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Clinical and Electrophysiological Heterogeneity of Orthostatic Tremor: An Individual Patient Data Analysis Cover

Clinical and Electrophysiological Heterogeneity of Orthostatic Tremor: An Individual Patient Data Analysis

Open Access
|Oct 2026

Figures & Tables

Figure 1

PRISMA flowchart.

Figure 2

Density plots of tremor frequency for isolated (blue) and non-isolated (orange) OT.

Table 1

Clinical spectrum of OT cases according to the MDS classification.

PARAMETERPRIMARY OTOT PLUSPSEUDO OTUNCLASSIFIABLE (FLUCTUATING FREQUENCY)UNCLASSIFIABLE (>18 Hz)
N1273961125
Age (SD)64.13 (11.25)64.41(13.55)58.56 (16.03)61.67 (17.48)61.80 (13.05)
Sex (M/F)50/7714/2525/364/83/2
Mean Tremor Frequency in Hz (SD)15.58 (1.26)15.46 (1.41)6.82 (2.36)13.12 (1.17)18.92 (0.71)
Familial history of OT132200
Table 2

Clinical spectrum of OT cases based on isolated/non-isolated phenotype.

PARAMETERISOLATED OTNON-ISOLATED OT
N14896
Age (SD)63.97 (11.35)60.51 (15.96)
Sex (M/F)60/8836/60
Mean Tremor Frequency in Hz (SD)14.80 (2.82)11.03 (4.81)
≥13 Hz/<13 Hz129/1949/47
Familial history of OT125
Table 3

Disorders associated with OT.

