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Different studies reported that glutathione (GSH) play a major role in modulating methylmercury (MeHg) neurotoxicity. Glutathione synthetase deficiency (GSD) and 5- Oxoprolinase deficiencies are two inborn errors of GSH metabolism; which were associated with physiological and pathological features resembling patients with Minamata disease (MD). The present study aimed to investigate whether there are similarities between the metabolic effects of MeHg and 5-oxoprolinase inhibitor (ICA) on both C6 Glial cells and PC12 neurons.