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Review
. 2006 Nov 27:1:48.
doi: 10.1186/1750-1172-1-48.

Early onset torsion dystonia (Oppenheim's dystonia)

Affiliations
Review

Early onset torsion dystonia (Oppenheim's dystonia)

Christoph Kamm. Orphanet J Rare Dis. .

Abstract

Early onset torsion dystonia (EOTD) is a rare movement disorder characterized by involuntary, repetitive, sustained muscle contractions or postures involving one or more sites of the body. A US study estimated the prevalence at approximately 1 in 30,000. The estimated prevalence in the general population of Europe seems to be lower, ranging from 1 in 330,000 to 1 in 200,000, although precise numbers are currently not available. The estimated prevalence in the Ashkenazi Jewish population is approximately five to ten times higher, due to a founder mutation. Symptoms of EOTD typically develop first in an arm or leg in middle to late childhood and progress in approximately 30% of patients to other body regions (generalized dystonia) within about five years. Distribution and severity of symptoms vary widely between affected individuals. The majority of cases from various ethnic groups are caused by an autosomal dominantly inherited deletion of 3 bp (GAG) in the DYT1 gene on chromosome 9q34. This gene encodes a protein named torsinA, which is presumed to act as a chaperone protein associated with the endoplasmic reticulum and the nuclear envelope. It may interact with the dopamine transporter and participate in intracellular trafficking, although its precise function within the cell remains to be determined. Molecular genetic diagnostic and genetic counseling is recommended for individuals with age of onset below 26 years, and may also be considered in those with onset after 26 years having a relative with typical early onset dystonia. Treatment options include botulinum toxin injections for focal symptoms, pharmacological therapy such as anticholinergics (most commonly trihexiphenydil) for generalized dystonia and surgical approaches such as deep brain stimulation of the internal globus pallidus or intrathecal baclofen application in severe cases. All patients have normal cognitive function, and despite a high rate of generalization of dystonia, 75% of those patients are able to maintain ambulation and independence, and therefore a comparatively good quality of life, with modern treatment modalities.

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References

    1. Bressman SB, de Leon D, Brin MF, Risch N, Burke RE, Greene PE, Shale H, Fahn S. Idiopathic dystonia among Ashkenazi Jews: evidence for autosomal dominant inheritance. Ann Neurol. 1989;26:612–620. doi: 10.1002/ana.410260505. - DOI - PubMed
    1. Cassetta E, Del Grosso N, Bentivoglio AR, Valente EM, Frontali M, Albanese A. Italian family with cranial cervical dystonia: clinical and genetic study. Mov Disord. 1999;14:820–825. doi: 10.1002/1531-8257(199909)14:5<820::AID-MDS1015>3.0.CO;2-I. - DOI - PubMed
    1. Bressman SB, Raymond D, Wendt K, Saunders-Pullman R, De Leon D, Fahn S, Ozelius L, Risch N. Diagnostic criteria for dystonia in DYT1 families. Neurology. 2002;59:1780–1782. - PubMed
    1. Nutt JG, Muenter MD, Aronson A, Kurland LT, Melton LJ., 3rd Epidemiology of focal and generalized dystonia in Rochester, Minnesota. Mov Disord. 1988;3:188–194. doi: 10.1002/mds.870030302. - DOI - PubMed
    1. Zeman W, Dyken P. Dystonia musculorum deformans. Clinical, genetic and pathoanatomical studies. Psychiatr Neurol Neurochir. 1967;70:77–121. - PubMed

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