Tolson Kristen P, Gemelli Terry, Meyer Donna, Yazdani Umar, Kozlitina Julia, Zinn Andrew R
McDermott Center for Human Growth and Development (K.P.T., T.G., D.M., U.Y., J.K., A.R.Z.) and Department of Internal Medicine (A.R.Z.), The University of Texas Southwestern Medical Center, Dallas, Texas 75390-8591.
Endocrinology. 2014 Jul;155(7):2436-44. doi: 10.1210/en.2013-2125. Epub 2014 Apr 28.
Germline haploinsufficiency of human or mouse Sim1 is associated with hyperphagic obesity. Sim1 encodes a transcription factor required for proper formation of the paraventricular (PVN), supraoptic, and anterior periventricular hypothalamic nuclei. Sim1 expression persists in these neurons in adult mice, raising the question of whether it plays a physiologic role in regulation of energy balance. We previously showed that Sim1 heterozygous mice had normal numbers of PVN neurons that were hyporesponsive to melanocortin 4 receptor agonism and showed reduced oxytocin expression. Furthermore, conditional postnatal neuronal inactivation of Sim1 also caused hyperphagic obesity and decreased hypothalamic oxytocin expression. PVN projections to the hindbrain, where oxytocin is thought to act to modulate satiety, were anatomically intact in both Sim1 heterozygous and conditional knockout mice. These experiments provided evidence that Sim1 functions in energy balance apart from its role in hypothalamic development but did not rule out effects of Sim1 deficiency on postnatal hypothalamic maturation. To address this possibility, we used a tamoxifen-inducible, neural-specific Cre transgene to conditionally inactivate Sim1 in adult mice with mature hypothalamic circuitry. Induced Sim1 inactivation caused increased food and water intake and decreased expression of PVN neuropeptides, especially oxytocin and vasopressin, with no change in energy expenditure. Sim1 expression was not required for survival of PVN neurons. The results corroborate previous evidence that Sim1 acts physiologically as well as developmentally to regulate body weight. Inducible knockout mice provide a system for studying Sim1's physiologic function in energy balance and identifying its relevant transcriptional targets in the hypothalamus.
人类或小鼠Sim1的种系单倍剂量不足与食欲亢进性肥胖有关。Sim1编码一种转录因子,该转录因子是室旁核(PVN)、视上核和下丘脑室周前核正常形成所必需的。Sim1在成年小鼠的这些神经元中持续表达,这就引发了一个问题,即它是否在能量平衡调节中发挥生理作用。我们之前表明,Sim1杂合小鼠的PVN神经元数量正常,但对黑皮质素4受体激动剂反应低下,且催产素表达减少。此外,Sim1的条件性出生后神经元失活也导致了食欲亢进性肥胖和下丘脑催产素表达降低。在Sim1杂合小鼠和条件性敲除小鼠中,PVN向后脑的投射在解剖学上都是完整的,而催产素被认为是在后脑发挥调节饱腹感的作用。这些实验提供了证据,表明Sim1在能量平衡中发挥作用,这与其在下丘脑发育中的作用无关,但并未排除Sim1缺乏对出生后下丘脑成熟的影响。为了解决这种可能性,我们使用了一种他莫昔芬诱导的、神经特异性的Cre转基因,在具有成熟下丘脑回路的成年小鼠中条件性失活Sim1。诱导的Sim1失活导致食物和水摄入量增加,PVN神经肽的表达减少,尤其是催产素和加压素,而能量消耗没有变化。PVN神经元的存活不需要Sim1表达。这些结果证实了之前的证据,即Sim1在生理和发育上均发挥作用来调节体重。诱导性敲除小鼠提供了一个系统,用于研究Sim1在能量平衡中的生理功能,并确定其在下丘脑中的相关转录靶点。