A case of chemotherapy-responsive paraneoplastic rubral tremor

ObjectivesTo further validate the diagnostic utility of 18F-AV-133 vesicular monoamine transporter type 2 (VMAT2) positron emission tomography (PET) in patients with clinically uncertain parkinsonian syndromes (CUPS) by comparison to clinical diagnosis at 3 years follow-up.Design, setting and participantsIn a previous study, we reported that 18F-AV-133 PET in community patients with CUPS changed diagnosis and management and increased diagnostic confidence. The current diagnosis of this cohort was obtained from the patient and treating specialist and compared with the diagnosis suggested 3 years earlier by the 18F-AV-133 PET. A second 18F-AV-133 PET was available in those with a discordant or inconclusive final diagnosis.Study outcome measuresThe primary end point was the proportion of patients who had a follow-up clinical diagnosis, which was concordant with their initial 18F-AV-133 PET scan. Secondary end points were the proportion of patients who had the same diagnosis at follow-up as that reached after the initial scan and the stability of diagnostic changes made after the first scan.Results81 of the 85 patients previously recruited to the CUPS study had follow-up of which 79 had a clinical diagnosis and 2 remained CUPS. The diagnosis was in agreement with the initial 18F-AV-133 PET scan result in 74 cases. Five patients had a discordant diagnosis; one patient with rubral tremor had a severely abnormal scan that had worsened when rescanned; four cases with normal initial and repeat scans had a clinical diagnosis of Parkinson’s disease. Two patients with suspected genetic disorders remained classified as CUPS and both had normal scans. In the 24 CUPS cohort patients where 18F-AV-133 PET initially changed diagnosis, this change was supported by follow-up diagnosis in all but the one rubral tremor case.Conclusion18F-AV-133 PET is a useful tool in improving diagnostic accuracy in CUPS providing results and diagnostic changes that remain robust after 3 years follow-up.

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Rubral tremor

Neurology ◽

10.1212/wnl.46.1.288-b ◽

1996 ◽

Vol 46 (1) ◽

pp. 288-288 ◽

Cited By ~ 4

Author(s):

B. Okuda ◽

H. Tachibana

Keyword(s):

Rubral Tremor

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Rubral tremor associated with cavernous angioma of the midbrain

Movement Disorders ◽

10.1002/1531-8257(199901)14:1<191::aid-mds1043>3.0.co;2-y ◽

1999 ◽

Vol 14 (1) ◽

pp. 191-193 ◽

Cited By ~ 11

Author(s):

G. K. K. Leung ◽

Y. W. Fan ◽

S. L. Ho

Keyword(s):

Cavernous Angioma ◽

Rubral Tremor

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Functional lesional neurosurgery for tremor: a systematic review and meta-analysis

Journal of Neurology Neurosurgery & Psychiatry ◽

10.1136/jnnp-2017-316302 ◽

2018 ◽

Vol 89 (7) ◽

pp. 717-726 ◽

Cited By ~ 7

Author(s):

Sebastian R Schreglmann ◽

Joachim K Krauss ◽

Jin Woo Chang ◽

Kailash P Bhatia ◽

Georg Kägi

Keyword(s):

Multiple Sclerosis ◽

Systematic Review ◽

Radiofrequency Ablation ◽

Side Effects ◽

Focused Ultrasound ◽

Meta Analysis ◽

Consistency Effect ◽

Rubral Tremor ◽

Ventral Intermediate Nucleus ◽

Mri Guided

BackgroundThis work evaluates the consistency, effect size and incidence of persistent side effects of lesional neurosurgical interventions in the treatment of tremor due to Parkinson’s disease (PD), essential tremor (ET), multiple sclerosis (MS) and midbrain lesions.MethodsSystematic review and meta-analysis according to PRISMA-P guidelines. Random effects meta-analysis of standardised mean difference based on a peer-reviewed protocol (PROSPERO no. CRD42016048049).ResultsFrom 1249 abstracts screened, 86 peer-reviewed studies reporting 102 cohorts homogeneous for tremor aetiology, surgical target and technique were included.Effect on PD tremor was better when targeted at the ventral intermediate nucleus (V.im.) by radiofrequency ablation (RF) (Hedge’s g: −4.15;) over V.im. by Gamma Knife (GK) (−2.2), subthalamic nucleus (STN) by RF (−1.12) and globus pallidus internus (GPi) by RF (−0.89). For ET MRI-guided focused ultrasound (MRIgFUS) ablation of the cerebellothalamic tract (CTT) (−2.35) and V.im. (−2.08) showed similar mean tremor reductions to V.im. ablation by RF (−2.42) or GK (−2.13). In MS V.im. ablation by GK (−1.96) and RF (−1.63) were similarly effective.Mean rates of persistent side effects after unilateral lesions in PD were 12.8% (RF V.im.), 13.6% (RF STN), 9.2% (RF GPi), 0.7% (GK V.im.) and 7.0% (MRIgFUS V.im.). For ET, rates were 9.3% (RF V.im.), 1.8% (GK V.im.), 18.7% (MRIgFUS V.im.) and 0.0% (MRIgFUS CTT), for MS 37.7% (RF V.im.) and for rubral tremor 30.3% (RF V.im.).ConclusionThis meta-analysis quantifies safety, consistency and efficacy of lesional neurosurgical interventions for tremor by target, technique and aetiology.

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