1Institute of Post Graduate Medical Education & Research and Bangur Institute of Neurosciences, West Bengal, India
2Department of Radiology, Apollo Multispeciality Hospitals, West Bengal, India
Copyright © 2024 The Korean Movement Disorder Society
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Conflicts of Interest
The authors have no financial conflicts of interest.
Funding Statement
None
Author contributions
Conceptualization: Amlan Kusum Datta, Adreesh Mukherjee. Data curation: Amlan Kusum Datta, Adreesh Mukherjee. Formal analysis: all authors. Investigation: Amlan Kusum Datta, Adreesh Mukherjee, Sudeshna Malakar. Methodology: Amlan Kusum Datta, Adreesh Mukherjee, Atanu Biswas. Project administration: Atanu Biswas, Amlan Kusum Datta. Resources: Amlan Kusum Datta, Adreesh Mukherjee, Atanu Biswas. Software: Amlan Kusum Datta, Adreesh Mukherjee, Atanu Biswas. Supervision: Atanu Biswas, Adreesh Mukherjee. Validation: Sudeshna Malakar, Adreesh Mukherjee, Atanu Biswas. Visualization: Amlan Kusum Datta, Adreesh Mukherjee, Atanu Biswas. Writing—original draft: Amlan Kusum Datta. Writing—review & editing: Adreesh Mukherjee, Atanu Biswas, Sudeshna Malakar.
Gajos et al., [2] 2010 | Raina et al., [3] 2016 | Nsengiyumva et al., [6] 2021 | Mishra et al., [22] 2022 | Present study | |
---|---|---|---|---|---|
Number of patients | 10 | 29 | 17 | 12 | 9 |
Gender distribution (male:female) | 5:5 | 13:16 | 9:8 | 11:1 | 4:5 |
Mean age of onset, yr | 44.8 ± 20.2 | 33.9 ± 20.1 | 45 ± 18.39 | 31.08 ± 10.1 | 53.3 ± 8.4 |
Latency between initia insult and onset of tremor | Mean: 6.3 months (range: 1 month–2 years) | Median: 2 months (range: 7 days–228 months) | Range: 8 weeks–14 years | Mean: 4.78 months (range: 7days–1 year) | Mean: 50.4 days (range: 21–90 days) |
Etiology | Vascular (n = 8; 80%) | Vascular (n = 14; 48.3%) | Vascular (n = 11; 64.7%) | Vascular (n = 8, 66.7%) | Vascular (100%) |
Head trauma (n = 2; 20%) | Head trauma (n = 5; 17.2%) | Head trauma (n = 2; 11.8%) | Head trauma (n = 2, 16.7%) | ||
Miscellaneous (n = 10; | Others (n = 4, 23.5%) | Tumor resection (n = 2, 16.7%) | |||
Neurological deficit(s) (apart from hyperkinetic movement) | Hemiparesis (n = 5; 50%) | Hemiparesis (n = 18; 62%) | Mild hemiparesis or monoparesis (n = 5; 29.41%) | Hemiparesis (n = 7; 58.3%) | Hemiparesis (n = 6; 66.6%) |
Cranial neuropathies (n = 7; 70%) | Ataxia (n = 15; 51.7%) | Cranial neuropathy (n = 6; 35.3%) | Ataxia (n = 7; 58.3%) | Monoparesis (n = 2; 22.2%) | |
Ataxia (n = 3; 30%) | Hypesthesia (n = 8; 27.58%) | Ataxia (n = 5; 29.4%) | Hemi-anesthesia (n = 2; 16.7%) | Abnormal proprioception (n = 4; 44.4%) | |
Atrophy of small muscles of hand (n = 1; 10%) | Cranial neuropathy (n = 7; 24.1%) | Proprioceptive sensory loss in affected limb (n = 4; 23.5%) | Quadriparesis (n = 2; 16.7%) | ||
Hemi-hypesthesia (n = 1; 10%) | Dysarthria (n = 7; 24.1%) | Dysarthria (n = 2; 16.7%) | Hemi-hypesthesia (n = 2; 22.2%) | ||
Depression (n = 1; 10%) | Vertical gaze abnormality (n = 2; 6.9%) | Dysarthria (n = 2; 11.8%) | Cognitive impairments (n = 2; 16.7%) | Cranial neuropathy (n = 2; 22.2%) | |