ASSOCIATED DISORDERSTOTAL NUMBER<13 Hz N (%)≥13 Hz N (%)MEAN TREMOR FREQUENCYSDMINIMUM FREQUENCYMAXIMUM FREQUENCY
Parkinson’s disease/Parkinsonism/PSP3822 (57.9)16 (42.1)10.075.171318
Essential tremor103 (30)7 (70)13.503.7526.519
Cerebellar disorders/lesions102 (20.0)8 (80.0)12.733.3824.515
Brainstem lesions62 (33.3)4 (66.7)12.375.2584.2517.5
Hyperthyroid states53 (60.0)2 (40.0)10.504.213615
Musculoskeletal injury21 (50)1 (50)10.757.4255.516
Hereditary spastic paraplegia31 (33.3)2 (66.7)14.004.583918
Stiff person syndrome20 (0.0)2 (100.0)16.000.0001616
Dementia20 (0.0)2 (100.0)15.002.8281317
Miscellaneous1813 (72.2)5 (27.8)8.984.5023.516
Isolated OT14819 (12.8)129 (87.2)14.802.8213.520
SL NOAUTHORYEAR OF PUBLICATIONAGESEXFREQUENCY (HZ)ISOLATED/NON-ISOLATEDASSOCIATED CONDITIONMDS CLASSIFICATION
1Tseng et al. [20]202534Male3Non-isolatedButane induced encephalopathy, ParkinsonismPseudo OT
2Swinnen et al. [21]202257Female4–5Isolated-Pseudo OT
3Grobe-Einsler et al. [22]202053Female14–16Isolated-Primary OT
4Verriello et al. [23]202082Male15–16Isolated-Primary OT
77Male15–16Isolated-Primary OT
5Yoshii et al. [24]202062Female5–6Isolated-Pseudo OT
6Yoo et al. [25]202069Female4–6Isolated-Pseudo OT
7Chaithra et al. [26]201972Female15–16Non-isolatedParkinson’s diseaseOT Plus
8Wadhwa et al. [27]201962Female17–18Isolated-Primary OT
9Iovino et al. [28]201923Male10.5Isolated-Pseudo OT
10Santus et al. [29]201973Female17Non-isolatedFrontotemporal dementiaOT Plus
11Mousele et al. [30]201866Male11–12Isolated-Pseudo OT
12Yoo et al. [31]201948Male4–6Non-isolatedParkinson’s diseasePseudo OT
13Wuehr et al. [32]201862Male14.5Isolated-Primary OT
55Female14Isolated-Primary OT
75Male17.5Isolated-Primary OT
75Male14Isolated-Primary OT
56Female17Isolated-Primary OT
75Female16.5Isolated-Primary OT
66Female14Isolated-Primary OT
75Male13.5Isolated-Primary OT
76Male15Isolated-Primary OT
14Evidente et al. [33]201873Male10–14Isolated-Unclassifiable(fluctuating)
68Male5–12Non-isolatedEssential TremorPseudo OT
15Shirai et al. [34]201860Male4–5Non-isolatedMachado-Joseph diseasePseudo OT
16Ruiz-Julián et al. [35]201875Female14Isolated-Primary OT
17Lehn et al. [36]201768Male16Isolated-Primary OT
18Larrosa Campo et al. [37]201981Male14–16Non-isolatedThyrotoxicosisPrimary OT
19Chiang et al. [38]201774Female~16Isolated-Primary OT
73Male~16Isolated-Primary OT
20Vidailhet et al. [39]201746Female17Isolated-Primary OT
21Schöberl et al. [40]201756Female17.2Isolated-Primary OT
64Female16Isolated-Primary OT
72Male14Isolated-Primary OT
73Male16.7Isolated-Primary OT
68Female15.5Isolated-Primary OT
74Male14.2Isolated-Primary OT
73Male13.5Isolated-Primary OT
73Female16.5Isolated-Primary OT
73Male14.5Isolated-Primary OT
73Female14.2Isolated-Primary OT
22Benito-Léon et al. [41]201674Female16–18Isolated-Primary OT
64Female10–12Isolated-Pseudo OT
57Male16–18Isolated-Primary OT
68Female16–18Non-isolatedParkinson’s diseaseOT Plus
78Female16–18Non-isolatedParkinson’s diseaseOT Plus
62Female12–16Isolated-Unclassifiable(fluctuating)
37Female16–18Isolated-Primary OT
76Female12–16Isolated-Unclassifiable(fluctuating)
75Female12–16Non-isolatedParkinson’s diseaseUnclassifiable(fluctuating)
77Female12–16Non-isolatedParkinson’s diseaseUnclassifiable(fluctuating)
38Male16–18Isolated-Primary OT
81Female12–16Non-isolatedParkinson’s diseaseUnclassifiable(fluctuating)
65Female16–18Isolated-Primary OT
57Female16–18Isolated-Primary OT
