2017
DOI: 10.1016/j.ymgmr.2016.12.008
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Gene therapy for a mouse model of glucose transporter-1 deficiency syndrome

Abstract: ObjectiveWe generated an adeno-associated virus (AAV) vector in which the human SLC2A1 gene was expressed under the synapsin I promoter (AAV-hSLC2A1) and examined if AAV-hSLC2A1 administration can lead to functional improvement in GLUT1-deficient mice.MethodsAAV-hSLC2A1 was injected into heterozygous knock-out murine Glut1 (GLUT1+/−) mice intraperitoneally (systemic; 1.85 × 1011 vg/mouse) or intra-cerebroventricularly (local; 1.85 × 1010 vg/mouse). We analyzed GLUT1 mRNA and protein expression, motor function … Show more

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Cited by 13 publications
(34 citation statements)
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“…Figure shows the results of the rota‐rod test at the age of 10 and 14 weeks. There was a significant deference between GLUT1 +/+ mice and control GLUT1 +/– mice (GLUT1 +/– mice after intracerebroventricular injection with saline) at 10 and 14 weeks, as reported previously . The rota‐rod test at the age of 10 and 14 weeks of the AAV‐GLUT1 injection group (AAV vector with GLUT1 promoter group) showed a trend for motor function improvement compared to the AAV‐h SLC2A1 injection group (AAV vector with synapsin I promoter group).…”
Section: Resultssupporting
confidence: 78%
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“…Figure shows the results of the rota‐rod test at the age of 10 and 14 weeks. There was a significant deference between GLUT1 +/+ mice and control GLUT1 +/– mice (GLUT1 +/– mice after intracerebroventricular injection with saline) at 10 and 14 weeks, as reported previously . The rota‐rod test at the age of 10 and 14 weeks of the AAV‐GLUT1 injection group (AAV vector with GLUT1 promoter group) showed a trend for motor function improvement compared to the AAV‐h SLC2A1 injection group (AAV vector with synapsin I promoter group).…”
Section: Resultssupporting
confidence: 78%
“…After intracerebroventricular injection, the average body weight of AAV‐treated GLUT1 +/– mice at the age of 14 weeks, corresponding to 8 weeks after intracerebroventricular injection, was 22.3 ± 1.6 g ( n = 12) (see Supporting information, Figure S9a). At the age of 14 weeks, the average cerebral hemisphere weight of GLUT1 +/+ mice (163.3 ± 5.5 mg, n = 6) and GLUT1 +/– mice (136.6 ± 6.6 mg, n = 6) were significantly different ( p < 0.05), as reported previously . After intracerebroventricular injection, the cerebral hemisphere weight of AAV‐treated GLUT1 +/– mice at the age of 14 weeks, corresponding to 8 weeks after intracerebroventricular injection, was 150 ± 4.4 mg ( n = 11) (see Supporting information, Figure S9b).…”
Section: Resultssupporting
confidence: 78%
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