Dementia (n = 1; 10%) | Seizures (n = 1; 3.4%) | Cognitive impairment (n = 2; 11.8%) | Seizures (n = 1; 8.3%) | Dysarthria (n = 1; 11.1%) | |
Psychiatric disorders (n = 1; 3.4%) | Parinaud’s syndrome (n = 1; 5.9%) | Vertical gaze restriction (n = 1; 8.3%) | Psychiatric abnormalities (n = 2; 22.2%) | ||
Homonymous hemianopia (n = 1; 8.3%) | |||||
Associated movements | None reported | Dystonia (n = 7; 24.1%); 3 patients had dystonic jerks | Dystonia of affected limb (n = 6; 20.7%). | Dystonia (n = 10; 83.3%) | Dystonia (n = 8; 88.8%) |
One patient experienced dystonic jerks “no-no” head tremor (n = 2; 11.8%) | Tongue dyskinesia (n = 1; 8.3%) | Chorea (n = 1; 11.1%) | |||
Myoclonus (n = 1; 3.4%) | Pseudoathetosis (n = 2; 11.8%) | Tongue protrusion tremor (n = 1; 8.3%) | Dystonic head tremor (n = 1; 11.1%) | ||
Distal choreiform movement of affected limb (n = 2; 11.8%) | Frontalis dystoina (n = 1; 11.1%) | ||||
Distribution of tremor phenotype | Unilateral tremor in all (n = 10; 100%) | One upper limb (n = 19; 65.5%) | All were unilateral | Unilateral upper limb (n = 6; 50%) | Distal predominance (n = 4; 44.4%) |
Upper and lower limb (n = 2; 20%) | Upper and lower limb (n = 6; 20.7%) | Single patient (n = 1; 5.9%) had involvement of ipsilateral lower limb (dystonia) | Unilateral upper and lower limb (n = 4; 33.3%) | Proximal predominance (n = 2; 22.2%) | |
Distal predominant (n = 1; 10%) | Both upper limbs (n = 4; 13.7%) | Bilateral upper limb tremor (n = 1; 8.3%) | Lower limb involvement (n = 2; 22.2%) | ||
Proximal predominant (n = 3; 30%) | Tremor involved proximal and distal segment of limbs in n = 16 (55.2%), with distal involvement in n = 3 (10.3%) | Myorhythmic rest tremor (n = 12; 70.6%) | Head tremor (n = 4; 33.3%; “nono” type in one, titubatory in one, non-specified in two) | Bilateral involvement (n = 2; 22.2%) | |
Proximal and distal (n = 7; 70%) | Distal more than proximal involvement in n = 1 (5.9%), with all others having proximal predominance | Distal predominance (n = 9; 75%) | Voice tremor (n = 1; 11.1%) | ||
Proximal predominance (n = 2; 16.7%) | |||||
Proximal and distal equal distribution (n = 1; 8.3%) | |||||
Neuroimaging results | MRI was done in n = 9 (90%) and CT in n = 1 (10%) patient(s) | MRI was performed in n = 28 (96.5%) patients while CT scan was done in n = 1 (3.4%) patient | Abnormal brain imaging (n = 16; 94.1%) | All had abnormal neuroimaging | n = 9 (100%) patients had abnormal neuroimaging |
SPECT was performed for 6 (n = 6; 60%) patients. Did not reveal any asymmetry | Midbrain (n = 5; 29.4%) | Midbrain (n = 4; 33.3%) | Thalamus (n = 9; 100%) | ||
Single region was involved in n = 6 (60%), more than one region n = 3 (30%) and no visible abnormality in n = 1 (10%) | Neuroimaging was abnormal in n = 28 (96.5%) | Thalamus (n = 5; 29.4%) | Thalamus (n = 4; 33.3%) | Basal ganglia (n =1; 10%) | |
Midbrain (n = 17; 58.6%), thalamus (n = 16; 55.2%), cerebellum (n = 8; 27.6%), pons (n = 6; 20.7%), medulla (n = 1; 3.4%) | Cerebellum and/or peduncles (n = 2; 11.7%) | Cerebellum (n = 3; 25%) | Occipital lobe (n = 1;10%) | ||
