Osmotic demyelination syndrome improving after immune-modulating treatment: Case report and literature review

Kalampokini, Stefania; Artemiadis, Artemios; Zis, Panagiotis; Hadjihannas, Linos; Parpas, Giorgos; Kyrri, Artemis; Hadjigeorgiou, Georgios M.

doi:10.1016/j.clineuro.2021.106811

Cited by 11 publications

(5 citation statements)

References 62 publications

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“…Clinical manifestation depends on the site of involvement: central pontine myelinolysis (CPM), extrapontine myelinolysis (EPM), or mixed, with typical symptoms appearing 1 -14 days after the initial insult [11]. Common clinical manifestations in CPM, as seen in our patient, include quadriparesis and bulbar weakness, which have been reported in 9.8-28.8% and 3.2 -11.5% cases, respectively.…”

Section: Discussionmentioning

confidence: 62%

Delayed Presentation of Osmotic Demyelination Syndrome Treated With Plasmapheresis

Sharif,

Singh,

Enabi

et al. 2023

Cureus

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We present a unique case of a 42-year-old gentleman with alcohol use disorder who developed osmotic demyelination syndrome (ODS) despite appropriate hyponatremia correction. This patient initially presented with severe hyponatremia (Na 97 mEq/L) due to beer potomania, which was corrected gradually over eight days, resulting in no observed neurological deficits upon discharge. However, he was readmitted with respiratory failure from aspiration pneumonia, leading to endotracheal intubation. Laboratory findings revealed a sodium level of 134 mEq/L and serum osmolality (293 mOsm/kg). The patient had neurological exam findings of spontaneous eye opening with left gaze preference and decreased power ⅕ in all extremities. Following extubation, he experienced a relapse with evolving subacute central pontine myelinolysis and bulbar weakness necessitating reintubation. Subsequently, five sessions of plasmapheresis were conducted, resulting in stable clinical findings. Despite remaining non-verbal, the patient demonstrated gradual neurological motor improvement, progressing from 1/5 power in all extremities to 4/5 on the right side and 3/5 on the left side. He was discharged with ventilator support, tracheostomy, and PEG tube placement to a long-term care facility. This case underscores the importance of vigilant monitoring in high-risk individuals following hyponatremia treatment because ODS presentation can be delayed.

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Section: Discussionmentioning

confidence: 62%

Delayed Presentation of Osmotic Demyelination Syndrome Treated With Plasmapheresis

Sharif,

Singh,

Enabi

et al. 2023

Cureus

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“…There have been some experimental therapies that have been tried with mixed results. All of them are based on immune modulation using plasmapheresis, intravenous immunoglobulin (IVIg), and corticosteroids or a combination of these [8][9][10].…”

Section: Discussionmentioning

confidence: 99%

Osmotic Demyelination After Rapid Correction of Hyperosmolar Hyperglycemia

Jain¹,

Kotwal²,

Gnanaraj³

et al. 2023

Cureus

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Osmotic demyelination syndrome (ODS) is seen due to an overt rise in serum osmolality, most often during rapid correction of chronic hyponatremia. We present the case of a 52-year-old patient who presented with polydipsia, polyuria, and elevated blood glucose with rapid correction of glucose levels under five hours and developed dysarthria, left-sided neglect, and unresponsiveness to light touch and pain in the left extremities on the second day of hospitalization. MRI revealed restricted diffusion in the central pons, extending into extrapontine areas suggestive of ODS. Our case highlights the importance of cautious correction of serum hyperglycemia and monitoring serum sodium levels in patients with a hyperosmolar hyperglycemic state (HHS).

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“…[146][147][148] Albeit many ODS fatalities, a substantial number resulted in favorable outcome due to immune-modulating treatment that can include, among others, plasmapheresis alone or combined with some anti-inflammatory treatment whether in rodents 148 or human. [149][150][151][152][153][154][155][156][157] Those treatments, using a concomitant slow recovery of osmolarity would reinstall circulating homeostasis and further ODS repairs in the milieu surrounding neuropil components to retrieve ephaptic functions. 158…”

Section: Discussionmentioning

confidence: 99%

Loss of Ephaptic Contacts in the Murine Thalamus during Osmotic Demyelination Syndrome

Gilloteaux

Swert

Suain

et al. 2023

Ultrastructural Pathology

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Background and aim:A murine model mimicking osmotic demyelination syndrome (ODS) revealed with histology in the relay posterolateral (VPL) and ventral posteromedial (VPM) thalamic nuclei adjoined nerve cell bodies in chronic hyponatremia, amongst the damaged 12 h and 48 h after reinstatement of osmolality. This report aims to verify and complement with ultrastructure other neurophysiology, immunohistochemistry, and molecular biochemistry data to assess the connexin-36 protein, as part of those hinted close contacts.This ODS investigation included four groups of mice: Sham (NN; n = 13), hyponatremic (HN; n = 11), those sacri ced 12 h after a fast restoration of normal natremia (ODS12h; n = 6) and mice sacri ced 48 h afterward, or ODS48 h (n = 9). Out of these, thalamic zones samples included NN (n = 2), HN (n = 2), ODS12h (n = 3) and ODS48h (n = 3). Results: Ultrastructure illustrated junctions between nerve cell bodies that were immunolabeled with connexin36 (Cx36) with light microscopy and Western blots. These cell's junctions were reminiscent of low resistance junctions characterized in other regions of the CNS with electrophysiology. Contiguous neurons showed neurolemma contacts in intact and damaged tissues according to their location in the ODS zones, at 12 h and 48 h post correction along with other demyelinating alterations. Neurons and ephaptic contact measurements indicated the highest alterations, including nerve cell necrosis in the ODS epicenter and damages decreased toward the outskirts of the demyelinated zone. Conclusion: Ephapses contained C × 36between intact or ODS injured neurons in the thalamus appeared to be resilient beyond the core degraded tissue injuries. These could maintain intercellular ionic and metabolite exchanges between these lesser injured regions and, thus, would partake to some brain plasticity repairs.

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Osmotic demyelination syndrome improving after immune-modulating treatment: Case report and literature review

Cited by 11 publications

References 62 publications

Delayed Presentation of Osmotic Demyelination Syndrome Treated With Plasmapheresis

Delayed Presentation of Osmotic Demyelination Syndrome Treated With Plasmapheresis

Osmotic Demyelination After Rapid Correction of Hyperosmolar Hyperglycemia

Loss of Ephaptic Contacts in the Murine Thalamus during Osmotic Demyelination Syndrome

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