Ferreira_EU Neurology 10/03/2010 10:38 Page 77
Pain and Quality of Life in the Treatment of Cervical Dystonia
dystonia and QoL. This idea is supported by another series of 101 significant improvement in the TWSTRS pain subscore 12 months after
patients with CD and 84 healthy controls: BoNTA treatment improved bilateral DBS-GPi.
60
After a mean follow-up of 32 months, Hung et al.
both scores of SF-36 (all domains) and TWSTRS, but without a found a 50% decrease in the TWSTRS pain subscore in 10 patients who
correlation between each other.
51
Depression was the main predictor underwent bilateral DBS-GPi for refractory CD.
61
Similar results were
of poor QoL, along with female gender, poor finance and living alone. reported by Krauss et al.
62
Only the Canadian study evaluated QoL, and
In contrast to the Hilker et al. study, a longer duration of BoNT therapy it found a significant improvement in SF-36 scores after surgery.
60
predicted better QoL score.
25,51
Physical Therapy
Oral Drugs Several rehabilitation techniques for the treatment of CD have been
Several oral drugs have been reported to be efficacious for the reported, but the data are generally of poor methodological quality.
63
treatment of CD, such as anticholinergics, antidopaminergics, In the single RCT available, neither pain nor QoL was evaluated.
64
dopaminergics, GABAergics, benzodiazepines and antiepileptics.
53,54
Anticholinergics have prevailed as the most prescribed in clinical Conclusions
practice, mainly before the advent of BoNT, although the evidence CD is a chronic disorder that has a severe impact on the QoL of
supporting their use is scarce.
55
There are no specific controlled trials patients,
24,25,27–29,31
comparable to that found in other chronic neurological
of anticholinergics for CD; however, one RCT compared BoNTA versus disorders such as multiple sclerosis.
24
Pain is present in almost two-
trihexyphenidyl in 66 patients with CD. In this small trial, BoNTA was thirds of patients with CD and represents one of the QoL domains that
found to be superior to trihexyphenidyl, and also was better is most affected in CD.
3,9
The main determinants for a poor QoL are
tolerated.
56
More patients in the BoNTA arm had pain relief (TWSTRS depression, anxiety and pain.
27–29,30,32,33
pain subscale) compared with those in the trihexyphenidyl arm, in
addition to having better scores in the QoL scale (General Health Although there are no specific trials that have evaluated the effect of
Perception Subscale of the Dutch MOS-Quality of Life Scale). pharmacological, surgical or physical treatments on pain or QoL as
primary outcomes, the best data available suggest that all
Surgical Interventions interventions that have improved dystonia have also alleviated pain
Peripheral Surgery and improved QoL domains.
25,27,29,40,47,48,51,52,58,59,60,61
Peripheral surgical techniques, either selective peripheral denervation
or myectomy, have been reported to be efficacious for the treatment The relevance of pain for the management of CD suggests that
of patients with CD refractory to BoNT.
54
Although controlled trials are treatments for CD that strictly target dystonic movements may not be
not available, large case series supported the approval of selective sufficient to have a major impact on QoL.
25,27,29,51,52
The therapeutic
peripheral denervation in patients with CD by the UK National Institute strategy to manage CD likely requires multiple pharmacological/
for Health and Clinical Excellence (NICE). These patients should be surgical interventions to target dystonia, pain, depression and anxiety
refractory to other pharmacological treatments, and the procedure in order to achieve a major decrease in the burden of disease. n
should be carried out in a specialist neurosurgical centre.
57–59
Only two
of the three case series available evaluated pain or QoL.
58,59
Munchau
et al. prospectively included 62 patients, and pain was assessed by
Miguel Coelho is an Attending Neurologist at the University Hospital of Santa Maria,
Lisbon and an investigator at the Neurological Clinical Research Unit of the Institute of
the TWSTRS pain subscale; this subscore for pain was reduced
Molecular Medicine, Lisbon. He has served as investigator for multiple clinical trials in
by 40 and 30%, respectively, at six and 12 months after surgery, movement disorders. His major research interests are Parkinson’s disease, atypical
although it increased afterwards as a possible consequence of muscle
parkinsonism, dystonia and botulinum toxin. Dr Coelho completed his neurological
training at the University Hospital of Santa Maria, Lisbon.
re-innervation.
59
In the study by Cohen-Gadol et al., 168 patients with
medically intractable CD were evaluated retrospectively.
58
Pain was
Anabela Ferreira Valadas is a Resident in Neurology at the University Hospital of Santa
measured by a patient self-assessment analogue scale, and 131
Maria, Lisbon and an investigator at The Neurological Clinical Research Unit of the
patients (81%) reported a moderate to marked benefit after surgery, Institute of Molecular Medicine, Lisbon. She has served as investigator for clinical trials
which was long-lasting (mean follow-up of 3.4 years) in 70 patients.
58 in movement disorders. Her major research interests are Parkinson’s disease, dystonia
and botulinum toxin. Dr Valadas obtained her medical degree at the Faculty of
Munchau et al. assessed changes in psychosocial function, including
Medicine, University of Coimbra in 2003.
the EuroQol Measure of Quality of Life, in which the score significantly
improved after surgery.
59
The study by Cohen-Gadol et al. did not
Tiago Mestre is a resident in neurology at the University Hospital of Santa Maria, Lisbon
measure QoL.
58
and an investigator at The Neurological Clinical Research Unit of the Institute of
Molecular Medicine, Lisbon. He has served as investigator for clinical trials in movement
Deep Brain Stimulation
disorders. He is the national co-ordinator of the Portuguese centres participating in the
EURO-Huntington´s Disease Network. Dr Mestre’s major research interests are
Deep brain stimulation (DBS) is largely replacing ablative surgery for
Parkinson’s disease, chorea, dystonia and botulinum toxin.
dystonia, particularly DBS of the globus pallidus internus (DBS-GPi). In
2003, the US Food and Drug Administration (FDA) approved the Activa
®
Joaquim J Ferreira is an Attending Physician in Neurology at the University Hospital of
Dystonia Therapy System under the Humanitarian Devices Exemption
Santa Maria, Lisbon and a Professor of Therapeutics and Neurology at the Lisbon
School of Medicine. He is also an investigator at the Neurological Clinical Research Unit
process for unilateral or bilateral stimulation of the GPi or the
of the Institute of Molecular Medicine, Lisbon and has served as principal investigator
subthalamic nucleus for the treatment of chronic and intractable (i.e. for multiple clinical trials in the field of movement disorders. In addition, he serves as
drug-refractory) primary dystonia (generalised, segmental, hemi- and
Chair of the Education Sub-committee of the European Movement Disorders Section
and Chair of the Liaison and Public Relations Committee of the Movement Disorders
cervical dystonia). Controlled studies of DBS in CD are not available, and
Society. Professor Ferreira’s major research interests are Parkinson’s disease, chorea,
most case series report on small-sized samples with heterogeneous dystonia, botulinum toxin and neuropharmacology. He obtained his medical degree
dystonic conditions. The Canadian multicentre, single-blind study
and performed neurological training at the University Hospital of Santa Maria, Lisbon.
prospectively evaluated 10 patients with disabling CD, and reported
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