Thalamus (n = 5; 50%), midbrain (n = 3; 30%) and pons (n = 1; 10%) | Lobar (n = 3; 25%, 2 in occipital and 1 in temporal lobe) | Capsule-thalamic (n = 1; 10%) | |||
Basal ganglia (n = 1; 8.3%) | |||||
Cerebello-pontine angle (n = 1; 8.3%) | |||||
Response to levodopa | n = 2 (out of 6; 33.3%) patients treated with levodopa experienced improvement at doses of 150 mg/day (additional treatment with piribedil 150 mg/day) and 400 mg/day (additional treatment with propranolol 40 mg/day) respectively | Levodopa was tried in n = 24 patients (82.8%) with improvement noted in n = 13 (out of 24; 54.2%) | Improvement noted in n = 5 (out of n = 16) patients who took levodopa (31.2%) | n = 12 (100%) patients were treated with levodopa | n = 9 (100%) patients were administered multi-drug therapy, with levodopa (up to 450 mg/day) added first and other drugs added sequentially in various combination and doses (trihexyphenidyl, clonazepam, topiramate and levetiracetam) |
n = 9 (75%) patients showed improvement | |||||
Two patients were additionally treated with topiramate (100 mg/day) and pramipexole (1.5 mg/day) respectively | n = 1 patient developed levodopa induced dyskinesia of upper limb which was treated with amantadine (100 mg, three times/day) | Improvement was noted in n = 5 (55.5%) patients |
Responders (n = 5) | Non-responders (n = 4) | p value | |
---|---|---|---|
Mean TETRAS (PS) before therapy | 19.0 ± 3.5 | 19.3 ± 3.8 | 0.920 |
Mean TETRAS (PS) after therapy | 9.6 ± 1.1 | 16.8 ± 4.4 | 0.014* |
Mean TETRAS (PS) percentage reduction | 48.8 ± 6.3 | 13.8 ± 8.4 | 0.014* |
Mean TETRAS (ADLS) before therapy | 31.2 ± 10.2 | 33.8 ± 12.5 | 0.623 |
Mean TETRAS (ADLS) after therapy | 17.8 ± 6.9 | 30.8 ± 13.4 | 0.142 |
Mean TETRAS (ADLS) percentage reduction | 44.4 ± 6.8 | 11.4 ± 11.2 | 0.014* |
Levodopa dose (mean), mg/day | 240 ± 54.7 | 400 ± 40.8 | 0.012* |
Latency (in weeks) | 7.4 ± 4.3 | 6.3 ± 4.0 | 0.705 |
Study (n = number of participants) | Aetiology and anatomy of lesion | Functional imaging and ligand used | Results of functional imaging | Response to therapy |
---|---|---|---|---|
Remy et al., [7] 1995 (n = 6) | Head injury (n = 2) | [18F-DOPA] PET | Marked decrease in ipsilateral striatal uptake | All patients responded to levodopa (dramatic response in n = 2, good response in n = 2, and partial response in n = 2) |
Bullet injury (n = 1) | [76Br]PET | |||
Haemorrhagic stroke (n = 3) | No asymmetry of D2 uptake | |||
All lesions in midbrain | ||||
Paviour et al., [29] 2006 (n = 1) | Midbrain cavernoma | DAT scan (ligand not specified) | Markedly decreased striatal uptake ipsilateral to lesion | No response to levodopa |
Hertel et al., [30] 2006 (n = 1) | Midbrain (aetiology not specified) | IBZM-SPECT | Normal post and pre-synaptic uptake | Responded well to surgical therapy (VP shunt followed by DBS) |
[123I]-beta-CIT-SPECT (DAT Scan) | ||||
Strecker et al., [31] 2007 (n = 1) | Midbrain abscess | DAT scan (123I SPECT) | Decreased uptake on putamen ipsilateral to lesion | Good response (almost complete amelioration of rest tremor with some persistence of kinetic tremor) |
Guedj et al., [32] 2007 (n = 1) | Head trauma | DAT scan (FP CIT SPECT) | No striatal uptake | Excellent response to DBS (target: VIM) |
