Dados do Trabalho

Título

CASE REPORT: ADOLESCENT WITH MIDBRAIN TUMOR AND HOLMES TREMOR PRESENTING SATISFACTORY RESPONSE TO PROPRANOLOL

Apresentação do caso único

A 17-year-old male presented to the pediatric oncology service in March 2023 with symptoms of right hemiparesis and paresthesias. A 4.4 x 3.2 x 3.0 cm expansive lesion in the left midbrain was identified via cranial MRI. The patient underwent radiotherapy and received oral and intravenous temozolomide. Due to tumor compression, he developed obstructive hydrocephalus necessitating ventriculoperitoneal shunt placement. The patient responded favorably to treatment, showing significant improvement in general condition, functionality, and lesion reduction on imaging. However, he developed a progressive, debilitating tremor in the right upper limb with large amplitude and low frequency, affecting daily and leisure activities and causing psychological distress. Evaluated by the pediatric neurology team, he was diagnosed with Holmes tremor. Initial treatment with propranolol 20mg/day yielded partial response. Subsequent trials of levetiracetam up to 3000mg/day with clonazepam 1mg at night proved insufficient with adverse effects. A second trial of propranolol at 60mg/day demonstrated good tolerance and significant symptom improvement, reducing tremor frequency and amplitude while enhancing voluntary limb control. The patient was able to resume activities such as going to the gym and using the computer.

Discussão

Holmes tremor, first described in 1904 by Gordon Holmes, is a rare movement disorder characterized by a slow (<4,5Hz), large-amplitude tremor that primarily affects the proximal extremities and is usually unilateral. This tremor is typically irregular and can manifest at rest, during action, and with posture. It often results from lesions in the dentato-rubro-thalamic pathway (classically in the midbrain), commonly caused by cerebrovascular accidents, tumors, infections, head trauma, or demyelinating diseases. In children, Holmes tremor is even rarer. The diagnosis is based on clinical and neuroimaging findings. Management of Holmes tremor is challenging and includes pharmacological interventions, such as trihexyphenidyl, levodopa, propranolol, clonazepam, amantadine, bromocriptine or levetiracetam, and, in some cases, neurosurgical procedures like stereotactic thalamotomy or deep brain stimulation may be considered.

Comentários finais

Early diagnosis and a multidisciplinary approach are critical for optimizing clinical outcomes and enhancing quality of life for affected patients.

Referências

PYRGELIS, Efstratios-Stylianos; AGAPIOU, Eleni; ANGELOPOULOU, Efthalia. Holmes tremor: an updated review. Neurological Sciences, v. 43, n. 12, p. 6731-6740, 2022.
ALQWAIFLY, Mohammed. Treatment responsive Holmes tremor: case report and literature review. International journal of health sciences, v. 10, n. 4, p. 558, 2016.
CHANG, Stephano J. et al. Treatment-responsive Holmes tremor in a child with low-pressure hydrocephalus: video case report and systematic review of the literature. Journal of Neurosurgery: Pediatrics, v. 29, n. 5, p. 520-527, 2022.

Palavras Chave

HOLMES-TREMOR; GLIOMA; BRAIN-CANCER