Objective: To report a rare case of episodic generalized dystonia with opisthotonus following anoxic brain injury, characterized by posterior limb of the internal capsule involvement and mild caudate and hippocampal changes.

Background: Opisthotonus posturing is typically linked to meningitis, tetanus, and drug poisoning but is rarely observed in neurodegenerative syndromes or post-hypoxic states. Post-anoxic movement disorders are uncommon after cardiac arrest, with MRI findings usually showing putaminal and pallidal damage. However, episodic, stimulus-induced generalized dystonia with opisthotonus has not been previously reported.

Method: We present a 27-year-old woman with heroin and alprazolam use disorder who suffered a non-shockable cardiac arrest (10-minute downtime). She was comatose upon arrival with preserved brainstem reflexes and no EEG findings of cortical injury. MRI and lumbar puncture ruled out infection or inflammation. Despite sedation, antiepileptics, gabapentin, and opioids, she developed spontaneous clonic movements and dystonic posturing with opisthotonus by day 6, showing limited response to treatment.

Results: MRI revealed restricted diffusion in the posterior limbs of the bilateral internal capsules and mild changes in the caudate heads and hippocampi. The patient exhibited episodic dystonia with truncal and neck opisthotonus, grimaces, and vital sign fluctuations. Hydromorphone and diazepam provided partial relief, while benztropine and amantadine were ineffective.

Conclusion: This case suggests that posterior limb involvement, along with mild caudate and hippocampal changes, may contribute to generalized dystonic posturing after anoxic injury. The episodic nature of opisthotonus was unique and did not follow expected responses to sedatives or antiepileptics. This case highlights the complexity of post-anoxic movement disorders and the need for further research into their mechanisms and management.

References: 1. Bhatt MH, Obeso JA, Marsden CD. Time course of postanoxic akinetic-rigid and dystonic syndromes. Neurology. 1993 Feb;43(2):314-7. doi: 10.1212/wnl.43.2.314. PMID: 8437695.

2. Davis, S. M., Murray, N. M., Diengdoh, J. V., Galea-Debono, A., & Kocen, R. S. (1981). Stimulus-sensitive spinal myoclonus. Journal of Neurology, Neurosurgery, and Psychiatry, 44(10), 884-888. https://doi.org/10.1136/jnnp.44.10.884

3. Stamelou M, Lai SC, Aggarwal A, Schneider SA, Houlden H, Yeh TH, Batla A, Lu CS, Bhatt M, Bhatia KP. Dystonic opisthotonus: a “red flag” for neurodegeneration with brain iron accumulation syndromes? Mov Disord. 2013 Sep;28(10):1325-9. doi: 10.1002/mds.25490. Epub 2013 Jun 4. PMID: 23736975; PMCID: PMC4208296.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/episodic-generalized-dystonia-with-opisthotonus-posturing-in-early-stages-of-anoxic-brain-injury-with-mild-degrees-of-caudate-involvement/