Cerebral peduncle | ||||
Seidel et al., [4] 2009 (n = 1) | Brainstem cavernoma bleed (left midbrain and pons) | DAT scan (Beta-CIT SPECT) | No striatal uptake | Good response to dopaminergic therapy |
Sung et al., [33] 2009 (n = 1) | Left thalamic haemorrhage | 99mTc-TRODAT-1 SPECT | Bilateral decrease in striatal uptake (more ipsilateral to lesion) | Poor response to dopaminergic therapy |
Gajos et al., [2] 2010 (n = 10) | Ischemic stroke-4 | [123I] DAT scan (SPECT) | No asymmetry of uptake | 6 patients treated with levodopa, 2 improved |
Hemorrhage-6 (2 TBI, 2 ICH, 1 cavernoma, 1 AVM) | ||||
4 thalamus | ||||
5 brainstem | ||||
1 no lesion visible | ||||
Reese et al., [34] 2011 (n = 1) | TBI (subdural hemorrhage) | DAT scan | Marked reduction in uptake in contralateral brain hemisphere | Transient response to dopaminergic therapy |
123I-FP-CIT SPECT | Treated with DBS (target VIM and STN) | |||
Good response to surgical therapy | ||||
Juri et al., [35] 2015 (n = 1) | Midbrain cavernoma bleed | 18F-PR04.MZ PET | Marked reduction in uptake at ipsilateral striatum | Marked response to levodopa/carbidopa |
Gajos et al., [36] 2017 (n = 3) | 2 thalamic ischemic stroke 1 TBI | [123I]-FP CIT—DaTSCAN | Normal pre and post synaptic uptake | 2 patients treated with levodopa did not improve |
IBZM SPECT |
18F-DOPA, 18-Fluorodopa; PET, positron emission tomogram; 76Br, bromolisuride; DAT, dopamine active transporter; IBZM, 123I-iodobenzamide; SPECT, single photon emission computed tomography; 123I-FP, ioflupane; 123I-FP-CIT, 123-radiolabeled 2β-carbomethoxy-3β-(4-iodophenyl)-N-(3-fluoropropyl) nortropane; DBS, deep brain stimulation; VIM, ventral intermediate nucleus of thalamus; TBI, traumatic brain injury; ICH, intracerebral hemorrhage; AVM, arterio-veinous malformation; STN, subthalamic nucleus.
Comments on this article
Gajos et al., [2] 2010 | Raina et al., [3] 2016 | Nsengiyumva et al., [6] 2021 | Mishra et al., [22] 2022 | Present study | |
---|---|---|---|---|---|
Number of patients | 10 | 29 | 17 | 12 | 9 |
Gender distribution (male:female) | 5:5 | 13:16 | 9:8 | 11:1 | 4:5 |
Mean age of onset, yr | 44.8 ± 20.2 | 33.9 ± 20.1 | 45 ± 18.39 | 31.08 ± 10.1 | 53.3 ± 8.4 |
Latency between initia insult and onset of tremor | Mean: 6.3 months (range: 1 month–2 years) | Median: 2 months (range: 7 days–228 months) | Range: 8 weeks–14 years | Mean: 4.78 months (range: 7days–1 year) | Mean: 50.4 days (range: 21–90 days) |
Etiology | Vascular (n = 8; 80%) | Vascular (n = 14; 48.3%) | Vascular (n = 11; 64.7%) | Vascular (n = 8, 66.7%) | Vascular (100%) |
Head trauma (n = 2; 20%) | Head trauma (n = 5; 17.2%) | Head trauma (n = 2; 11.8%) | Head trauma (n = 2, 16.7%) | ||
Miscellaneous (n = 10; | Others (n = 4, 23.5%) | Tumor resection (n = 2, 16.7%) | |||
Neurological deficit(s) (apart from hyperkinetic movement) | Hemiparesis (n = 5; 50%) | Hemiparesis (n = 18; 62%) | Mild hemiparesis or monoparesis (n = 5; 29.41%) | Hemiparesis (n = 7; 58.3%) | Hemiparesis (n = 6; 66.6%) |
Cranial neuropathies (n = 7; 70%) | Ataxia (n = 15; 51.7%) | Cranial neuropathy (n = 6; 35.3%) | Ataxia (n = 7; 58.3%) | Monoparesis (n = 2; 22.2%) | |
