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Hideki Oshima, Yoichi Katayama and Teruyasu Hirayama

Object. The collateral vessels in moyamoya disease appear to retain their ability to constrict during hypocapnia but not to dilate during hypercapnia. It has been claimed that hypercapnia, as well as hypocapnia, decreases the blood flow in regions perfused by collateral vessels, presumably because of intracerebral steal. If this holds true, the decrease in blood flow may be proportional to the global hyperemia in the brain. To establish appropriate hemodynamic control during revascularization surgery, the authors monitored the jugular bulb oxygen saturation (SjO2) intraoperatively, a method that could sensitively detect global hyperemia.

Methods. A total of 17 patients, most of whom presented with transient ischemic attacks or fluctuating neurological deficits, underwent intraoperative monitoring of their SjO2 and end-tidal carbon dioxide (ETCO2) after a state of anesthesia had been induced with isoflurane (Group 1) or propofol (Group 2). In eight of these patients, the regional cerebral blood flow (rCBF) of the collateral vessel territory was also monitored by laser Doppler flowmetry during the period of cortical exposure, and a total of 113 data sets (averaged values during 2.5-minute intervals) was collected. There was fluctuation in the ETCO2 levels ranging from 36 to 44 mm Hg. The mean SjO2 level was clearly greater (p < 0.01) in Group 1 (71.8 ± 2.2%) than in Group 2 (63.3 ± 2.1%). An episodic fall in rCBF was observed in association with a transient increase in SjO2. Such an event was not uncommon in Group 1 and there was a greater risk of rCBF decreasing when SjO2 exceeded a cutoff level of 76% (p < 0.01). This level could sometimes be reached at a broad range of ETCO2 readings (37–44 mm Hg). In Group 2, similar events sometimes occurred when SjO2 increased beyond 70%. However, this level could be reached only with a higher ETCO2 (42–44 mm Hg). The rCBF level was negatively correlated to SjO2 (p < 0.01), but not always to ETCO2, indicating that the episodic fall in rCBF was closely related to global hyperemia rather than the absolute level of hypercapnia.

Conclusions. The observed association between a fall in rCBF and global hyperemia supports the intracerebral steal hypothesis and indicates that it is prudent to avoid excessive global hyperemia. The optimal range of CO2 for isoflurane is more restricted than that for propofol, presumably because isoflurane induces hyperemia by itself. Monitoring of SjO2 appears to represent the most practical technique for detecting global hyperemia as well as global ischemia, both of which may cause ischemic complications in moyamoya disease.

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Tetsuya Takahata, Yoichi Katayama, Takashi Tsubokawa, Hideki Oshima and Atsuo Yoshino

✓ Intracranial ectopic pituitary adenoma occurs most frequently in the suprasellar cistern, usually in continuity with the pituitary stalk. Such tumors probably originate from cells of the pars tuberalis located above the diaphragma sellae or from aberrant anterior pituitary cells of the pituitary stalk. The authors report the case of a 37-year-old woman with Cushing's syndrome caused by an ectopic pituitary adenoma of unique location: the tumor was separate from the pituitary stalk and confined within the interpeduncular cistern. After surgical removal of the tumor, continued improvement in the patient's laboratory results and disappearance of her endocrine symptoms strongly indicated the absence of adenoma cells in the pituitary gland or stalk. The tumor in the present case appears to have arisen from aberrant pituitary cells that were present in the leptomeninges of the basal surface of the hypothalamus.

