ArticleThe Journal of clinical investigation2012
Cyclocreatine treatment improves cognition in mice with creatine transporter deficiency.
Article in The Journal of clinical investigation, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
43 citing papers in PubMed, 101 citations in OpenAlex.
- Dietary formaldehyde: a silent aggravator of diabetes and cognitive impairments.Nutrition & diabetes · 2025Review
- Effects of SLC6A8 mutation-induced creatine deficiency on cellular function in fibroblasts.Scientific reports · 2025Article
- Rescue of myocytes and locomotion throughMolecular therapy. Methods & clinical development · 2024Article
- TREM2 deficiency aggravates renal injury by promoting macrophage apoptosis and polarization via the JAK-STAT pathway in mice.Cell death & disease · 2024Article
- TREM2-IGF1 Mediated Glucometabolic Enhancement Underlies Microglial Neuroprotective Properties During Ischemic Stroke.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2024Article
- Article
- "Heads Up" for Creatine Supplementation and its Potential Applications for Brain Health and Function.Sports medicine (Auckland, N.Z.) · 2023Review
- Diagnosis and Treatment of X-Linked Creatine Transporter Deficiency: Case Report and Literature Review.Brain sciences · 2023Review
- Magnetic resonance in medical sciences : MRMS : an official journal of Japan Society of Magnetic Resonance in Medicine · 2022Article
- Creatine transporter-deficient rat model shows motor dysfunction, cerebellar alterations, and muscle creatine deficiency without muscle atrophy.Journal of inherited metabolic disease · 2022Article
- Effects of Delivering Guanidinoacetic Acid or Its Prodrug to the Neural Tissue: Possible Relevance for Creatine Transporter Deficiency.Brain sciences · 2022Article
- Article
- Evaluation of chronic toxicity of cyclocreatine in beagle dogs after oral gavage administration for up to 23 weeks.Toxicology and applied pharmacology · 2021Article
- Creatine transporter deficiency impairs stress adaptation and brain energetics homeostasis.JCI insight · 2021Article
- Review
- A new rat model of creatine transporter deficiency reveals behavioral disorder and altered brain metabolism.Scientific reports · 2021Article
- The influence of the R47H triggering receptor expressed on myeloid cells 2 variant on microglial exosome profiles.Brain communications · 2021Article
- Cyclocreatine treatment ameliorates the cognitive, autistic and epileptic phenotype in a mouse model of Creatine Transporter Deficiency.Scientific reports · 2020Article
- Cyclocreatine Transport by SLC6A8, the Creatine Transporter, in HEK293 Cells, a Human Blood-Brain Barrier Model Cell, and CCDSs Patient-Derived Fibroblasts.Pharmaceutical research · 2020Article
- The Creatine Transporter Unfolded: A Knotty Premise in the Cerebral Creatine Deficiency Syndrome.Frontiers in synaptic neuroscience · 2020Review
Corrections and comments
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Authors and funding
9 authors at 1 institution in 1 country.
Funding
Abstract
The second-largest cause of X-linked mental retardation is a deficiency in creatine transporter (CRT; encoded by SLC6A8), which leads to speech and language disorders with severe cognitive impairment. This syndrome, caused by the absence of creatine in the brain, is currently untreatable because CRT is required for creatine entry into brain cells. Here, we developed a brain-specific Slc6a8 knockout mouse (Slc6a8-/y) as an animal model of human CRT deficiency in order to explore potential therapies for this syndrome. The phenotype of the Slc6a8-/y mouse was comparable to that of human patients. We successfully treated the Slc6a8-/y mice with the creatine analog cyclocreatine. Brain cyclocreatine and cyclocreatine phosphate were detected after 9 weeks of cyclocreatine treatment in Slc6a8-/y mice, in contrast to the same mice treated with creatine or placebo. Cyclocreatine-treated Slc6a8-/y mice also exhibited a profound improvement in cognitive abilities, as seen with novel object recognition as well as spatial learning and memory tests. Thus, cyclocreatine appears promising as a potential therapy for CRT deficiency.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.