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Movement Disorders in Patients with Subacute Sclerosing Panencephalitis: A Systematic Review
Divyani Garg1, Sahil Patel2, Charulata S Sankhla2
1Department of Neurology, All India Institute of Medical Sciences, New Delhi, India.
Background:
Subacute sclerosing panencephalitis (SSPE) is a complication of measles, occurring after a latency of 4-10 years. It continues to occur in developing countries although resurgence is being reported from developed countries. Characteristic features include progressive neuropsychiatric issues, myoclonus, seizures, movement disorders and visual impairment. Electroencephalography (EEG) typically shows periodic generalized discharges, and elevated CSF anti-measles antibodies are diagnostic. Movement disorders are being increasingly recognized as part of the clinical spectrum, and range from hyperkinetic (chorea, dystonia, tremor, tics) to hypokinetic (parkinsonism) disorders and ataxia.
Objectives:
This article aims to comprehensively review the spectrum of movement disorders associated with SSPE.
Methods:
A literature search was conducted in PubMed and EMBASE databases in December 2023 and articles were identified for review.
Results:
Movement disorders reported in SSPE included hyperkinetic (chorea, dystonia, tremor and tics), hypokinetic (parkinsonism), ataxia and extraocular movement disorders. Myoclonus, a core clinical feature, was the most frequent "abnormal movement." Movement disorders were observed in all clinical stages, and could also be a presenting feature, even sans myoclonus. Hyperkinetic movement disorders were more common than hypokinetic movement disorders. An evolution of movement disorders was observed, with ataxia, chorea and dystonia occurring earlier, and parkinsonism later in the disease. Neuroradiological correlates of movement disorders remained unclear.
Conclusion:
A wide spectrum of movement disorders was observed throughout the clinical stages of SSPE. Most data were derived from case reports and small case series. Multicentric longitudinal studies are required to better delineate the spectrum and evolution of movement disorders in SSPE.
Insights
Subacute sclerosing panencephalitis (SSPE) causes a wide range of movement disorders, including hyperkinetic and hypokinetic types, throughout its clinical stages. Further research is needed to fully understand their evolution.
Area of Science:
- Neurology
- Infectious Diseases
Background:
- Subacute sclerosing panencephalitis (SSPE) is a rare, progressive neurological complication of measles infection.
- It presents with diverse symptoms including neuropsychiatric decline, seizures, and increasingly recognized movement disorders.
- While historically associated with developing nations, SSPE resurgence is noted globally.
Approach:
- A comprehensive literature review was performed using PubMed and EMBASE databases.
- The search focused on identifying and analyzing reported movement disorders in SSPE patients.
- Data collection occurred in December 2023.
Key Points:
- SSPE encompasses a broad spectrum of movement disorders, including hyperkinetic (chorea, dystonia, tremor, tics), hypokinetic (parkinsonism), and ataxia.
- Myoclonus, a hallmark of SSPE, is the most common abnormal movement observed.
- Movement disorders manifest across all disease stages and can even be an initial presentation, sometimes without myoclonus.
Conclusions:
- Movement disorders are integral to the clinical presentation of SSPE, varying in type and timing throughout disease progression.
- Current understanding relies heavily on case reports and small series, limiting definitive conclusions.
- Multicentric, longitudinal studies are essential to comprehensively map the spectrum and evolution of SSPE-associated movement disorders.
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