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Tomoya Yamashita, Hironobu Sakaura, Kazuya Oshima, Motoki Iwasaki and Hideki Yoshikawa

The authors report on the case of a 64-year-old man with solitary intradural extramedullary non-Hodgkin lymphoma of the cervical spine. The lesion mimicked the appearance of meningioma on MR imaging. Positron emission tomography showed increased accumulation of fluorine-18–labeled fluorodeoxyglucose only in the cervical lesion. Serum levels of C-reactive protein and soluble interleukin-2 receptor were mildly elevated. At surgery, the intradural tumor in the subarachnoid space was totally extirpated. Based on histopathological findings, diffuse, large B-cell type non-Hodgkin lymphoma was diagnosed. Postoperatively, the patient was treated with 2 courses of chemotherapy by intrathecal injection of methotrexate, cytarabine, and prednisolone and 4 courses of intravenous rituximab, an antibody binding to CD20 on the surface of B cells. All preoperative symptoms completely resolved after surgery. Two years postoperatively, the patient was faring well with no evidence of local recurrence or new lesions at any other site. To the best of the authors' knowledge, this case is the first reported instance of solitary intradural extramedullary non-Hodgkin lymphoma of the cervical spine.

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Takamitsu Yamamoto, Yoichi Katayama, Toshikazu Kano, Kazutaka Kobayashi, Hideki Oshima and Chikashi Fukaya

Object. The tremor-suppression effect resulting from long-term stimulation of the thalamic nucleus ventralis intermedius (Vim) and the nucleus ventralis oralis posterior (Vop) was examined in the treatment of parkinsonian, essential, and poststroke tremor.

Methods. After identifying the accurate anterior border of the nucleus ventrocaudalis (Vc), deep brain stimulation (DBS) electrodes with four contacts were inserted into the Vim—Vop region at an angle of between 40 and 50° from the horizontal plane of the anterior commissure—posterior commissure line. Two distal contacts were placed on the Vim side and two proximal contacts on the Vop side. The best sites of stimulation and parameters of bipolar stimulation were selected in each case and follow-up examinations were conducted for at least 2 years.

In all 15 cases of parkinsonian tremor (18 sides) and in 14 of 15 cases of essential tremor (24 of 25 sides), cathodal stimulation of the Vim side with anodal stimulation of the Vop side was determined to be the best choice to suppress the tremor. In poststroke tremor, however, six of 12 cases (six of 12 sides) were selected for cathodal stimulation of the Vop side with anodal stimulation of the Vim side. The average stimulation intensity 1 month after initiation of DBS was 1.61 V in cases of parkinsonian tremor, 1.99 V in cases of essential tremor, and 2.39 V in cases of poststroke tremor. A comparison of stimulation intensities required at 1 and 24 months after initiation of DBS revealed that the lowest effective stimulation intensity increased 24.2% in cases of parkinsonian tremor, 21% in cases of poststroke tremor, and 46.9% in cases of essential tremor. Suppression of tremor was achieved in all cases (42 cases, 55 sides) during a period of 2 years. Nevertheless, two cases of poststroke tremor required dual-lead stimulation at the unilateral Vim—Vop region from the start of DBS, and two cases of essential tremor and one case of poststroke tremor required a stimulation intensity that was high enough to evoke unpleasant paresthesia and slight motor contraction during the follow-up period.

Conclusions. Effective stimulation sites and stimulation intensities differ in different kinds of tremor; Vim and Vop stimulation is necessary in many cases. Interactions of the Vim and Vop under the control of interconnected areas of the motor circuitry may play an important role in both the development and DBS-induced suppression of tremor.

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Takamitsu Yamamoto, Yoichi Katayama, Chikashi Fukaya, Hideki Oshima, Masahiko Kasai and Kazutaka Kobayashi

✓ Reversibility and adaptability are preferred features of long-term therapeutic deep brain stimulation (DBS). In such therapy, a permanent stimulating electrode with four contact points is placed at the stimulation site and, generally speaking, bipolar stimulation is induced by various pairs of adjacent contact points on one electrode. The stimulation sites are thus all located along the trajectory of the implanted electrode. In a patient with unilateral severe essential tremor, the authors implanted two electrodes side by side and parallel to each other in the unilateral thalamic ventralis intermedius nucleus. Using these electrodes, the authors were able to deliver current flow not only along the electrode trajectory, but also between the two electrodes in a direction parallel to the anterior commissure—posterior commissure line. Although individual stimulations, delivered by each of the two electrodes using all parameters and all stimulation points, were unable to stop the patient's tremor completely without adverse effects, the new stimulation method, in which electrical currents passed between the two electrodes, effected complete abolition of the tremor without adverse effects. With the aid of this method, one can use two electrodes, implanted in parallel and side by side, to achieve maximum efficacy and to reduce adverse effects in some instances of DBS therapy.

