Locked-in Syndrome due to Central Pontine Myelinolysis: Case Report

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Abstract

Central pontine myelinolysis (CPM) classically occurs in alcoholics, malnourished individuals, chronic liver diseases, and rapid correction of hyponatremia. This report presents locked-in syndrome due to CPM following rapid correction of hyponatremia. A 44-year-old male came to the hospital due to a short period of loss of consciousness. He was alert and had no focal neurological abnormalities at admission. The serum sodium concentration was 118 mEq/L and was corrected to 134 mEq/L in the first 18 hours. One week later, progressive weakness in limbs developed and he progressed to a complete quadriplegic state and bulbar palsy, with only eye blinking preserved. Brain magnetic resonance imaging revealed a characteristic hyperintense signal abnormality in both pons, so he was diagnosed to locked-in syndrome caused by CPM. The patient gradually improved following continuous intensive rehabilitation for more than 2 years. He was able to move all joint muscles against gravity in generally and he could gait under supervision.

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Osmotic demyelination syndrome following correction of hyponatremia.

Richard Sterns, J Riggs, S Schochet (1986)

The treatment of hyponatremia is controversial: some authorities have cautioned that rapid correction causes central pontine myelinolysis, and others warn that severe hyponatremia has a high mortality rate unless it is corrected rapidly. Eight patients treated over a five-year period at our two institutions had a neurologic syndrome with clinical or pathological findings typical of central pontine myelinolysis, which developed after the patients presented with severe hyponatremia. Each patient's condition worsened after relatively rapid correction of hyponatremia (greater than 12 mmol of sodium per liter per day)--a phenomenon that we have called the osmotic demyelination syndrome. Five of the patients were treated at one hospital, and accounted for all the neurologic complications recorded among 60 patients with serum sodium concentrations below 116 mmol per liter; no patient in whom the sodium level was raised by less than 12 mmol per liter per day had any neurologic sequelae. Reviewing published reports on patients with very severe hyponatremia (serum sodium less than 106 mmol per liter) revealed that neurologic sequelae were associated with correction of hyponatremia by more than 12 mmol per liter per day; when correction proceeded more slowly, patients had uneventful recoveries. We suggest that the osmotic demyelination syndrome is a preventable complication of overly rapid correction of chronic hyponatremia.

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Central Pontine Myelinolysis

RAYMOND ADAMS (1959)

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Clinical and functional outcome and factors predicting prognosis in osmotic demyelination syndrome (central pontine and/or extrapontine myelinolysis) in 25 patients.

Ashalatha Radhakrishnan, Chandrasekharan Kesavadas, Ramesha Kallakatta … (2011)

To assess the functional and clinical outcome in a sizeable cohort of patients with osmotic demyelination syndrome (ODS) and to characterise the factors which could predict the final outcome. Twenty five consecutive patients with ODS formed the study cohort. The diagnosis of ODS was based on clinical features with corroborating imaging findings. Two functional scales--Functional Independent Measure (FIM) and Disability Rating Scale (DRS)--were applied to assess the functional status at the time of admission, discharge and last follow-up. Patients who became independent for activities of daily living (ADL) at last follow-up were classified as favourable outcome, and those who died or became dependent for ADL were classified as a poor outcome group respectively. The Fisher exact test and Mann-Whitney U test were used to assess categorical and continuous variables respectively. The mean age at diagnosis was 53 ± 14 years. Five (20%) had central pontine myelinolysis, seven (28%) had extrapontine myelinolysis, and 13 (52%) had both. Hyponatraemia and hypokalaemia were noted in 20 (80%) and 10 (40%) patients respectively. Six (24%) received intravenous methylprednisolone. Eleven (46%) had a favourable outcome at a mean follow-up of 2.2 ± 2.5 years. Hyponatraemia ≤ 115 mEq (p=0.04), associated hypokalaemia (p=0.04) and low Glasgow Coma Scale (GCS) (p=0.008) at presentation were predictive of poor outcome. The mean FIM score at admission (p=0.05) and at discharge (p=0.01), and mean DRS at admission (p=0.05) were predictive of poor outcome. Higher GCS scores, better scores in functional scales in hospital, less severe hyponatraemia and absence of superadded hypokalaemia predicted favourable outcome.

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Author and article information

Journal

Journal ID (nlm-ta): Ann Rehabil Med

Journal ID (iso-abbrev): Ann Rehabil Med

Journal ID (publisher-id): ARM

Title: Annals of Rehabilitation Medicine

Publisher: Korean Academy of Rehabilitation Medicine

ISSN (Print): 2234-0645

ISSN (Electronic): 2234-0653

Publication date (Print): October 2014

Publication date (Electronic): 30 October 2014

Volume: 38

Issue: 5

Pages: 702-706

Affiliations

Department of Rehabilitation Medicine, Chungnam National University School of Medicine, Daejeon, Korea.

Author notes

Corresponding author: Jin Hee Nam. Department of Rehabilitation Medicine, Chungnam National University Hospital, 282 Munhwa-ro, Jung-gu, Daejeon 301-721, Korea. Tel: +82-42-338-2460, Fax: +82-42-338-2461, shapil21@ 123456cnuh.co.kr

Article

DOI: 10.5535/arm.2014.38.5.702

PMC ID: 4221401

PubMed ID: 25379502

SO-VID: 3aba740c-e529-423a-a201-2ad962706a36

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This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Locked-in Syndrome due to Central Pontine Myelinolysis: Case Report

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Most cited references 10

Osmotic demyelination syndrome following correction of hyponatremia.

Central Pontine Myelinolysis

Clinical and functional outcome and factors predicting prognosis in osmotic demyelination syndrome (central pontine and/or extrapontine myelinolysis) in 25 patients.

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