| Summary: | Abstract Background Lithium is primarily used to treat bipolar disorder and is known to cause several acute neurological complications. Reversible splenial lesions (RSLs) may be evident in antiepileptic drug toxicity or withdrawal, infections, and other phenomena. We report two cases of RSL presenting as neuroleptic malignant syndrome-like symptoms (NMSLS) with lithium associated neurotoxicity. Case presentation A 28-year-old woman was admitted after taking increased dosages of lithium for schizophrenia. She experienced generalized tremor, rigidity, dysarthria, high fever, and tachycardia. Symptoms and brain lesion recovered 2 weeks after discontinuation of lithium. The second case involved a 59-year-old woman who was receiving treatment for bipolar disorder since 1988. When lithium was administered for impatience and aggressive behavior, her mental state deteriorated and fever developed, along with generalized tremor in the extremities. Brain magnetic resonance imaging (MRI) in both patients showed a reversible oval-shaped lesion localized to the splenium of the corpus callosum. Both patients were defined as neuroleptic malignant syndrome-like symptoms (NMSLS) based on the DSM-5 diagnostic criteria for neuroleptic malignant syndrome. The suspected etiology of our cases was lithium associated neurotoxicity according to their clinical course and medical information. Our patients fully recovered in 10–14 days after the discontinuation of lithium. Conclusions The patients experienced similar clinical courses and had similar radiological findings of RSL. Manifestations in both cases were related to lithium associated neurotoxicity and this should be considered in patients with RSL and NMSLS.
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