23Blahak et al. [42]201658Female15Non-isolatedDiabetic neuropathyPrimary OT
72Male14Isolated-Primary OT
73Female17Isolated-Primary OT
67Male17Isolated-Primary OT
24Coleman et al. [43]201661Female14Isolated-Primary OT
74Female13–18Isolated-Primary OT
25Athauda et al. [44]201746Female16Isolated-Primary OT
73Male16Isolated-Primary OT
66Male17Non-isolatedParkinsonismOT Plus
26Contarino et al. [45]201570Female16Isolated-Primary OT
27Erro et al. [46]201463Female15Non-isolatedHereditary spastic paraparesisOT Plus
28Mazzucchi et al. [47]201470Female8Non-isolatedGraves’ diseasePseudo OT
29Adebayo et al. [48]201457Female16Isolated-Primary OT
30Vetrugno et al. [49]201377Female16Non-isolatedStiff Person SyndromeOT Plus
55Female16Non-isolatedStiff Person SyndromeOT Plus
31Bhattacharyya et al. [50]201312Male15–16Non-isolatedPoliomyelitisPrimary OT
7Male14–15Non-isolatedEssential tremorOT Plus
32Coffeng et al. [17]201386Male10–11Isolated-Pseudo OT
33Lin et al. [51]201326Male8–9Non-isolatedGraves’ diseasePseudo OT
34Lyons et al. [52]201275Male13Isolated-Primary OT
35Virmani et al. [53]201279Male13.5Isolated-Primary OT
75Female13.3Isolated-Primary OT
36Lee et al. [54]201264Male11–13Non-isolatedSpinal SchwannomaUnclassifiable(fluctuating)
37Lee et al. [55]201181Female4–9Non-isolatedParkinson’s diseasePseudo OT
38Kumar et al. [56]201160Male16Isolated-Primary OT
39Hegde et al. [57]201169Male3–4Isolated-Pseudo OT
40Yaltho et al. [58]201178Male14Isolated-Primary OT
41Magariños-Ascone et al. [59]201068Female12–18Isolated-Unclassifiable(fluctuating)
42Williams et al. [60]201070Female9Non-isolatedCerebellar atrophyPseudo OT
43Vetrugno et al. [61]201058Female16Non-isolatedLeft midbrain lesionOT Plus
44Raudino et al. [62]200959Male13–14Isolated-Primary OT
45Baker et al. [63]200938Female8–15Non-isolatedMultiple SclerosisUnclassifiable(fluctuating)
46Kang et al. [64]200961Female6Non-isolatedParkinson’s diseasePseudo OT
60Female6–7Non-isolatedParkinson’s diseasePseudo OT
61Female4–5Non-isolatedParkinson’s diseasePseudo OT
45Female3–4Non-isolatedParkinson’s diseasePseudo OT
47Female16–17Non-isolatedParkinson’s diseaseOT Plus
47Ramtahal et al. [65]200878Male13–14Isolated-Primary OT
65Male15–16Isolated-Primary OT
65Female15Isolated-Primary OT
48Wegner et al. [66]200861Male16–18Isolated-Primary OT
61Female16–18Isolated-Primary OT
49Espay et al. [67]200873Female15Isolated-Primary OT
67Female15Isolated-Primary OT
50Guridi et al. [68]200855Female16Isolated-Primary OT
51Trocello et al. [69]200849Male16Isolated-Primary OT
76Female17Isolated-Primary OT
55Female17Isolated-Primary OT
66Male16Isolated-Primary OT
66Female15Isolated-Primary OT
49Female15Isolated-Primary OT
75Female16Isolated-Primary OT
62Female16Isolated-Primary OT
74Female16Isolated-Primary OT
78Female17Isolated-Primary OT
70Female18Isolated-Primary OT
70Male15Isolated-Primary OT
52Stich et al. [70]200957Female17.5Non-isolatedDemyelinating lesion involving the right brainstem, Biclonal gammopathy of undetermined significanceOT Plus
53Tan et al. [71]200850Female14–16Non-isolatedGraves’ diseasePrimary OT
54Leu-Semenescu et al. [72]200772Female18Non-isolatedParkinson’s diseaseOT Plus
77Female16Non-isolatedParkinson’s diseaseOT Plus
79Male13.5Non-isolatedParkinson’s diseaseOT Plus
75Female17Non-isolatedParkinson’s diseaseOT Plus
76Female7.5–9Non-isolatedParkinson’s diseasePseudo OT
85Female5Non-isolatedParkinson’s diseasePseudo OT
57Female5–6Non-isolatedParkinson’s diseasePseudo OT