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Takamitsu Yamamoto, Yoichi Katayama, Kazutaka Kobayashi, Hideki Oshima and Chikashi Fukaya

✓ Using a new perforator, the authors have developed a new dual-floor burr-hole method for use in deep brain stimulation therapy. The modification is called “dual-floor” because the usual 15-mm-diameter burr hole, which is located centrally and reaches the dura mater, is surrounded by a 4-mm-wide rim that is also planed downward by the new perforator to a depth of 4 mm. This dual-floor burr hole is adjusted to fit the burr-hole ring and cap that are are supplied by the electrode manufacturer. Such a method eliminates bulging of the scalp just over the burr-hole ring and cap. In addition, it is helpful for securing a tight fixation between the burr-hole ring and the skull.

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Yoichi Katayama, Masahiko Kasai, Hideki Oshima, Chikashi Fukaya, Takamitsu Yamamoto, Katsuhiko Ogawa and Tomohiko Mizutani

Object. A blinded evaluation of the effects of subthalamic nucleus (STN) stimulation was performed in levodopaintolerant patients with Parkinson disease (PD). These patients (Group I, seven patients) were moderately or severely disabled (Hoehn and Yahr Stages III–V during the off period), but were receiving only a small dose of medication (levodopa-equivalent dose [LED] 0–400 mg/day) because they suffered unbearable side effects. The results were analyzed in comparison with those obtained in patients with advanced PD (Group II, seven patients) who were severely disabled (Hoehn and Yahr Stages IV and V during the off period), but were treated with a large dose of medication (500–990 mg/day).

Methods. The patients were evaluated twice at 6 to 8 months after surgery. To determine the actual benefits afforded by STN stimulation to their overall daily activities, the patients were maintained on their medication regimen with optimal doses and schedules. Stimulation was turned off overnight for at least 12 hours. It was turned on in the morning (or remained turned off), and each patient's best and worst scores on the Unified Parkinson's Disease Rating Scale during waking daytime activity were recorded as on- and off-period scores, respectively. The order of assessment with respect to whether stimulation was occurring was determined randomly.

The STN stimulation markedly improved daily activity and total motor scores in Group I patients. The percentage time of immobility (Hoehn and Yahr Stages IV and V) became 0% in patients who were intermittently immobile while not receiving stimulation. Improvements were demonstrated in tremor, rigidity, akinesia, and gait subscores. The STN stimulation produced less marked but still noticeable improvements in the daily activity and total motor scores in Group II patients. The percentage time of immobility as well as the LED was reduced in patients who displayed intermittent immobility with pronounced motor fluctuations while not receiving stimulation. Improvements were demonstrated in tremor, rigidity, and dyskinesia subscores in these patients. In contrast, STN stimulation did not improve the overall daily activities at all in patients who had become unresponsive to a tolerable dose of levodopa and were continuously immobile, even though these patients' tremor and rigidity subscores were still improved by stimulation.

Conclusions. Consistent with earlier findings, the great benefit of STN stimulation in levodopa-intolerant patients is that STN stimulation can reduce the level of required levodopa medication. This suggests that STN stimulation could be a therapeutic option for patients with less-advanced PD by allowing levodopa medication to be maintained at as low a dose as possible, and to prevent adverse reactions to the continued use of large-dose levodopa.

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Chikashi Fukaya, Yoichi Katayama, Toshikazu Kano, Takafumi Nagaoka, Kazutaka Kobayashi, Hideki Oshima and Takamitsu Yamamoto

Object

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.

Methods

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.

Results

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.

Conclusions

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.