Ataxia (n = 3; 30%) | Hypesthesia (n = 8; 27.58%) | Ataxia (n = 5; 29.4%) | Hemi-anesthesia (n = 2; 16.7%) | Abnormal proprioception (n = 4; 44.4%) | |
Atrophy of small muscles of hand (n = 1; 10%) | Cranial neuropathy (n = 7; 24.1%) | Proprioceptive sensory loss in affected limb (n = 4; 23.5%) | Quadriparesis (n = 2; 16.7%) | ||
Hemi-hypesthesia (n = 1; 10%) | Dysarthria (n = 7; 24.1%) | Dysarthria (n = 2; 16.7%) | Hemi-hypesthesia (n = 2; 22.2%) | ||
Depression (n = 1; 10%) | Vertical gaze abnormality (n = 2; 6.9%) | Dysarthria (n = 2; 11.8%) | Cognitive impairments (n = 2; 16.7%) | Cranial neuropathy (n = 2; 22.2%) | |
Dementia (n = 1; 10%) | Seizures (n = 1; 3.4%) | Cognitive impairment (n = 2; 11.8%) | Seizures (n = 1; 8.3%) | Dysarthria (n = 1; 11.1%) | |
Psychiatric disorders (n = 1; 3.4%) | Parinaud’s syndrome (n = 1; 5.9%) | Vertical gaze restriction (n = 1; 8.3%) | Psychiatric abnormalities (n = 2; 22.2%) | ||
Homonymous hemianopia (n = 1; 8.3%) | |||||
Associated movements | None reported | Dystonia (n = 7; 24.1%); 3 patients had dystonic jerks | Dystonia of affected limb (n = 6; 20.7%). | Dystonia (n = 10; 83.3%) | Dystonia (n = 8; 88.8%) |
One patient experienced dystonic jerks “no-no” head tremor (n = 2; 11.8%) | Tongue dyskinesia (n = 1; 8.3%) | Chorea (n = 1; 11.1%) | |||
Myoclonus (n = 1; 3.4%) | Pseudoathetosis (n = 2; 11.8%) | Tongue protrusion tremor (n = 1; 8.3%) | Dystonic head tremor (n = 1; 11.1%) | ||
Distal choreiform movement of affected limb (n = 2; 11.8%) | Frontalis dystoina (n = 1; 11.1%) | ||||
Distribution of tremor phenotype | Unilateral tremor in all (n = 10; 100%) | One upper limb (n = 19; 65.5%) | All were unilateral | Unilateral upper limb (n = 6; 50%) | Distal predominance (n = 4; 44.4%) |
Upper and lower limb (n = 2; 20%) | Upper and lower limb (n = 6; 20.7%) | Single patient (n = 1; 5.9%) had involvement of ipsilateral lower limb (dystonia) | Unilateral upper and lower limb (n = 4; 33.3%) | Proximal predominance (n = 2; 22.2%) | |
Distal predominant (n = 1; 10%) | Both upper limbs (n = 4; 13.7%) | Bilateral upper limb tremor (n = 1; 8.3%) | Lower limb involvement (n = 2; 22.2%) | ||
Proximal predominant (n = 3; 30%) | Tremor involved proximal and distal segment of limbs in n = 16 (55.2%), with distal involvement in n = 3 (10.3%) | Myorhythmic rest tremor (n = 12; 70.6%) | Head tremor (n = 4; 33.3%; “nono” type in one, titubatory in one, non-specified in two) | Bilateral involvement (n = 2; 22.2%) | |
Proximal and distal (n = 7; 70%) | Distal more than proximal involvement in n = 1 (5.9%), with all others having proximal predominance | Distal predominance (n = 9; 75%) | Voice tremor (n = 1; 11.1%) | ||
Proximal predominance (n = 2; 16.7%) | |||||
Proximal and distal equal distribution (n = 1; 8.3%) | |||||
Neuroimaging results | MRI was done in n = 9 (90%) and CT in n = 1 (10%) patient(s) | MRI was performed in n = 28 (96.5%) patients while CT scan was done in n = 1 (3.4%) patient | Abnormal brain imaging (n = 16; 94.1%) | All had abnormal neuroimaging | n = 9 (100%) patients had abnormal neuroimaging |
