Syndrome of fixed dystonia in adolescents – Short term outcome in 4 cases

Majumdar, Anirban; López-Casas, J; Póo, Pilar; Colomer, J.; Galván, Marta Ferrer; Lingappa, Lokesh; Short, Clare; Jardine, Philip; Fernández-Álvarez, E

doi:10.1016/j.ejpn.2008.09.005

Cited by 14 publications

(16 citation statements)

References 16 publications

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“…A history of injury is almost always demonstrated in affected children and the lower limbs are more often affected than the upper limbs, while the upper extremity was affected more frequently than the lower extremity in adults. Abnormal movements and dystonia have also been frequently reported in adults and children with CRPS and may complicate the clinical picture 19 20…”

Section: Introductionmentioning

confidence: 99%

Complex regional pain syndrome in children: incidence and clinical characteristics

Abu-Arafeh

Abu‐Arafeh

2016

Arch Dis Child

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Section: Introductionmentioning

confidence: 99%

Complex regional pain syndrome in children: incidence and clinical characteristics

Abu-Arafeh

Abu‐Arafeh

2016

Arch Dis Child

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“…In 1993, Bhatia et al 14 reported 18 patients who developed fixed painful limb dystonia triggered by trivial peripheral injuries. 18 In their original description of the causalgia-dystonia syndrome, Bhatia et al 14 concluded "at present it is impossible to decide whether this distressing syndrome is a true functional disorder of the CNS, or is of psychogenic origin." Like patients with traumatic cervical dystonia, these patients had fixed postures of the limbs, prominent pain, and poor response to therapy.…”

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confidence: 99%

Psychogenic dystonia and peripheral trauma

Hawley¹,

Weiner²

2011

Neurology

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Dystonia in association with peripheral trauma is a well-described clinical syndrome. The syndrome goes by many names--"traumatic" dystonia, "fixed" dystonia, peripherally induced dystonia, or complex region pain syndrome (CRPS) dystonia. We reviewed the role of peripheral trauma in the development of dystonia, focusing on 4 subtypes--cervical dystonia, focal limb dystonia, CRPS dystonia, and psychogenic dystonia. We show that peripheral trauma inducing, provoking, or precipitating structural changes within the CNS leading to dystonia is not an accepted concept, and current evidence supporting a pathophysiologic mechanism is virtually nonexistent. A better approach to this clinical syndrome is to define it as fixed abnormal posturing that is most commonly psychogenic. While symptomatic treatment of pain and spasms with medication can be beneficial, early psychological evaluation and patient-specific treatment is important. Modalities such as physical and occupational therapy should be utilized early. Finally, it should be emphasized that like many psychogenic movement disorders, it remains a highly disabling and distressing disorder.

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“…Basic research has revealed the role of GABA-B receptors in the central nervous system in the regulation of pain [16]. Some clinical studies have also demonstrated the analgesic effect of ITB in treating intractable pain syndromes after spinal cord injury or cerebral infarction [13,14,15]. ITB may reduce neuropathic pain by modulating spinal GABA mechanisms, and thus, ITB can be an important option for patients with CRPS whose conditions are refractory to other methods of pain control [11,16,17].…”

Section: Discussionmentioning

confidence: 99%

“…Recently, some reports have suggested that ITB therapy can enhance the effect of SCS [11,12,13]. Furthermore, ITB therapy has been reported to be an effective modality for treating fixed dystonia related to CRPS [14,15]. Therefore, ITB may have the potential as an adjunctive agent for treating patients with CRPS showing severe and complex symptomatology.…”

Section: Introductionmentioning

confidence: 99%

Spinal Cord Stimulation and Intrathecal Baclofen Therapy: Combined Neuromodulation for Treatment of Advanced Complex Regional Pain Syndrome

Goto¹,

Taira²,

Horisawa³

et al. 2013

Stereotact Funct Neurosurg

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Objective: Spinal cord stimulation (SCS) is one of the widely used procedures for the treatment of complex regional pain syndrome (CRPS). However, complete pain relief is rarely achieved, and the SCS effect diminishes over time. Recently intrathecal baclofen (ITB) therapy is reported to be a modality for treating fixed dystonia related to CRPS. Other reports have suggested that ITB therapy can enhance the effect of SCS in patients with neuropathic pain. We report the effectiveness of combined SCS and ITB therapy, focusing on the role of ITB therapy as an adjunctive therapy for controlling symptoms of advanced CRPS. Methods: Five affected extremities of 4 patients with CRPS (2 male; mean age, 32.5 years) refractory to conservative treatment were evaluated retrospectively. Three patients underwent SCS implantation first, with ITB pumps being implanted a few years later. Bolus ITB injection was administered under temporary percutaneous SCS in 1 patient. Pain intensity was evaluated using the visual analogue scale (VAS) before and after ITB administration. Results: Pain relief of more than 50% was observed in the upper extremity of 1 patient and one of more than 30% was observed in 2 patients. The mean pain reduction rate in all 4 patients was 28.9% before and 43.8% after treatment. All patients, including those without any improvements in VAS score, showed decreased postural abnormalities after combined SCS and ITB therapy. Improvement in postural abnormalities, such as fixed dystonia or paroxysmal tremor-like movements, resulted in overall pain relief by reducing pain fluctuations. Conclusions: Combined SCS and ITB neuromodulation decreases pain intensity in refractory CRPS cases or improves associated abnormal dystonic posture and movement disorders and reduces pain fluctuations.

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Syndrome of fixed dystonia in adolescents – Short term outcome in 4 cases

Cited by 14 publications

References 16 publications

Complex regional pain syndrome in children: incidence and clinical characteristics

Complex regional pain syndrome in children: incidence and clinical characteristics

Psychogenic dystonia and peripheral trauma

Spinal Cord Stimulation and Intrathecal Baclofen Therapy: Combined Neuromodulation for Treatment of Advanced Complex Regional Pain Syndrome

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