Abstract
We report a case study of a 60-year-old man with bipolar disorder on stable lithium treatment who developed severe toxicity while admitted to ICU with sepsis and multiorgan failure. Despite unchanged lithium administration, his serum levels escalated due to renal dysfunction, resulting in lithium toxicity. After regaining consciousness, he exhibited a cerebellar syndrome marked by ataxia, tremor, and scanning speech. MRI revealed cerebellar atrophy. Following discontinuation of lithium and hemodialysis, the patient’s symptoms remained static. The patient was diagnosed with syndrome of irreversible lithium-effectuated neurotoxicity (SILENT), a chronic cerebellar disorder characterized by persistent ataxia, nystagmus, and gait abnormalities extending beyond two months post-lithium exposure. The disorder has a predilection for cerebellar and basal ganglia dysfunction. MRI findings include cerebellar gliosis and atrophy and leptomeningeal enhancement. This case report highlights that SILENT is both preventable and permanent, urging heightened awareness among clinicians to facilitate early detection and intervention. Patients on lithium with compromised renal function or fever necessitate vigilant lithium level monitoring, dose adjustment, or cessation, to forestall enduring morbidity. This case emphasizes the significance of recognizing and managing SILENT, particularly in critical care settings, to mitigate long-term cerebellar impairment and optimize patient outcomes.
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References
Martínez-Martín Á, Sánchez-Larsen Á, Sánchez-Mora C, Sáez-Povedano R, Segura T. Intoxicación por litio: La amenaza SILENTiosa. Revista de Psiquiatría y Salud Mental. 2021;14(4):233–234. https://www.sciencedirect.com/science/article/pii/S1888989120300756. https://doi.org/10.1016/j.rpsm.2020.06.006.
Adityanjee, Munshi KR, Thampy A. The syndrome of irreversible lithium-effectuated neurotoxicity. Clin Neuropharmacol. 2005;28(1):38–49. https://www.ncbi.nlm.nih.gov/pubmed/15714160. https://doi.org/10.1097/01.wnf.0000150871.52253.b7.
Fernandes Santos C, Gomes R. Syndrome of irreversible lithium-effectuated neurotoxicity (SILENT): A review. Eur Psychiatry. 2022;65(S1):S717. https://search.proquest.com/docview/2708707189. https://doi.org/10.1192/j.eurpsy.2022.1850.
Verdoux H, Debruyne A, Queuille E, De Leon J. A reappraisal of the role of fever in the occurrence of neurological sequelae following lithium intoxication: a systematic review. Expert Opin Drug Saf. 2021;20(7):827–838. https://www.tandfonline.com/doi/abs/10.1080/14740338.2021.1912011. https://doi.org/10.1080/14740338.2021.1912011.
Donaldson IM, Cuningham J. Persisting neurologic sequelae of lithium carbonate therapy. Arch Neurol. 1983;40(12):747–51. https://doi.org/10.1001/archneur.1983.04050110065011.
Schou M. Long-lasting neurological sequelae after lithium intoxication. Acta Psychiatr Scand. 1984;70(6):594–602. https://doi.org/10.1111/j.1600-0447.1984.tb01254.x.
Niethammer M, Ford B. Permanent lithium-induced cerebellar toxicity: three cases and review of literature. Mov Disord. 2007;22(4):570–3. https://doi.org/10.1002/mds.21318.10.1002/mds.21318.
Dethy S, Manto M, Bastianelli E, Gangji V, Laute MA, Goldman S, Hildebrand J. Cerebellar spongiform degeneration induced by acute lithium intoxication in the rat. Neurosci Lett. 1997;224(1):25–8. https://doi.org/10.1016/s0304-3940(97)13461-9.
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Sarah Marmol and Nestor Beltre wrote the main manuscript texts. Sarah Marmol provided the figures and video. Nestor Beltre provided video editing. Jason Margolesky provided editorial review and critique.
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Sarah Marmol and Nestor Beltre co-first authorship.
Lithium is a mood-stabilizing medication used to treat bipolar disorder that has been known to cause both acute and chronic neurotoxicity. We present a case of a patient in their 60s who experienced syndrome of irreversible lithium-effectuated neurotoxicity (SILENT) after serum lithium levels became toxic in the ICU due to renal failure. Despite discontinuation of lithium and hemodialysis, the patient suffered permanent cerebellar symptoms.
SILENT, a preventable though permanent cerebellar syndrome, underscores the need for vigilance and management of lithium levels in ICU patients, particularly with renal impairment. Recognizing this risk, monitoring drug levels, and discontinuing lithium if needed, can mitigate the risk, and reduce morbidity. .
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Marmol, S., Beltre, N. & Margolesky, J. Syndrome of Irreversible Lithium-Effectuated Neurotoxicity (SILENT): A Preventable Cerebellar Disorder. Cerebellum (2024). https://doi.org/10.1007/s12311-024-01668-z
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DOI: https://doi.org/10.1007/s12311-024-01668-z