54Female4.3–5Non-isolatedParkinson’s diseasePseudo OT
55Fischer et al. [73]200769Male16–17Isolated-Primary OT
68Male16–17Isolated-Primary OT
63Male16–17Isolated-Primary OT
56Thomas et al. [10]200747Female6–7Non-isolatedParkinsonismPseudo OT
52Male6–7Non-isolatedParkinsonismPseudo OT
52Male6–7Non-isolatedParkinsonism, Kearns-Sayre syndromePseudo OT
26Female6–7Non-isolatedParkinsonismPseudo OT
57de Bie et al. [74]200767Female16–17Non-isolatedProgressive supranuclear palsyOT Plus
58Krauss et al. [75]200658Female15–16Isolated-Primary OT
72Male14Isolated-Primary OT
59Contarino et al. [76]200668Male13Isolated-Primary OT
68Male13Isolated-Primary OT
60Gilhuis et al. [77]200555Female3–5Non-isolatedSmall cell carcinoma of lungPseudo OT
61Karlberg et al. [78]200546Female12Isolated-Pseudo OT
36Male8.5Isolated-Pseudo OT
63Female12.5Isolated-Pseudo OT
64Female15.5Isolated-Primary OT
73Female18Isolated-Primary OT
62Bacsi et al. [79]200559Female15.9Isolated-Primary OT
73Male17.3Isolated-Primary OT
50Female15.2Isolated-Primary OT
57Female16.5Isolated-Primary OT
64Female18.1Isolated-Unclassifiable(>18 Hz)
68Female17.3Isolated-Primary OT
51Female17.3Isolated-Primary OT
69Female16.8Isolated-Primary OT
75Female15.6Isolated-Primary OT
66Male15.7Isolated-Primary OT
63Rodrigues et al. [12]200568Female19Isolated-Unclassifiable(>18 Hz)
70Female14Isolated-Primary OT
74Female16Isolated-Primary OT
53Male15Isolated-Primary OT
63Female14Isolated-Primary OT
65Female13Isolated-Primary OT
64Trip et al. [80]200343Female16Isolated-Primary OT
65Apartis et al. [81]200175Female18Non-isolatedParkinson’s diseaseOT Plus
66Finkel et al. [13]200076Male18–19Isolated-Unclassifiable(>18 Hz)
67Cavallazi et al. [14]200042Female16Isolated-Primary OT
41Male20Isolated-Unclassifiable(>18 Hz)
59Female17Isolated-Primary OT
48Female15Isolated-Primary OT
68Benito-Leon et al. [82]200068Male15Non-isolatedB12 deficiencyOT Plus
69Manto et al. [83]199949Male14Non-isolatedPancerebellar atrophyOT Plus
62Female15Non-isolatedPancerebellar atrophyOT Plus
64Female14Non-isolatedPancerebellar atrophyOT Plus
70Wills et al. [84]199974Male14.5Non-isolatedParkinson’s diseaseOT Plus
71Sander et al. [85]199879Female15–17Isolated-Primary OT
64Male15–17Isolated-Primary OT
72Setta F et al. [86]199848Male14Non-isolatedCerebellar atrophyOT Plus
63Female14Non-isolatedCerebellar atrophyOT Plus
61Female15Non-isolatedCerebellar atrophyOT Plus
73Tsai et al. [87]199858Female16Isolated-Primary OT
68Female16Isolated-Primary OT
74Benito-Leon et al. [88]199749Female7–8Non-isolatedRight pontine lesionPseudo OT
39Male14–15Non-isolatedLeft pontine lesionOT Plus
75Hanna et al. [89]199733Female17.5Isolated-Primary OT
76Poersch et al. [90]199460Female14Isolated-Primary OT
77Britton et al. [91]199263Female16–18Isolated-Primary OT
25Female8–16Isolated-Unclassifiable(fluctuating)
68Male16–17Isolated-Primary OT
57Female15–16Isolated-Primary OT
47Female16–18Isolated-Primary OT
64Male15–16Isolated-Primary OT
78Willeit et al. [92]199177Male14–16Isolated-Primary OT
57Male14–16Isolated-Primary OT
79FitzGerald et al. [15]199160Male18–20Non-isolatedEssential tremorUnclassifiable(>18 Hz)
46Male12–14Non-isolatedEssential tremorUnclassifiable(fluctuating)
70Male12Non-isolatedEssential tremorPseudo OT
56Female14Non-isolatedEssential tremorOT Plus
80Walker et al. [93]199079Male15Isolated-Primary OT
81Gabellini et al. [94]199068Female14–16Isolated-Primary OT
56Female14–16Isolated-Primary OT
45Female14–16Isolated-Primary OT
