A Case of Central Pontine Myelinolysis Caused by Hypophosphatemia Secondary to Refeeding Syndrome

Yamashita, Chikara; Sekiya, Hiroshi; Maeda, Norihisa; Torii, Takako; Ohyagi, Yasumasa; Kira, Jun Ichi

doi:10.1159/000440711

Cited by 8 publications

(7 citation statements)

References 15 publications

(17 reference statements)

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“…Hypokalaemia was present during the course of their illness in 14 (93.4%) and was corrected, but the rate was not documented 15 . Hypophosphataemia was present in nine (60%), of which eight were moderate to severe (<0.64); this finding has been reported in case reports only 16–18 …”

Section: Discussionsupporting

confidence: 61%

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Osmotic demyelination syndrome: novel risk factors and proposed pathophysiology

Ambati

Kho

Prentice

et al. 2022

Internal Medicine Journal

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Background: Osmotic demyelination syndrome (ODS) is non-inflammatory demyelination in response to an osmotic challenge. It can be pontine or extrapontine in presentation.Aims: To retrospectively review cases involving ODS and define the spectrum of causes, risk factors, clinical and radiological presentations, and functional outcomes. Results:The study utilised data from 15 patients with a mean age of 53.6 years. Malnutrition (9; 60%) and chronic alcoholism (10; 66.7%) were the most common associated disorders. Two (13.3%) patients had severe hyponatraemia (<120 mmol/L). The average highest single-day change was 5.1 mmol/L. Radiologically, 14 (93.3%) had pontine and 6 (40%) had extra-pontine lesions. Hypokalaemia (14; 93.3%) and hypophosphataemia (9; 60%) were commonly associated. Common clinical manifestations include altered consciousness/encephalopathy (9; 60%), dysphagia (4; 26.7%) and limb weakness (4; 26.7%). At 3 months, two (14.3%) had died and six (40%) were functionally independent (modified Rankin scale 0-2). Conclusion:We found that ODS occurred despite appropriate correction rates of hyponatraemia. Factors such as malnutrition, chronic alcoholism, hypokalaemia and hypophosphataemia are thought to play a role in its pathogenesis. Approximately half of the patients survived and became functionally independent.

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Section: Discussionsupporting

confidence: 61%

“…15 Hypophosphataemia was present in nine (60%), of which eight were moderate to severe (<0.64); this finding has been reported in case reports only. [16][17][18] The clinical features were non-specific and included confusion, reduced conscious state, ataxia, paranoid ideation and stroke-like presentation with monoparesis.…”

Section: Discussionmentioning

confidence: 99%

Osmotic demyelination syndrome: novel risk factors and proposed pathophysiology

Ambati

Kho

Prentice

et al. 2022

Internal Medicine Journal

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“…Leukoencephalopathy has been caused by toxic, acquired vascular, metabolic, and infectious/inflammatory insults, as well as inherited disorders [4]. Two reports described patients with hypophosphatemia who had central pontine and extrapontine myelinolysis (osmotic myelinolysis) [5, 6]. These patients did not have leukoencephalopathy or lesions in the cerebral white matter, unlike our patient.…”

Section: Discussionmentioning

confidence: 50%

“…Why the phosphate levels did not parallel her consciousness levels or MRI findings remains to be elucidated, but hypophosphatemia might have triggered myelinolysis, the symptoms of which lasted after the correction of phosphate levels. Similar remaining symptoms after normalization of the phosphate level have been reported in a patient with hypophosphatemia-associated osmotic myelinolysis [6]. We also cannot completely deny the coincidental occurrence of hypophosphatemia and leukoencephalopathy, and therefore, accumulation of cases is apparently needed to draw firm conclusion of their link.…”

Section: Discussionmentioning

confidence: 62%

Treatable Leukoencephalopathy in a Patient with Hypophosphatemia

et al. 2017

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We report the first patient with pathologically proven leukoencephalopathy associated with hypophosphatemia. A 61-year-old woman had repetitive episodes of decreased consciousness with pontine and pallidal lesions and extensive leukoencephalopathy on MRI, later found to be associated with hypophosphatemia. Although hypophosphatemia has been linked to central pontine and extrapontine myelinolysis (osmotic myelinolysis), lesions in the deep white matter have not been reported. Brain biopsy performed during the first diagnosis process revealed nonspecific demyelination with gliosis, a finding similar to that of chronic osmotic myelinolysis. After normalization of phosphate levels, her consciousness completely improved and MRI abnormalities partly resolved. We should consider that leukoencephalopathy can be associated with hypophosphatemia, which is often treatable.

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“…This indicates that, for the development of CPM, brain damage caused by the underlying condition itself can be sufficient in some cases, whereas the sodium event is not always a requirement. Other osmolyte dysregulation, such as hypokalemia [ 12 ] and hypophosphatemia [ 13 ], are also reported to mediate CPM.…”

Section: Introductionmentioning

confidence: 99%

A 44-Year-Old Alcohol-Dependent Man Who Recovered from Central Pontine Myelinolysis with Supportive Physical Therapy

Tobiume¹,

Iha²,

Miyahira³

et al. 2022

Am J Case Rep

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Patient: Male, 44-year-old Final Diagnosis: Central pontine myelinolysis Symptoms: Bulbar paralysis • locked-in syndrome • tetraplegia Medication: — Clinical Procedure: Brain MRI Specialty: Neurology • Rehabilitation Objective: Unusual clinical course Background: Central pontine myelinolysis (CPM) includes symmetric demyelination of the central pons. CPM is a rare neurological disorder that generally develops after rapid correction of hyponatremia in individuals having underlying conditions, such as malnutrition, alcoholism, and severe burns. It can cause severe long-term disabilities. However, there is currently no pharmacotherapy capable of promoting remyelination, a process crucial for recovery from CPM. We present the case of a patient with alcoholism and malnutrition-related CPM, which developed following rapid correction of hyponatremia but then improved remarkably with supportive physical therapy. Case Report: A 44-year-old alcoholic and malnourished man was admitted to an emergency hospital for disorientation due to overdrinking, but later developed bulbar palsy after hyponatremia was unexpectedly, but rapidly, corrected. Axial scans of the diffusion-weighted brain MRI revealed a characteristic lesion known as a piglet sign in the central pons. Based on his underlying conditions, present episode of sodium correction, and MRI finding, the patient was diagnosed as having CPM, which progressively worsened, resulting in locked-in syndrome after 12 days. The patient was then transferred to a long-term care unit and received simple motion exercise daily, but no specific medication. His symptoms gradually improved, achieving discontinuation of tube feeding on day 21, independent walking on day 110, and discharge after 6 months. Conclusions: This report highlights the importance of physical therapy, the potential of which is often underestimated despite its broad benefits for human health, as a readily applicable intervention for patients with CPM. Further understanding of mechanisms underlying exercise-induced myelination should contribute to establishing novel therapies for a wide spectrum of brain disorders.

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A Case of Central Pontine Myelinolysis Caused by Hypophosphatemia Secondary to Refeeding Syndrome

Cited by 8 publications

References 15 publications

Osmotic demyelination syndrome: novel risk factors and proposed pathophysiology

Osmotic demyelination syndrome: novel risk factors and proposed pathophysiology

Treatable Leukoencephalopathy in a Patient with Hypophosphatemia

A 44-Year-Old Alcohol-Dependent Man Who Recovered from Central Pontine Myelinolysis with Supportive Physical Therapy

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