SPECT was performed for 6 (n = 6; 60%) patients. Did not reveal any asymmetry | Midbrain (n = 5; 29.4%) | Midbrain (n = 4; 33.3%) | Thalamus (n = 9; 100%) | ||
Single region was involved in n = 6 (60%), more than one region n = 3 (30%) and no visible abnormality in n = 1 (10%) | Neuroimaging was abnormal in n = 28 (96.5%) | Thalamus (n = 5; 29.4%) | Thalamus (n = 4; 33.3%) | Basal ganglia (n =1; 10%) | |
Midbrain (n = 17; 58.6%), thalamus (n = 16; 55.2%), cerebellum (n = 8; 27.6%), pons (n = 6; 20.7%), medulla (n = 1; 3.4%) | Cerebellum and/or peduncles (n = 2; 11.7%) | Cerebellum (n = 3; 25%) | Occipital lobe (n = 1;10%) | ||
Thalamus (n = 5; 50%), midbrain (n = 3; 30%) and pons (n = 1; 10%) | Lobar (n = 3; 25%, 2 in occipital and 1 in temporal lobe) | Capsule-thalamic (n = 1; 10%) | |||
Basal ganglia (n = 1; 8.3%) | |||||
Cerebello-pontine angle (n = 1; 8.3%) | |||||
Response to levodopa | n = 2 (out of 6; 33.3%) patients treated with levodopa experienced improvement at doses of 150 mg/day (additional treatment with piribedil 150 mg/day) and 400 mg/day (additional treatment with propranolol 40 mg/day) respectively | Levodopa was tried in n = 24 patients (82.8%) with improvement noted in n = 13 (out of 24; 54.2%) | Improvement noted in n = 5 (out of n = 16) patients who took levodopa (31.2%) | n = 12 (100%) patients were treated with levodopa | n = 9 (100%) patients were administered multi-drug therapy, with levodopa (up to 450 mg/day) added first and other drugs added sequentially in various combination and doses (trihexyphenidyl, clonazepam, topiramate and levetiracetam) |
n = 9 (75%) patients showed improvement | |||||
Two patients were additionally treated with topiramate (100 mg/day) and pramipexole (1.5 mg/day) respectively | n = 1 patient developed levodopa induced dyskinesia of upper limb which was treated with amantadine (100 mg, three times/day) | Improvement was noted in n = 5 (55.5%) patients |
Case | Latency | Other hyperkinetic movements | Location and type of lesion | Tremor predominance and distribution | Other neurological deficits | Treatment | Response to therapy |
|
---|---|---|---|---|---|---|---|---|
Before TETRAS | After TETRAS | |||||||
1 | 3 weeks | Chorea, dystonia, of right upper limb > lower limb | Left thalamic infarct (thalamo-geniculate territory) | Distal = Proximal | Right hemiparesis | LDP (400 mg/day), THP (6 mg/day), TOP (100 mg/day), CLON (1 mg/day) | ADLS: 30 | ADLS: 28 |
Upper limb | Proprioceptive deficits of ipsilateral limb | PS: 16 | PS: 14 | |||||
2 | 6 weeks | Dystonia of right upper limb, dystonic head tremor | Left thalamic gliosis, post hemorrhage | Proximal > Distal | Right upper limb monoparesis | LDP (350 mg/day), CLON (1 mg/day), THP (8 mg/day), LVT (1 gm/day) | ADLS: 46 | ADLS: 42 |
Upper limb | Proprioceptive deficits of right hand | PS: 22 | PS: 21 | |||||
3 | 6 weeks | Left upper limb dystonia | Right sided thalamic (posterior choroidal territory) and occipital infarction | Distal > Proximal | Proprioceptive deficits in affected limb | LDP (300 mg/day), CLON (1 mg/day), THP (6 mg/day) | ADLS:13 | ADLS: 6 |
