Objective: To  describe a challenging clinical case of akinetic-rigid syndrome in the setting of delayed post-hypoxic leukoencephalopathy (DPHL).

Background: The susceptibility of basal ganglia to hypoxic-ischemic injury can lead to immediate and, less frequently, delayed cortico-striato-thalamo-cortical circuit damage. Akinetic-rigid syndromes resembling catatonia, parkinsonism or mixed phenotipes occur in DPHL, a demyelinating process that manifests days to weeks after apparent recovery from arrest.

Method: Clinical case presentation.

Results: We present a 20-year-old male with haemorrhagic shock and cardiac arrest secondary to a thoracic stabbing wound and lacerated ventricle. Three weeks later, he developed behavioural disturbance and visual hallucinations, for which he was started on Olanzapine by Psychiatry. Initial MRI was reported as normal. During the following week he developed severe akinesia, perplexity, postural maintenance, dysarthria, and affective flattening which evolved to severe rigidity and mutism. He was admitted under Psychiatry for assessment of suspected catatonia. At neurology consultation, patient presented with severe bradykinesia, axial rigidity with dystonic hand posture, anarthria, hypomimia, ocular apraxia and frontal signs. MRI five weeks after hypoxic event (Image 1: A,B) showed bilateral symmetrical T2/FLAIR/ADC hyperintensities in lenticular nuclei and caudates, with slight T1 hyperintensity.  Levodopa and trihexyphenidyl were started for suspected akinetic mutism with parkinsonism due to delayed post-hypoxic leukoencephalopathy. He was discharged with ongoing motor and speech recovery and sustained stable clinical and imaging (Image 1: C,D) findings at the four-month follow-up.

Conclusion: Clinicians should be aware of the diverse neurological and psychiatric presentations of DPHL, a rare but potentially severe demyelinating syndrome. Prompt recognition of DPHL following sudden cardiac arrest and timely initiation of supportive care and rehabilitations improves patient care and clinical outcome.

References: Beeskow, A. B., Oberstadt, M., Saur, D., Hoffmann, K. T., & Lobsien, D. (2018). DelayedPost-hypoxicLeukoencephalopathy(DPHL)-An UncommonVariant ofHypoxicBrain Damage in Adults.Frontiers in neurology,9, 708. https://doi.org/10.3389/fneur.2018.00708.
Jang, S. H., & Kwon, H. G. (2024). Akineticmutismand gaitdisturbancein a patientwithdelayedpost-hypoxicleukoencephalopathy.Neurocase,30(1), 29–31. https://doi.org/10.1080/13554794.2024.2353125.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/when-motion-ceases-twice-delayed-post-hypoxic-leukoencephalopathy-with-parkinsonism-a-case-report/