Objective: To provide a systematic literature overview of orthostatic tremor (OT).

Background: Orthostatic tremor (OT) is characterized by a high-frequency tremor of the leg muscles during stance, resulting in an unsteady feeling. The pathophysiology is still not understood. Treatment options are limited and are often unsatisfactory. A systematic literature review on primary OT is lacking the literature.

Methods: Here we review data of a total of 617 OT-patients, with a focus on primary OT, retrieved after a systematic search in Pubmed (583 individual patients from 45 case reports, 20 case series, and 7 therapeutic trials) and from a questionnaire-based study in Dutch OT-patients (n=34).

Results: Overall, 67% of identified OT patients is female; mean age at onset is 57 (17-81) years. Most patients were primary OT cases, however this distinction is not always made. Six percent (22 of 390 cases) has a positive family history for OT. Mean delay to diagnosis is 7.7 years (n=268). A substantial number of patients reports falls. Quality of life is affected. Clonazepam is the most prescribed drug but is not always effective. Trials report a possible positive effect of gabapentin. Deep brain stimulation (DBS), performed in a limited number of patients, has shown a positive effect.

Conclusions: OT can be disabling and is under-recognized. The most effective treatment remains uncertain, with some evidence for clonazepam and gabapentin. DBS provides an alternative for drug-resistant and disabling OT, although long term effects are still unknown. To gain more insight in OT, it is of importance to differentiate primary OT from OT-plus and secondary OT. Increasing awareness for OT will shorten the delay in time to diagnosis and possibly lead to development of better treatment options.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/shaky-legs-the-clinical-spectrum-and-treatment-of-orthostatic-tremor-a-systematic-review/