Objective: To report a rare case of Holmes Tremor (HT) secondary to HIV-Associated Hydrocephalus and its successful resolution through a combined surgical and pharmacological approach

Background: Holmes tremor (HT) is characterised by rest, postural, and kinetic tremors, secondary to lesion of the dentato-rubro-olivary or cerebello-thalamo-cortical tract involving midbrain, thalamus, and cerebellum.[1] While typically caused by structural lesions, its association with HIV-related hydrocephalus is rarely documented. Hydrocephalus is a common complication of HIV-related central nervous system opportunistic infection.[2] HT secondary to hydrocephalus may arise from axonal dysfunction secondary to mechanical compression of periventricular structures, such as thalamus and midbrain, due to raised ICP and mechanical distention of periventricular white matter tracts due to ventriculomegaly.[3] HT is classically thought to be resistant to pharmacotherapy, although some success has been reported with antiparkinsonian agents (e.g. levodopa), antiepileptics (e.g. levetiracetam)(3), and long-acting benzodiazepines (e.g. clonazepam)[4]

Method: A 42-year-old man living with HIV on TLD Regiment (Tenofovir, Lamivudin, Dolutegravir), presented with subacute fever, headache, and generalised seizures. He had low-frequency (3-4 Hz), coarse-amplitude tremors involving all extremities at rest, raising hands, reaching goal, consistent with HT. Head CT Scan showed meningoencephalitis with moderate communicating hydrocephalus. The CSF analysis showed slight pleocytosis (10 cells/mm³), normal protein and glucose level (9,1 mg/dL; 72 mg/dL), and negative GeneXpert MTB, suggesting viral etiology

Results: The patient underwent Ventriculoperitoneal (VP)-shunting. Brain MRI evaluation showed a remarkable reduction in the degree of hydrocephalus with no other abnormalities, aligned with partial tremor reduction. Clonazepam was added to the therapy. Within the 3-day follow-up, tremors subsided significantly, allowing the patient to regain functional independence

Conclusion: This case demonstrates significant clinical improvement of Holmes Tremor in the context of Hydrocephalus through a multimodal strategy via combination of clonazepam therapy, due to its GABAergic effect, and VP-shunting, which reverses the underlying Hydrocephalus-induced midbrain pathology

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References: 1. Raisa N, Pratiwi AC, Rianawati SB, Munir B. Holmes Tremor and Dystonia as Manifestation of Benedikt Syndrome in HIV-Related Cerebral Toxoplasmosis : A Case Report. Mov Disord. 2023;38(Suppl 1):S585.
2. Loan JJM, Poon MTC, Tominey S, Mankahla N, Meintjes G, Fieggen AG. Ventriculoperitoneal shunt insertion in human immunodeficiency virus infected adults: a systematic review and meta-analysis. BMC Neurol. 2020 Apr;20(1):141.
3. Chang SJ, Mitchell R, Hukin J, Singhal A. Treatment-responsive Holmes tremor in a child with low-pressure hydrocephalus: video case report and systematic review of the literature. J Neurosurg Pediatr [Internet]. 2022;29(5):520–7. Available from: https://thejns.org/pediatrics/view/journals/j-neurosurg-pediatr/29/5/article-p520.xml
4. Jacob PC, Pratap Chand R. Posttraumatic rubral tremor responsive to clonazepam. Mov Disord. 1998 Nov;13(6):977–8.

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