Upper limb | PS: 15 | PS: 8 | ||||||
4 | 3 months | No | Left sided thalamic infarct (thalamo-geniculate territory) | Proximal > Distal | Right upper limb monoparesis | LDP (450 mg/day), THP (6 mg/day), CLON (1 mg/day), LVT (1 gm/day) | ADLS: 41 | ALDS: 40 |
Upper limb | Abnormal proprioception in affected hand | PS: 23 | PS: 20 | |||||
5 | 4 weeks | Right upper limb dystonia | Left sided thalamic infarct (thalamo-geniculate) | Distal > Proximal | Right sided hemiparesis | LDP (300 mg/day), CLON (1 mg/day), THP (3 mg/day) | ADLS: 35 | ADLS: 18 |
Bilateral upper limb tremor (right > left) | PS: 21 | PS: 11 | ||||||
6 | 3 weeks | Right upper limb dystonia | Left sided capsule-thalamic hemorrhagic stroke | Distal > Proximal | Right sided hemiparesis, right hemi-hypesthesia right lower facial weakness | LDP (200 mg/day), THP (6 mg/day) | ADLS: 36 | ADLS: 22 |
Upper limb | PS: 17 | PS: 10 | ||||||
7 | 1 month | Left upper limb dystonia | Right sided capsule-thalamic hemorrhagic stroke | Distal > Proximal | Left sided hemiparesis, left hemi-hypesthesia left lower facial weakness | LDP (400 mg/day), CLON (1 mg/day), THP (12 mg/day) | ADLS: 18 | ADLS: 13 |
Upper limb | PS: 16 | PS: 12 | ||||||
8 | 3 months | Choreodystonia (bilateral involvement, asymmetric) | Right sided thalamic and left sided putaminal gliosis (post ischemic) | Distal = Proximal | Left upper limb monoparesis, left hemi-hypesthesia | LDP (200 mg/day), THP (4 mg/day), CLON (0.5 mg/day), MIRT (7.5 mg/day) | ADLS: 35 | ADLS: 20 |
Postural tremor of bilateral upper limb with more amplitude on left | Depression and psychosomatic complaints | PS: 24 | PS: 10 | |||||
Intention tremor of left upper limb | Cerebellar (“scanning”) speech | |||||||
9 | 3 months | Dystonia of right hand | Left thalamic hemorrhage | Distal = Proximal | Right sided spastic hemiparesis | LDP (200 mg/day), THP (4 mg/day), CLON (1 mg/day) | ADLS: 37 | ADLS: 23 |
Right upper limb | Frontalis dystonia | PS: 18 | PS: 9 | |||||
Depression |
Responders (n = 5) | Non-responders (n = 4) | p value | |
---|---|---|---|
Mean TETRAS (PS) before therapy | 19.0 ± 3.5 | 19.3 ± 3.8 | 0.920 |
Mean TETRAS (PS) after therapy | 9.6 ± 1.1 | 16.8 ± 4.4 | 0.014 |
Mean TETRAS (PS) percentage reduction | 48.8 ± 6.3 | 13.8 ± 8.4 | 0.014 |
Mean TETRAS (ADLS) before therapy | 31.2 ± 10.2 | 33.8 ± 12.5 | 0.623 |
Mean TETRAS (ADLS) after therapy | 17.8 ± 6.9 | 30.8 ± 13.4 | 0.142 |
Mean TETRAS (ADLS) percentage reduction | 44.4 ± 6.8 | 11.4 ± 11.2 | 0.014 |
Levodopa dose (mean), mg/day | 240 ± 54.7 | 400 ± 40.8 | 0.012 |
Latency (in weeks) | 7.4 ± 4.3 | 6.3 ± 4.0 | 0.705 |
Study (n = number of participants) | Aetiology and anatomy of lesion | Functional imaging and ligand used | Results of functional imaging | Response to therapy |
---|---|---|---|---|
Remy et al., [7] 1995 (n = 6) | Head injury (n = 2) | [18F-DOPA] PET | Marked decrease in ipsilateral striatal uptake | All patients responded to levodopa (dramatic response in n = 2, good response in n = 2, and partial response in n = 2) |
Bullet injury (n = 1) | [76Br]PET | |||
Haemorrhagic stroke (n = 3) | No asymmetry of D2 uptake | |||