75Female6–7Isolated-Pseudo OT
64Male6–7Isolated-Pseudo OT
66Male6–7Isolated-Pseudo OT
59Male6–7Non-isolatedAqueduct stenosis hydrocephalusPseudo OT
47Female6–7Non-isolatedChronic relapsing radiculopathyPseudo OT
82Oda et al. [95]198979Female4Non-isolatedVascular ParkinsonismPseudo OT
83Uncini et al. [96]198973Male8–10Isolated-Pseudo OT
64Female16Isolated-Primary OT
84Papa et al. [97]198863Female16Non-isolatedEssential tremorOT Plus
72Female16Non-isolatedEssential tremorOT Plus
68Female16Non-isolatedEssential tremorOT Plus
85Thompson et al. [9]198655Male8–16Isolated-Unclassifiable(fluctuating)
86Kim et al. [98]202318Male5–6Non-isolatedKnee contusionPseudo OT
87Park et al. [99]202079Female4–4.5Non-isolatedRight pontine lesionPseudo OT
78Female6.5Non-isolatedParkinsonismPseudo OT
74Female8Non-isolatedBrain lesionPseudo OT
46Female9Non-isolatedParkinson’s diseasePseudo OT
63Male13.5–14Non-isolatedRestless leg syndromeOT Plus
62Female6Non-isolatedDrug induced parkinsonismPseudo OT
59Male5–6Non-isolatedTardive dyskinesiaPseudo OT
68Female15Isolated-Primary OT
49Male4–5Non-isolatedArteriovenous malformationPseudo OT
65Female4Non-isolatedREM sleep behaviour disorderPseudo OT
67Male8–8.5Non-isolatedParkinsonism with Normal Pressure hydrocephalusPseudo OT
58Female16Isolated-Primary OT
72Male13Non-isolatedDementiaOT Plus
61Male13Isolated-Primary OT
56Female13Isolated-Primary OT
72Male8Non-isolatedParkinson’s disease with Essential TremorPseudo OT
68Male6–7Non-isolatedEssential tremorPseudo OT
65Female4–4.5Non-isolatedParkinson’s diseasePseudo OT
88Comolli et al. [100]202465Female6Non-isolatedGraves’ diseasePseudo OT
89Li et al. [101]202222Male6–8Non-isolatedHyperphenylalaninemiaPseudo OT
90Lv et al. [102]202267Male16Non-isolatedPrimary malignant melanoma of the spinal cordOT Plus
91Bhattacharyya et al. [103]200370Male18Isolated-Primary OT
92Jones et al. [104]201165Male16Isolated-Primary OT
93Manrique-Huarte et al. [105]201265Female14–16Isolated-Primary OT
69Female2–9Non-isolatedPsychogenic dizzinessPseudo OT
45Female10Isolated-Pseudo OT
63Female8Non-isolatedMigraine with auraPseudo OT
94Yague et al. [106]201147Female16Non-isolatedSurgery to left meniscus following traumaPrimary OT
95Sanitate et al. [107]199372Female14–15Non-isolatedHead trauma, demyelinating lesions in the periventricular region and subcortical white matter, Ischemic changesin the right pons and the right cerebellar hemisphereOT Plus
96Marechal et al. [108]202473Male14Non-isolatedRestless leg syndrome, PSPOT Plus
97Myers et al. [109]200372Female16Isolated-Primary OT
98Sarva et al. [110]201681Male13.8–15.9Non-isolatedCerebellar degenerationOT Plus
57Male13Non-isolatedSpinocerebellar ataxiaOT Plus
99Picillo et al. [111]201774Female18Non-isolatedHereditary spastic paraplegia (SPG31)OT Plus
30Female9Non-isolatedHereditary spastic paraplegia (SPG15)Pseudo OT
100Brotman et al. [112]200065Male3–4Non-isolatedNeurosyphillisPseudo OT
DOI: https://doi.org/10.5334/tohm.1278 | Journal eISSN: 2160-8288
Language: English
Page range: 55 - 55
Submitted on: Aug 5, 2026
Accepted on: Sep 26, 2026
Published on: Oct 8, 2026
Published by: Ubiquity Press
In partnership with: Paradigm Publishing Services

© 2026 Leroy D’Souza, Niharika B. R., Julián Benito-León, Samarth B. R., Rishika Das, Muskan Kotwal, Kruthika Reddy, Pranav Anil Katti, C. S. Pranav Bhat, published by Ubiquity Press
This work is licensed under the Creative Commons Attribution 4.0 License.