Orthostatic Tremor: An Update on a Rare Entity

Benito‐León, Julián; Domingo-Santos, Ángela

doi:10.7916/d81n81bt

Cited by 24 publications

(9 citation statements)

References 105 publications

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“…We suspect that although hydrocephalus strengthens the symptoms, the melanoma played a more important role in this patient, because gait disturbance occurring 6 months later proved to be a sign due to worsening of spinal cord lesions. Electromyography also showed a tremor of approximately 16 Hz in the patient's legs while he was standing (Figure 1c); however, in previous studies and cases, the OT caused by hydrocephalus had a frequency of 6-7 Hz [9,18]. On the last admission, the physicians found a sensory impairment at the T7-T8 level and dysuria, with weakness of muscle and disappearance of tendon reflex, indicating lesions in the spinal cord.…”

Section: Discussion and Con Clus Ionsmentioning

confidence: 81%

“…Because OT is a rare condition and remains poorly understood, most patients are diagnosed with idiopathic OT [7,8]. Illness associated with OT reportedly has been observed in patients with vitamin B12 deficiency, nontumoral aqueduct stenosis, brainstem lesions, cerebellar degeneration, head trauma, metabolic diseases, and other neurological disorders [9]. Although OT belongs to the variant of essential tremor, it is distinguished from essential tremor or other types of tremor by its unique electrophysiological characteristics [5,10].…”

Section: Discussion and Con Clus Ionsmentioning

confidence: 99%

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Orthostatic tremor secondary to primary malignant melanoma of the spinal cord: A case report

Song

et al. 2022

Euro J of Neurology

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Background and purpose Orthostatic tremor and spinal cord melanoma are rare entities and seem unlikely to be associated. Herein, we report a patient diagnosed with orthostatic tremor secondary to primary malignant melanoma of the spinal cord. Case Presentation We report the case of a 67‐year‐old man who experienced tremor when he was standing, which disappeared when he was sitting or walking. He also reported gait disturbance and cognitive dysfunction. Electromyography revealed a regular and symmetric high‐frequency tremor in the lower extremities. The patient was admitted to a hospital several times and was diagnosed with primary orthostatic tremor and later hydrocephalus; thus, he received a ventriculoperitoneal shunt. Finally, he showed symptoms of the presence of melanoma in the spinal cord, which was supported by spinal cord magnetic resonance imaging findings. Primary malignant melanoma of the spinal cord was confirmed postoperatively. Conclusions Orthostatic tremor is a rare entity that can be characterized by specific high‐frequency tremors when the subject is standing. Considering that it remains unknown why this condition appears, some possible associations, such as primary spinal cord melanoma, should be considered. Thus, a comprehensive assessment of these types of patients is required. Our case report may facilitate the understanding of the pathophysiology and clinical symptoms of this disease.

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Section: Discussion and Con Clus Ionsmentioning

confidence: 81%

Section: Discussion and Con Clus Ionsmentioning

confidence: 99%

Orthostatic tremor secondary to primary malignant melanoma of the spinal cord: A case report

Song

et al. 2022

Euro J of Neurology

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“… 1 , 2 Patients complain of instability on standing, with a normal to minimally affected gait. 3 , 4 Patients with POT are often severely disabled, there is no cure and treatment with medication or deep brain stimulation is often unsatisfactory. 3 …”

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confidence: 99%

“… 3 , 4 Patients with POT are often severely disabled, there is no cure and treatment with medication or deep brain stimulation is often unsatisfactory. 3 …”

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confidence: 99%

“…These include the helicopter sign, tremor of the knees, and fine‐amplitude rippling of leg muscles on standing. 3 , 5 A postural arm tremor with features reminiscent of essential tremor is present in the majority (50%–90%) of patients. 6 , 7 , 8 Recently, the hem sign (ie, fast trembling of the hem of the skirt or long shirt covering the thigh on standing) has been reported in POT, but its prevalence is unknown.…”

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confidence: 99%

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The Phenomenology of Primary Orthostatic Tremor

Swinnen

Waal

Buijink

et al. 2022

Movement Disord Clin Pract

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Background The presence and prevalence of several neurological signs in patients with primary orthostatic tremor have not been systematically studied. Objectives To assess the prevalence of clinical features of primary orthostatic tremor. Methods Video‐based assessment by four raters of standardized neurological examination of 11 patients with primary orthostatic tremor. Results On standing, bent knees (7/11), hem sign (6/10), and a broad base of support (6/11) were the three most prevalent signs. Examination of gait revealed abnormal tandem gait (9/11) and bent knees (6/11) as the most prevalent clinical signs. In the arms, none of the patients displayed bradykinesia, ataxia, or dystonia. In the legs, ataxia was absent in all patients and bradykinesia was present in only one patient. Conclusions Abnormal tandem gait, bent knees, hem sign, and broad base on standing are the most prevalent clinical signs in primary orthostatic tremor. We did not encounter clear extrapyramidal or unequivocal cerebellar signs.

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A data mining approach for classification of orthostatic and essential tremor based on MRI‐derived brain volume and cortical thickness

Benito‐León

Louis

Mato‐Abad

et al. 2019

Ann Clin Transl Neurol

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ObjectiveOrthostatic tremor (OT) is an extremely rare, misdiagnosed, and underdiagnosed disorder affecting adults in midlife. There is debate as to whether it is a different condition or a variant of essential tremor (ET), or even, if both conditions coexist. Our objective was to use data mining classification methods, using magnetic resonance imaging (MRI)‐derived brain volume and cortical thickness data, to identify morphometric measures that help to discriminate OT patients from those with ET.MethodsMRI‐derived brain volume and cortical thickness were obtained from 14 OT patients and 15 age‐, sex‐, and education‐matched ET patients. Feature selection and machine learning methods were subsequently applied.ResultsFour MRI features alone distinguished the two, OT from ET, with 100% diagnostic accuracy. More specifically, left thalamus proper volume (normalized by the total intracranial volume), right superior parietal volume, right superior parietal thickness, and right inferior parietal roughness (i.e., the standard deviation of cortical thickness) were shown to play a key role in OT and ET characterization. Finally, the left caudal anterior cingulate thickness and the left caudal middle frontal roughness allowed us to separate with 100% diagnostic accuracy subgroups of OT patients (primary and those with mild parkinsonian signs).ConclusionsA data mining approach applied to MRI‐derived brain volume and cortical thickness data may differentiate between these two types of tremor with an accuracy of 100%. Our results suggest that OT and ET are distinct conditions.

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Orthostatic Tremor: An Update on a Rare Entity

Cited by 24 publications

References 105 publications

Orthostatic tremor secondary to primary malignant melanoma of the spinal cord: A case report

Orthostatic tremor secondary to primary malignant melanoma of the spinal cord: A case report

The Phenomenology of Primary Orthostatic Tremor

A data mining approach for classification of orthostatic and essential tremor based on MRI‐derived brain volume and cortical thickness

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