
| Jazyk: | angličtina |
| Rok vydání: | 2020 |
| Předmět: |
0301 basic medicine
medicine.medical_specialty Melanin-concentrating hormone Lateral hypothalamus Hypothalamus Context (language use) Biology 03 medical and health sciences chemistry.chemical_compound Mice 0302 clinical medicine Neurodevelopmental disorder Internal medicine medicine Premovement neuronal activity Animals RNA Small Nucleolar Melanins Neurons Orexins Hypothalamic Hormones Behavior Animal Epigenetic General Medicine Feeding Behavior medicine.disease Orexin Disease Models Animal Pituitary Hormones 030104 developmental biology Endocrinology chemistry 030220 oncology & carcinogenesis Hypothalamic Area Lateral Genomic imprinting Sleep Prader-Willi Syndrome Neuroscience Research Article |
| Zdroj: | JCI Insight |
| Popis: | Imprinted genes are highly expressed in the hypothalamus; however, whether specific imprinted genes affect hypothalamic neuromodulators and their functions is unknown. It has been suggested that Prader–Willi syndrome (PWS), a neurodevelopmental disorder caused by lack of paternal expression at chromosome 15q11–q13, is characterized by hypothalamic insufficiency. Here, we investigate the role of the paternally expressed Snord116 gene within the context of sleep and metabolic abnormalities of PWS, and we report a significant role of this imprinted gene in the function and organization of the 2 main neuromodulatory systems of the lateral hypothalamus (LH) — namely, the orexin (OX) and melanin concentrating hormone (MCH) — systems. We observed that the dynamics between neuronal discharge in the LH and the sleep-wake states of mice with paternal deletion of Snord116 (PWScr(m+/p–)) are compromised. This abnormal state–dependent neuronal activity is paralleled by a significant reduction in OX neurons in the LH of mutant mice. Therefore, we propose that an imbalance between OX- and MCH-expressing neurons in the LH of mutant mice reflects a series of deficits manifested in the PWS, such as dysregulation of rapid eye movement (REM) sleep, food intake, and temperature control. |
| Databáze: | OpenAIRE |
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