Ciarlone · Neurobiology of disease 2016 · controlled animal experiment · n=?

Ketone ester supplementation attenuates seizure activity, and improves behavior and hippocampal synaptic plasticity in an Angelman syndrome mouse model.

Cited 110 times in the scientific literature.

Level 5 - mechanism / opinion, no new human data

Preclinical animal model study (Oxford CEBM Level 5).

PubMed 27546058 · doi:10.1016/j.nbd.2016.08.002 · record verified 2026-08-30

What was done

Angelman syndrome (AS) model mice were administered the ketone ester R,S-1,3-butanediol acetoacetate diester ad libitum for eight weeks to test whether exogenous ketone supplementation could mimic a ketogenic diet. Outcomes measured included motor coordination, learning and memory, hippocampal synaptic plasticity, seizure activity, and brain amino acid metabolism.

What was found

Ketone ester supplementation induced sustained ketosis, exerted anticonvulsant effects, altered brain amino acid metabolism, and improved motor coordination, learning, memory, and hippocampal synaptic plasticity in AS mice. The abstract reports no numerical values, effect sizes, or statistical metrics.

Why it matters

Because strict ketogenic diets are challenging for patients with Angelman syndrome to maintain, exogenous ketone esters may offer a more manageable therapeutic approach for managing seizures and neurological deficits if translated to humans.

Limits

This study was conducted entirely in an animal model and cannot directly establish clinical efficacy or safety in humans. The abstract omits sample size, dosage quantities consumed under ad libitum access, quantitative comparisons to control groups, and potential adverse effects.

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