All lesions in midbrain | ||||
Paviour et al., [29] 2006 (n = 1) | Midbrain cavernoma | DAT scan (ligand not specified) | Markedly decreased striatal uptake ipsilateral to lesion | No response to levodopa |
Hertel et al., [30] 2006 (n = 1) | Midbrain (aetiology not specified) | IBZM-SPECT | Normal post and pre-synaptic uptake | Responded well to surgical therapy (VP shunt followed by DBS) |
[123I]-beta-CIT-SPECT (DAT Scan) | ||||
Strecker et al., [31] 2007 (n = 1) | Midbrain abscess | DAT scan (123I SPECT) | Decreased uptake on putamen ipsilateral to lesion | Good response (almost complete amelioration of rest tremor with some persistence of kinetic tremor) |
Guedj et al., [32] 2007 (n = 1) | Head trauma | DAT scan (FP CIT SPECT) | No striatal uptake | Excellent response to DBS (target: VIM) |
Cerebral peduncle | ||||
Seidel et al., [4] 2009 (n = 1) | Brainstem cavernoma bleed (left midbrain and pons) | DAT scan (Beta-CIT SPECT) | No striatal uptake | Good response to dopaminergic therapy |
Sung et al., [33] 2009 (n = 1) | Left thalamic haemorrhage | 99mTc-TRODAT-1 SPECT | Bilateral decrease in striatal uptake (more ipsilateral to lesion) | Poor response to dopaminergic therapy |
Gajos et al., [2] 2010 (n = 10) | Ischemic stroke-4 | [123I] DAT scan (SPECT) | No asymmetry of uptake | 6 patients treated with levodopa, 2 improved |
Hemorrhage-6 (2 TBI, 2 ICH, 1 cavernoma, 1 AVM) | ||||
4 thalamus | ||||
5 brainstem | ||||
1 no lesion visible | ||||
Reese et al., [34] 2011 (n = 1) | TBI (subdural hemorrhage) | DAT scan | Marked reduction in uptake in contralateral brain hemisphere | Transient response to dopaminergic therapy |
123I-FP-CIT SPECT | Treated with DBS (target VIM and STN) | |||
Good response to surgical therapy | ||||
Juri et al., [35] 2015 (n = 1) | Midbrain cavernoma bleed | 18F-PR04.MZ PET | Marked reduction in uptake at ipsilateral striatum | Marked response to levodopa/carbidopa |
Gajos et al., [36] 2017 (n = 3) | 2 thalamic ischemic stroke 1 TBI | [123I]-FP CIT—DaTSCAN | Normal pre and post synaptic uptake | 2 patients treated with levodopa did not improve |
IBZM SPECT |
MRI, magnetic resonance imaging; CT, computed tomography; SPECT, single photon emission computed tomography.
TETRAS, the essential tremor ratings assessment scale; LDP, levodopa; THP, trihexyphenidyl; TOP, topiramate; CLON, clonazepam; ADLS, activities of daily living score; PS, performance score; LVT, levetiracetam; MIRT, mirtazapine.
Values are presented as mean ± standard deviation unless otherwise indicated. TETRAS, the essential tremor ratings assessment scale; PS, performance score; ADLS, activities of daily living score.
18F-DOPA, 18-Fluorodopa; PET, positron emission tomogram; 76Br, bromolisuride; DAT, dopamine active transporter; IBZM, 123I-iodobenzamide; SPECT, single photon emission computed tomography; 123I-FP, ioflupane; 123I-FP-CIT, 123-radiolabeled 2β-carbomethoxy-3β-(4-iodophenyl)-N-(3-fluoropropyl) nortropane; DBS, deep brain stimulation; VIM, ventral intermediate nucleus of thalamus; TBI, traumatic brain injury; ICH, intracerebral hemorrhage; AVM, arterio-veinous malformation; STN, subthalamic nucleus.