Delayed post-hypoxic leukoencephalopathy (DPHL) is a poorly recognized syndrome characterized by neuropsychiatric symptoms following recovery from an acute hypoxic episode. Although most cases are related to carbon monoxide poisoning, some have been linked to excessive opioid use. Opioid intoxication has recently become known for manifesting the characteristic imaging findings involving cerebellar, hippocampal, and basal nuclei transient edema with restricted diffusion (CHANTER) syndrome. Herein, we present a patient with severe disturbances in consciousness who was initially diagnosed with CO poisoning but was later found to have taken excessive tramadol. Magnetic resonance imaging (MRI) in the acute phase revealed abnormal intensities in the bilateral globus pallidus and the cerebellum, indicative of CHANTER syndrome. After intensive care, his level of consciousness was restored. However, around the 3rd week after hospitalization, his consciousness gradually deteriorated and he developed severe neurological symptoms. Another MRI on day 25 revealed a new diffuse white matter abnormality; DPHL was suspected. Cerebrospinal fluid collected on day 28 revealed significantly elevated myelin basic protein levels. Although it was challenging to decide on a treatment plan, hyperbaric oxygen (HBO) therapy trials were initiated on day 58; the patient's condition improved after a series of HBO sessions. MRI revealed gradual shrinkage of the white matter abnormality. A total of 63 consecutive HBO sessions were performed, leading to the successful resolution of the serious neurological symptoms. While the effectiveness of HBO therapy for DPHL remains inconclusive, especially in opioid-related cases, this patient made a remarkable recovery, likely due to the therapeutic effect of improved cerebral blood flow and oxygenation.

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http://dx.doi.org/10.3389/fmed.2024.1364038DOI Listing

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Article Synopsis
  • CHANTER syndrome is a newly identified neurological condition often triggered by polysubstance or opioid abuse, presenting with acute unresponsiveness or coma.
  • Patients typically show specific MRI findings, particularly restricted diffusion in the cerebellar cortices, hippocampi, and variable changes in the basal ganglia, which may lead to dangerous complications like hydrocephalus.
  • A case study of a 37-year-old man who exhibited these symptoms demonstrated the importance of recognizing CHANTER features early; he improved after treatment and rehabilitation, with follow-up MRIs showing significant recovery.
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Article Synopsis
  • * CHANTER syndrome (cerebellar, hippocampal, and basal nuclei transient edema with restricted diffusion) is identified when all these regions are affected, with the first histopathologic findings linked to this syndrome reported in a patient who died from fentanyl overdose.
  • * Post-mortem examinations revealed various brain injuries, including microhemorrhage and neuronal necrosis, suggesting that both hypoxic-ischemic and cytotoxic mechanisms contribute to CHANTER syndrome, but further research is necessary for targeted treatments. *
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CHANTER syndrome in the context of pain medication: a case report.

BMC Neurol

July 2024

Department of Neuroradiology, Ev. Klinikum Bethel, University Hospital OWL, Campus Bielefeld-Bethel, Bielefeld, Germany.

Background: CHANTER (Cerebellar Hippocampal and Basal Nuclei Transient Edema with Restricted diffusion) is a recently described syndrome occurring in the context of drug abuse. While clinical findings are rather unspecific (disorientation, unresponsiveness), MR imaging (MRI) discloses a characteristic pattern (restricted diffusion in the basal ganglia and hippocampi, cerebellar oedema and haemorrhage), allowing for timely diagnosis before complications such as cerebellar swelling and herniation do occur. Here we report a case of CHANTER primarily based on imaging findings, as there was no evidence of drug abuse on admission.

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Before Diagnosing CHANTER Syndrome, All Possible Differential Diagnoses Must Be Carefully Excluded.

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