Chikashi Fukaya, Yoichi Katayama, Toshikazu Kano, Takafumi Nagaoka, Kazutaka Kobayashi, Hideki Oshima and Takamitsu Yamamoto
Writer's cramp is a type of idiopathic focal hand dystonia characterized by muscle cramps that accompany execution of the writing task specifically. In this report, the authors describe the clinical outcome after thalamic deep brain stimulation (DBS) therapy in patients with writer's cramp and present an illustrative case with which they compare the effects of pallidal and thalamic stimulation. In addition to these results for the clinical effectiveness, they also examine the best point and pattern for therapeutic stimulation of the motor thalamus, including the nucleus ventrooralis (VO) and the ventralis intermedius nucleus (VIM), for writer's cramp.
The authors applied thalamic DBS in five patients with writer's cramp. The inclusion criteria for the DBS trial in this disorder were a diagnosis of idiopathic writer's cramp and the absence of a positive response to medication. The exclusion criteria included significant cognitive dysfunction, active psychiatric symptoms, and evidence of other central nervous system diseases or other medical disorders. In one of the cases, DBS leads were implanted into both the globus pallidus internus and the VO/VIM, and test stimulation was performed for 1 week. The authors thus had an opportunity to compare the effects of pallidal and thalamic stimulation in this patient.
Immediately after the initiation of thalamic stimulation, the neurological deficits associated with writer's cramp were improved in all five cases. Postoperatively all preoperative scale scores indicating the seriousness of the writer's cramp were significantly lower (p < 0.001). In the patient in whom two DBS leads were implanted, the clinical effect of thalamic stimulation was better than that of pallidal stimulation. During the thalamic stimulation, the maximum effect was obtained when stimulation was applied to both the VO and the VIM widely, compared with being applied only within the VO.
The authors successfully treated patients with writer's cramp by thalamic DBS. Insofar as they are aware, this is the first series in which writer's cramp has been treated with DBS. Thalamic stimulation appears to be a safe and valuable therapeutic option for writer's cramp.
Hideki Oshima, Yoichi Katayama, Chikashi Fukaya, Toshikazu Kano, Kazutaka Kobayashi, Takamitsu Yamamoto and Yutaka Suzuki
✓Beginning-of-dose motor deterioration (BDMD) is a complication of levodopa medications in Parkinson disease (PD) that is presumably caused by inhibitory effects of levodopa. Only limited experience of BDMD has been described in the literature. The authors report the case of a patient with PD who demonstrated a marked BDMD while being treated with standard levodopa medications. This 55-year-old woman had a 12-year history of PD and a 10-year history of levodopa treatment. Marked exacerbation of symptoms occurred 15 to 20 minutes after every dose of levodopa at 100 mg and lasted approximately 15 minutes. The PD symptoms, particularly tremor and rigidity, were exacerbated more markedly during this period than during the wearing-off deterioration. The BDMD could be controlled very well by subthalamic nucleus (STN) stimulation without any change in the regimen of levodopa medications. These observations suggest that the BDMD was inhibited by STN stimulation through a direct effect.
Kazutaka Kobayashi, Yoichi Katayama, Hideki Oshima, Mitsuru Watanabe, Koichiro Sumi, Toshiki Obuchi, Chikashi Fukaya and Takamitsu Yamamoto
Holmes' tremor (HT) is generally considered to be a symptomatic tremor associated with lesions of the cerebellum, midbrain, or thalamus. Deep brain stimulation (DBS) therapy for essential tremor and parkinsonian tremor has proved quite successful. In contrast, surgical treatment outcomes for HT have often been disappointing. The use of 2 ipsilateral DBS electrodes implanted in parallel within the thalamus for severe essential tremor has been reported. Since dual-lead stimulation within a single target can cover a wider area than single-lead stimulation, it produces greater effects. On the other hand, DBS of the subthalamic area (SA) was recently reported to be effective for refractory tremor.
The authors implanted 2 DBS electrodes (one at the nucleus ventralis oralis/nucleus ventralis intermedius and the other at the SA) in 4 patients with HT. For more than 2 years after implantation, each patient's tremor was evaluated using a tremor rating scale under the following 4 conditions of stimulation: “on” for both thalamus and SA DBS; “off” for both thalamus and SA DBS; “on” for thalamus and “off” for SA DBS; and “on” for SA and “off” for thalamus DBS.
The tremor in all patients was improved for more than 2 years (mean 25.8 ± 3.5 months). Stimulation with 2 electrodes exerted greater effect on the tremor than did 1-electrode stimulation. Interestingly, in all patients progressive effects were observed, and in one patient treated with DBS for 1 year, tremor did not appear even while stimulation was temporarily switched off, suggesting irreversible improvement effects.
The presence of both resting and intentional/action tremor implies combined destruction of the pallidothalamic and cerebellothalamic pathways in HT. A larger stimulation area may thus be required for HT patients. Multitarget, dual-lead stimulation permits coverage of the wide area needed to suppress the tremor without adverse effects of stimulation. Some reorganization of the neural network may be involved in the development of HT because the tremor appears several months after the primary insult. The mechanism underlying the absence of tremor while stimulation was temporarily off remains unclear, but the DBS may have normalized the abnormal neural network.
The authors successfully treated patients with severe HT by using dual-electrode DBS over a long period. Such DBS may offer an effective and safe treatment modality for intractable HT.
Hideki Oshima, Yoichi Katayama, Takashi Morishita, Koichiro Sumi, Toshiharu Otaka, Kazutaka Kobayashi, Yutaka Suzuki, Chikashi Fukaya and Takamitsu Yamamoto
The objective of this study was to evaluate the efficacy of chronic subthalamic nucleus (STN) stimulation for alleviating pain related to Parkinson disease (PD).
Among 163 consecutive patients undergoing STN stimulation, 69 were identified as experiencing pain preoperatively that was related to their PD. All 69 patients suffering from pain were followed up prospectively for 12 months after surgery. All patients described the severity of their pain according to a visual analog scale (VAS) preoperatively and at 2 weeks, 6 months, and 12 months postoperatively. Pain unrelated to PD was not studied.
Several types of pain related to PD, the categories of which were based on a modification of 2 previous classifications (Ford and Honey), can occur in such patients: 1) musculoskeletal pain, 2) dystonic pain, 3) somatic pain exacerbated by PD, 4) radicular/peripheral neuropathic pain, and 5) central pain. The overall mean VAS score was significantly decreased postoperatively by 75% and 69% at 2 weeks and 6 months, respectively (p < 0.001). The mean VAS score at 12 months was also decreased by 80%, but 6 instances of pain (3 reports of somatic back pain and 3 reports of radicular/peripheral neuropathic pain) required additional spinal surgery to alleviate the pain severity. The results were analyzed using the Wilcoxon signed-rank test and demonstrated a significant reduction in VAS scores at all follow-up assessments (p < 0.001). Musculoskeletal pain and dystonic pain were well alleviated by STN stimulation. In contrast, somatic pain exacerbated by PD and peripheral neuropathic pain originating from lumbar spinal diseases, such as spondylosis deformans and/or canal stenosis, often deteriorated postoperatively despite attenuation of the patients' motor disability. Patients with central pain were poor responders.
This study found that STN stimulation produced significant improvement of overall pain related to PD in patients with advanced PD, and the efficacy continued for at least 1 year. The present results indicate that musculoskeletal pain and dystonic pain responded well to STN stimulation, but patients with back pain (somatic pain) and radicular/peripheral neuropathic pain originating from spinal disease have a potential risk for postoperative deterioration of their pain.